Evaluating the efficacy of the Tübingen Palatal Plate in managing upper airway obstruction in Pierre Robin Sequence: Experience from a tertiary hospital in Sydney, Australia

Evaluating the efficacy of the Tübingen Palatal Plate in managing upper airway obstruction in Pierre Robin Sequence: Experience from a tertiary hospital in Sydney, Australia | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Research Article Evaluating the efficacy of the Tübingen Palatal Plate in managing upper airway obstruction in Pierre Robin Sequence: Experience from a tertiary hospital in Sydney, Australia Nathan Lieu, Lisa Theis, Coral Yeung, Richard Widmer, Winey Wan, and 3 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-8912918/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 8 You are reading this latest preprint version Abstract Study objective: To evaluate the efficacy of the Tübingen Palatal Plate in managing upper airway obstruction in Pierre Robin Sequence during its introduction into a tertiary hospital in Sydney, Australia. Methods We evaluated polysomnography and treatment outcomes in children diagnosed with PRS and managed with TPP from October 2023 - March 2025 at the Children’s Hospital in Westmead. A level 1 polysomnography was performed at baseline, prior to discharge from hospital and at the end of treatment. Sleep study parameters were compared between initial and follow-up studies. Results From 14 admissions of infants with PRS, ten infants completed TPP treatment. At baseline all ten had severe obstructive sleep apnea (OSA) with a median apnea-hypopnea index (AHI) of 53.9 events/hr, and mixed and obstructive apnea index (MOAI) of 37.8 events/hr. There was evidence of impaired gas exchange with an oxygen nadir of 88% (71–91%) and peak carbon dioxide (CO 2 ) level of 51.7mmHg (46.1–65.8). The final median AHI of 8.1 (2.9–13.3) events/hr, and MOAI of 2.9 (1.3–8.8) events/hr. When compared to baseline there were statistically significant improvements in AHI, OAHI, MOAI and peak CO 2 levels. Excluded Infants included one patient with an intact soft palate, and three others with clinical and/or practical issues preventing implementation of the TPP. Conclusions This study demonstrates that when first introduced in a new health setting, TPP therapy is effective for treating airway obstruction in infants with PRS. Pierre Robin Sequence Tübingen Palatal Plate obstructive sleep apnoea paediatrics Brief Summary Current Knowledge/Study Rationale: PRS is a congenital triad of micrognathia, glossoptosis and upper airway obstruction with a range of management options. This study assesses the effectiveness of TPP for treating airway obstruction in neonates with PRS during the establishment of the method in a new center. Study Impact: As the first Australian center to introduce the TPP, the Children’s Hospital at Westmead has demonstrated that the TPP is an effective non-invasive treatment modality for managing OSA in infants with PRS while introducing the treatment. Introduction Pierre Robin sequence (PRS) is a congenital triad of micrognathia, glossoptosis and airway obstruction[ 1 ]. Up to 50% of PRS have an underlying genetic component, of which there are currently 34 known genetic syndromes, including Stickler, Treacher Collins and Velocardiofacial syndrome[ 2 , 3 ]. Cleft palate is also present in up to 90% of cases[ 3 ]. The incidence of PRS ranges from 1/8500 to 1/14,000 births[ 4 ], with a higher incidence of 1/5500reported in Australia[ 5 ]. First line management of airway obstruction in PRS varies worldwide and according to the severity of disease. Graded approaches for managing airway problems usually start with non-invasive approaches that include prone positioning, nasopharyngeal airway (NPA), continuous positive airway pressure (CPAP) and orthodontic devices such as palatal plates. Prone positioning has been shown to be an effective method of relieving obstruction by allowing the mandible and tongue to fall forwards[ 6 ]. However prone positioning alongside prematurity and smoking are well recognized risk factors for sudden infant death syndrome (SIDS). Centers that use positioning treatment may recommend home monitoring such as apnea devices, and polysomnogram studies to confirm the effect of sleep position on airway obstruction[ 7 , 8 ]. This treatment is used infrequently at the Children’s Hospital at Westmead (CHW), where infants with PRS are generally referred for treatment because of significant airway and feeding issues, and the severity of their airway obstruction requires more aggressive intervention. Surgical treatment options and tracheostomy are reserved for infants with the most severe airway disturbances. Orthodontic appliances have been in use to treat PRS for the last 15 years but have gained increasing popularity as evidence for their effectiveness as a non-invasive approach increases and technological developments make their implementation easier. The group in Tübingen Germany have published broadly on the use of the Tübingen Palate Plate (TPP), which features a palatal base plate covering the hard palate and cleft, and a velopharyngeal spur which works by displacing the tongue anteriorly to widen the hypopharynx and thereby relieve upper airway obstruction[ 9 , 10 ]. They have documented positive outcomes with improvement of obstructive events on PSG within 2 weeks of treatment, with normalization of PSG scores at 6–8 months when treatment is discontinued [ 11 ]. Technological advances allowing use of 3D scanning and 3D printing have facilitated the wider adoption of this therapy. At CHW, we were able to introduce the TPP because, in addition to the technologies, we had access to dental specialists and a specialist dental center with a well-appointed dental laboratory (Westmead Centre for Oral Health). This study was undertaken to review the clinical experiences and outcomes in our initial cohort of patients as we introduced the therapy, being the first center in Australia to implement the TPP as an alternate non-surgical intervention for PRS. Methods Electronic medical records for all infants under 6 months of age who were diagnosed with PRS from June 2023 to March 2025 were reviewed retrospectively. Specific analysis was undertaken for infants who underwent TPP treatment at the Children’s Hospital at Westmead (CHW). Ethics approval was provided by the Sydney Children’s Hospital Network (SCHN) Human Research Ethics Committee (ETH01484), which operates in accordance with the National Health and Medical Research Council’s National Statement on Ethical Conduct in Human Research and CPMP/ICH Note for Guidance on Good Clinical Practice. For each infant, we collated the following information: demographic data, comorbid conditions, hospital length of stay, interventions undertaken, any recorded adverse events, and outcomes of sleep studies for the duration of their TPP therapy. One clinician (KW) scored and reported all PSGs to maintain consistency. Our clinical team developed a standardized workflow plan for patients undergoing TPP treatment including the following eligibility and exclusion criteria: Inclusion criteria Pierre Robin Sequence Under 6 months of age Cleft palate Obstructive sleep apnea (OSA) Exclusion criteria Multi-level airway obstruction Multi-organ/system involvement Bulbar dysfunction, impaired swallow Presence of teeth Our primary outcome was the efficacy of TPP in managing severe obstructive sleep apnea in this cohort of patients. This was documented by performing polysomnography at baseline, prior to initial hospital discharge, following placement of a new TPP when infants outgrew their first plate, and at the conclusion of treatment when they outgrew their second plate and polysomnography confirmed resolution of their OSA. Respiratory parameters from the sleep study include AHI, obstructive apnea-hypopnea index (OAHI), obstructive apnea index (OAI), mixed and obstructive apnea index (MOAI), mean oxygen saturations and peak carbon dioxide levels. The OAHI includes hypopneic events and indices which are less consistent among analysts and potentially attributable to central arousals, rather than airway events, so the MOAI is the recommended marker to compare assessments of obstructive events as infants are known to have a higher number of central events at baseline [ 12 – 15 ]. Outcomes are presented as descriptive statistics, or for statistical analyses, the Wilcoxon signed-ranked test. A statistically significant difference was defined by a p-value of < 0.05. Results Over the study period, fourteen infants were admitted to CHW with PRS for airway management. Three were excluded from the TPP pathway, one each due to the following: complex multi-level airway obstruction, previous MDO complicated by infection and unexpected staff absences precluding use of the TPP. One patient, with an intact soft palate, did not tolerate the plate and developed worse clinical symptoms so the TPP was ceased after a 1-week trial. Ten infants completed TPP treatment, including 3 referred from interstate centers. Results are presented for the ten infants who completed their therapy. Table 1 summarizes the demographic data. The majority were male (6/10; 60%), had an antenatal diagnosis of PRS (6/10; 60%) and were born at term (10/11; 91%; 32–40 + 2 weeks). All infants required respiratory support therapies to manage airway obstruction while the TPP was manufactured and fitted and included CPAP (7/10; 70%) and nasopharyngeal airway (NPA) (5/10; 50%; some infants had both). After the TPP was established, these interventions were able to be ceased within 0.5 (0–5, mean range) days. The median length of stay (LOS) in hospital was 36 days with the bulk of the patient management occurring in the outpatient setting. The therapy finished when the infant outgrew their second plate (around 6 months of age) and polysomnography confirmed that OSA had improved/resolved. The median duration of treatment for our cohort was 131.5 (58–208) days (approximately 4–5 months) which is comparably shorter than published data (6–8 months) in Tübingen [ 12 ]. Table 1 Demographic data of patients who underwent TPP (n = 10) Male 6/10 (60%) Female 4/10 (40%) Antenatal diagnosis 6/10 (60%) Gestation 38 + 5 (32–40 + 2; 1 + 2) weeks Birth weight 3030g (2144-3400g; 636g) Comorbidities Cleft palate 10/10 (100%) VOUS 2/10 (20%) Other symptoms/events 4/10 (40%) Intervention CPAP 7/10 (70%) NPA 5/10 (50%) TPP Age at intervention (n = 10) 23 (6–63; 39) days Length of stay (n = 10) 36 (8–45; 17) days Adverse events 8/10 (80%) Duration of treatment (n = 10) 131.5 (58–208; 55.75) days Data expressed in median, range and interquartile range where possible VOUS = genetic variant of unknown significance NPA = nasopharyngeal airway Other symptoms/events included: congenital heart defect, aspiration, scaphocephaly, hip dysplasia. Table 2 shows the polysomnography results, pre and post TPP treatment. At baseline all patients had severe OSA with a median OAHI of 46.7 events/hr and MOAI of 37.8 events/hr. There was evidence of impaired gas exchange with an oxygen nadir of 88% (71–91%) and peak carbon dioxide (CO 2 ) level of 51.7mmHg (46.1–65.8). PSG with the plate in-situ prior to the first hospital discharge demonstrated statistically significant improvement to AHI and OAHI with a median difference of 25.1 and 24.6 events/hr respectively. At the conclusion of TPP treatment, these 10 patients had a median AHI of 8.1 events/hr, OAHI 3.2 events/hr, and MOAI 2.5 events/hr and improvements other PSG parameters included a median drop in AHI (45.2 events/hr), OAHI (43.4 events/hr), OAI (33.8 events/hr), MOAI (37.2 events/hr) and in peak CO 2 levels (10.9mmHg). Small numbers did not allow statistical comparison of oxygen nadir levels. Table 2 Summary of polysomnography data at baseline and post intervention (n = 10) AHI (events/hr) Baseline (n = 10) TPP in (n = 10) Difference Post TPP treatment (n = 10) Difference 53.9 (40-88.5; 21.8) 23.5 (4.6–58.1; 25.9) 25.1 (W = 2; p < 0.05) 8.1 (1.3–13.3; 7.2) 45.2 [W = 0; p < 0.05) OAHI (events/hr) 46.7 (15.5–88.5; 25.2) 21.8 (25.9–76.1; 26.5) 24.6 [W = 6; p < 0.05) 3.2 (0.1–12.4; 5.2) 43.4 [W = 0; p < 0.05] OAI (events/hr) 35 (10.1–79; 35.9) 19.3 (2.1–37.2; 18.2) 20.9 [W = 10; p = 0.08] 2.5 (0-8.3; 3.5) 33.8 [W = 0; p 0.09] 2.9 (0-8.8; 5.1) 37.2 [W = 0; p < 0.05] Oxygen nadir (%) 88 (71–91; 7) 91 (87–95; 4.3) -4.1 [W = 12; p = 0.11) 90.0 (76–94; 7) * CO 2 peak (mmHg) 51.7 (46.1–65.8; 8.6) 46.5 (39-60.2; 12.5) 9.5 [W = 16; p = 0.24] 44.3 (36-55.9; 5.6) 10.9 [W = 0; p < 0.05] Data expressed in median, range and interquartile range *unable to run analysis due to insufficient power, as one patient had the same median oxygen nadir before and after TPP. AHI = apnoea-hypopnoea index; OAHI = obstructive apnoea-hypopnoea index; OAI = obstructive apnoea index; MOAI = mixed and obstructive apnoea index; CO 2 = carbon dioxide The adverse events we noted are summarized in Table 3 . These occurred in 80% of patients and the most common was pressure areas secondary to the plate that required time with the plate out. The occurrence of pressure areas is not unexpected due to the sensitivity of the neonatal oral mucosa, but it was likely more common as we developed expertise in fitting and trimming the plates. The second most common was hospital-acquired bronchiolitis, but during these respiratory infections no infant required additional respiratory support above ongoing use of the TPP. As mentioned above, an infant with an intact soft palate did not tolerate the TPP; this infant developed increased oral secretions and more obstructive episodes clinically with the plate in place. These responses were attributed to intact sensation in the soft palate. Social stressors resulted in another family discharging against medical advice (DAMA) however they attended all outpatient follow-ups and went on to complete the TPP treatment although this child had sufficient residual OSA after TPP treatment that home CPAP therapy was recommended. Contact dermatitis (25%) secondary to steri-strips (3M™, Solventum, United States of America) were managed with topical hydrocortisone cream. Two patients developed oral candidiasis of which one had persistent symptoms requiring a prolonged course of oral nystatin, combined with regular disinfection (benzalkonium chloride) and ultrasonic cleaning of the plate. Nystatin prophylaxis is now prescribed for all patients for the duration of their TPP therapy. Table 3 List of recorded adverse events (n = 8) Mucosal pressure area 6/8 (75%) Bronchiolitis~ 3/8 (38%) Oral candidiasis 2/8 (25%) Contact dermatitis 2/8 (25%) Persistent OSA* 1/8 (13%) DAMA 1/8 (13%) DAMA = discharge against medical advice. This patient discharged against medical advice for social reasons however continued to treatment as an outpatient. ~ 2 patients had rhinovirus; 1 had influenza A *patient had significant improvement from baseline, however had sufficient residual obstructive symptoms to warrant CPAP Discussion This review covers the initial period of implementing TPP therapy to treat infants with PRS at CHW, in Sydney. The data from our first ten infants demonstrated substantial resolution of OSA at the completion of their therapy, confirming its effectiveness for managing airway obstruction in PRS, even as the method was first introduced to our center, with results consistent with those from the team in Tübingen where the TPP originated and is well established [ 12 , 16 ]. Other treatment options available at our hospital in Australia, include CPAP and NPA, with surgical intervention such as mandibular distraction osteogenesis (MDO) and tracheostomy reserved for infants who fail these therapies. NPAs bypass obstruction by placing a modified endotracheal tube through the nasal passage with the tip ending at the distal oropharynx. They offer an interim airway and allow the infant to feed while awaiting mandibular growth [ 1 , 6 , 8 ]. CPAP has been the primary, non-invasive approach traditionally offered at CHW, with worldwide evidence of demonstrating improvement and avoidance of surgical intervention[ 10 , 17 , 18 ]. Factors reported to limit the use of CPAP in other centers include poor patient adherence, as well as limited availability of infant masks that can form a good seal with minimal leak [ 6 , 8 ]. However, at CHW we have demonstrated that the treatment is clinically effective and most infants are able to cease therapy by age 12 months [ 19 ]. Elsewhere in Australia, Queensland Children’s Hospital reported 96% success rates with non-surgical management (primarily NPA and CPAP) of their patients with PRS over a 5-year period[ 1 ]. Perth Children’s Hospital recently reported implementing a standardized approach to managing upper airway obstruction in PRS [ 14 ]. The advantages of the TPP that led us to implement the therapy in Australia include its ability to relieve upper airway obstruction, promote mandibular advancement via the velopharyngeal spur, and to be managed by parents in the community. The velopharyngeal spur facilitates early oral feeding but also offers an alternate option to avoid surgical intervention via MDO [ 16 ]. The TPP also allows greater freedom of movement than a CPAP device that is dependent on access to a power source[ 12 , 20 ]. However, effective implementation of this treatment pathway requires a multi-disciplinary team of orthodontists, pediatric dentists, neonatologists, sleep physicians, ear nose and throat surgeons, speech pathologists, dietitians and specialized nursing staff experienced in managing PRS. It also require access to specialized technicians and equipment such as 3D scanners and printers and an accredited dental laboratory to fabricate the TPP. The team then needs to develop the expertise to manage the workflow and any issues that arise during treatment. We have now developed a clinical model of care and workflow for our center. Inclusion criteria are designed to maximize patient safety and minimize adverse events, which for our cohort were present in 8 patients (Table 3 ). Similar to Tübingen, mucosal pressure areas were most common [ 16 ]. These were managed conservatively with prescribed plate-free time and relieving the pressure areas on the plate without discontinuation of treatment. As highlighted earlier, one patient with an intact soft palate had worsening signs of obstruction within 1 week of starting TPP, thought to be due to intact sensation in the soft palate resulting in irritation and increased oral secretions so we now include an intact palate as an exclusion criterion. It is anticipated that with increasing expertise and experience our team will be able to investigate novel initiatives to overcome some of these problems, as has occurred in Tübingen [ 21 ]. However, parent preferences are an increasing component of management-decision pathways, and as previously mentioned our center maintains expertise in the use of nasal CPAP in the home. One significant change that has already occurred in our center is a reduction in the requests for MDO, with a total of 7 patients between 2019–2023, compared to only 1 over the study period. Ongoing throughput of sufficient patient numbers will be important for maintaining and developing our expertise. Limitations to this study are primarily due to our small patient cohort, limiting statistical power. It also increased the impact of outlying events such as one patient having an intercurrent viral infection at the time of their pre-discharge PSG causing ‘worse’ parameters (Table 2 ). Nonetheless, this study indicates the capacity for a tertiary center that is remote from Europe, such as CHW, to successfully implement this highly specialized therapy. Moving forward, we expect to continue to offer the TPP as a non-invasive treatment option for infants with PRS and significant airway dysfunction, who are referred to our center for management. Progressive expansion of expertise within our specialist teams will facilitate the workflow. Long-term, we can also follow-up and review of patient outcomes to provide objective evidence of whether the significant improvements in upper airway obstruction are maintained. Future plans and conclusions This study demonstrates that we have been able to successfully establish a TPP service in Australia with assistance and guidance from Professor Poets and his team in Tübingen. While this report focuses on patient outcomes regarding upper airway obstruction, ongoing data collection and further studies will assess secondary outcomes such as growth, feed establishment and long-term outcomes. With the implementation of TPP therapy in other centers, data from larger patient numbers should enable future reviews and cohort studies comparing TPP against other therapies as well as consolidating data regarding the utility of TPP as a suitable management option for patients with PRS. Abbreviations CHW The Children’s Hospital at Westmead CO 2 carbon dioxide CPAP continuous positive airway pressure DAMA discharge against medical advice LOS length of stay MAI mixed apnoea index MDO mandibular distraction osteogenesis MOAI mixed and obstructive apnoea index NPA nasopharyngeal airway OAHI obstructive apnoea–hypopnoea index OAI obstructive apnoea index PRS Pierre Robin Sequence SCHN Sydney Children’s Hospital Network TLA tongue–lip adhesion TPP Tübingen palatal plate VOUS variant of unknown significance Declarations Author contributions All authors contributed to the study conception and design. Material preparation, data collection and analysis were performed by Nathan Lieu. The first draft manuscript was written by Nathan Lieu and all authors reviewed and edited previous versions and approved the final manuscript. Supervision was provided by Karen Waters. Disclosure statement: All authors have seen and approved the manuscript. This study was performed at the Children’s Hospital at Westmead, Australia. The authors report no conflicts of interest. Funding: The authors declare that no funds, grants, or other support were received during the preparation of this manuscript. Ethics: Ethics approval was provided by the Sydney Children’s Hospital Network (SCHN) Human Research Ethics Committee (ETH01484), which operates in accordance with the National Health and Medical Research Council’s National Statement on Ethical Conduct in Human Research and CPMP/ICH Note for Guidance on Good Clinical Practice. Consent was waived given this was a retrospective analysis of de-identified data. Acknowledgments We would like to acknowledge the expertise and guidance from Professor Christian Poets from the University Hospital Tübingen in helping establish our TPP service at the Children’s Hospital at Westmead. As well as the ever-willing support of the technical staff and use of the dental laboratory at the Westmead Centre of Oral Health is gratefully acknowledged. We also acknowledge the data provided by the Agency for Clinical Innovation-Neonatal Intensive and Special Care Units' Data Registry regarding incidence of MDO. References Theile H et al (2024) Conservative Airway Management Successful in Majority of Infants With Pierre-Robin Sequence at Queensland Children's Hospital: A Retrospective Review. J Craniofac Surg Karempelis P et al (2020) Associated syndromes in patients with Pierre Robin Sequence. Int J Pediatr Otorhinolaryngol 131:109842 Caouette-Laberge L, Bayet B, Larocque Y (1994) The Pierre Robin sequence: review of 125 cases and evolution of treatment modalities. Plast Reconstr Surg 93(5):934–942 Gangopadhyay N, Mendonca DA, Woo AS (2012) Pierre robin sequence. 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Chest 117(3):916–918 Daniel M et al (2013) Airway, feeding and growth in infants with Robin sequence and sleep apnoea. Int J Pediatr Otorhinolaryngol 77(4):499–503 Poets CF et al (2019) The Tübingen palatal plate approach to Robin sequence: Summary of current evidence. J Craniomaxillofac Surg 47(11):1699–1705 Lim K et al (2022) Should obstructive hypopneas be included when analyzing sleep studies in infants with Robin Sequence? Sleep Med 98:9–12 Goel D et al (2025) Robin Sequence: From Dilemmas to Developing an Adaptable Standardized Stepwise Approach. Acta Paediatr 114(8):1760–1777 Daftary AS et al (2019) Polysomnography Reference Values in Healthy Newborns. J Clin Sleep Med 15(3):437–443 Buchenau W et al (2017) Functional treatment of airway obstruction and feeding problems in infants with Robin sequence. Archives Disease Child - Fetal Neonatal Ed 102(2):F142 Buchenau W et al (2007) A randomized clinical trial of a new orthodontic appliance to improve upper airway obstruction in infants with Pierre Robin sequence. J Pediatr 151(2):145–149 Murage KP et al (2014) Complications associated with neonatal mandibular distraction osteogenesis in the treatment of Robin sequence. J Craniofac Surg 25(2):383–387 Singh J et al (2022) Polysomnography in infants with clinical suspicion of sleep-related breathing disorders. J Clin Sleep Med 18(12):2803–2812 Wiechers C et al (2019) Mandibular growth in infants with Robin sequence treated with the Tübingen palatal plate. Head Face Med 15(1):17 Aretxabaleta M et al (2025) Complete Protocol and Guidelines for the Implementation and Manufacturing of the Tübingen Palatal Plate-An Interdisciplinary Technical Note on the Tübingen Approach for Infants with Robin Sequence. Bioeng (Basel), 12(10) Additional Declarations No competing interests reported. Cite Share Download PDF Status: Under Review Version 1 posted Editorial decision: Revision requested 27 Mar, 2026 Reviews received at journal 26 Mar, 2026 Reviewers agreed at journal 10 Mar, 2026 Reviewers agreed at journal 03 Mar, 2026 Reviewers invited by journal 03 Mar, 2026 Editor assigned by journal 03 Mar, 2026 Submission checks completed at journal 02 Mar, 2026 First submitted to journal 18 Feb, 2026 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-8912918","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":601411359,"identity":"75574d6d-72c5-448f-9707-4cf6d5e48c93","order_by":0,"name":"Nathan Lieu","email":"data:image/png;base64,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","orcid":"","institution":"Children's Hospital at Westmead","correspondingAuthor":true,"prefix":"","firstName":"Nathan","middleName":"","lastName":"Lieu","suffix":""},{"id":601411360,"identity":"3233d658-dc1c-40b3-a7bc-1653c9a6edaf","order_by":1,"name":"Lisa Theis","email":"","orcid":"","institution":"Children's Hospital at Westmead","correspondingAuthor":false,"prefix":"","firstName":"Lisa","middleName":"","lastName":"Theis","suffix":""},{"id":601411377,"identity":"3f61f1c7-a6e0-4267-9e78-cb27e97b4234","order_by":2,"name":"Coral Yeung","email":"","orcid":"","institution":"Children's Hospital at Westmead","correspondingAuthor":false,"prefix":"","firstName":"Coral","middleName":"","lastName":"Yeung","suffix":""},{"id":601411392,"identity":"19f784a9-2427-4b4d-9654-f65bb3cf8d3d","order_by":3,"name":"Richard Widmer","email":"","orcid":"","institution":"Children's Hospital at Westmead","correspondingAuthor":false,"prefix":"","firstName":"Richard","middleName":"","lastName":"Widmer","suffix":""},{"id":601411395,"identity":"323ebd07-9265-490e-a46d-bb5356055581","order_by":4,"name":"Winey Wan","email":"","orcid":"","institution":"Children's Hospital at Westmead","correspondingAuthor":false,"prefix":"","firstName":"Winey","middleName":"","lastName":"Wan","suffix":""},{"id":601411398,"identity":"f2069ca3-826c-4f14-9992-52bf32d156aa","order_by":5,"name":"Cecile Jones","email":"","orcid":"","institution":"Westmead Centre for Oral Health","correspondingAuthor":false,"prefix":"","firstName":"Cecile","middleName":"","lastName":"Jones","suffix":""},{"id":601411400,"identity":"14fb48a8-a207-4905-a008-0376e993efb8","order_by":6,"name":"Bhavesh Mehta","email":"","orcid":"","institution":"Children's Hospital at Westmead","correspondingAuthor":false,"prefix":"","firstName":"Bhavesh","middleName":"","lastName":"Mehta","suffix":""},{"id":601411411,"identity":"8552b0b5-93f5-4b2b-a4b2-81a1e393732d","order_by":7,"name":"Karen A. Waters","email":"","orcid":"","institution":"Children's Hospital at Westmead","correspondingAuthor":false,"prefix":"","firstName":"Karen","middleName":"A.","lastName":"Waters","suffix":""}],"badges":[],"createdAt":"2026-02-19 00:38:17","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-8912918/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-8912918/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":104403644,"identity":"a3a170d3-9c9c-403d-94c5-96df236f77cc","added_by":"auto","created_at":"2026-03-11 12:18:45","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":639319,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-8912918/v1/242afcbd-d6e6-4357-9489-f8c3b3b9bcae.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Evaluating the efficacy of the Tübingen Palatal Plate in managing upper airway obstruction in Pierre Robin Sequence: Experience from a tertiary hospital in Sydney, Australia","fulltext":[{"header":"Brief Summary","content":"\u003cp\u003e\u003cstrong\u003eCurrent Knowledge/Study Rationale:\u0026nbsp;\u003c/strong\u003ePRS is a congenital triad of micrognathia, glossoptosis and upper airway obstruction with a range of management options. This study assesses the effectiveness of TPP for treating airway obstruction in neonates with PRS during the establishment of the method in a new center.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eStudy Impact:\u003c/strong\u003e As the first Australian center to introduce the TPP, the Children\u0026rsquo;s Hospital at Westmead has demonstrated that the TPP is an effective non-invasive treatment modality for managing OSA in infants with PRS while introducing the treatment.\u003c/p\u003e"},{"header":"Introduction","content":"\u003cp\u003ePierre Robin sequence (PRS) is a congenital triad of micrognathia, glossoptosis and airway obstruction[\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e]. Up to 50% of PRS have an underlying genetic component, of which there are currently 34 known genetic syndromes, including Stickler, Treacher Collins and Velocardiofacial syndrome[\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e, \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e]. Cleft palate is also present in up to 90% of cases[\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e]. The incidence of PRS ranges from 1/8500 to 1/14,000 births[\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e], with a higher incidence of 1/5500reported in Australia[\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eFirst line management of airway obstruction in PRS varies worldwide and according to the severity of disease. Graded approaches for managing airway problems usually start with non-invasive approaches that include prone positioning, nasopharyngeal airway (NPA), continuous positive airway pressure (CPAP) and orthodontic devices such as palatal plates. Prone positioning has been shown to be an effective method of relieving obstruction by allowing the mandible and tongue to fall forwards[\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e]. However prone positioning alongside prematurity and smoking are well recognized risk factors for sudden infant death syndrome (SIDS). Centers that use positioning treatment may recommend home monitoring such as apnea devices, and polysomnogram studies to confirm the effect of sleep position on airway obstruction[\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e, \u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e]. This treatment is used infrequently at the Children\u0026rsquo;s Hospital at Westmead (CHW), where infants with PRS are generally referred for treatment because of significant airway and feeding issues, and the severity of their airway obstruction requires more aggressive intervention. Surgical treatment options and tracheostomy are reserved for infants with the most severe airway disturbances.\u003c/p\u003e \u003cp\u003eOrthodontic appliances have been in use to treat PRS for the last 15 years but have gained increasing popularity as evidence for their effectiveness as a non-invasive approach increases and technological developments make their implementation easier. The group in T\u0026uuml;bingen Germany have published broadly on the use of the T\u0026uuml;bingen Palate Plate (TPP), which features a palatal base plate covering the hard palate and cleft, and a velopharyngeal spur which works by displacing the tongue anteriorly to widen the hypopharynx and thereby relieve upper airway obstruction[\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e, \u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e]. They have documented positive outcomes with improvement of obstructive events on PSG within 2 weeks of treatment, with normalization of PSG scores at 6\u0026ndash;8 months when treatment is discontinued [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eTechnological advances allowing use of 3D scanning and 3D printing have facilitated the wider adoption of this therapy. At CHW, we were able to introduce the TPP because, in addition to the technologies, we had access to dental specialists and a specialist dental center with a well-appointed dental laboratory (Westmead Centre for Oral Health). This study was undertaken to review the clinical experiences and outcomes in our initial cohort of patients as we introduced the therapy, being the first center in Australia to implement the TPP as an alternate non-surgical intervention for PRS.\u003c/p\u003e"},{"header":"Methods","content":"\u003cp\u003eElectronic medical records for all infants under 6 months of age who were diagnosed with PRS from June 2023 to March 2025 were reviewed retrospectively. Specific analysis was undertaken for infants who underwent TPP treatment at the Children\u0026rsquo;s Hospital at Westmead (CHW). Ethics approval was provided by the Sydney Children\u0026rsquo;s Hospital Network (SCHN) Human Research Ethics Committee (ETH01484), which operates in accordance with the National Health and Medical Research Council\u0026rsquo;s National Statement on Ethical Conduct in Human Research and CPMP/ICH Note for Guidance on Good Clinical Practice.\u003c/p\u003e \u003cp\u003eFor each infant, we collated the following information: demographic data, comorbid conditions, hospital length of stay, interventions undertaken, any recorded adverse events, and outcomes of sleep studies for the duration of their TPP therapy. One clinician (KW) scored and reported all PSGs to maintain consistency.\u003c/p\u003e \u003cp\u003eOur clinical team developed a standardized workflow plan for patients undergoing TPP treatment including the following eligibility and exclusion criteria:\u003c/p\u003e \u003cp\u003eInclusion criteria\u003c/p\u003e \u003cp\u003e \u003cul\u003e \u003cli\u003e \u003cp\u003ePierre Robin Sequence\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eUnder 6 months of age\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eCleft palate\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eObstructive sleep apnea (OSA)\u003c/p\u003e \u003c/li\u003e \u003c/ul\u003e \u003c/p\u003e \u003cp\u003eExclusion criteria\u003c/p\u003e \u003cp\u003e \u003cul\u003e \u003cli\u003e \u003cp\u003eMulti-level airway obstruction\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eMulti-organ/system involvement\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003eBulbar dysfunction, impaired swallow\u003c/p\u003e \u003c/li\u003e \u003cli\u003e \u003cp\u003ePresence of teeth\u003c/p\u003e \u003c/li\u003e \u003c/ul\u003e \u003c/p\u003e \u003cp\u003eOur primary outcome was the efficacy of TPP in managing severe obstructive sleep apnea in this cohort of patients. This was documented by performing polysomnography at baseline, prior to initial hospital discharge, following placement of a new TPP when infants outgrew their first plate, and at the conclusion of treatment when they outgrew their second plate and polysomnography confirmed resolution of their OSA. Respiratory parameters from the sleep study include AHI, obstructive apnea-hypopnea index (OAHI), obstructive apnea index (OAI), mixed and obstructive apnea index (MOAI), mean oxygen saturations and peak carbon dioxide levels. The OAHI includes hypopneic events and indices which are less consistent among analysts and potentially attributable to central arousals, rather than airway events, so the MOAI is the recommended marker to compare assessments of obstructive events as infants are known to have a higher number of central events at baseline [\u003cspan additionalcitationids=\"CR13 CR14\" citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eOutcomes are presented as descriptive statistics, or for statistical analyses, the Wilcoxon signed-ranked test. A statistically significant difference was defined by a p-value of \u0026lt;\u0026thinsp;0.05.\u003c/p\u003e"},{"header":"Results","content":"\u003cp\u003eOver the study period, fourteen infants were admitted to CHW with PRS for airway management. Three were excluded from the TPP pathway, one each due to the following: complex multi-level airway obstruction, previous MDO complicated by infection and unexpected staff absences precluding use of the TPP. One patient, with an intact soft palate, did not tolerate the plate and developed worse clinical symptoms so the TPP was ceased after a 1-week trial. Ten infants completed TPP treatment, including 3 referred from interstate centers.\u003c/p\u003e \u003cp\u003eResults are presented for the ten infants who completed their therapy. Table\u0026nbsp;\u003cspan refid=\"Tab1\" class=\"InternalRef\"\u003e1\u003c/span\u003e summarizes the demographic data. The majority were male (6/10; 60%), had an antenatal diagnosis of PRS (6/10; 60%) and were born at term (10/11; 91%; 32\u0026ndash;40\u0026thinsp;+\u0026thinsp;2 weeks). All infants required respiratory support therapies to manage airway obstruction while the TPP was manufactured and fitted and included CPAP (7/10; 70%) and nasopharyngeal airway (NPA) (5/10; 50%; some infants had both). After the TPP was established, these interventions were able to be ceased within 0.5 (0\u0026ndash;5, mean range) days. The median length of stay (LOS) in hospital was 36 days with the bulk of the patient management occurring in the outpatient setting. The therapy finished when the infant outgrew their second plate (around 6 months of age) and polysomnography confirmed that OSA had improved/resolved. The median duration of treatment for our cohort was 131.5 (58\u0026ndash;208) days (approximately 4\u0026ndash;5 months) which is comparably shorter than published data (6\u0026ndash;8 months) in T\u0026uuml;bingen [\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\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 data of patients who underwent TPP (n\u0026thinsp;=\u0026thinsp;10)\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"2\"\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 \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMale\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003e6/10 (60%)\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eFemale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e4/10 (40%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAntenatal diagnosis\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e6/10 (60%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eGestation\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e38\u0026thinsp;+\u0026thinsp;5 (32\u0026ndash;40\u0026thinsp;+\u0026thinsp;2; 1\u0026thinsp;+\u0026thinsp;2) weeks\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBirth weight\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3030g (2144-3400g; 636g)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cb\u003eComorbidities\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCleft palate\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e10/10 (100%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eVOUS\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2/10 (20%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOther symptoms/events\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e4/10 (40%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cb\u003eIntervention\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCPAP\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e7/10 (70%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNPA\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e5/10 (50%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cb\u003eTPP\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAge at intervention (n\u0026thinsp;=\u0026thinsp;10)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e23 (6\u0026ndash;63; 39) days\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eLength of stay (n\u0026thinsp;=\u0026thinsp;10)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e36 (8\u0026ndash;45; 17) days\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAdverse events\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e8/10 (80%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eDuration of treatment (n\u0026thinsp;=\u0026thinsp;10)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e131.5 (58\u0026ndash;208; 55.75) days\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\u003eData expressed in median, range and interquartile range where possible\u003c/p\u003e \u003cp\u003eVOUS\u0026thinsp;=\u0026thinsp;genetic variant of unknown significance\u003c/p\u003e \u003cp\u003eNPA\u0026thinsp;=\u0026thinsp;nasopharyngeal airway\u003c/p\u003e \u003cp\u003eOther symptoms/events included: congenital heart defect, aspiration, scaphocephaly, hip dysplasia.\u003c/p\u003e \u003cp\u003eTable\u0026nbsp;\u003cspan refid=\"Tab2\" class=\"InternalRef\"\u003e2\u003c/span\u003e shows the polysomnography results, pre and post TPP treatment. At baseline all patients had severe OSA with a median OAHI of 46.7 events/hr and MOAI of 37.8 events/hr. There was evidence of impaired gas exchange with an oxygen nadir of 88% (71\u0026ndash;91%) and peak carbon dioxide (CO\u003csub\u003e2\u003c/sub\u003e) level of 51.7mmHg (46.1\u0026ndash;65.8). PSG with the plate in-situ prior to the first hospital discharge demonstrated statistically significant improvement to AHI and OAHI with a median difference of 25.1 and 24.6 events/hr respectively. At the conclusion of TPP treatment, these 10 patients had a median AHI of 8.1 events/hr, OAHI 3.2 events/hr, and MOAI 2.5 events/hr and improvements other PSG parameters included a median drop in AHI (45.2 events/hr), OAHI (43.4 events/hr), OAI (33.8 events/hr), MOAI (37.2 events/hr) and in peak CO\u003csub\u003e2\u003c/sub\u003e levels (10.9mmHg). Small numbers did not allow statistical comparison of oxygen nadir levels.\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\u003eSummary of polysomnography data at baseline and post intervention (n\u0026thinsp;=\u0026thinsp;10)\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=\"char\" char=\".\" 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=\"char\" char=\".\" 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\" morerows=\"1\" rowspan=\"2\"\u003e \u003cp\u003eAHI (events/hr)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eBaseline (n\u0026thinsp;=\u0026thinsp;10)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eTPP in (n\u0026thinsp;=\u0026thinsp;10)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eDifference\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003ePost TPP treatment (n\u0026thinsp;=\u0026thinsp;10)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c6\"\u003e \u003cp\u003eDifference\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003e53.9 (40-88.5; 21.8)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003e23.5 (4.6\u0026ndash;58.1; 25.9)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003e25.1 (W\u0026thinsp;=\u0026thinsp;2; p\u0026thinsp;\u0026lt;\u0026thinsp;0.05)\u003c/b\u003e\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003e8.1 (1.3\u0026ndash;13.3; 7.2)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c6\"\u003e \u003cp\u003e\u003cb\u003e45.2 [W\u0026thinsp;=\u0026thinsp;0; p\u0026thinsp;\u0026lt;\u0026thinsp;0.05)\u003c/b\u003e\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOAHI (events/hr)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e46.7 (15.5\u0026ndash;88.5; 25.2)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e21.8 (25.9\u0026ndash;76.1; 26.5)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003e24.6 [W\u0026thinsp;=\u0026thinsp;6; p\u0026thinsp;\u0026lt;\u0026thinsp;0.05)\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c5\"\u003e \u003cp\u003e3.2 (0.1\u0026ndash;12.4; 5.2)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e\u003cb\u003e43.4 [W\u0026thinsp;=\u0026thinsp;0; p\u0026thinsp;\u0026lt;\u0026thinsp;0.05]\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOAI (events/hr)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e35 (10.1\u0026ndash;79; 35.9)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e19.3 (2.1\u0026ndash;37.2; 18.2)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e20.9 [W\u0026thinsp;=\u0026thinsp;10; p\u0026thinsp;=\u0026thinsp;0.08]\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c5\"\u003e \u003cp\u003e2.5 (0-8.3; 3.5)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e\u003cb\u003e33.8 [W\u0026thinsp;=\u0026thinsp;0; p\u0026thinsp;\u0026lt;\u0026thinsp;0.05]\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMOAI (events/hr)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e37.8 (11.4\u0026ndash;86; 32.8)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e23.4 (3.2\u0026ndash;41.1; 29.5)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e19.9 [W\u0026thinsp;=\u0026thinsp;11; p\u0026thinsp;\u0026gt;\u0026thinsp;0.09]\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c5\"\u003e \u003cp\u003e2.9 (0-8.8; 5.1)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e\u003cb\u003e37.2 [W\u0026thinsp;=\u0026thinsp;0; p\u0026thinsp;\u0026lt;\u0026thinsp;0.05]\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOxygen nadir (%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e88 (71\u0026ndash;91; 7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e91 (87\u0026ndash;95; 4.3)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e-4.1 [W\u0026thinsp;=\u0026thinsp;12; p\u0026thinsp;=\u0026thinsp;0.11)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c5\"\u003e \u003cp\u003e90.0 (76\u0026ndash;94; 7)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e*\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCO\u003csub\u003e2\u003c/sub\u003e peak (mmHg)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e51.7 (46.1\u0026ndash;65.8; 8.6)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e46.5 (39-60.2; 12.5)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e9.5 [W\u0026thinsp;=\u0026thinsp;16; p\u0026thinsp;=\u0026thinsp;0.24]\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c5\"\u003e \u003cp\u003e44.3 (36-55.9; 5.6)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e\u003cb\u003e10.9 [W\u0026thinsp;=\u0026thinsp;0; p\u0026thinsp;\u0026lt;\u0026thinsp;0.05]\u003c/b\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\u003eData expressed in median, range and interquartile range\u003c/p\u003e \u003cp\u003e*unable to run analysis due to insufficient power, as one patient had the same median oxygen nadir before and after TPP.\u003c/p\u003e \u003cp\u003eAHI\u0026thinsp;=\u0026thinsp;apnoea-hypopnoea index; OAHI\u0026thinsp;=\u0026thinsp;obstructive apnoea-hypopnoea index; OAI\u0026thinsp;=\u0026thinsp;obstructive apnoea index; MOAI\u0026thinsp;=\u0026thinsp;mixed and obstructive apnoea index; CO\u003csub\u003e2\u003c/sub\u003e\u0026thinsp;=\u0026thinsp;carbon dioxide\u003c/p\u003e \u003cp\u003eThe adverse events we noted are summarized in Table \u003cspan refid=\"Tab3\" class=\"InternalRef\"\u003e3\u003c/span\u003e. These occurred in 80% of patients and the most common was pressure areas secondary to the plate that required time with the plate out. The occurrence of pressure areas is not unexpected due to the sensitivity of the neonatal oral mucosa, but it was likely more common as we developed expertise in fitting and trimming the plates. The second most common was hospital-acquired bronchiolitis, but during these respiratory infections no infant required additional respiratory support above ongoing use of the TPP. As mentioned above, an infant with an intact soft palate did not tolerate the TPP; this infant developed increased oral secretions and more obstructive episodes clinically with the plate in place. These responses were attributed to intact sensation in the soft palate. Social stressors resulted in another family discharging against medical advice (DAMA) however they attended all outpatient follow-ups and went on to complete the TPP treatment although this child had sufficient residual OSA after TPP treatment that home CPAP therapy was recommended. Contact dermatitis (25%) secondary to steri-strips (3M\u0026trade;, Solventum, United States of America) were managed with topical hydrocortisone cream. Two patients developed oral candidiasis of which one had persistent symptoms requiring a prolonged course of oral nystatin, combined with regular disinfection (benzalkonium chloride) and ultrasonic cleaning of the plate. Nystatin prophylaxis is now prescribed for all patients for the duration of their TPP therapy.\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\u003eList of recorded adverse events (n\u0026thinsp;=\u0026thinsp;8)\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"2\"\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 \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMucosal pressure area\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003e6/8 (75%)\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eBronchiolitis~\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3/8 (38%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOral candidiasis\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2/8 (25%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eContact dermatitis\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2/8 (25%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePersistent OSA*\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1/8 (13%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eDAMA\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1/8 (13%)\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\u003eDAMA\u0026thinsp;=\u0026thinsp;discharge against medical advice. This patient discharged against medical advice for social reasons however continued to treatment as an outpatient.\u003c/p\u003e \u003cp\u003e~\u0026thinsp;2 patients had rhinovirus; 1 had influenza A\u003c/p\u003e \u003cp\u003e*patient had significant improvement from baseline, however had sufficient residual obstructive symptoms to warrant CPAP\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eThis review covers the initial period of implementing TPP therapy to treat infants with PRS at CHW, in Sydney. The data from our first ten infants demonstrated substantial resolution of OSA at the completion of their therapy, confirming its effectiveness for managing airway obstruction in PRS, even as the method was first introduced to our center, with results consistent with those from the team in T\u0026uuml;bingen where the TPP originated and is well established [\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e, \u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eOther treatment options available at our hospital in Australia, include CPAP and NPA, with surgical intervention such as mandibular distraction osteogenesis (MDO) and tracheostomy reserved for infants who fail these therapies. NPAs bypass obstruction by placing a modified endotracheal tube through the nasal passage with the tip ending at the distal oropharynx. They offer an interim airway and allow the infant to feed while awaiting mandibular growth [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e, \u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e]. CPAP has been the primary, non-invasive approach traditionally offered at CHW, with worldwide evidence of demonstrating improvement and avoidance of surgical intervention[\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e, \u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e, \u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e]. Factors reported to limit the use of CPAP in other centers include poor patient adherence, as well as limited availability of infant masks that can form a good seal with minimal leak [\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e, \u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e]. However, at CHW we have demonstrated that the treatment is clinically effective and most infants are able to cease therapy by age 12 months [\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e]. Elsewhere in Australia, Queensland Children\u0026rsquo;s Hospital reported 96% success rates with non-surgical management (primarily NPA and CPAP) of their patients with PRS over a 5-year period[\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e]. Perth Children\u0026rsquo;s Hospital recently reported implementing a standardized approach to managing upper airway obstruction in PRS [\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe advantages of the TPP that led us to implement the therapy in Australia include its ability to relieve upper airway obstruction, promote mandibular advancement via the velopharyngeal spur, and to be managed by parents in the community. The velopharyngeal spur facilitates early oral feeding but also offers an alternate option to avoid surgical intervention via MDO [\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e]. The TPP also allows greater freedom of movement than a CPAP device that is dependent on access to a power source[\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e, \u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e]. However, effective implementation of this treatment pathway requires a multi-disciplinary team of orthodontists, pediatric dentists, neonatologists, sleep physicians, ear nose and throat surgeons, speech pathologists, dietitians and specialized nursing staff experienced in managing PRS. It also require access to specialized technicians and equipment such as 3D scanners and printers and an accredited dental laboratory to fabricate the TPP. The team then needs to develop the expertise to manage the workflow and any issues that arise during treatment.\u003c/p\u003e \u003cp\u003e We have now developed a clinical model of care and workflow for our center. Inclusion criteria are designed to maximize patient safety and minimize adverse events, which for our cohort were present in 8 patients (Table\u0026nbsp;\u003cspan refid=\"Tab3\" class=\"InternalRef\"\u003e3\u003c/span\u003e). Similar to T\u0026uuml;bingen, mucosal pressure areas were most common [\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e]. These were managed conservatively with prescribed plate-free time and relieving the pressure areas on the plate without discontinuation of treatment. As highlighted earlier, one patient with an intact soft palate had worsening signs of obstruction within 1 week of starting TPP, thought to be due to intact sensation in the soft palate resulting in irritation and increased oral secretions so we now include an intact palate as an exclusion criterion. It is anticipated that with increasing expertise and experience our team will be able to investigate novel initiatives to overcome some of these problems, as has occurred in T\u0026uuml;bingen [\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e]. However, parent preferences are an increasing component of management-decision pathways, and as previously mentioned our center maintains expertise in the use of nasal CPAP in the home. One significant change that has already occurred in our center is a reduction in the requests for MDO, with a total of 7 patients between 2019\u0026ndash;2023, compared to only 1 over the study period. Ongoing throughput of sufficient patient numbers will be important for maintaining and developing our expertise.\u003c/p\u003e \u003cp\u003eLimitations to this study are primarily due to our small patient cohort, limiting statistical power. It also increased the impact of outlying events such as one patient having an intercurrent viral infection at the time of their pre-discharge PSG causing \u0026lsquo;worse\u0026rsquo; parameters (Table \u003cspan refid=\"Tab2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). Nonetheless, this study indicates the capacity for a tertiary center that is remote from Europe, such as CHW, to successfully implement this highly specialized therapy. Moving forward, we expect to continue to offer the TPP as a non-invasive treatment option for infants with PRS and significant airway dysfunction, who are referred to our center for management. Progressive expansion of expertise within our specialist teams will facilitate the workflow. Long-term, we can also follow-up and review of patient outcomes to provide objective evidence of whether the significant improvements in upper airway obstruction are maintained.\u003c/p\u003e\n\u003ch3\u003eFuture plans and conclusions\u003c/h3\u003e\n\u003cp\u003eThis study demonstrates that we have been able to successfully establish a TPP service in Australia with assistance and guidance from Professor Poets and his team in T\u0026uuml;bingen. While this report focuses on patient outcomes regarding upper airway obstruction, ongoing data collection and further studies will assess secondary outcomes such as growth, feed establishment and long-term outcomes. With the implementation of TPP therapy in other centers, data from larger patient numbers should enable future reviews and cohort studies comparing TPP against other therapies as well as consolidating data regarding the utility of TPP as a suitable management option for patients with PRS.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cdiv class=\"DefinitionList\"\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCHW\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eThe Children\u0026rsquo;s Hospital at Westmead\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCO\u003csub\u003e2\u003c/sub\u003e\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003ecarbon dioxide\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCPAP\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003econtinuous positive airway pressure\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eDAMA\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003edischarge against medical advice\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eLOS\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003elength of stay\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eMAI\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003emixed apnoea index\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eMDO\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003emandibular distraction osteogenesis\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eMOAI\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003emixed and obstructive apnoea index\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eNPA\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003enasopharyngeal airway\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eOAHI\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eobstructive apnoea\u0026ndash;hypopnoea index\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eOAI\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eobstructive apnoea index\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003ePRS\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003ePierre Robin Sequence\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eSCHN\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eSydney Children\u0026rsquo;s Hospital Network\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eTLA\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003etongue\u0026ndash;lip adhesion\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eTPP\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eT\u0026uuml;bingen palatal plate\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eVOUS\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003evariant of unknown significance\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003c/div\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eAuthor contributions\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eAll authors contributed to the study conception and design. Material preparation, data collection and analysis were performed by Nathan Lieu. The first draft manuscript was written by Nathan Lieu and all authors reviewed and edited previous versions and approved the final manuscript. Supervision was provided by Karen Waters.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eDisclosure statement:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eAll authors have seen and approved the manuscript. This study was performed at the Children\u0026rsquo;s Hospital at Westmead, Australia. The authors report no conflicts of interest.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding:\u0026nbsp;\u003c/strong\u003eThe authors declare that no funds, grants, or other support were received during the preparation of this manuscript.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eEthics:\u0026nbsp;\u003c/strong\u003eEthics approval was provided by the Sydney Children\u0026rsquo;s Hospital Network (SCHN) Human Research Ethics Committee (ETH01484), which operates in accordance with the National Health and Medical Research Council\u0026rsquo;s National Statement on Ethical Conduct in Human Research and CPMP/ICH Note for Guidance on Good Clinical Practice. Consent was waived given this was a retrospective analysis of de-identified data.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgments\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWe would like to acknowledge the expertise and guidance from Professor Christian Poets from the University Hospital Tübingen in helping establish our TPP service at the Children’s Hospital at Westmead. As well as the ever-willing support of the technical staff and use of the dental laboratory at the Westmead Centre of Oral Health is gratefully acknowledged. We also acknowledge the data provided by the Agency for Clinical Innovation-Neonatal Intensive and Special Care Units' Data Registry regarding incidence of MDO.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eTheile H et al (2024) Conservative Airway Management Successful in Majority of Infants With Pierre-Robin Sequence at Queensland Children's Hospital: A Retrospective Review. J Craniofac Surg\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eKarempelis P et al (2020) Associated syndromes in patients with Pierre Robin Sequence. Int J Pediatr Otorhinolaryngol 131:109842\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eCaouette-Laberge L, Bayet B, Larocque Y (1994) The Pierre Robin sequence: review of 125 cases and evolution of treatment modalities. Plast Reconstr Surg 93(5):934\u0026ndash;942\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGangopadhyay N, Mendonca DA, Woo AS (2012) Pierre robin sequence. Semin Plast Surg 26(2):76\u0026ndash;82\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eJunaid M et al (2022) Epidemiology of Rare Craniofacial Anomalies: Retrospective Western Australian Population Data Linkage Study. J Pediatr 241:162\u0026ndash;172e9\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGomez OJ, Lopez A, Gutierrez E (2024) Pierre Robin Sequence: An Updated Evidence-Based Treatment Proposal. J Craniofac Surg\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eC\u0026ocirc;t\u0026eacute; A et al (2015) Pierre Robin sequence: review of diagnostic and treatment challenges. Int J Pediatr Otorhinolaryngol 79(4):451\u0026ndash;464\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eWaters KA (2021) Positioning as a conservative treatment option in infants with micrognathia and/or cleft. Semin Fetal Neonatal Med 26(6):101282\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eAbel F et al (2012) The successful use of the nasopharyngeal airway in Pierre Robin sequence: an 11-year experience. Arch Dis Child 97(4):331\u0026ndash;334\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLi KK, Riley RW, Guilleminault C (2000) An unreported risk in the use of home nasal continuous positive airway pressure and home nasal ventilation in children: mid-face hypoplasia. Chest 117(3):916\u0026ndash;918\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDaniel M et al (2013) Airway, feeding and growth in infants with Robin sequence and sleep apnoea. Int J Pediatr Otorhinolaryngol 77(4):499\u0026ndash;503\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003ePoets CF et al (2019) The T\u0026uuml;bingen palatal plate approach to Robin sequence: Summary of current evidence. J Craniomaxillofac Surg 47(11):1699\u0026ndash;1705\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLim K et al (2022) Should obstructive hypopneas be included when analyzing sleep studies in infants with Robin Sequence? Sleep Med 98:9\u0026ndash;12\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGoel D et al (2025) Robin Sequence: From Dilemmas to Developing an Adaptable Standardized Stepwise Approach. Acta Paediatr 114(8):1760\u0026ndash;1777\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDaftary AS et al (2019) Polysomnography Reference Values in Healthy Newborns. J Clin Sleep Med 15(3):437\u0026ndash;443\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBuchenau W et al (2017) Functional treatment of airway obstruction and feeding problems in infants with Robin sequence. Archives Disease Child - Fetal Neonatal Ed 102(2):F142\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBuchenau W et al (2007) A randomized clinical trial of a new orthodontic appliance to improve upper airway obstruction in infants with Pierre Robin sequence. J Pediatr 151(2):145\u0026ndash;149\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMurage KP et al (2014) Complications associated with neonatal mandibular distraction osteogenesis in the treatment of Robin sequence. J Craniofac Surg 25(2):383\u0026ndash;387\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSingh J et al (2022) Polysomnography in infants with clinical suspicion of sleep-related breathing disorders. J Clin Sleep Med 18(12):2803\u0026ndash;2812\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eWiechers C et al (2019) Mandibular growth in infants with Robin sequence treated with the T\u0026uuml;bingen palatal plate. Head Face Med 15(1):17\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eAretxabaleta M et al (2025) Complete Protocol and Guidelines for the Implementation and Manufacturing of the T\u0026uuml;bingen Palatal Plate-An Interdisciplinary Technical Note on the T\u0026uuml;bingen Approach for Infants with Robin Sequence. Bioeng (Basel), 12(10)\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":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"european-journal-of-pediatrics","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"ejpe","sideBox":"Learn more about [European Journal of Pediatrics](https://www.springer.com/journal/431)","snPcode":"431","submissionUrl":"https://submission.nature.com/new-submission/431/3","title":"European Journal of Pediatrics","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false},"keywords":"Pierre Robin Sequence, Tübingen Palatal Plate, obstructive sleep apnoea, paediatrics","lastPublishedDoi":"10.21203/rs.3.rs-8912918/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-8912918/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003eStudy objective:\u003c/h2\u003e \u003cp\u003eTo evaluate the efficacy of the T\u0026uuml;bingen Palatal Plate in managing upper airway obstruction in Pierre Robin Sequence during its introduction into a tertiary hospital in Sydney, Australia.\u003c/p\u003e\u003ch2\u003eMethods\u003c/h2\u003e \u003cp\u003eWe evaluated polysomnography and treatment outcomes in children diagnosed with PRS and managed with TPP from October 2023 - March 2025 at the Children\u0026rsquo;s Hospital in Westmead. A level 1 polysomnography was performed at baseline, prior to discharge from hospital and at the end of treatment. Sleep study parameters were compared between initial and follow-up studies.\u003c/p\u003e\u003ch2\u003eResults\u003c/h2\u003e \u003cp\u003eFrom 14 admissions of infants with PRS, ten infants completed TPP treatment. At baseline all ten had severe obstructive sleep apnea (OSA) with a median apnea-hypopnea index (AHI) of 53.9 events/hr, and mixed and obstructive apnea index (MOAI) of 37.8 events/hr. There was evidence of impaired gas exchange with an oxygen nadir of 88% (71\u0026ndash;91%) and peak carbon dioxide (CO\u003csub\u003e2\u003c/sub\u003e) level of 51.7mmHg (46.1\u0026ndash;65.8). The final median AHI of 8.1 (2.9\u0026ndash;13.3) events/hr, and MOAI of 2.9 (1.3\u0026ndash;8.8) events/hr. When compared to baseline there were statistically significant improvements in AHI, OAHI, MOAI and peak CO\u003csub\u003e2\u003c/sub\u003e levels. Excluded Infants included one patient with an intact soft palate, and three others with clinical and/or practical issues preventing implementation of the TPP.\u003c/p\u003e\u003ch2\u003eConclusions\u003c/h2\u003e \u003cp\u003eThis study demonstrates that when first introduced in a new health setting, TPP therapy is effective for treating airway obstruction in infants with PRS.\u003c/p\u003e","manuscriptTitle":"Evaluating the efficacy of the Tübingen Palatal Plate in managing upper airway obstruction in Pierre Robin Sequence: Experience from a tertiary hospital in Sydney, Australia","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2026-03-08 07:53:42","doi":"10.21203/rs.3.rs-8912918/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2026-03-28T00:18:00+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2026-03-26T19:50:08+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"22009965126050653478272205423968611883","date":"2026-03-11T02:23:56+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"301475303337076982665864161029156380647","date":"2026-03-04T02:52:19+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2026-03-03T14:27:56+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2026-03-03T10:52:37+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2026-03-02T23:37:26+00:00","index":"","fulltext":""},{"type":"submitted","content":"European Journal of Pediatrics","date":"2026-02-19T00:26:44+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"european-journal-of-pediatrics","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"ejpe","sideBox":"Learn more about [European Journal of Pediatrics](https://www.springer.com/journal/431)","snPcode":"431","submissionUrl":"https://submission.nature.com/new-submission/431/3","title":"European Journal of Pediatrics","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"Springer Hybrid","inReviewEnabled":true,"inReviewRevisionsEnabled":false}}],"origin":"","ownerIdentity":"075eaa1f-7d18-471c-89d9-1246de9c2f15","owner":[],"postedDate":"March 8th, 2026","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2026-05-18T19:23:24+00:00","versionOfRecord":[],"versionCreatedAt":"2026-03-08 07:53:42","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-8912918","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-8912918","identity":"rs-8912918","version":["v1"]},"buildId":"XKTyCvWXoU3ODBz1xrDgd","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}