Morphometric Measurements of Intraoral Anatomy in Children with Beckwith-Wiedemann Syndrome: A Prospective Study and Novel Approach

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Romeo, Andrew M. George, Jonathan H. Sussman, Manisha Banala, and 7 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-4132656/v1 This work is licensed under a CC BY 4.0 License Status: Published Journal Publication published 17 Oct, 2024 Read the published version in Orphanet Journal of Rare Diseases → Version 1 posted 5 You are reading this latest preprint version Abstract Objectives An easy-to-use tool to objectively measure intraoral anatomy with meaningful clinical correlations may improve care for patients with Beckwith-Wiedemann syndrome (BWS), who commonly have symptomatic macroglossia. This study aims to address this gap by assessing correlations between tongue morphometrics and clinical characteristics including BWS clinical score, percentage mosaicism, BWS index of macroglossia (BIG) score, sleep apnea, and surgical incidence in patients with BWS. Methods Children aged 2–17 years with BWS were prospectively enrolled between 12/2021-01/2024. Digital intraoral photographs with a laser ruler were taken, and morphometric measurements were made using ImageJ software. Relationships between morphometrics and outcomes including BWS clinical score, percentage mosaicism, and incidence of tongue reduction surgery were examined using t-tests and multivariate linear models. Results Pharyngeal morphometric measurements were obtained in 49 patients with BWS. Mouth area, width, and height differed across BWS molecular subtypes (p < 0.05). Right-to-left tongue width (p = 0.022) and mouth width (p = 0.049) were larger in those with loss of methylation at imprinting control region 2 (IC2 LOM) than other BWS variants. Patients with paternal uniparental isodisomy of chromosome 11p15 (pUPD11) had narrower mouths than others (p = 0.040). Those with tongue reduction surgery had more tongue ridging than those without surgery (p < 0.01). There were correlations between mouth area and BWS clinical score (p = 0.046), tongue width and BWS clinical score (p = 0.040), and tongue length and percentage mosaicism (p = 0.012). Conclusion Intraoral morphometric measurements are associated with phenotypic burden in BWS. Tongue morphology varies across the BWS spectrum, with IC2 LOM having wider tongues and mouths, and pUPD11 having narrower mouths. Tongue ridging is more common in those selected for surgery. Intraoral morphometric measurements may be safely obtained at low costs across centers caring for children with BWS or others at risk of upper airway obstruction. Beckwith-Wiedemann Syndrome BWS macroglossia morphometrics Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Introduction Beckwith-Wiedemann syndrome (BWS) is a congenital overgrowth and cancer predisposition disorder caused by (epi)genetic changes on chromosome 11p15 1–3 that presents with macroglossia in roughly 85% of patients. 4 Macroglossia can negatively affect patients, 1,2,5–10 and is often treated by a tongue reduction surgery, which has been shown to improve symptoms. 11–15 The decision to perform surgery on these patients is not standardized across centers, 10,16,17 and more data are needed to better understand macroglossia in BWS. A scoring system was developed to quantify phenotypic severity in BWS, 2,7 under which patients were assigned two points for “cardinal” features such as macroglossia and one point for “suggestive” features such as macrosomia. Confirmatory blood testing is recommended when patients have at least one cardinal feature, and those with scores of four or more including a cardinal feature are clinically diagnosed with BWS. 2 Patients diagnosed with BWS undergo methylation analysis of either blood or an affected tissue 18 that examines imprinting control regions 1 and 2 (IC1 and IC2) on chromosome 11p15, CDKN1C gene analysis, and/or copy number analysis or chromosome microarray analysis. 2,7 Roughly 50% of patients with molecularly confirmed BWS have a loss of methylation at IC2 (IC2 LOM), 7,19 5–10% have gain of methylation at IC1 (IC1 GOM), 20% have paternal uniparental isodisomy of chromosome 11p15 (pUPD11), 5% have CDKN1C variants, and less than 5% have other chromosomal abnormalities in the 11p15 region. 7,20,21 BWS is a mosaic disease, meaning that patients have a mixture of unaffected cells and those with genetic or epigenetic changes. 6,7,19,22–26 Blood mosaicism data can provide useful information about overall disease burden and can be obtained non-invasively then compared across patients with different molecular causes of BWS. 18 A common complication and surgical indication in patients with macroglossia and BWS is obstructive sleep apnea (OSA), 27 which has an estimated prevalence of 48%. 28 Upper airway soft tissue enlargement exacerbates breathing difficulties, 29–32 and puts these patients at risk for OSA. When measured on magnetic resonance imaging (MRI) and/or computed tomography (CT), larger tongues and increased pharyngeal soft tissue volume impart an increased risk of developing OSA. 30,31,33–35 However, MRI and CT scans are expensive, often require sedation in pediatric populations, and expose patients to radiation, 36–39 making them impractical screening tools for pediatric patients with BWS who rarely have indications for cranial imaging. A low-cost and easy-to-use way to objectively measure the tongue with meaningful clinical correlations would improve risk stratification in patients with BWS. Recently, digital morphometric measurements using a laser ruler have been shown to quantify tongue size, airway visibility, and Mallampati scores in controls and patients with obstructive sleep apnea (OSA). 40 However, the digital morphometric measurements have not been used to quantify macroglossia severity in pediatric patients or in those with BWS. This study aims to address this gap by assessing correlations between tongue morphometrics and clinical characteristics including BWS clinical score, percentage mosaicism, BWS index of macroglossia (BIG) score, sleep apnea, and surgical incidence in patients with BWS. We hypothesize that increased morphometric measurements are associated with more severe clinical outcomes in patients with BWS. Methods This was a single-center prospective cohort study. Institutional review board approval (IRB 13-010658) and consent from each patient were obtained. Children with Beckwith-Wiedemann syndrome able to sit upright who were seen by the team geneticist (JMK) for regular clinical visits were prospectively enrolled in this study. Demographic variables and history of tongue reduction surgery were recorded. Blood methylation testing was reviewed, and the BWS molecular subtypes which were recorded as either 11p15 duplication, CDKN1C variant, genome-wide paternal uniparental isodisomy (GWpUPD), IC1 GOM, IC2 LOM, and pUPD11. Patients with a clinical diagnosis of BWS were labeled accordingly. A BWS clinical score was calculated according to the International Consensus Scoring system. 7 Blood mosaicism percentage was calculated using established methodology (Supplementary Table 2) in eligible patients. 18,41 Beckwith-Wiedemann syndrome index (BIG) scores were recorded in both the pre- and post-surgical cohorts. Polysomnography variables included evaluation age, obstructive apnea-hypopnea index (OAHI), and oxyhemoglobin saturation (SpO 2 ) nadir. Polysomnographic data that were obtained prior to tongue reduction surgery were excluded. Previously, our team analyzed 459 patients with BWS and developed a macroglossia severity scoring system, the BWS Index of macroGlossia (BIG) (manuscript under review). Patients were classified from BIG0 to BIG3, where BIG0 included those without macroglossia; BIG1 included those with macroglossia not protruding beyond the teeth/alveolus; BIG2 included those with tongue protrusion past the teeth/alveolus to the lips but that can be contained within the mouth; and BIG3 included those with tongues that protrude beyond the teeth/alveolus and lips but that cannot be closed within the mouth. BIG score was found to have significant correlations with phenotypic severity and tongue reduction surgical incidence. To obtain photographs for morphometric measures, each subject was seated with their head in a neutral position and line of sight parallel to the floor, as has been validated. 42 A camera and laser ruler were placed roughly one meter from subjects who were instructed to open their mouths. Images were obtained using a Canon Rebel EOS T3 digital camera and an intraoral laser composed of a right angle beam splitter and mirror that were aligned such that the two parallel beams projected forward 1 cm apart, as has been described (Fig. 1 ). 40 A camera was attached to the laser ruler and digital photographs were taken to capture the laser beams near anatomical regions of interest for further study. The distance between the lasers was used to calculate quantitative measures from the photographs. Three intraoral photographs were taken, two anteriorly and one laterally. The two anterior positions included open mouth no phonation (OMNP), tongue extended maximally anterior (TEMA), and tongue extended maximally lateral (TEML). Regarding OMNP, the distance between lasers (scale), mouth area (MArea), mouth width (MWidth), mouth height (MHeight), and tongue width (TWidth) were measured. For TEMA, distance between lasers (scale), mouth width (MWidth), tongue area (TArea), tongue width (TWidth), and tongue length (TLength) were measured. Regarding TEML, distance between laser (scale), tongue area (TArea), tongue curvature (TCurve), tongue length (TLength), and tongue thickness (TThick) were measured (Fig. 2 ). Categorical measures of pharyngeal airway visibility, Mallampati scores, tongue ridging, tongue curvature, tonsil hypertrophy grade, and pharyngeal narrowing were also obtained. Morphometric measurements were calculated using ImageJ software (Fig. 3 ). 43 Relationships among morphometric measurements, BWS clinical score, level of affected cells in the blood, BIG score, polysomnography findings, and surgical incidence were examined using R v4.3.2. Continuous variables were reported as median (interquartile range) or mean ± SD and compared using Student’s t-tests or analysis of variance analysis. Categorical variables were compared using Chi-square tests. Pearson’s correlation and multivariate linear models were utilized to analyze associations between continuous variables. Multivariate logistic regression was used to model risk of surgery. Statistical tests used for each comparison are indicated in the corresponding tables and figures. P-values less than 0.05 were considered statistically significant. Results Forty-nine children with Beckwith-Wiedemann syndrome underwent the morphometric measurements. All patients (100%) had at least one photograph able to be analyzed on ImageJ. The cohort was comprised of 25 (51%) males and 24 (49%) females with a median (interquartile range) age of 6.13 (4.66–7.85) years. BWS diagnoses were: 1 (1.9%) 11p15 duplication, 2 (3.7%) CDKN1C variants, 3 (5.6%) GWpUPD, 3 (5.6%) IC1 GOM, 21 (38.9%) IC2 LOM, 15 (27.8%) pUPD11, and 9 (16.7%) clinical diagnoses. The median BWS clinical score was 8 ( 2 – 10 ) and the median blood mosaicism percentage was 57% (51–64%). Regarding BWS index of macroglossia scores (BIG), 13 (36.1%) patients were assigned a BIG0, 10 (27.8%) were BIG1, 12 (33.3%) were BIG2, and 1 (2.8%) was BIG3. The OAHI was 3.9 events/hour (1.8–7.0) and SpO 2 nadir was 86% (85–90). Nineteen patients (36.5%) underwent tongue reduction surgery at a median age of 1.54 (0.82–0.90) years old. The time range between surgery and morphometric measurements was 5.28 (3.27–8.06) years. All categorical and quantitative morphometric measurements were recorded (Table 1 ). Table 1 Demographic and morphometric measurement overview Category Quantification Category Quantification Category Quantification Patients 49 Grade Ridging (n = 53) TEMA Tongue Visibility (n = 53) Morphometric Measurements 54 0 16 (30.2%) 1 32 (60.4%) Sex 1 29 (54.7%) 2 1 (1.9%) Male 25 (51.0%) 2 6 (11.3%) 3 19 (35.8%) Female 24 (49.0%) 3 2 (3.8%) 4 1 (1.9%) Age (years) 6.13 (4.66–7.85) Tonsil Grade (n = 16) TEMA Laser Quality (n = 53) BMI (kg/m 2 ) 17.74 (16.02–18.96) 0 6 (37.5%) 1 52 (98.1%) BWS Diagnosis (n = 54) 1 2 (12.5%) 3 1 (1.9%) 11p15 Duplication 1 (1.9%) 2 3 (18.8%) TEML Tongue Quality (n = 29) CDKN1C 2 (3.7%) 3 5 (31.2%) 1 28 (96.6%) GWpUPD 3 (5.6%) Lateral Wall Grade (n = 16) 2 1 (3.4%) IC1 GOM 3 (5.6%) 1 1 (6.2%) TEML Tongue Angle (n = 29) IC2 LOM 21 (38.9%) 2 12 (75.0%) 1 22 (75.9%) pUPD11 15 (27.8%) 3 3 (18.8%) 2 1 (3.4%) Clinical 9 (16.7%) OMNP Mallampati Score (n = 8) 3 6 (20.7%) BWS Clinical Score (n = 49) 8.00 (2.00–10.00) 1 2 (25.0%) TEML Laser Quality (n = 29) Percent Mosaicism (n = 21) 57% (51–64%) 2 5 (62.5%) 1 29 (100.0%) BIG Score 3 1 (12.5%) OMNP 0 13 (36.1%) OMNP Tongue Visibility (n = 42) Scale (n = 42) 290.7 (253.9–330) 1 10 (27.8%) 1 1 (2.4%) MArea (n = 34) 1084000 (820300–1355000) 2 12 (33.3%) 2 1 (2.4%) MWidth (n = 40) 1205 (1058–1430) 3 1 (2.8%) 3 27 (64.3%) MHeight (n = 35) 1138 (945.9–1305) Polysomnography (n = 18) 4 13 (31.0%) TWidth (n = 27) 1110 (934.3–1232) AHI (n = 18) 3.85 (1.82–7.04) OMNP Laser Quality (n = 42) TEMA SpO2 (n = 18) 0.86 (0.85–0.90) 1 42 (100.0%) Scale (n = 53) 277 (236.3–319) Prior Glossectomy TEMA Mallampati Score (n = 16) MWidth (n = 48) 1256 (1085–1344) Yes 19 (36.5%) 1 1 (6.2%) TArea (n = 33) 1075000 (803700–1443000) Age (years; n = 16) 1.54 (0.82–2.48) 2 13 (81.2%) TWidth (n = 53) 1072 (894–1210) No 33 (63.5%) 3 2 (12.5%) TLength (n = 32) 1214 (1009–1414) TEMA Tongue Quality (n = 52) TEML 1 46 (88.5%) Scale (n = 29) 259 (233.1–309) 2 3 (5.8%) TArea (n = 28) 452300 (376900–590600) 3 1 (1.9%) TCurve (n = 27) 1564 (1317–1686) 4 2 (3.8%) TLength (n = 27) 1158 (1045–1295) TThick (n = 28) 478.8 (437.3–608.5) Values are reported as median (interquartile range) BMI: body mass index, BWS: Beckwith-Wiedemann Syndrome, BIG: BWS Index of Macroglossia, AHI: apnea hypoxia index, SpO 2 nadir: oxygen saturation nadir, OMNP: open mouth no phonation, TEMA: tongue extended maximally anterior, TEML: tongue extended maximally lateral Examining morphometric measurements showed that in the open mouth no phonation photographs (OMNP), MArea, MWidth, and MHeight differed across the various molecular diagnoses (p 0.05). Further subgroup analysis revealed that in the TEMA view, tongue width (p = 0.023; Fig. 4 ) and mouth width (p = 0.049; Fig. 5 ) were significantly larger in the IC2 LOM cohort than the remaining patients. Additionally, those with pUPD11 had significantly narrower mouths than others (p = 0.040). Table 2 Associations between molecular diagnoses and various clinical outcomes IC1 GOM (n = 3) IC2 LOM (n = 21) pUPD11 (n = 15) CDKN1C (n = 2) GWpUPD (n = 3) 11p15 Duplication (n = 1) Clinical Diagnosis (n = 9) p BWS Clinical Score 3.333 ± 5.774 8.333 ± 3.23 5.80 ± 4.99 4.00 ± 5.66 2.33 ± 4.04 0 7.00 ± 0.82 0.7 Percent Mosaicism 55 ± 1% 55 ± 17% 66 ± 7% - - - - 0.137 BIG Score 0 ± 0 1.769 ± 0.5991 1.00 ± 0.94 1 0.33 ± 0.58 1 **0.004 AHI 10.9 4.79 ± 5.34 5.63 ± 4.34 3.9 - - 4.05 ± 1.74 0.759 SpO 2 0.79 0.84 ± 0.06 0.87 ± 0.06 0.95 - - 0.88 ± 0.03 0.296 Ridging Grade 1 ± 1 1.19 ± 0.8729 0.79 ± 0.43 0.00 ± 0.00 1.00 ± 1.00 1 0.44 ± 0.53 0.334 Tonsil Grade - 1.111 ± 1.269 1.67 ± 1.53 3 0 2 3 0.645 Lateral Wall Grade 2 2.25 ± 0.4629 2.00 ± 0.00 1.50 ± 0.71 - 3 2 0.206 OMNP Mallampati - 1.8 ± 0.8367 2.00 2 - 2 - 0.824 Scale 263.53 ± 21.92 277.35 ± 75.18 303.9 ± 64.34 301.8 ± 31.46 357.8 ± 13.01 327.03 256.9 ± 66.86 0.566 MArea 9.285E + 5 ± 2.628E + 5 1.0400E + 6 ± 5.522E + 5 1.055E + 6 ± 4.289E + 5 8.388E + 5 8.187E + 6 ± 9.823E + 6 2.999E + 7 9.035E + 6 ± 4.594E + 6 **<0.0001 MWidth 1128 ± 56.82 1219 ± 369.9 1226 ± 266.0 1070 1526 ± 398.5 2740 896.9 ± 538.0 **0.00059 MHeight 1030 ± 276.8 1152 ± 477.3 1114 ± 246.0 1384 ± 524.3 1339 2918 971.8 ± 337.4 **0.0112 TWidth 934.3 ± 46.27 1009 ± 403.9 1113 ± 299.8 1102 ± 170.7 1363 ± 328.1 1627 953.6 ± 288.6 0.518 TEMA Mallampati 2 ± 0 2 ± 0.5774 2.33 ± 0.58 2 - 2 2.00 ± 0.00 0.977 Scale 228.7 ± 75.79 290.4 ± 86.73 245.0 ± 58.05 330.7 ± 34.90 258.2 ± 53.06 201.68 278.7 ± 62.30 0.35 MWidth 944.7 ± 201.6 1318 ± 366.9 1054 ± 229.7 1499 ± 211.2 1217 ± 56.63 1195 1210 ± 310.0 0.154 TArea 1.085E + 6 ± 2.407E + 4 1.072E + 6 ± 5.504E + 6 1.109E + 6 ± 5.282E + 5 1.397E + 6 ± 1.578E + 5 8.281E + 5 ± 1.355E + 5 9.750E + 5 1.194E + 06 ± 4.861E + 05 0.947 TWidth 827.2 ± 192.0 1179 ± 349.1 942.0 ± 263.3 1154 ± 42.12 992.3 ± 156.2 1470 1022 ± 229.1 0.135 TLength 1324 ± 110.8 1088 ± 380.6 1269 ± 391.8 1446 ± 218.3 1104 1298 1309 ± 398.8 0.835 TEML Scale 258.6 ± 0.56 267.4 ± 59.36 255.6 ± 59.36 281.0 ± 39.6 311.1 204.5 274.3 ± 53.99 0.884 TArea 4.035E + 5 ± 8.446E + 4 4.610E + 5 ± 2.220E + 5 4.896E + 5 ± 1.704E + 5 4.841E + 5 ± 1.519E + 5 3.986E + 5 5.110E + 5 7.632E + 5 ± 3.007E + 5 0.481 TCurve 1564 1365 ± 326.8 1560 ± 340 1542 ± 446 1422 1844 1965 ± 367.9 0.236 TLength 1190 ± 68.54 1063 ± 389.0 1199 ± 179.8 1340 ± 343.5 1184 1466 1423 ± 270.5 0.588 TThick 407.6 ± 57.44 532.0 ± 161.1 496.0 ± 80.99 442.2 ± 2.044 440.4 427.8 652.7 ± 158.5 0.472 BMI: body mass index, BWS: Beckwith-Wiedemann Syndrome, BIG: BWS Index of Macroglossia, AHI: apnea hypoxia index, SpO 2 nadir: oxygen saturation nadir, OMNP: open mouth no phonation, TEMA: tongue extended maximally anterior, TEML: tongue extended maximally lateral Values are reported as mean ± standard deviation Red: analysis of variance analysis Blue: chi-squared test Comparing those who had tongue reduction surgery to those who did not, revealed more ridging in individuals with surgery (1.37 ± 0.76 vs. 0.62 ± 0.61, p < 0.01). There was no difference in any morphometric measurement between those who had surgery in the last five years and those who had it longer than five years ago. Additionally, tongues of those who had surgery had less curvature than those in patients who did not have surgery (p = 0.01). Patients who underwent tongue reduction surgery also had higher BIG scores (1.86 ± 0.53 vs. 0.50 ± 0.67, p < 0.01) and BWS clinical scores (9.21 ± 2.80 vs. 4.79 ± 4.31, p < 0.01) than those who did not (Table 3 ). Table 3 Outcome variance between surgery and non-surgery cohorts History of Tongue Reduction Surgery No Yes p BWS Clinical Score 4.79 ± 4.31 9.21 ± 2.80 **0.009 Percent Mosaicism 64 ± 8% 57 ± 15% 0.210 BIG Score 0.50 ± 0.67 1.86 ± 0.53 **<0.001 AHI 6.59 ± 3.80 4.07 ± 4.62 0.232 SpO 2 0.87 ± 0.05 0.85 ± 0.06 0.411 Ridging Grade 0.62 ± 0.61 1.37 ± 0.76 **0.008 Tonsil Grade 1.78 ± 1.39 1.00 ± 1.15 0.268 Lateral Wall Grade 2.00 ± 0.47 2.33 ± 0.52 0.411 OMNP Mallampati 1.75 ± 0.50 2.00 ± 0.82 0.549 Scale 288.2 ± 57.97 278.7 ± 82.94 0.674 MArea 3.120E + 6 ± 7.081E + 6 1.097E + 6 ± 5.831E + 5 0.315 MWidth 1242 ± 494.8 1208 ± 386 0.816 MHeight 1162 ± 506.3 1212 ± 494.8 0.775 TWidth 1064 ± 319.7 1064 ± 397.4 1 TEMA Mallampati 2.10 ± 0.32 2.00 ± 0.63 0.359 Scale 272.2 ± 62.8 267.2 ± 93.48 0.82 MWidth 1190 ± 275.8 1213 ± 403.9 0.817 Tarea 1.090E + 6 ± 3.907E + 5 1.154E + 6 ± 7.253E + 5 0.754 TWidth 1018 ± 235.6 1141 ± 399.4 0.175 TLength 1255 ± 330.3 1131 ± 469.7 0.431 TEML Scale 270.7 ± 43.09 251.2 ± 62.09 0.335 TArea 5.252E + 5 ± 1.470E + 5 4.104E + 5 ± 1.983E + 5 0.096 TCurve 1621 ± 259 1320 ± 320 **0.014 TLength 1232 ± 168 1062 ± 407.8 0.144 TThick 518.6 ± 125.1 485.6 ± 131.3 0.511 AHI: apnea-hypoxia index, SpO 2 : oxygen saturation nadir, BWS: Beckwith-Wiedmann Syndrome, BIG: BWS Index of Macroglossia, OMNP: open mouth no phonation, TEMA: tongue extended maximally anterior, TEML: tongue extended maximally lateral All values reported as mean ± standard deviation Red: two-sampled Student’s t-test Blue: chi-squared test Correlation analysis showed significant correlations between OMNP mouth area and BWS clinical score (p = 0.046). Additionally, there were correlations between TEMA tongue width and BWS clinical score (p = 0.040), TEMA tongue length and percentage mosaicism (p = 0.012), as well as between TEML tongue curvature and percentage mosaicism (p = 0.045; Table 4 ). Between the two independent image raters (AMG and MB) assessing categorical variables in photographs, there was a high degree of accuracy (64%) that was significantly greater than random chance (p = 0.007). For continuous variables, a Pearson’s correlation also revealed a high agreement between raters (p < 0.001). Table 4 Linear models controlling for age at morphometric measurements and body mass index AHI SpO2 Percent Mosaicism BIG Score BWS Clinical Score Coefficient p Coefficient p Coefficient p Coefficient p Coefficient p BIG Score 3.78E + 00 -6.07E-02 0.230 -3.09E-02 0.569 - - 4.15E + 00 < 0.0001 AHI - - -9.92E-03 0. 003 3.08E-02 0.039 4.91E-02 0.286 2.10E-01 0.256 SpO2 -4.74E + 01 0.003 - - -1.94E + 00 0.163 -3.78E + 00 0.230 -2.42E + 01 0.047 Percent Mosaicism 2.25E + 01 0.039 -2.18E-01 0.163 - - -1.09E + 00 0.569 -6.55E + 00 0.505 BWS Clinical Score 4.34E-01 0.256 -1.05E-02 0.047 -4.06E-03 0.505 1.43E-01 < 0.0001 - - OMNP MArea -4.30E-07 0.868 2.29E-08 0.618 -2.10E-08 0.765 -1.89E-07 0.100 -3.74E-07 0.046 MWidth -7.68E-04 0.879 1.91E-05 0.784 -3.41E-05 0.811 5.60E-04 0.300 -4.84E-04 0.820 MHeight 1.29E-03 0.749 -3.23E-05 0.587 -5.11E-05 0.676 6.78E-04 0.173 -5.72E-04 0.738 TWidth -7.97E-04 0.849 -2.64E-05 0.754 -8.12E-05 0.490 3.41E-04 0.723 1.24E-03 0.648 TEMA TArea -3.69E-06 0.359 -3.56E-08 0.641 7.85E-08 0.236 2.01E-07 0.581 1.70E-06 0.361 TWidth 2.07E-03 0.618 -1.05E-04 0.076 -2.01E-05 0.822 6.82E-04 0.175 4.36E-03 0.040 TLength -5.20E-03 0.208 -4.31E-06 0.958 1.89E-04 0.012 -1.57E-04 0.742 2.38E-04 0.921 MWidth -9.42E-04 0.873 -9.26E-05 0.293 -3.64E-05 0.736 3.04E-04 0.527 2.07E-03 0.345 TEPM TArea 1.03E-05 0.241 9.69E-08 0.448 5.73E-07 0.140 -3.00E-07 0.812 -9.85E-07 0.810 TCurve 5.05E-03 0.273 4.04E-05 0.538 4.20E-04 0.045 -6.83E-04 0.324 -2.06E-03 0.403 TLength 4.48E-03 0.327 1.88E-05 0.777 9.54E-05 0.560 -3.28E-04 0.645 -2.47E-03 0.339 TThick 2.51E-02 0.099 2.83E-05 0.904 9.80E-04 0.088 5.13E-04 0.812 4.44E-03 0.483 Discussion BWS commonly presents with macroglossia, but objectively quantifying the severity of macroglossia is challenging. This prospective study demonstrates that easily obtained morphometric measurements using a laser and digital camera can objectively assess the intraoral anatomy of children with BWS and can provide meaningful clinical insights. The findings presented here add distinctions to emerging discussions about the spectrum of intraoral morphology across the different BWS molecular diagnoses. Finally, this study highlights that characteristics such as tongue ridging are more common in patients who had tongue reduction surgery than in those who did not. Many studies have examined associations between intraoral anatomy and clinical outcomes, but these often require expensive equipment, use calibers which are difficult to maneuver in the mouth, are inaccurate, or exclude pediatric populations. 40,42,44–49 It is important to characterize soft tissue morphology and risk factors in children predisposed to diseases such as those with BWS or OSA. Still, objective soft tissue data are classically obtained via CT or MR imaging, exposing children to radiation or requiring sedation, and are therefore usually not obtained. 36–39 In their landmark study of 860 patients (542 with OSA and 318 controls), Schwab et. al demonstrated that tongue morphometric measurements using a digital camera and laser ruler provided accurate and reproducible measurements of the tongue and mouth that were significantly associated with polysomnographic outcomes. 40 However, this previous study did not include children or patients with BWS, limiting its generalizability. Our study builds on this previous work by showing that tongue and mouth measurements are significantly correlated by genetic subtype and with measures of phenotypic severity in BWS including clinical score and percentage mosaicism. This information is useful because while BWS clinical scores allow objective assessment of phenotypic burden, obtaining this information requires comprehensive patient assessment. Further, the BWS clinical scoring system views macroglossia as a dichotomous variable that is either present or absent, failing to account for the range of macroglossia across patients with BWS. 7 Additionally, while the BIG scores proposed by our group correlate with clinical outcomes such as surgical incidence, this system did not involve objective measurements of the oral anatomy and may be challenging to reproduce across clinical settings. The morphometric findings presented here reveal correlations between oral measurements, BWS clinical scores, and BIG scores, offering a quick and reproducible technique to quantify macroglossia severity in patients with BWS. Obtaining morphometric measurements is inexpensive, does not require sedation, and can be performed in most clinical settings. Children are advised to sit down with their tongues extended for a few seconds while photographs are taken. Images from the three positions are captured, and the entire process takes less than five minutes to complete. As we obtained these images, we found that children as young as two years of age can sit long enough for accurate photographs to be taken. Previously, obtaining tongue morphometric measurements had been shown to be feasible in adults, but not in children. This demonstrates that this modality can augment care not only for children with BWS, but more broadly in all patients at risk of upper airway obstruction, such as in children with Down Syndrome or obesity. 50,51 Assessing these photographs in patients with BWS, we found different morphological features across the various molecular diagnoses. It is well established that the incidence of certain clinical features is higher in some BWS molecular subtypes than other. For instance, patients with IC2 LOM more commonly present with omphalocele, patients with pUPD11 more often have lateralized overgrowth, and most of the patients with IC2 LOM have macroglossia. 6 There is little published literature reporting measurable differences in tongue morphology across those with different BWS molecular subtypes. Our study shows that there are objective differences between the intraoral anatomical measurements between the different BWS molecular subtypes. Anecdotally, we have observed that those with IC2 LOM tend to have wide and long tongues, and patients with pUPD11 appear to have thick and wide tongues. Testing this hypothesis, we observed that those with IC2 LOM had wider tongues and mouths than others. Interestingly, we did not find a difference between the tongue width or thickness in patients with pUPD11 compared to others with BWS (p > 0.05), though we found that they had smaller mouths than others. A recent study from our center assessed a large cohort of patients who underwent tongue reduction surgery, finding that those with pUPD11 made up a large proportion of patients selected for surgery, and that they more commonly had repeat surgery than patients with other BWS subtypes. 14 We initially speculated that a reason for these findings could be larger tongues in those with pUPD11, but morphometrics data demonstrated that patients with pUPD11 did not have larger tongues in the cohort presented here. A possible explanation for the discrepancy between our expectations and measurements is that we were underpowered to make statistically significant conclusions in this patient population using morphometrics. Another possibility is that tongue morphology alone is only one of many factors contributing to the need for surgery, and that the entire clinical picture of patients is needed to determine if a patient requires surgical intervention. At our center, all patients with Beckwith-Wiedemann syndrome are seen by the team geneticist (JMK) who orders molecular testing and assigns both a BWS clinical score and BIG score. As part of our macroglossia research evaluation, patients also have tongue morphometric photographs captured. Those with symptomatic macroglossia are referred to the pediatric plastic surgeon, with approximately 25% of referred patients determined to be surgical candidates. Surgery is considered when patients have a primary indication of breathing and feeding concerns (typically in younger patients) or craniodental development concerns (typically in those older than 1 year of age). Comparing the morphometric measurements between patients who had surgery to those who did not, we found that tongues were more ridged in individuals with a surgical history. Ridging, which is primarily a measure of posterior lingual crowding, is likely unchanged pre- and postoperatively given that the posterior tongue is not surgically excised. Therefore, the finding of an association between increased ridging and surgical selection may be useful information for surgeons considering intervention. Specifically, if minimal ridging is present, then surgeons may be less inclined to consider surgical intervention, and vice versa. There are several limitations to this study. First, sleep studies and morphometric measurements were conducted at different times throughout patients’ treatment courses, but when we controlled for age between polysomnography and morphometric measurements, there was minimal change in outcomes. Further, we used morphometric measurement age when conducting statistical analysis. Additionally, we do not currently have pre- and post-tongue reduction morphometric data on patients, and this will be an area of future study. Conclusion Objective intraoral morphometric measurements are associated with phenotypic burden in BWS. Tongue morphology varies across the BWS spectrum, with IC2 LOM presenting with wider tongues and mouths, and pUPD11 having narrower mouths. Tongue ridging is more common in those selected for surgery. These measurements are easy to obtain and may be adopted at low costs across centers caring for children with BWS or others at risk of upper airway obstruction. Declarations Ethics approval and consent to participate: institutional review board approval (IRB 13-010658) and consent from each patient were obtained. Consent for publication: all patients have provided consent for the sharing of their photographs in this manuscript. Availability of data and materials: available on reasonable request. Competing interests: none. Funding: none. Authors' contributions: DJR participated in the conception and design of the project, data analysis, interpretation of data, drafting of the manuscript, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. AMG participated in the conception and design of the project, interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. JHS participated in the conception and design of the project, interpretation of data, drafting of the manuscript, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work MB participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. AW participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. MW participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. JJN participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. JAT participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. RJS participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. CMC participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. JMK participated in conception and design of the project, interpretation of data, drafting of the manuscript, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work. Financial Disclosure Statement: The authors declare no conflicts of interest. Acknowledgements: This work is supported by the Victoria Fertitta Fund through the Lorenzo “Turtle” Sartini Jr. Endowed Chair in Beckwith–Wiedemann Syndrome Research (JMK). Data availability: Data used in this study are not openly available due to reasons of sensitivity and patient privacy but are available from the corresponding author upon reasonable request. These data are in a controlled data storage drive at the Children’s Hospital of Philadelphia. 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Obesity and obstructive sleep apnea in children. Paediatr Respir Rev. 2006;7(4):247–59. 10.1016/j.prrv.2006.08.003 . Cite Share Download PDF Status: Published Journal Publication published 17 Oct, 2024 Read the published version in Orphanet Journal of Rare Diseases → Version 1 posted Editorial decision: Major revision 15 Jul, 2024 Reviewers agreed at journal 05 Jul, 2024 Reviewers invited by journal 19 May, 2024 Editor assigned by journal 03 Apr, 2024 First submitted to journal 26 Mar, 2024 You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-4132656","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":304240744,"identity":"c5f53bb7-4329-43b6-90f9-1f24011e78cc","order_by":0,"name":"Dominic J. 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legend\u003c/p\u003e","description":"","filename":"5.png","url":"https://assets-eu.researchsquare.com/files/rs-4132656/v1/e61dc59cf570074cadac77f9.png"},{"id":67149084,"identity":"50606922-a364-4e7a-9fbe-662c38e0ac85","added_by":"auto","created_at":"2024-10-21 16:11:46","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":59998887,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4132656/v1/fc3bef0a-b41f-448e-b2f6-045ab93480e8.pdf"}],"financialInterests":"","formattedTitle":"Morphometric Measurements of Intraoral Anatomy in Children with Beckwith-Wiedemann Syndrome: A Prospective Study and Novel Approach","fulltext":[{"header":"Introduction","content":"\u003cp\u003eBeckwith-Wiedemann syndrome (BWS) is a congenital overgrowth and cancer predisposition disorder caused by (epi)genetic changes on chromosome 11p15\u003csup\u003e1\u0026ndash;3\u003c/sup\u003e that presents with macroglossia in roughly 85% of patients.\u003csup\u003e4\u003c/sup\u003e Macroglossia can negatively affect patients,\u003csup\u003e1,2,5\u0026ndash;10\u003c/sup\u003e and is often treated by a tongue reduction surgery, which has been shown to improve symptoms.\u003csup\u003e11\u0026ndash;15\u003c/sup\u003e The decision to perform surgery on these patients is not standardized across centers,\u003csup\u003e10,16,17\u003c/sup\u003e and more data are needed to better understand macroglossia in BWS.\u003c/p\u003e \u003cp\u003eA scoring system was developed to quantify phenotypic severity in BWS,\u003csup\u003e2,7\u003c/sup\u003e under which patients were assigned two points for \u0026ldquo;cardinal\u0026rdquo; features such as macroglossia and one point for \u0026ldquo;suggestive\u0026rdquo; features such as macrosomia. Confirmatory blood testing is recommended when patients have at least one cardinal feature, and those with scores of four or more including a cardinal feature are clinically diagnosed with BWS.\u003csup\u003e2\u003c/sup\u003e Patients diagnosed with BWS undergo methylation analysis of either blood or an affected tissue\u003csup\u003e18\u003c/sup\u003e that examines imprinting control regions 1 and 2 (IC1 and IC2) on chromosome 11p15, \u003cem\u003eCDKN1C\u003c/em\u003e gene analysis, and/or copy number analysis or chromosome microarray analysis.\u003csup\u003e2,7\u003c/sup\u003e Roughly 50% of patients with molecularly confirmed BWS have a loss of methylation at IC2 (IC2 LOM),\u003csup\u003e7,19\u003c/sup\u003e 5\u0026ndash;10% have gain of methylation at IC1 (IC1 GOM), 20% have paternal uniparental isodisomy of chromosome 11p15 (pUPD11), 5% have \u003cem\u003eCDKN1C\u003c/em\u003e variants, and less than 5% have other chromosomal abnormalities in the 11p15 region.\u003csup\u003e7,20,21\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eBWS is a mosaic disease, meaning that patients have a mixture of unaffected cells and those with genetic or epigenetic changes.\u003csup\u003e6,7,19,22\u0026ndash;26\u003c/sup\u003e Blood mosaicism data can provide useful information about overall disease burden and can be obtained non-invasively then compared across patients with different molecular causes of BWS.\u003csup\u003e18\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eA common complication and surgical indication in patients with macroglossia and BWS is obstructive sleep apnea (OSA),\u003csup\u003e27\u003c/sup\u003e which has an estimated prevalence of 48%.\u003csup\u003e28\u003c/sup\u003e Upper airway soft tissue enlargement exacerbates breathing difficulties,\u003csup\u003e29\u0026ndash;32\u003c/sup\u003e and puts these patients at risk for OSA. When measured on magnetic resonance imaging (MRI) and/or computed tomography (CT), larger tongues and increased pharyngeal soft tissue volume impart an increased risk of developing OSA.\u003csup\u003e30,31,33\u0026ndash;35\u003c/sup\u003e However, MRI and CT scans are expensive, often require sedation in pediatric populations, and expose patients to radiation,\u003csup\u003e36\u0026ndash;39\u003c/sup\u003e making them impractical screening tools for pediatric patients with BWS who rarely have indications for cranial imaging.\u003c/p\u003e \u003cp\u003eA low-cost and easy-to-use way to objectively measure the tongue with meaningful clinical correlations would improve risk stratification in patients with BWS. Recently, digital morphometric measurements using a laser ruler have been shown to quantify tongue size, airway visibility, and Mallampati scores in controls and patients with obstructive sleep apnea (OSA).\u003csup\u003e40\u003c/sup\u003e However, the digital morphometric measurements have not been used to quantify macroglossia severity in pediatric patients or in those with BWS. This study aims to address this gap by assessing correlations between tongue morphometrics and clinical characteristics including BWS clinical score, percentage mosaicism, BWS index of macroglossia (BIG) score, sleep apnea, and surgical incidence in patients with BWS. We hypothesize that increased morphometric measurements are associated with more severe clinical outcomes in patients with BWS.\u003c/p\u003e"},{"header":"Methods","content":"\u003cp\u003eThis was a single-center prospective cohort study. Institutional review board approval (IRB 13-010658) and consent from each patient were obtained. Children with Beckwith-Wiedemann syndrome able to sit upright who were seen by the team geneticist (JMK) for regular clinical visits were prospectively enrolled in this study. Demographic variables and history of tongue reduction surgery were recorded. Blood methylation testing was reviewed, and the BWS molecular subtypes which were recorded as either 11p15 duplication, \u003cem\u003eCDKN1C\u003c/em\u003e variant, genome-wide paternal uniparental isodisomy (GWpUPD), IC1 GOM, IC2 LOM, and pUPD11. Patients with a clinical diagnosis of BWS were labeled accordingly. A BWS clinical score was calculated according to the International Consensus Scoring system.\u003csup\u003e7\u003c/sup\u003e Blood mosaicism percentage was calculated using established methodology (Supplementary Table\u0026nbsp;2) in eligible patients.\u003csup\u003e18,41\u003c/sup\u003e Beckwith-Wiedemann syndrome index (BIG) scores were recorded in both the pre- and post-surgical cohorts. Polysomnography variables included evaluation age, obstructive apnea-hypopnea index (OAHI), and oxyhemoglobin saturation (SpO\u003csub\u003e2\u003c/sub\u003e) nadir. Polysomnographic data that were obtained prior to tongue reduction surgery were excluded.\u003c/p\u003e \u003cp\u003e Previously, our team analyzed 459 patients with BWS and developed a macroglossia severity scoring system, the BWS Index of macroGlossia (BIG) (manuscript under review). Patients were classified from BIG0 to BIG3, where BIG0 included those without macroglossia; BIG1 included those with macroglossia not protruding beyond the teeth/alveolus; BIG2 included those with tongue protrusion past the teeth/alveolus to the lips but that can be contained within the mouth; and BIG3 included those with tongues that protrude beyond the teeth/alveolus and lips but that cannot be closed within the mouth. BIG score was found to have significant correlations with phenotypic severity and tongue reduction surgical incidence.\u003c/p\u003e \u003cp\u003eTo obtain photographs for morphometric measures, each subject was seated with their head in a neutral position and line of sight parallel to the floor, as has been validated.\u003csup\u003e42\u003c/sup\u003e A camera and laser ruler were placed roughly one meter from subjects who were instructed to open their mouths. Images were obtained using a Canon Rebel EOS T3 digital camera and an intraoral laser composed of a right angle beam splitter and mirror that were aligned such that the two parallel beams projected forward 1 cm apart, as has been described (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e).\u003csup\u003e40\u003c/sup\u003e A camera was attached to the laser ruler and digital photographs were taken to capture the laser beams near anatomical regions of interest for further study. The distance between the lasers was used to calculate quantitative measures from the photographs.\u003c/p\u003e \u003cp\u003eThree intraoral photographs were taken, two anteriorly and one laterally. The two anterior positions included open mouth no phonation (OMNP), tongue extended maximally anterior (TEMA), and tongue extended maximally lateral (TEML). Regarding OMNP, the distance between lasers (scale), mouth area (MArea), mouth width (MWidth), mouth height (MHeight), and tongue width (TWidth) were measured. For TEMA, distance between lasers (scale), mouth width (MWidth), tongue area (TArea), tongue width (TWidth), and tongue length (TLength) were measured. Regarding TEML, distance between laser (scale), tongue area (TArea), tongue curvature (TCurve), tongue length (TLength), and tongue thickness (TThick) were measured (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). Categorical measures of pharyngeal airway visibility, Mallampati scores, tongue ridging, tongue curvature, tonsil hypertrophy grade, and pharyngeal narrowing were also obtained. Morphometric measurements were calculated using ImageJ software (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e).\u003csup\u003e43\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eRelationships among morphometric measurements, BWS clinical score, level of affected cells in the blood, BIG score, polysomnography findings, and surgical incidence were examined using R v4.3.2. Continuous variables were reported as median (interquartile range) or mean \u0026plusmn; SD and compared using Student\u0026rsquo;s t-tests or analysis of variance analysis. Categorical variables were compared using Chi-square tests. Pearson\u0026rsquo;s correlation and multivariate linear models were utilized to analyze associations between continuous variables. Multivariate logistic regression was used to model risk of surgery. Statistical tests used for each comparison are indicated in the corresponding tables and figures. P-values less than 0.05 were considered statistically significant.\u003c/p\u003e"},{"header":"Results","content":"\u003cp\u003eForty-nine children with Beckwith-Wiedemann syndrome underwent the morphometric measurements. All patients (100%) had at least one photograph able to be analyzed on ImageJ. The cohort was comprised of 25 (51%) males and 24 (49%) females with a median (interquartile range) age of 6.13 (4.66\u0026ndash;7.85) years. BWS diagnoses were: 1 (1.9%) 11p15 duplication, 2 (3.7%) \u003cem\u003eCDKN1C\u003c/em\u003e variants, 3 (5.6%) GWpUPD, 3 (5.6%) IC1 GOM, 21 (38.9%) IC2 LOM, 15 (27.8%) pUPD11, and 9 (16.7%) clinical diagnoses. The median BWS clinical score was 8 (\u003cspan additionalcitationids=\"CR3 CR4 CR5 CR6 CR7 CR8 CR9\" citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e) and the median blood mosaicism percentage was 57% (51\u0026ndash;64%). Regarding BWS index of macroglossia scores (BIG), 13 (36.1%) patients were assigned a BIG0, 10 (27.8%) were BIG1, 12 (33.3%) were BIG2, and 1 (2.8%) was BIG3. The OAHI was 3.9 events/hour (1.8\u0026ndash;7.0) and SpO\u003csub\u003e2\u003c/sub\u003e nadir was 86% (85\u0026ndash;90). Nineteen patients (36.5%) underwent tongue reduction surgery at a median age of 1.54 (0.82\u0026ndash;0.90) years old. The time range between surgery and morphometric measurements was 5.28 (3.27\u0026ndash;8.06) years. All categorical and quantitative morphometric measurements were recorded (Table\u0026nbsp;\u003cspan refid=\"Tab1\" class=\"InternalRef\"\u003e1\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab1\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 1\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eDemographic and morphometric measurement overview\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"6\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c6\" colnum=\"6\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCategory\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eQuantification\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eCategory\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eQuantification\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eCategory\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c6\"\u003e \u003cp\u003eQuantification\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePatients\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e49\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u003cb\u003eGrade Ridging (n\u0026thinsp;=\u0026thinsp;53)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e\u003cb\u003eTEMA Tongue Visibility (n\u0026thinsp;=\u0026thinsp;53)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMorphometric Measurements\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e54\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e16 (30.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e32 (60.4%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSex\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e29 (54.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1 (1.9%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e25 (51.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e6 (11.3%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e19 (35.8%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eFemale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e24 (49.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2 (3.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e4\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1 (1.9%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAge (years)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e6.13 (4.66\u0026ndash;7.85)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u003cb\u003eTonsil Grade (n\u0026thinsp;=\u0026thinsp;16)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e\u003cb\u003eTEMA Laser Quality (n\u0026thinsp;=\u0026thinsp;53)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBMI (kg/m\u003csup\u003e2\u003c/sup\u003e)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e17.74 (16.02\u0026ndash;18.96)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e6 (37.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e52 (98.1%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBWS Diagnosis (n\u0026thinsp;=\u0026thinsp;54)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2 (12.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1 (1.9%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e11p15 Duplication\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1 (1.9%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e3 (18.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e\u003cb\u003eTEML Tongue Quality (n\u0026thinsp;=\u0026thinsp;29)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCDKN1C\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2 (3.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e5 (31.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e28 (96.6%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eGWpUPD\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3 (5.6%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e\u003cb\u003eLateral Wall Grade (n\u0026thinsp;=\u0026thinsp;16)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1 (3.4%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eIC1 GOM\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3 (5.6%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1 (6.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e\u003cb\u003eTEML Tongue Angle (n\u0026thinsp;=\u0026thinsp;29)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eIC2 LOM\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e21 (38.9%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e12 (75.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e22 (75.9%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003epUPD11\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e15 (27.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e3 (18.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1 (3.4%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eClinical\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e9 (16.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e\u003cb\u003eOMNP Mallampati Score (n\u0026thinsp;=\u0026thinsp;8)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e6 (20.7%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBWS Clinical Score (n\u0026thinsp;=\u0026thinsp;49)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e8.00 (2.00\u0026ndash;10.00)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2 (25.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e\u003cb\u003eTEML Laser Quality (n\u0026thinsp;=\u0026thinsp;29)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePercent Mosaicism (n\u0026thinsp;=\u0026thinsp;21)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e57% (51\u0026ndash;64%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e5 (62.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e29 (100.0%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBIG Score\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1 (12.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e\u003cb\u003eOMNP\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e13 (36.1%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e\u003cb\u003eOMNP Tongue Visibility (n\u0026thinsp;=\u0026thinsp;42)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eScale (n\u0026thinsp;=\u0026thinsp;42)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e290.7 (253.9\u0026ndash;330)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e10 (27.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1 (2.4%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eMArea (n\u0026thinsp;=\u0026thinsp;34)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1084000 (820300\u0026ndash;1355000)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e12 (33.3%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1 (2.4%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eMWidth (n\u0026thinsp;=\u0026thinsp;40)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1205 (1058\u0026ndash;1430)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1 (2.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e27 (64.3%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eMHeight (n\u0026thinsp;=\u0026thinsp;35)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1138 (945.9\u0026ndash;1305)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePolysomnography (n\u0026thinsp;=\u0026thinsp;18)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e4\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e13 (31.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eTWidth (n\u0026thinsp;=\u0026thinsp;27)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1110 (934.3\u0026ndash;1232)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAHI (n\u0026thinsp;=\u0026thinsp;18)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3.85 (1.82\u0026ndash;7.04)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e\u003cb\u003eOMNP Laser Quality (n\u0026thinsp;=\u0026thinsp;42)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e\u003cb\u003eTEMA\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSpO2 (n\u0026thinsp;=\u0026thinsp;18)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0.86 (0.85\u0026ndash;0.90)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e42 (100.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eScale (n\u0026thinsp;=\u0026thinsp;53)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e277 (236.3\u0026ndash;319)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePrior Glossectomy\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e\u003cb\u003eTEMA Mallampati Score (n\u0026thinsp;=\u0026thinsp;16)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eMWidth (n\u0026thinsp;=\u0026thinsp;48)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1256 (1085\u0026ndash;1344)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eYes\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e19 (36.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1 (6.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eTArea (n\u0026thinsp;=\u0026thinsp;33)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1075000 (803700\u0026ndash;1443000)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAge (years; n\u0026thinsp;=\u0026thinsp;16)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1.54 (0.82\u0026ndash;2.48)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e13 (81.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eTWidth (n\u0026thinsp;=\u0026thinsp;53)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1072 (894\u0026ndash;1210)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNo\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e33 (63.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2 (12.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eTLength (n\u0026thinsp;=\u0026thinsp;32)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1214 (1009\u0026ndash;1414)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e\u003cb\u003eTEMA Tongue Quality (n\u0026thinsp;=\u0026thinsp;52)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e\u003cb\u003eTEML\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e46 (88.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eScale (n\u0026thinsp;=\u0026thinsp;29)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e259 (233.1\u0026ndash;309)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e3 (5.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eTArea (n\u0026thinsp;=\u0026thinsp;28)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e452300 (376900\u0026ndash;590600)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1 (1.9%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eTCurve (n\u0026thinsp;=\u0026thinsp;27)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1564 (1317\u0026ndash;1686)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e4\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2 (3.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eTLength (n\u0026thinsp;=\u0026thinsp;27)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1158 (1045\u0026ndash;1295)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eTThick (n\u0026thinsp;=\u0026thinsp;28)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e478.8 (437.3\u0026ndash;608.5)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"6\" nameend=\"c6\" namest=\"c1\"\u003e \u003cp\u003e\u003cem\u003eValues are reported as median (interquartile range)\u003c/em\u003e\u003c/p\u003e \u003cp\u003e\u003cem\u003eBMI: body mass index, BWS: Beckwith-Wiedemann Syndrome, BIG: BWS Index of Macroglossia, AHI: apnea hypoxia index, SpO\u003c/em\u003e\u003csub\u003e\u003cem\u003e2\u003c/em\u003e\u003c/sub\u003e \u003cem\u003enadir: oxygen saturation nadir, OMNP: open mouth no phonation, TEMA: tongue extended maximally anterior, TEML: tongue extended maximally lateral\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003eExamining morphometric measurements showed that in the open mouth no phonation photographs (OMNP), MArea, MWidth, and MHeight differed across the various molecular diagnoses (p\u0026thinsp;\u0026lt;\u0026thinsp;0.05; Table\u0026nbsp;\u003cspan refid=\"Tab2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). Similar trends were observed in tongue and mouth width in the TEMA view, though significance was not achieved (p\u0026thinsp;\u0026gt;\u0026thinsp;0.05). Further subgroup analysis revealed that in the TEMA view, tongue width (p\u0026thinsp;=\u0026thinsp;0.023; Fig.\u0026nbsp;\u003cspan refid=\"Fig5\" class=\"InternalRef\"\u003e4\u003c/span\u003e) and mouth width (p\u0026thinsp;=\u0026thinsp;0.049; Fig.\u0026nbsp;\u003cspan refid=\"Fig6\" class=\"InternalRef\"\u003e5\u003c/span\u003e) were significantly larger in the IC2 LOM cohort than the remaining patients. Additionally, those with pUPD11 had significantly narrower mouths than others (p\u0026thinsp;=\u0026thinsp;0.040).\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab2\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 2\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eAssociations between molecular diagnoses and various clinical outcomes\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"9\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c6\" colnum=\"6\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c7\" colnum=\"7\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c8\" colnum=\"8\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c9\" colnum=\"9\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eIC1 GOM (n\u0026thinsp;=\u0026thinsp;3)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eIC2 LOM (n\u0026thinsp;=\u0026thinsp;21)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003epUPD11 (n\u0026thinsp;=\u0026thinsp;15)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eCDKN1C (n\u0026thinsp;=\u0026thinsp;2)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c6\"\u003e \u003cp\u003eGWpUPD (n\u0026thinsp;=\u0026thinsp;3)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c7\"\u003e \u003cp\u003e11p15 Duplication (n\u0026thinsp;=\u0026thinsp;1)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c8\"\u003e \u003cp\u003eClinical Diagnosis (n\u0026thinsp;=\u0026thinsp;9)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c9\"\u003e \u003cp\u003e\u003cem\u003ep\u003c/em\u003e\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBWS Clinical Score\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3.333\u0026thinsp;\u0026plusmn;\u0026thinsp;5.774\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e8.333\u0026thinsp;\u0026plusmn;\u0026thinsp;3.23\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e5.80\u0026thinsp;\u0026plusmn;\u0026thinsp;4.99\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e4.00\u0026thinsp;\u0026plusmn;\u0026thinsp;5.66\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e2.33\u0026thinsp;\u0026plusmn;\u0026thinsp;4.04\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e7.00\u0026thinsp;\u0026plusmn;\u0026thinsp;0.82\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.7\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePercent Mosaicism\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e55\u0026thinsp;\u0026plusmn;\u0026thinsp;1%\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e55\u0026thinsp;\u0026plusmn;\u0026thinsp;17%\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e66\u0026thinsp;\u0026plusmn;\u0026thinsp;7%\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e\u003cb\u003e0.137\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBIG Score\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0\u0026thinsp;\u0026plusmn;\u0026thinsp;0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.769\u0026thinsp;\u0026plusmn;\u0026thinsp;0.5991\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1.00\u0026thinsp;\u0026plusmn;\u0026thinsp;0.94\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e0.33\u0026thinsp;\u0026plusmn;\u0026thinsp;0.58\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e\u003cb\u003e**0.004\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAHI\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e10.9\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e4.79\u0026thinsp;\u0026plusmn;\u0026thinsp;5.34\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e5.63\u0026thinsp;\u0026plusmn;\u0026thinsp;4.34\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e3.9\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e4.05\u0026thinsp;\u0026plusmn;\u0026thinsp;1.74\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.759\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSpO\u003csub\u003e2\u003c/sub\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0.79\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0.84\u0026thinsp;\u0026plusmn;\u0026thinsp;0.06\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.87\u0026thinsp;\u0026plusmn;\u0026thinsp;0.06\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0.95\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e0.88\u0026thinsp;\u0026plusmn;\u0026thinsp;0.03\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.296\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eRidging Grade\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1\u0026thinsp;\u0026plusmn;\u0026thinsp;1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.19\u0026thinsp;\u0026plusmn;\u0026thinsp;0.8729\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.79\u0026thinsp;\u0026plusmn;\u0026thinsp;0.43\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0.00\u0026thinsp;\u0026plusmn;\u0026thinsp;0.00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1.00\u0026thinsp;\u0026plusmn;\u0026thinsp;1.00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e0.44\u0026thinsp;\u0026plusmn;\u0026thinsp;0.53\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.334\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTonsil Grade\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.111\u0026thinsp;\u0026plusmn;\u0026thinsp;1.269\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1.67\u0026thinsp;\u0026plusmn;\u0026thinsp;1.53\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e 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colname=\"c9\"\u003e \u003cp\u003e0.206\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOMNP\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMallampati\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.8\u0026thinsp;\u0026plusmn;\u0026thinsp;0.8367\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2.00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.824\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eScale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e263.53\u0026thinsp;\u0026plusmn;\u0026thinsp;21.92\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e 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colname=\"c2\"\u003e \u003cp\u003e9.285E\u0026thinsp;+\u0026thinsp;5\u0026thinsp;\u0026plusmn;\u0026thinsp;2.628E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.0400E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;5.522E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1.055E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;4.289E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e8.388E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e8.187E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;9.823E\u0026thinsp;+\u0026thinsp;6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e2.999E\u0026thinsp;+\u0026thinsp;7\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e9.035E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;4.594E\u0026thinsp;+\u0026thinsp;6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e\u003cb\u003e**\u0026lt;0.0001\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMWidth\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1128\u0026thinsp;\u0026plusmn;\u0026thinsp;56.82\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1219\u0026thinsp;\u0026plusmn;\u0026thinsp;369.9\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1226\u0026thinsp;\u0026plusmn;\u0026thinsp;266.0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1070\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1526\u0026thinsp;\u0026plusmn;\u0026thinsp;398.5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e2740\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e896.9\u0026thinsp;\u0026plusmn;\u0026thinsp;538.0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e\u003cb\u003e**0.00059\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMHeight\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1030\u0026thinsp;\u0026plusmn;\u0026thinsp;276.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1152\u0026thinsp;\u0026plusmn;\u0026thinsp;477.3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1114\u0026thinsp;\u0026plusmn;\u0026thinsp;246.0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1384\u0026thinsp;\u0026plusmn;\u0026thinsp;524.3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1339\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e2918\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e971.8\u0026thinsp;\u0026plusmn;\u0026thinsp;337.4\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e\u003cb\u003e**0.0112\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTWidth\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e934.3\u0026thinsp;\u0026plusmn;\u0026thinsp;46.27\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1009\u0026thinsp;\u0026plusmn;\u0026thinsp;403.9\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1113\u0026thinsp;\u0026plusmn;\u0026thinsp;299.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1102\u0026thinsp;\u0026plusmn;\u0026thinsp;170.7\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1363\u0026thinsp;\u0026plusmn;\u0026thinsp;328.1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e1627\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e953.6\u0026thinsp;\u0026plusmn;\u0026thinsp;288.6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.518\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTEMA\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMallampati\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2\u0026thinsp;\u0026plusmn;\u0026thinsp;0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2\u0026thinsp;\u0026plusmn;\u0026thinsp;0.5774\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2.33\u0026thinsp;\u0026plusmn;\u0026thinsp;0.58\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e2.00\u0026thinsp;\u0026plusmn;\u0026thinsp;0.00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.977\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eScale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e228.7\u0026thinsp;\u0026plusmn;\u0026thinsp;75.79\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e290.4\u0026thinsp;\u0026plusmn;\u0026thinsp;86.73\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e245.0\u0026thinsp;\u0026plusmn;\u0026thinsp;58.05\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e330.7\u0026thinsp;\u0026plusmn;\u0026thinsp;34.90\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e258.2\u0026thinsp;\u0026plusmn;\u0026thinsp;53.06\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e201.68\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e278.7\u0026thinsp;\u0026plusmn;\u0026thinsp;62.30\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.35\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMWidth\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e944.7\u0026thinsp;\u0026plusmn;\u0026thinsp;201.6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1318\u0026thinsp;\u0026plusmn;\u0026thinsp;366.9\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1054\u0026thinsp;\u0026plusmn;\u0026thinsp;229.7\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1499\u0026thinsp;\u0026plusmn;\u0026thinsp;211.2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1217\u0026thinsp;\u0026plusmn;\u0026thinsp;56.63\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e1195\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e1210\u0026thinsp;\u0026plusmn;\u0026thinsp;310.0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.154\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTArea\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1.085E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;2.407E\u0026thinsp;+\u0026thinsp;4\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.072E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;5.504E\u0026thinsp;+\u0026thinsp;6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1.109E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;5.282E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1.397E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;1.578E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e8.281E\u0026thinsp;+\u0026thinsp;5\u0026thinsp;\u0026plusmn;\u0026thinsp;1.355E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e9.750E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e1.194E\u0026thinsp;+\u0026thinsp;06\u0026thinsp;\u0026plusmn;\u0026thinsp;4.861E\u0026thinsp;+\u0026thinsp;05\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.947\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTWidth\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e827.2\u0026thinsp;\u0026plusmn;\u0026thinsp;192.0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1179\u0026thinsp;\u0026plusmn;\u0026thinsp;349.1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e942.0\u0026thinsp;\u0026plusmn;\u0026thinsp;263.3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1154\u0026thinsp;\u0026plusmn;\u0026thinsp;42.12\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e992.3\u0026thinsp;\u0026plusmn;\u0026thinsp;156.2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e1470\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e1022\u0026thinsp;\u0026plusmn;\u0026thinsp;229.1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.135\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTLength\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1324\u0026thinsp;\u0026plusmn;\u0026thinsp;110.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1088\u0026thinsp;\u0026plusmn;\u0026thinsp;380.6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1269\u0026thinsp;\u0026plusmn;\u0026thinsp;391.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1446\u0026thinsp;\u0026plusmn;\u0026thinsp;218.3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1104\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e1298\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e1309\u0026thinsp;\u0026plusmn;\u0026thinsp;398.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.835\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTEML\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eScale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e258.6\u0026thinsp;\u0026plusmn;\u0026thinsp;0.56\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e267.4\u0026thinsp;\u0026plusmn;\u0026thinsp;59.36\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e255.6\u0026thinsp;\u0026plusmn;\u0026thinsp;59.36\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e281.0\u0026thinsp;\u0026plusmn;\u0026thinsp;39.6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e311.1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e204.5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e274.3\u0026thinsp;\u0026plusmn;\u0026thinsp;53.99\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.884\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTArea\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e4.035E\u0026thinsp;+\u0026thinsp;5\u0026thinsp;\u0026plusmn;\u0026thinsp;8.446E\u0026thinsp;+\u0026thinsp;4\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e4.610E\u0026thinsp;+\u0026thinsp;5\u0026thinsp;\u0026plusmn;\u0026thinsp;2.220E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e4.896E\u0026thinsp;+\u0026thinsp;5\u0026thinsp;\u0026plusmn;\u0026thinsp;1.704E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e4.841E\u0026thinsp;+\u0026thinsp;5\u0026thinsp;\u0026plusmn;\u0026thinsp;1.519E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e3.986E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e5.110E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e7.632E\u0026thinsp;+\u0026thinsp;5\u0026thinsp;\u0026plusmn;\u0026thinsp;3.007E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.481\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTCurve\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1564\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1365\u0026thinsp;\u0026plusmn;\u0026thinsp;326.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1560\u0026thinsp;\u0026plusmn;\u0026thinsp;340\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1542\u0026thinsp;\u0026plusmn;\u0026thinsp;446\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1422\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e1844\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e1965\u0026thinsp;\u0026plusmn;\u0026thinsp;367.9\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.236\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTLength\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1190\u0026thinsp;\u0026plusmn;\u0026thinsp;68.54\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1063\u0026thinsp;\u0026plusmn;\u0026thinsp;389.0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1199\u0026thinsp;\u0026plusmn;\u0026thinsp;179.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e1340\u0026thinsp;\u0026plusmn;\u0026thinsp;343.5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e1184\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e1466\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e1423\u0026thinsp;\u0026plusmn;\u0026thinsp;270.5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.588\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTThick\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e407.6\u0026thinsp;\u0026plusmn;\u0026thinsp;57.44\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e532.0\u0026thinsp;\u0026plusmn;\u0026thinsp;161.1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e496.0\u0026thinsp;\u0026plusmn;\u0026thinsp;80.99\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e442.2\u0026thinsp;\u0026plusmn;\u0026thinsp;2.044\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e440.4\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e427.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e652.7\u0026thinsp;\u0026plusmn;\u0026thinsp;158.5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.472\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"9\" nameend=\"c9\" namest=\"c1\"\u003e \u003cp\u003e\u003cem\u003eBMI: body mass index, BWS: Beckwith-Wiedemann Syndrome, BIG: BWS Index of Macroglossia, AHI: apnea hypoxia index, SpO\u003c/em\u003e\u003csub\u003e\u003cem\u003e2\u003c/em\u003e\u003c/sub\u003e \u003cem\u003enadir: oxygen saturation nadir, OMNP: open mouth no phonation, TEMA: tongue extended maximally anterior, TEML: tongue extended maximally lateral\u003c/em\u003e\u003c/p\u003e \u003cp\u003e\u003cem\u003eValues are reported as mean\u0026thinsp;\u0026plusmn;\u0026thinsp;standard deviation\u003c/em\u003e\u003c/p\u003e \u003cp\u003eRed: analysis of variance analysis\u003c/p\u003e \u003cp\u003eBlue: chi-squared test\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003eComparing those who had tongue reduction surgery to those who did not, revealed more ridging in individuals with surgery (1.37\u0026thinsp;\u0026plusmn;\u0026thinsp;0.76 vs. 0.62\u0026thinsp;\u0026plusmn;\u0026thinsp;0.61, p\u0026thinsp;\u0026lt;\u0026thinsp;0.01). There was no difference in any morphometric measurement between those who had surgery in the last five years and those who had it longer than five years ago. Additionally, tongues of those who had surgery had less curvature than those in patients who did not have surgery (p\u0026thinsp;=\u0026thinsp;0.01). Patients who underwent tongue reduction surgery also had higher BIG scores (1.86\u0026thinsp;\u0026plusmn;\u0026thinsp;0.53 vs. 0.50\u0026thinsp;\u0026plusmn;\u0026thinsp;0.67, p\u0026thinsp;\u0026lt;\u0026thinsp;0.01) and BWS clinical scores (9.21\u0026thinsp;\u0026plusmn;\u0026thinsp;2.80 vs. 4.79\u0026thinsp;\u0026plusmn;\u0026thinsp;4.31, p\u0026thinsp;\u0026lt;\u0026thinsp;0.01) than those who did not (Table\u0026nbsp;\u003cspan refid=\"Tab3\" class=\"InternalRef\"\u003e3\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab3\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 3\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eOutcome variance between surgery and non-surgery cohorts\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"4\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colspan=\"3\" nameend=\"c4\" namest=\"c2\"\u003e \u003cp\u003eHistory of Tongue Reduction Surgery\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e\u003cb\u003eNo\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u003cb\u003eYes\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003ep\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBWS Clinical Score\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e4.79\u0026thinsp;\u0026plusmn;\u0026thinsp;4.31\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e9.21\u0026thinsp;\u0026plusmn;\u0026thinsp;2.80\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003e**0.009\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePercent Mosaicism\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e64\u0026thinsp;\u0026plusmn;\u0026thinsp;8%\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e57\u0026thinsp;\u0026plusmn;\u0026thinsp;15%\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003e0.210\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBIG Score\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0.50\u0026thinsp;\u0026plusmn;\u0026thinsp;0.67\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.86\u0026thinsp;\u0026plusmn;\u0026thinsp;0.53\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003e**\u0026lt;0.001\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAHI\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e6.59\u0026thinsp;\u0026plusmn;\u0026thinsp;3.80\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e4.07\u0026thinsp;\u0026plusmn;\u0026thinsp;4.62\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.232\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSpO\u003csub\u003e2\u003c/sub\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0.87\u0026thinsp;\u0026plusmn;\u0026thinsp;0.05\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0.85\u0026thinsp;\u0026plusmn;\u0026thinsp;0.06\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.411\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eRidging Grade\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0.62\u0026thinsp;\u0026plusmn;\u0026thinsp;0.61\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.37\u0026thinsp;\u0026plusmn;\u0026thinsp;0.76\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003e**0.008\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTonsil Grade\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1.78\u0026thinsp;\u0026plusmn;\u0026thinsp;1.39\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.00\u0026thinsp;\u0026plusmn;\u0026thinsp;1.15\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.268\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eLateral Wall Grade\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2.00\u0026thinsp;\u0026plusmn;\u0026thinsp;0.47\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2.33\u0026thinsp;\u0026plusmn;\u0026thinsp;0.52\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.411\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOMNP\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMallampati\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1.75\u0026thinsp;\u0026plusmn;\u0026thinsp;0.50\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2.00\u0026thinsp;\u0026plusmn;\u0026thinsp;0.82\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.549\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eScale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e288.2\u0026thinsp;\u0026plusmn;\u0026thinsp;57.97\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e278.7\u0026thinsp;\u0026plusmn;\u0026thinsp;82.94\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.674\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMArea\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3.120E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;7.081E\u0026thinsp;+\u0026thinsp;6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.097E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;5.831E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.315\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMWidth\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1242\u0026thinsp;\u0026plusmn;\u0026thinsp;494.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1208\u0026thinsp;\u0026plusmn;\u0026thinsp;386\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.816\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMHeight\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1162\u0026thinsp;\u0026plusmn;\u0026thinsp;506.3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1212\u0026thinsp;\u0026plusmn;\u0026thinsp;494.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.775\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTWidth\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1064\u0026thinsp;\u0026plusmn;\u0026thinsp;319.7\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1064\u0026thinsp;\u0026plusmn;\u0026thinsp;397.4\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTEMA\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMallampati\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2.10\u0026thinsp;\u0026plusmn;\u0026thinsp;0.32\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2.00\u0026thinsp;\u0026plusmn;\u0026thinsp;0.63\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.359\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eScale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e272.2\u0026thinsp;\u0026plusmn;\u0026thinsp;62.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e267.2\u0026thinsp;\u0026plusmn;\u0026thinsp;93.48\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.82\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMWidth\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1190\u0026thinsp;\u0026plusmn;\u0026thinsp;275.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1213\u0026thinsp;\u0026plusmn;\u0026thinsp;403.9\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.817\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTarea\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1.090E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;3.907E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1.154E\u0026thinsp;+\u0026thinsp;6\u0026thinsp;\u0026plusmn;\u0026thinsp;7.253E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.754\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTWidth\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1018\u0026thinsp;\u0026plusmn;\u0026thinsp;235.6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1141\u0026thinsp;\u0026plusmn;\u0026thinsp;399.4\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.175\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTLength\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1255\u0026thinsp;\u0026plusmn;\u0026thinsp;330.3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1131\u0026thinsp;\u0026plusmn;\u0026thinsp;469.7\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.431\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTEML\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eScale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e270.7\u0026thinsp;\u0026plusmn;\u0026thinsp;43.09\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e251.2\u0026thinsp;\u0026plusmn;\u0026thinsp;62.09\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.335\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTArea\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e5.252E\u0026thinsp;+\u0026thinsp;5\u0026thinsp;\u0026plusmn;\u0026thinsp;1.470E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e4.104E\u0026thinsp;+\u0026thinsp;5\u0026thinsp;\u0026plusmn;\u0026thinsp;1.983E\u0026thinsp;+\u0026thinsp;5\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.096\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTCurve\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1621\u0026thinsp;\u0026plusmn;\u0026thinsp;259\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1320\u0026thinsp;\u0026plusmn;\u0026thinsp;320\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003e**0.014\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTLength\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1232\u0026thinsp;\u0026plusmn;\u0026thinsp;168\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1062\u0026thinsp;\u0026plusmn;\u0026thinsp;407.8\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.144\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTThick\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e518.6\u0026thinsp;\u0026plusmn;\u0026thinsp;125.1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e485.6\u0026thinsp;\u0026plusmn;\u0026thinsp;131.3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0.511\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"4\" nameend=\"c4\" namest=\"c1\"\u003e \u003cp\u003eAHI: apnea-hypoxia index, SpO\u003csub\u003e2\u003c/sub\u003e: oxygen saturation nadir, \u003cem\u003eBWS: Beckwith-Wiedmann Syndrome, BIG: BWS Index of Macroglossia, OMNP: open mouth no phonation, TEMA: tongue extended maximally anterior, TEML: tongue extended maximally lateral\u003c/em\u003e\u003c/p\u003e \u003cp\u003eAll values reported as mean\u0026thinsp;\u0026plusmn;\u0026thinsp;standard deviation\u003c/p\u003e \u003cp\u003eRed: two-sampled Student\u0026rsquo;s t-test\u003c/p\u003e \u003cp\u003eBlue: chi-squared test\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003eCorrelation analysis showed significant correlations between OMNP mouth area and BWS clinical score (p\u0026thinsp;=\u0026thinsp;0.046). Additionally, there were correlations between TEMA tongue width and BWS clinical score (p\u0026thinsp;=\u0026thinsp;0.040), TEMA tongue length and percentage mosaicism (p\u0026thinsp;=\u0026thinsp;0.012), as well as between TEML tongue curvature and percentage mosaicism (p\u0026thinsp;=\u0026thinsp;0.045; Table\u0026nbsp;\u003cspan refid=\"Tab4\" class=\"InternalRef\"\u003e4\u003c/span\u003e). Between the two independent image raters (AMG and MB) assessing categorical variables in photographs, there was a high degree of accuracy (64%) that was significantly greater than random chance (p\u0026thinsp;=\u0026thinsp;0.007). For continuous variables, a Pearson\u0026rsquo;s correlation also revealed a high agreement between raters (p\u0026thinsp;\u0026lt;\u0026thinsp;0.001).\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab4\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 4\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eLinear models controlling for age at morphometric measurements and body mass index\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"11\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c6\" colnum=\"6\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c7\" colnum=\"7\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c8\" colnum=\"8\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c9\" colnum=\"9\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c10\" colnum=\"10\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c11\" colnum=\"11\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e \u003cp\u003eAHI\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c5\" namest=\"c4\"\u003e \u003cp\u003eSpO2\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c7\" namest=\"c6\"\u003e \u003cp\u003ePercent Mosaicism\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c9\" namest=\"c8\"\u003e \u003cp\u003eBIG Score\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c11\" namest=\"c10\"\u003e \u003cp\u003eBWS Clinical Score\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e\u003cb\u003eCoefficient\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u003cb\u003ep\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003eCoefficient\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e\u003cb\u003ep\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e\u003cb\u003eCoefficient\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e\u003cb\u003ep\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e\u003cb\u003eCoefficient\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e\u003cb\u003ep\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e \u003cp\u003e\u003cb\u003eCoefficient\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e\u003cb\u003ep\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBIG Score\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3.78E\u0026thinsp;+\u0026thinsp;00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e-6.07E-02\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0.230\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-3.09E-02\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e0.569\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e \u003cp\u003e4.15E\u0026thinsp;+\u0026thinsp;00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e\u0026lt;\u0026thinsp;0.0001\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAHI\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e-9.92E-03\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0.\u003cb\u003e003\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e3.08E-02\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e\u003cb\u003e0.039\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e4.91E-02\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.286\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e \u003cp\u003e2.10E-01\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e0.256\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSpO2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e-4.74E\u0026thinsp;+\u0026thinsp;01\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u003cb\u003e0.003\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-1.94E\u0026thinsp;+\u0026thinsp;00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e0.163\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e-3.78E\u0026thinsp;+\u0026thinsp;00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.230\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e \u003cp\u003e-2.42E\u0026thinsp;+\u0026thinsp;01\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e0.047\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePercent Mosaicism\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2.25E\u0026thinsp;+\u0026thinsp;01\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u003cb\u003e0.039\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e-2.18E-01\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0.163\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e-1.09E\u0026thinsp;+\u0026thinsp;00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.569\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e \u003cp\u003e-6.55E\u0026thinsp;+\u0026thinsp;00\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e0.505\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBWS Clinical Score\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e4.34E-01\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0.256\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e-1.05E-02\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e\u003cb\u003e0.047\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-4.06E-03\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e0.505\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e1.43E-01\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e\u003cb\u003e\u0026lt;\u0026thinsp;0.0001\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e-\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOMNP\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMArea\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e-4.30E-07\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0.868\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2.29E-08\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0.618\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e-2.10E-08\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e0.765\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e-1.89E-07\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.100\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e \u003cp\u003e-3.74E-07\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e\u003cb\u003e0.046\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMWidth\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e-7.68E-04\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0.879\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1.91E-05\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" 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align=\"left\" colname=\"c10\"\u003e \u003cp\u003e-2.06E-03\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e0.403\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTLength\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e4.48E-03\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0.327\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1.88E-05\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0.777\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e9.54E-05\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e0.560\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e-3.28E-04\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.645\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e \u003cp\u003e-2.47E-03\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e0.339\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTThick\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2.51E-02\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0.099\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2.83E-05\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0.904\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e9.80E-04\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003e0.088\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c8\"\u003e \u003cp\u003e5.13E-04\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c9\"\u003e \u003cp\u003e0.812\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c10\"\u003e \u003cp\u003e4.44E-03\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c11\"\u003e \u003cp\u003e0.483\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eBWS commonly presents with macroglossia, but objectively quantifying the severity of macroglossia is challenging. This prospective study demonstrates that easily obtained morphometric measurements using a laser and digital camera can objectively assess the intraoral anatomy of children with BWS and can provide meaningful clinical insights. The findings presented here add distinctions to emerging discussions about the spectrum of intraoral morphology across the different BWS molecular diagnoses. Finally, this study highlights that characteristics such as tongue ridging are more common in patients who had tongue reduction surgery than in those who did not.\u003c/p\u003e \u003cp\u003eMany studies have examined associations between intraoral anatomy and clinical outcomes, but these often require expensive equipment, use calibers which are difficult to maneuver in the mouth, are inaccurate, or exclude pediatric populations.\u003csup\u003e40,42,44\u0026ndash;49\u003c/sup\u003e It is important to characterize soft tissue morphology and risk factors in children predisposed to diseases such as those with BWS or OSA. Still, objective soft tissue data are classically obtained via CT or MR imaging, exposing children to radiation or requiring sedation, and are therefore usually not obtained.\u003csup\u003e36\u0026ndash;39\u003c/sup\u003e In their landmark study of 860 patients (542 with OSA and 318 controls), Schwab et. al demonstrated that tongue morphometric measurements using a digital camera and laser ruler provided accurate and reproducible measurements of the tongue and mouth that were significantly associated with polysomnographic outcomes.\u003csup\u003e40\u003c/sup\u003e However, this previous study did not include children or patients with BWS, limiting its generalizability. Our study builds on this previous work by showing that tongue and mouth measurements are significantly correlated by genetic subtype and with measures of phenotypic severity in BWS including clinical score and percentage mosaicism.\u003c/p\u003e \u003cp\u003eThis information is useful because while BWS clinical scores allow objective assessment of phenotypic burden, obtaining this information requires comprehensive patient assessment. Further, the BWS clinical scoring system views macroglossia as a dichotomous variable that is either present or absent, failing to account for the range of macroglossia across patients with BWS.\u003csup\u003e7\u003c/sup\u003e Additionally, while the BIG scores proposed by our group correlate with clinical outcomes such as surgical incidence, this system did not involve objective measurements of the oral anatomy and may be challenging to reproduce across clinical settings. The morphometric findings presented here reveal correlations between oral measurements, BWS clinical scores, and BIG scores, offering a quick and reproducible technique to quantify macroglossia severity in patients with BWS.\u003c/p\u003e \u003cp\u003eObtaining morphometric measurements is inexpensive, does not require sedation, and can be performed in most clinical settings. Children are advised to sit down with their tongues extended for a few seconds while photographs are taken. Images from the three positions are captured, and the entire process takes less than five minutes to complete. As we obtained these images, we found that children as young as two years of age can sit long enough for accurate photographs to be taken. Previously, obtaining tongue morphometric measurements had been shown to be feasible in adults, but not in children. This demonstrates that this modality can augment care not only for children with BWS, but more broadly in all patients at risk of upper airway obstruction, such as in children with Down Syndrome or obesity.\u003csup\u003e50,51\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eAssessing these photographs in patients with BWS, we found different morphological features across the various molecular diagnoses. It is well established that the incidence of certain clinical features is higher in some BWS molecular subtypes than other. For instance, patients with IC2 LOM more commonly present with omphalocele, patients with pUPD11 more often have lateralized overgrowth, and most of the patients with IC2 LOM have macroglossia.\u003csup\u003e6\u003c/sup\u003e There is little published literature reporting measurable differences in tongue morphology across those with different BWS molecular subtypes. Our study shows that there are objective differences between the intraoral anatomical measurements between the different BWS molecular subtypes.\u003c/p\u003e \u003cp\u003eAnecdotally, we have observed that those with IC2 LOM tend to have wide and long tongues, and patients with pUPD11 appear to have thick and wide tongues. Testing this hypothesis, we observed that those with IC2 LOM had wider tongues and mouths than others. Interestingly, we did not find a difference between the tongue width or thickness in patients with pUPD11 compared to others with BWS (p\u0026thinsp;\u0026gt;\u0026thinsp;0.05), though we found that they had smaller mouths than others. A recent study from our center assessed a large cohort of patients who underwent tongue reduction surgery, finding that those with pUPD11 made up a large proportion of patients selected for surgery, and that they more commonly had repeat surgery than patients with other BWS subtypes.\u003csup\u003e14\u003c/sup\u003e We initially speculated that a reason for these findings could be larger tongues in those with pUPD11, but morphometrics data demonstrated that patients with pUPD11 did not have larger tongues in the cohort presented here. A possible explanation for the discrepancy between our expectations and measurements is that we were underpowered to make statistically significant conclusions in this patient population using morphometrics. Another possibility is that tongue morphology alone is only one of many factors contributing to the need for surgery, and that the entire clinical picture of patients is needed to determine if a patient requires surgical intervention.\u003c/p\u003e \u003cp\u003eAt our center, all patients with Beckwith-Wiedemann syndrome are seen by the team geneticist (JMK) who orders molecular testing and assigns both a BWS clinical score and BIG score. As part of our macroglossia research evaluation, patients also have tongue morphometric photographs captured. Those with symptomatic macroglossia are referred to the pediatric plastic surgeon, with approximately 25% of referred patients determined to be surgical candidates. Surgery is considered when patients have a primary indication of breathing and feeding concerns (typically in younger patients) or craniodental development concerns (typically in those older than 1 year of age). Comparing the morphometric measurements between patients who had surgery to those who did not, we found that tongues were more ridged in individuals with a surgical history. Ridging, which is primarily a measure of posterior lingual crowding, is likely unchanged pre- and postoperatively given that the posterior tongue is not surgically excised. Therefore, the finding of an association between increased ridging and surgical selection may be useful information for surgeons considering intervention. Specifically, if minimal ridging is present, then surgeons may be less inclined to consider surgical intervention, and vice versa.\u003c/p\u003e \u003cp\u003eThere are several limitations to this study. First, sleep studies and morphometric measurements were conducted at different times throughout patients\u0026rsquo; treatment courses, but when we controlled for age between polysomnography and morphometric measurements, there was minimal change in outcomes. Further, we used morphometric measurement age when conducting statistical analysis. Additionally, we do not currently have pre- and post-tongue reduction morphometric data on patients, and this will be an area of future study.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eObjective intraoral morphometric measurements are associated with phenotypic burden in BWS. Tongue morphology varies across the BWS spectrum, with IC2 LOM presenting with wider tongues and mouths, and pUPD11 having narrower mouths. Tongue ridging is more common in those selected for surgery. These measurements are easy to obtain and may be adopted at low costs across centers caring for children with BWS or others at risk of upper airway obstruction.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003eEthics approval and consent to participate: institutional review board approval (IRB 13-010658) and consent from each patient were obtained.\u003c/p\u003e\n\u003cp\u003eConsent for publication: all patients have provided consent for the sharing of their photographs in this manuscript.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eAvailability of data and materials: available on reasonable request.\u003c/p\u003e\n\u003cp\u003eCompeting interests: none.\u003c/p\u003e\n\u003cp\u003eFunding: none.\u003c/p\u003e\n\u003cp\u003eAuthors\u0026apos; contributions:\u003c/p\u003e\n\u003cp\u003eDJR participated in the conception and design of the project, data analysis, interpretation of data, drafting of the manuscript, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003eAMG participated in the conception and design of the project, interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003eJHS participated in the conception and design of the project, interpretation of data, drafting of the manuscript, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work\u003c/p\u003e\n\u003cp\u003eMB participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003eAW participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003eMW participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003eJJN participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003eJAT participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003eRJS participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003eCMC participated in interpretation of data, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003eJMK participated in conception and design of the project, interpretation of data, drafting of the manuscript, critical revision of the manuscript, final approval of the version to be published, and agreement to be accountable for all aspects of the work.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFinancial Disclosure Statement:\u003c/strong\u003e The authors declare no conflicts of interest.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgements:\u0026nbsp;\u003c/strong\u003eThis work is supported by the\u003cstrong\u003e\u0026nbsp;\u003c/strong\u003eVictoria Fertitta Fund through the Lorenzo \u0026ldquo;Turtle\u0026rdquo; Sartini Jr. Endowed Chair in Beckwith\u0026ndash;Wiedemann Syndrome Research (JMK).\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eData availability: Data used in this study are not openly available due to reasons of sensitivity and patient privacy but are available from the corresponding author upon reasonable request. These data are in a controlled data storage drive at the Children\u0026rsquo;s Hospital of Philadelphia.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eMussa A, Russo S, De Crescenzo A, et al. (Epi)genotype\u0026ndash;phenotype correlations in Beckwith\u0026ndash;Wiedemann syndrome: a paradigm for genomic medicine. Eur J Hum Genet. 2016;24(2):183\u0026ndash;90. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.1038/ejhg.2015.88\u003c/span\u003e\u003cspan address=\"10.1038/ejhg.2015.88\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eWang KH, Kupa J, Duffy KA, Kalish JM. Diagnosis and Management of Beckwith-Wiedemann Syndrome. 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Am J Respir Crit Care Med. 2001;163(4):947\u0026ndash;50. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.1164/ajrccm.163.4.2005136\u003c/span\u003e\u003cspan address=\"10.1164/ajrccm.163.4.2005136\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGastelum E, Cummins M, Singh A, Montoya M, Urbano G, Tablizo M. Treatment Considerations for Obstructive Sleep Apnea in Pediatric Down Syndrome. Children. 2021;8(11):1074. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.3390/children8111074\u003c/span\u003e\u003cspan address=\"10.3390/children8111074\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eTauman R, Gozal D. Obesity and obstructive sleep apnea in children. Paediatr Respir Rev. 2006;7(4):247\u0026ndash;59. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.1016/j.prrv.2006.08.003\u003c/span\u003e\u003cspan address=\"10.1016/j.prrv.2006.08.003\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":true,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"orphanet-journal-of-rare-diseases","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"ojrd","sideBox":"Learn more about [Orphanet Journal of Rare Diseases](http://ojrd.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/ojrd/default.aspx","title":"Orphanet Journal of Rare Diseases","twitterHandle":"@bmc","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"BMC/SO AJ","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"Beckwith-Wiedemann Syndrome, BWS, macroglossia, morphometrics","lastPublishedDoi":"10.21203/rs.3.rs-4132656/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4132656/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003eObjectives\u003c/h2\u003e \u003cp\u003eAn easy-to-use tool to objectively measure intraoral anatomy with meaningful clinical correlations may improve care for patients with Beckwith-Wiedemann syndrome (BWS), who commonly have symptomatic macroglossia. This study aims to address this gap by assessing correlations between tongue morphometrics and clinical characteristics including BWS clinical score, percentage mosaicism, BWS index of macroglossia (BIG) score, sleep apnea, and surgical incidence in patients with BWS.\u003c/p\u003e\u003ch2\u003eMethods\u003c/h2\u003e \u003cp\u003eChildren aged 2\u0026ndash;17 years with BWS were prospectively enrolled between 12/2021-01/2024. Digital intraoral photographs with a laser ruler were taken, and morphometric measurements were made using ImageJ software. Relationships between morphometrics and outcomes including BWS clinical score, percentage mosaicism, and incidence of tongue reduction surgery were examined using t-tests and multivariate linear models.\u003c/p\u003e\u003ch2\u003eResults\u003c/h2\u003e \u003cp\u003ePharyngeal morphometric measurements were obtained in 49 patients with BWS. Mouth area, width, and height differed across BWS molecular subtypes (p\u0026thinsp;\u0026lt;\u0026thinsp;0.05). Right-to-left tongue width (p\u0026thinsp;=\u0026thinsp;0.022) and mouth width (p\u0026thinsp;=\u0026thinsp;0.049) were larger in those with loss of methylation at imprinting control region 2 (IC2 LOM) than other BWS variants. Patients with paternal uniparental isodisomy of chromosome 11p15 (pUPD11) had narrower mouths than others (p\u0026thinsp;=\u0026thinsp;0.040). Those with tongue reduction surgery had more tongue ridging than those without surgery (p\u0026thinsp;\u0026lt;\u0026thinsp;0.01). There were correlations between mouth area and BWS clinical score (p\u0026thinsp;=\u0026thinsp;0.046), tongue width and BWS clinical score (p\u0026thinsp;=\u0026thinsp;0.040), and tongue length and percentage mosaicism (p\u0026thinsp;=\u0026thinsp;0.012).\u003c/p\u003e\u003ch2\u003eConclusion\u003c/h2\u003e \u003cp\u003eIntraoral morphometric measurements are associated with phenotypic burden in BWS. Tongue morphology varies across the BWS spectrum, with IC2 LOM having wider tongues and mouths, and pUPD11 having narrower mouths. Tongue ridging is more common in those selected for surgery. Intraoral morphometric measurements may be safely obtained at low costs across centers caring for children with BWS or others at risk of upper airway obstruction.\u003c/p\u003e","manuscriptTitle":"Morphometric Measurements of Intraoral Anatomy in Children with Beckwith-Wiedemann Syndrome: A Prospective Study and Novel Approach","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-06-04 18:58:52","doi":"10.21203/rs.3.rs-4132656/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Major revision","date":"2024-07-16T02:56:41+00:00","index":"","fulltext":""},{"type":"reviewerAgreed","content":"","date":"2024-07-05T05:54:49+00:00","index":0,"fulltext":""},{"type":"reviewersInvited","content":"","date":"2024-05-19T18:22:28+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2024-04-03T16:12:41+00:00","index":"","fulltext":""},{"type":"submitted","content":"Orphanet Journal of Rare Diseases","date":"2024-03-26T08:03:03+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"orphanet-journal-of-rare-diseases","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"ojrd","sideBox":"Learn more about [Orphanet Journal of Rare Diseases](http://ojrd.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/ojrd/default.aspx","title":"Orphanet Journal of Rare Diseases","twitterHandle":"@bmc","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"BMC/SO AJ","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"5376ae7b-9c5d-4863-9e13-d884c0455611","owner":[],"postedDate":"June 4th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"published-in-journal","subjectAreas":[],"tags":[],"updatedAt":"2024-10-21T16:03:56+00:00","versionOfRecord":{"articleIdentity":"rs-4132656","link":"https://doi.org/10.1186/s13023-024-03350-3","journal":{"identity":"orphanet-journal-of-rare-diseases","isVorOnly":false,"title":"Orphanet Journal of Rare Diseases"},"publishedOn":"2024-10-17 15:58:01","publishedOnDateReadable":"October 17th, 2024"},"versionCreatedAt":"2024-06-04 18:58:52","video":"","vorDoi":"10.1186/s13023-024-03350-3","vorDoiUrl":"https://doi.org/10.1186/s13023-024-03350-3","workflowStages":[]},"version":"v1","identity":"rs-4132656","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4132656","identity":"rs-4132656","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}