Intrapelvic Tumor Height (ITH) as a Promising Novel Prognostic Indicator for long-term functional outcomes in Sacrococcygeal Teratomas

Intrapelvic Tumor Height (ITH) as a Promising Novel Prognostic Indicator for long-term functional outcomes in Sacrococcygeal Teratomas | Authorea try { document.documentElement.classList.add('js'); } catch (e) { } var _gaq = _gaq || []; _gaq.push(['_setAccount', 'G-8VDV14Y67G']); _gaq.push(['_trackPageview']); (function() { var ga = document.createElement('script'); ga.type = 'text/javascript'; ga.async = true; ga.src = ('https:' == document.location.protocol ? 'https://ssl' : 'http://www') + '.google-analytics.com/ga.js'; var s = document.getElementsByTagName('script')[0]; s.parentNode.insertBefore(ga, s); })(); Skip to main content Preprints Collections Wiley Open Research IET Open Research Ecological Society of Japan All Collections About About Authorea FAQs Contact Us Quick Search anywhere Search for preprint articles, keywords, etc. Search Search ADVANCED SEARCH SCROLL This is a preprint and has not been peer reviewed. Data may be preliminary. 12 February 2025 V1 Latest version Share on Intrapelvic Tumor Height (ITH) as a Promising Novel Prognostic Indicator for long-term functional outcomes in Sacrococcygeal Teratomas Authors : Sébastien Faraj 0000-0002-5875-6121 [email protected] , Yousuf Al-shaqsi , Pauline Lallemant-Dudek , Eleonore Blondiaux , Aurore COULOMB L'HERMINE , Sabine Irtan , and Georges Audry Authors Info & Affiliations https://doi.org/10.22541/au.173934582.27528960/v1 Published Pediatric Blood & Cancer Version of record Peer review timeline 188 views 152 downloads Contents Abstract Information & Authors Metrics & Citations View Options References Figures Tables Media Share Abstract Background: Sacrococcygeal teratoma (SCT) is considered the most common congenital neoplasm in neonates. This study aimed to assess long-term impacts on urinary and anorectal functions and quality of life (QoL) in patients who underwent surgery for SCT and to identify predictive factors for these outcomes, with a focus on the Intrapelvic Tumor Height (ITH). Methods : This retrospective study included patients treated surgically for SCT, between 1984 and 2017 at the Armand Trousseau Hospital. Urologic and anorectal functions were evaluated through clinical assessments and patient-reported outcomes through questionnaires. Statistical analysis was performed to explore correlations between these factors and long-term functional outcomes. Results: Twenty-six patients (median age=17 years) were identified. With a median follow-up of 180 months [96-444 months], 12 patients (46%) presented functional impairments: seven had isolated anorectal dysfunction and five had both urinary and anorectal dysfunctions, including neurogenic bladder (n=2) and/or detrusor-sphincter dyssynergia (n=3). A significant correlation was observed between adverse outcomes and intrapelvic tumor height (ITH) (median ITH was 27 mm in patients with impairments versus 15 mm in those without, p<0.05). Of the 12 questionnaire respondents, two patients experienced urinary and anorectal dysfunctions and five experienced isolated anorectal dysfunction. Most patients reported a good QoL, only one patient reporting a severe impact on daily life. Conclusions: This study highlights that long-term urologic and anorectal dysfunctions are prevalent in patients treated for SCT, although the overall impact on quality of life remains moderate. Importantly, ITH emerged as a more objective and accurate predictor of functional outcomes than tumor size or Altman classification. Title: Intrapelvic Tumor Height (ITH) as a Promising Novel Prognostic Indicator for long-term functional outcomes in Sacrococcygeal Teratomas Author names: Sébastien Faraj a* , Yousuf Al-shaqsi a , Pauline Lallemant-Dudek b , Eleonore Blondiaux c , Aurore Coulomb-Lhermine d , Sabine Irtan a , Georges Audry a Author affiliations: 1. Sorbonne University, AP-HP, Armand Trousseau Hospital, Department of Visceral and Neonatal Pediatric Surgery, 26 Avenue du Dr Arnold Netter, 75012 Paris, France 2. Sorbonne University, AP-HP, Armand Trousseau Hospital, Department of Physical Medicine and Rehabilitation, 26 Avenue du Dr Arnold Netter, 75012 Paris, France 3. Sorbonne University, AP-HP, Armand Trousseau Hospital, Department of Radiology, 26 Avenue du Dr Arnold Netter, 75012 Paris, France 4. Sorbonne University, AP-HP, Armand Trousseau Hospital, Department of Pathology, 26 Avenue du Dr Arnold Netter, 75012 Paris, France Corresponding author: Sébastien Faraj Author’s contribution: • Study conception and design: Yousuf Al-shaqsi, Georges Audry, Sabine Irtan • Data acquisition: Yousuf Al-shaqsi, Eleonore Blondiaux, Aurore Coulomb-Lhermine, Pauline Lallemant • Analysis and data interpretation: Yousuf Al-shaqsi, Pauline Lallemant, Sébastien Faraj • Drafting of the manuscript: Yousuf Al-shaqsi, Sébastien Faraj • Critical revision: Sabine Irtan, Georges Audry, Sébastien Faraj Competing interests: No conflict of interest to disclose Category of the manuscript: Original article Previous communication: No previous communication Financial support statement: No source of financial support Ethics statements: Patient consent for publication Not required Ethics approval This study is a retrospective study, conducted in accordance with the principles of Declaration of Helsinki. The study protocol was exempted by the Institutional Board of Armand Trousseau Hospital because it is a non-experimental medical study but cohort and quality-improvement study, the personal identities cannot be identified and anonymous questionnaires were used. Word count Abstract : words Original manuscript: words ABSTRACT Background: Sacrococcygeal teratoma (SCT) is considered the most common congenital neoplasm in neonates. This study aimed to assess long-term impacts on urinary and anorectal functions and quality of life (QoL) in patients who underwent surgery for SCT and to identify predictive factors for these outcomes, with a focus on the Intrapelvic Tumor Height (ITH). Methods : This retrospective study included patients treated surgically for SCT, between 1984 and 2017 at the Armand Trousseau Hospital. Urologic and anorectal functions were evaluated through clinical assessments and patient-reported outcomes through questionnaires. Statistical analysis was performed to explore correlations between these factors and long-term functional outcomes. Results: Twenty-six patients (median age=17 years) were identified. With a median follow-up of 180 months [96-444 months], 12 patients (46%) presented functional impairments: seven had isolated anorectal dysfunction and five had both urinary and anorectal dysfunctions, including neurogenic bladder (n=2) and/or detrusor-sphincter dyssynergia (n=3). A significant correlation was observed between adverse outcomes and intrapelvic tumor height (ITH) (median ITH was 27 mm in patients with impairments versus 15 mm in those without, p<0.05). Of the 12 questionnaire respondents, two patients experienced urinary and anorectal dysfunctions and five experienced isolated anorectal dysfunction. Most patients reported a good QoL, only one patient reporting a severe impact on daily life. Conclusions: This study highlights that long-term urologic and anorectal dysfunctions are prevalent in patients treated for SCT, although the overall impact on quality of life remains moderate. Importantly, ITH emerged as a more objective and accurate predictor of functional outcomes than tumor size or Altman classification. Keywords: teratoma, bladder dysfunction, constipation, quality of life, outcome, predictive factors Level of evidence: III Abbreviations: CT: computed tomography; IQR: interquartile range; ITH: intrapelvic tumor height; MRI: magnetic resonance imaging; QoL: quality of life; SCT: sacrococcygeal teratoma; UTI: urinary tract infection; VUR: vesicoureteral reflux. INTRODUCTION Sacrococcygeal teratoma (SCT) is a rare congenital tumor with an incidence of 1 in 14,000–28,000 live births and a female predominance of 4:1[1]. However, it is the most common neonatal solid tumor[2]. SCT may be diagnosed during antenatal, neonatal, infancy, or early childhood periods[3]. The treatment of SCT involves surgical resection of the tumor and the coccyx, which extends variably into the presacral space and pelvis. Postoperative morbidity following SCT surgery is likely multifactorial, potentially resulting from local compression of pelvic nerves and pelvic floor musculature caused by the original tumor, possible iatrogenic injury during surgical resection, or associated urogenital anomalies, such as urethral stenosis or urethrovaginal fistula[4]. Few studies have evaluated the long-term impacts on urinary and anorectal functions or the quality of life (QoL) of patients treated for SCT and were either based on medical records alone or questionnaire-based surveys. Several factors such as the Altman classification, tumor size, abdominoperineal resection, and tumor histopathology have been heterogeneously reported as risk factors for unfavorable outcomes[5–8]. However, the intrapelvic tumor height (ITH) in relation to the sacro-lumbar vertebrae has not been considered as a potential predictive factor. Therefore, this study aimed to assess long-term impacts on urinary and anorectal functions and QoL in patients who underwent surgery for SCT at our institution and identify predictive factors for these outcomes. METHODS In this retrospective study, we included all patients who underwent surgery for SCT between 1984 and 2017 at Armand Trousseau Hospital (Assitance Publique des Hôpitaux de Paris). We reviewed their medical records to collect demographic, clinical, radiological, operative, and histological data to determine long-term functional outcomes and associated predictive factors. Urinary and anorectal functional outcomes were assessed based on the medical records and by questionnaires sent to patients if they were over 18 years of age or to their parents if they were younger. Urinary functional outcomes were assessed using the validated questionnaire developed by Shalaby et al[9]. Anorectal functional outcomes were evaluated using the international Krickenbeck classification for the assessment of postoperative results following surgeries for anorectal malformations[10]. The QoL of patients treated for SCT was assessed using the pediatric incontinence questionnaire (PinQ) designed by Bower et al[11]. Written informed consent was obtained from the patients or their parents prior to participation. Urodynamic studies (UDSs) performed for all suspected cases of bladder dysfunction following surgical treatment of SCT were reviewed and analyzed in collaboration with the physical medicine and rehabilitation department. We investigated potential predictive factors associated with functional impairments following SCT resection by analyzing patients’ demographic and clinical information, such as sex, age at diagnosis, Altman classification, radiological measurement of ITH relative to the sacro-lumbar vertebrae, radiological and histological characteristics of the tumor, surgical approach, completeness of resection, and the need for radiotherapy or chemotherapy. The ITH was evaluated retrospectively by reviewing all preoperative radiological images obtained using magnetic resonance imaging (MRI) or computer tomography (CT). Radiological images were reviewed by a radiologist blinded to the long-term outcomes of the patients. ITH was measured on a sagittal view from the pubococcygeal line to the maximum height of the tumor relative to the sacro-lumbar vertebra (Figure 1). Statistical analysis was conducted using GraphPad Software version 8.4.3 (San Diego, California, USA). Data are reported as medians, ranges, interquartile ranges (IQR), and percentages. The Mann-Whitney U test and Fisher’s exact test were used for data analysis. Additionally, the Kruskal-Wallis test was applied to compare variables across multiple independent groups. A p-value <0.05 was considered statistically significant. Population We identified 26 patients (21 female and 5 male [4:1 female-to-male ratio]) who met the inclusion criteria. The median follow-up period until last clinical examination was 180 months [96-444 months]. Sixteen patients had a prenatal diagnosis, while 10 patients were diagnosed postnatally. The Altman classification was determined based on radiological and/or operative reports. Seven patients underwent surgery for type I SCT, 10 for type II, six for type III, and three for type IV. All patients underwent surgical excision, by perineal approach (20 patients) or abdomino-perineal approach (6 patients ; three Altman type I-II, two type III and one type IV). Pathological examination showed that 21 patients were diagnosed with a mature teratoma, four with an immature teratoma, and one with a localized malignant mixed germ cell tumor. We observed four cases of recurrence. One patient initially diagnosed with an immature teratoma experienced recurrence as a yolk sac tumor and was treated by chemotherapy alone. Another patient had local recurrence of an immature teratoma with an intramedullary extension and required reoperation followed by chemotherapy. The two other patients, initially diagnosed with mature teratomas, underwent subsequent surgeries (and follow-up chemotherapy for one) due to incomplete resection (Table 1). Functional outcomes Long-term functional impairments were observed in 12 patients (Tables 1 and 2). Seven patients had isolated anorectal dysfunction and five patients had both urinary and anorectal dysfunctions. Anorectal dysfunction included chronic constipation in 11 patients and soiling in eight patients. Urinary dysfunction manifested as voiding difficulties or urinary incontinence, often associated with recurrent urinary tract infections (UTIs). All five patients with urinary dysfunction underwent urodynamic studies (UDS), which revealed neurogenic bladder in two cases: one patient had overactive bladder with low compliance and was treated with clean intermittent catheterization and bladder augmentation; another had overactive bladder associated with detrusor-sphincter dyssynergia and was managed with clean intermittent catheterization and anticholinergic medication. Two other patients presented with isolated detrusor-sphincter dyssynergia and were treated symptomatically. One patient experienced recurrent UTIs and urinary incontinence but had normal findings on UDS. Vesicoureteral reflux (VUR) was observed in three out of these five patients. Recurrent febrile UTIs were also observed in four other patients, without urinary dysfunction found on exploration and without the need for correction. On univariate analysis, there was no significant difference between patients with or without anorectal or urinary functional impairments regarding sex distribution, Altman classification, surgical approach, tumor histology, recurrence, or treatment with chemotherapy (Table 1). ITH and functional outcomes The ITH was measured in 18 patients with preoperative MR and/or CT images. Eight patients, who were operated on immediately after birth based on antenatal ultrasonography findings (n=6) or due to a large postnatally detected tumor (n=2), did not have ITH measurements and were therefore excluded from the statistical analysis. The median ITH was 27 mm in the group with functional impairments and 15 mm in the group without functional impairment (p<0.05) (Figure 2). ITH was significantly correlated with functional dysfunction (p = 0.007) in univariate non-parametric analysis and remained independent of other variables such as Altman classification (p = 0.08), sex distribution (p = 0.16), surgical approach (p = 0.43), tumor histology (p = 0.53), recurrence (p = 0.11), or treatment with chemotherapy (p = 0.10), in multifactorial analysis. Questionnaires’ outcomes Twelve patients completed the questionnaires. The demographic and clinical characteristics of the patients are shown in Table 3. The median age of respondents was 13 years [4–37 years] with a median follow-up of 13 years [4-37 years]. Five out of the 12 respondents experienced no urinary or anorectal dysfunction. Recurrent UTIs were noted in five patients, including two patients with urinary incontinence and voiding difficulties due to underlying neurogenic bladder or detrusor sphincter dyssynergia. Anorectal complications occurred in seven patients and included chronic constipation that ranged from grade I (five patients, treated conservatively through dietary regimens) to grade III (2 patients, requiring disimpaction or colonic irrigation). Fecal soiling was reported in three patients. Among the seven patients with anorectal complications, two also had neurogenic bladder. All patients with severe urinary or anorectal dysfunction had already been identified during routine follow-up. The QoL of patients who underwent surgery for SCT was assessed using the PinQ. Most patients (92%) experienced mild impacts on their daily life due to the functional outcomes described above. The patients generally reported very good outcomes regarding mental, social, and physical aspects. Only one patient reported severe mental and social impacts. DISCUSSION In this study, 46% of patients who underwent surgery for SCT experienced long-term urologic and anorectal dysfunction with minor impact on their QoL. Our findings suggest that the ITH may be a predictive factor for urologic or anorectal impairment in these patients. Anorectal impairments were the most common type of dysfunction in our cohort (n=12/26, 46% of patients), manifesting as high grade constipation and/or fecal incontinence. Detrusor-sphincter dyssynergia and neurogenic bladder were the most frequent urologic dysfunction identified (n=5/26, 19% of patients). These findings align with the variability reported in the literature[5–9, 12–19], with frequency of anorectal and urologic problems following SCT excision ranging from 8-47% and 7–37% respectively (Table 4). These studies did not clearly distinguish between combined or isolated dysfunctions. Whether these functional disorders result from the tumor’s mass effect, surgical damage to intrapelvic nerves and muscles, or a combination of both remains controversial[5, 6]. Several predictive factors for functional impairments in patients operated on for SCT resection have been studied, such as age at surgery or high-grade Altman classification[5, 6, 12, 20]. With a total of 41% and 40% of functional sequelae reported respectively, Malone et al[5] and Partridge et al[12] showed that patients with Altman type III or higher-grade tumors had a higher incidence of fecal and/or urinary incontinence. We did not find a significant correlation between the Altman grade and the occurrence of functional impairment, which is consistent with findings of other studies[13, 21, 22]. Similarly, Shalaby et al[9] did not find correlations between a high Altman classification, histopathological characteristics of tumors, and dysfunctional outcomes. Tumor size has also been evocated as predictive factor for dysfunctional outcomes, with variable findings between studies. Hambraeus et al[6] and Masahata et al[13] reported that larger tumor size was significantly associated with a higher incidence of dysfunctional outcomes, while Güler et al[7] found no significant difference between tumors smaller or bigger than 10cm. In our study, we did not consider tumor size as a valid predictive factor, as we deemed tumor size difficult to compare across cases, given that the intrapelvic and extrapelvic portions may have different potential functional consequences. However, we did observe a significant correlation between Intrapelvic Tumor Height (ITH) and functional impairment (p and reliable predictor of postoperative dysfunction. These findings could be explained by the innervation patterns of the anorectal and bladder regions. The parasympathetic fibers of the inferior hypogastric plexus originate between the second and fourth sacral segments of the spinal cord, while sympathetic fibers that innervate the bladder originate between spinal cord segments T11 and L2. Additionally, fibers from L5 supply the rectum and internal anal sphincter[23, 24]. These anatomical relationships support our observation that functional impairments in patients who underwent surgery for SCT were significantly correlated to ITH and may explain why all patients with urinary dysfunction also experienced anorectal dysfunction. Therefore, ITH may serve as a more accurate predictive factor than Altman classification for urologic and anorectal dysfunctions in patients who underwent surgery for SCT. The QoL of our patients was assessed using the PinQ, which showed that most of the patients (92%) reported that urologic and anorectal functional outcomes only mildly impacted their daily life activities. Although previous studies have suggested that QoL of patients treated for SCT may be impaired due to urinary and anorectal dysfunctions[8, 21], no such correlation was found in our study. This may be related to the management approach, as QoL mainly depends on achieving continence, regardless of the treatment used. Management strategies also vary across studies depending on nationality. Similarly, Hambraeus et al. were unable to establish a statistical correlation between physical function and QoL[14]. The results of this study emphasize the need for long-term monitoring of rectal and urological function in children treated surgically for sacrococcygeal teratomas. We propose a structured follow-up schedule, including regular clinical examinations, fecal function with constipation assessment, voiding diary and urinary system ultrasounds at 1 year, 3 years, and 5 years postoperatively, followed by assessments every 5 years. For patients presenting urinary symptoms or unfavorable prognostic factors such as a high intrapelvic tumor height (ITH), urodynamic studies should also be considered to ensure early detection and management of potential complications. Uroflowmetry may be considered in all patients as a non-invasive bladder drainage function evaluation. This study has several limitations, which include its retrospective and single-center design and small sample size. The number of patients was insufficient to establish significant correlations for various outcomes. We did not study the long term cosmetic result. Moreover, only a few patients reported both urological and anorectal dysfunction, with some reporting only isolated anorectal dysfunction. Thus, a larger sample size would be required to explore these findings. Nevertheless, this preliminary study is one of the few that combine medical records and questionnaire data to evaluate long-term functional outcomes and predictive factors of urologic and anorectal dysfunctions in patients treated for SCT. Our results underscore the need for multi-institutional collaborative research to identify the key predictive factors of functional impairment in this population. In conclusion , long-term urinary and anorectal dysfunctions are common outcomes in patients treated for SCT, although their impact on quality of life remains moderate. This study highlights a new predictive factor, intrapelvic tumor height (ITH), which appears to be more objective and accurate than tumor size or Altman grade. The value of ITH as a predictive marker warrants further investigation in future studies to validate its utility across other patient cohorts. Therefore, thorough preoperative imaging evaluation and specialized follow-up of urinary and anorectal functions are essential for patients undergoing SCT surgery to optimize long-term functional outcomes. ACKNOWLEDGMENTS We thank all the parents and patients who participated in this study and completed the questionnaires. REFERENCES 1. Altman RP, Randolph JG, Lilly JR. Sacrococcygeal teratoma: American Academy of Pediatrics Surgical Section Survey-1973. J Pediatr Surg, 1974 9: 389–398. 2. Makin EC, Hyett J, Ade-Ajayi N, Patel S, Nicolaides K, Davenport M. Outcome of antenatally diagnosed sacrococcygeal teratomas: single-center experience (1993-2004). J Pediatr Surg, 2006 41: 388–393. 3. Tuladhar R, Patole SK, Whitehall JS. Sacrococcygeal teratoma in the perinatal period. Postgrad Med J, 2000 76: 754–759. 4. Shalaby MS, O’Toole S, Driver C, Bradnock T, Lam J, Carachi R. Urogenital anomalies in girls with sacrococcygeal teratoma: a commonly missed association. 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Derikx JPM, De Backer A, van de Schoot L, Aronson DC, de Langen ZJ, van den Hoonaard TL, Bax NMA, van der Staak F, van Heurn LWE. Long-term functional sequelae of sacrococcygeal teratoma: a national study in The Netherlands. J Pediatr Surg, 2007 42: 1122–1126. 22. Havránek P, Rubenson A, Güth D, Frenckner B, Olsen L, Kornfält SA, Hansson G. Sacrococcygeal teratoma in Sweden: a 10-year national retrospective study. J Pediatr Surg, 1992 27: 1447–1450. 23. Carmichael JC, Mills S. Anatomy and Embryology of the Colon, Rectum, and Anus [Internet], in Steele SR, Hull TL, Hyman N, Maykel JA, Read TE, Whitlow CB (eds): The ASCRS Textbook of Colon and Rectal Surgery. Cham, Springer International Publishing, 2022, pp 3–27[cited 2025 Jan 16] Available from: https://doi.org/10.1007/978-3-030-66049-9_124. Mahadevan V. Anatomy of the lower urinary tract. Surg Oxf, 2019 37: 351–358. Table 1. Demographic and clinical characteristics of patients in the entire cohort (n=26) Yes (n=12) 46% No (n=14) 54% p-value Sex Male Female 5 (19%) 21 (81%) 2 (17%) 10 (83%) 3 (21%) 11 (79%) 1.0000 1.0000 Age at diagnosis Prenatal (WGA) Postnatal (day) 16 (62%) 10 (38%) 6 (50%) 6 (50%) 10 (71%) 4 (29%) 0.7501 0.7112 Weight of birth (g) median 3200 (1420­–4500) 2980 (1700–4500) 3300 (1420–3730) 1.0000 Altman Classification I II III IV 7 (27%) 10 (38%) 6 (23%) 3 (12%) 3 (25%) 3 (25%) 4 (33%) 2 (17%) 4 (29%) 7 (50%) 2 (14%) 1 (7%) 1.0000 0.4682 0.6880 0.5977 Radiology characteristic Tissue Cystic Mixed 2 (8%) 3 (11%) 21 (81%) 2 (17%) 1 (8%) 9 (75%) 0 2 (14%) 12 (86%) 0.4815 1.0000 0.7761 AFP (median/IQR) 15420/25492 18200/38359 13709/39298 0.3223 Chemotherapy 5 (19%) 4 (33%) 1 (7%) 0.1824 Age at surgery (days) Median IQR 2.5 (0–4320) 68 8 (0–4320) 53 2.5 (1–3600) 101 0.1326 Surgical approach Perineal Abdominoperineal 20 (77%) 6 (23%) 7 (58%) 5 (42%) 13 (93%) 1 (7%) 0.5510 0.1779 Resection Complete Incomplete 24 (92%) 2 (8%) 10 (83%) 2 (17%) 14 (100%) 0 0.7827 0.4815 Histology Mature Immature Malignant 21 (81%) 4 (15%) 1 (4%) 7 (58%) 4 (33%) 1 (8%) 14 (100%) 0 0 0.5506 0.1029 0.4815 Recurrence 4 (15%) 3 (25%) 1 (7%) 0.5977 Reoperated 3 (11%) 2 (17%) 1 (7%) 0.5977 IQR: interquartile range; WGA: weeks of gestational age. AFP: alpha fetoprotein Table 2 . Demographic and clinical characteristics of patients with functional impairments (n=12) Sex Male Female 0 5 (100%) 2 (29%) 5 (71%) Age at diagnosis Prenatal (WGA) Postnatal (day) 4 (80%) 1 (20%) 2 (29%) 5 (71 %) Weight of birth (g) median 3100 (1700–3420) 2650 (2200–4500) Altman classification I II III IV 0 2 (40%) 2 (40%) 1 (20%) 3 (43%) 1 (14%) 2 (29%) 1 (14%) Radiology characteristics Tissue Cystic Mixed 0 0 5 (100%) 2 (29%) 1 (14%) 4 (57%) AFP (median/IQR) 19515/43489 15420/41652 Chemotherapy 2 (40%) 2 (29%) Age at surgery (day) Median IQR 1 (0–30) 22 36 (0–4320) 329 Surgical approach Perineal Abdominoperineal 2 (40%) 3 (60%) 5 (71%) 2 (29%) Resection Complete Incomplete 3 (60%) 2 (40%) 7 (100%) 0 (0%) Histology Mature Immature Malignant 3 (60%) 2 (40%) 0 4 (57%) 2 (29%) 1 (14%) Recurrence 1 (20%) 2 (29%) Reoperated 1 (20%) 1 (14%) IQR: interquartile range; WGA: weeks of gestational age; AFP: alpha fetoprotein. Table 3. Demographic and clinical characteristics of the questionnaire respondents (n=12) Sex Male Female 2 (17%) 10 (83) 0 (0%) 2 (100%) 1 (20%) 4 (80%) 1 (20%) 4 (80%) Age of diagnosis Prenatal (WGA) Postnatal (day) 7 (58%) 5 (42%) 1 (50%) 1 (50%) 2 (40%) 3 (60%) 4 (80%) 1 (20%) Weight of birth (g) median 3258 (1420–4500) 3260 (3100–3420) 2650 (2200–4500) 3150 (1420–3635) Altman classification I II III IV 2 (17%) 7 (58%) 2 (17%) 1 (8%) 0 (0%) 1 (50%) 0 (0%) 1 (50%) 2 (40%) 1 (20%) 2 (40%) 0 (0%) 0 (0%) 5 (100%) 0 (0%) 0 (0%) Radiology characteristic Tissue Cystic Mixed 1 (8%) 1 (8%) 10 (83%) 0 (0%) 0 (0%) 2 (100%) 1 (20%) 1 (20%) 3 (60%) 0 (0%) 0 (0%) 5 (100%) AFP (median/IQR) 18638/50043 16430/30240 15420/30923 33516/54940 Chemotherapy 2 (17%) 1(50%) 1 (20%) 0 (0%) Age of intervention (day) Median IQR 1.5 (1–330) 23 16 (1–30) 29 1 (1–330) 59 2 (2–7) 4.5 Surgical approach Perineal Abdominoperineal 10 (83%) 2 (17%) 1 (50%) 1 (50%) 4 (80%) 1 (20%) 5 (100%) 0 (0%) Resection Complete Incomplete 11 (92%) 1 (8%) 1 (50%) 1 (50%) 5 (100%) 0 (0%) 5 (100%) 0 (0%) Histology Mature Immature Malignant 10 (83%) 2 (17%) 0 (0%) 1(50%) 1 (50%) 0 (0%) 4 (80%) 1 (20%) 0 (0%) 5 (100%) 0 (0%) 0 (0%) Recurrence 2 (17%) 0 (0%) 2 (40%) 0 (0%) Reoperated 1 (8%) 0 (0%) 1 (20 %) 0 (0%) IQR: interquartile range; WGA: weeks of gestational age. AFP: alpha fetoprotein Table 4. Literature review of functional outcomes in patients treated for SCT Malone et al[5] A monocentric, retrospective study 33 5 years (2–12) I-II 56% (19) III-IV 44% (14) Benign 79 % Malignant 21% 24% (8) 24% (8) 7% (2) -Altman III / IV -Abdominoperineal approach Hambraeus et al[6] A monocentric, controlled study 17 Median age 7.3 years I=41% (23) II=18% (11) III=29% (11) IV=12% (8) Mature 82% Immature 18% 47% 29% 29% -Tumor size Güler et al[7] A monocentric, retrospective study 27 out of 40 Mean f/u 78.5 months (26–206) I=22.5% II=25% III=37.5% IV=15% Mature 65% Immature 25% Malignant 10% 25.9% (7) 3.7% (1) 7.4% (2) -Neither Altman nor tumor size is statistically significant Rintala et al[8] A monocentric, retrospective study 26 Mean age 30 years (19–45) I 46% (12) II 30% (8) III-IV 23% (6) 34.6% (9) 11.5% (3) 19% (5) -Altman has no statistical significance Shalaby et al[9] Multicentric, retrospective study 31 Median age 12 years (5–35) I-II 59% III-IV 41% 39% (9) 19% (6) 29% (9) -No correlation to sex, age, histology, or Altman Partridge et al[12] A monocentric, retrospective study 45 Median 41.5 months (12–124) I=20% II=53% III=17.8% IV=8.9% 29.9 % 8.9% 15.6% -Altman classification Masahata et al[13] A monocentric, retrospective study 29 ≥ 3 years III-IV 41.4% Mature 79.3% Immature 20.7 % 17.2% 10.3% 13.8% -Tumor size -Age at surgery Hambraeus et al[14] A multicentric, retrospective study 49 Median age 8.9 years (3.6–28.8) I = 43% II= 21% III= 21% IV=15% Mature 72% Immature 26% Malignant 2% 20% 23% 28% -Gestational age Cozzi et al[15] A monocentric, retrospective study 13 Mean age 25 years (17–38) I= 56% II= 11% III=11% IV=22% Not evaluated 38% (5) 8% (1) 15%stress incontinence 23% enuresis -Not evaluated Berger et al[16] A monocentric, retrospective study 24 Mean age at f/u 79 months I = 21% (5) II= 21% (5) III= 21% (5) IV = 37% (9) Mature 71% (17) Immature29% (7) 8% (2) 4% (1) 8% (2) -Altman has no statistical significance Khalil et al[17] A monocentric, retrospective study 12 Average f/u 10.6 years (1–17) Malignant only 23% 8% -Not evaluated Kremer et al[18] A multicentric, retrospective study 47 Mean age 26.2 years I-II =73 % III = 16 % IV= 11% Mature 64 % Immature 11% Malignant 25% 21.7% (10) 23.4% (11) 19.6 % (9) -Altman has no statistical significance Villamil et al[19] A monocentric, retrospective study 8 Mean age 23 years (4–37) I = 37.5% II = 25% III = 25% IV = 12 % Mature 37.5% Immature 62.5% 25% 12 % 37% -Not evaluated f/u: follow-up Figure 1 : Measurement of the ITH (Intrapelvic Tumor Height) on sagittal MRI Sagittal MRI images illustrating the measurement of the intrapelvic tumor height (ITH). The pubococcygeal line extends from the pubis to the coccyx. The ITH measurement represents the maximum height of the tumor relative to the sacro-lumbar vertebra. The two images demonstrate the measurement process on different cases. Figure 2 : Boxplots showing the distribution of Intratumoral Height (ITH) across patient groups based on functional outcomes This figure presents the distribution of ITH measurements in three patient groups: those without functional impairments (”No Trouble”), those with isolated anorectal dysfunction, and those with combined uro-anorectal dysfunction. The distribution is displayed as boxplots. Statistical significance between groups is indicated by the * symbol. Information & Authors Information Version history V1 Version 1 12 February 2025 Peer review timeline Published Pediatric Blood & Cancer Version of Record 17 Mar 2026 Published Copyright This work is licensed under a Non Exclusive No Reuse License. Keywords germ cell tumors quality of life surgery Authors Affiliations Sébastien Faraj 0000-0002-5875-6121 [email protected] Sorbonne Universite View all articles by this author Yousuf Al-shaqsi Sorbonne Universite View all articles by this author Pauline Lallemant-Dudek Sorbonne Universite View all articles by this author Eleonore Blondiaux Sorbonne Universite View all articles by this author Aurore COULOMB L'HERMINE Sorbonne Universite View all articles by this author Sabine Irtan Sorbonne Universite View all articles by this author Georges Audry Sorbonne Universite View all articles by this author Metrics & Citations Metrics Article Usage 188 views 152 downloads .FvxKWukQNSOunydq8rnd { width: 100px; } Citations Download citation Sébastien Faraj, Yousuf Al-shaqsi, Pauline Lallemant-Dudek, et al. Intrapelvic Tumor Height (ITH) as a Promising Novel Prognostic Indicator for long-term functional outcomes in Sacrococcygeal Teratomas. Authorea . 12 February 2025. 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