Skip to main content
Key Specialties
Cardiology Diabetology Endocrinology Gastroenterology Geriatrics and Gerontology Gynecology and Obstetrics Hematology Infectious Disease Internal Medicine Nephrology Neurology Oncology Pediatrics Respiratory Medicine Rheumatology Surgery
Congress Coverage
2024 ACC Congress 2023 ESMO Congress 2023 EASD Congress 2023 ERS Congress 2023 ESC Congress
Clinical Collections
Advances in Alzheimer’s

Keynote webinar | Spotlight on medication adherence

LIVE: Thursday 27th June 2024, 18:00-19:30 (CEST)
Join our expert panel to discover why you need to understand the drivers of non-adherence in your patients, and how you can optimize medication adherence in your clinics to drastically improve patient outcomes.
ADVANCED SEARCH
Log in
Top
World Journal of Pediatrics

Open Access 05-04-2024 | Original Article

Prospective cohort study on facial profile changes in infants with Robin sequence and healthy controls

Authors: Cornelia Wiechers, Julian Sowula, Karen Kreutzer, Christoph E. Schwarz, Christina Weismann, Michael Krimmel, Christian F. Poets, Bernd Koos

Published in: World Journal of Pediatrics

Login to get access

Abstract

Background

Various conservative and surgical approaches exist to treat Robin sequence (RS), but their effects on facial profile and mandibular catch-up growth are unclear. A functional treatment concept, used in our centre for 25 years, includes an individualized palatal plate with a velo-pharyngeal extension and intensive feeding training.

Methods

We performed a prospective study to objectively describe facial profiles in infants with RS treated with the above concept. Infants with isolated RS were admitted to our tertiary perinatal and national referral centre for craniofacial malformations between May 2018 and Nov 2019. Infants with RS received 3D-photographs during clinically indicated visits. Healthy controls were recruited from Dec 2018 to Sep 2019 and received 3D-photographs every 3 months. The digitally measured jaw index (JI), defined as alveolar overjet (O) x maxillary arch (U)/mandibular arch (L), and the soft tissue reference points A’-point, Nasion’, B’-point angle (ANB’), describing the relative position of maxilla to mandible, were evaluated. Linear mixed models were used to examine time trajectories in JI and ANB’.

Results

A total of 207 3D images, obtained in 19 infants with RS and 32 controls, were analysed. JI and ANB’ decreased over time in both groups [for JI − 0.18 (95% CI − 0.25 to − 0.10); for ANB’: − 0.40° per month [(95% CI − 0.48 to − 0.32)]] but remained lower in controls [for JI − 2.5 (95% CI − 3.2 to − 1.8); for ANB’-1.7° (95% CI − 2.4 to − 1.0)]. Also, the ANB’ model showed a significant effect of the interaction term diagnosis x age.

Conclusions

Based on longitudinal 3D images, we describe changes in objective parameters of facial profile in infants with and without RS during the first year of life. Our findings indicate catch-up growth in infants treated for RS.
Video Abstract
Appendix
Available only for authorised users
Literature
1.
Evans KN, Sie KC, Hopper RA, Glass RP, Hing AV, Cunningham ML. Robin sequence: from diagnosis to development of an effective management plan. Pediatrics. 2011;127:936–48.CrossRefPubMedPubMedCentral
2.
Poets CF, Bacher M. Treatment of upper airway obstruction and feeding problems in robin-like phenotype. J Pediatr. 2011;159:887–92.CrossRefPubMed
3.
van Lieshout MJ, Joosten KF, Mathijssen IM, Koudstaal MJ, Hoeve HL, van der Schroeff MP, et al. Robin sequence: A European survey on current practice patterns. J Craniomaxillofac Surg. 2015;43:1626–31. CrossRefPubMed
4.
Breugem CC, Evans KN, Poets CF, Suri S, Picard A, Filip C, et al. Best practices for the diagnosis and evaluation of infants with robin sequence: a clinical consensus report. JAMA Pediatr. 2016;170:894–902.  CrossRefPubMed
5.
Purnell CA, Janes LE, Klosowiak JL, Gosain AK. Mandibular catch-up growth in Pierre Robin sequence: a systematic review. Cleft Palate Craniofac J. 2019;56:168–76.CrossRefPubMed
6.
Eriksen J, Hermann NV, Darvann TA, Kreiborg S. Early postnatal development of the mandible in children with isolated cleft palate and children with nonsyndromic Robin sequence. Cleft Palate Craniofac J. 2006;43:160–7. CrossRefPubMed
7.
Hermann NV, Kreiborg S, Darvann TA, Jensen BL, Dahl E, Bolund S. Craniofacial morphology and growth comparisons in children with Robin Sequence, isolated cleft palate, and unilateral complete cleft lip and palate. Cleft Palate Craniofac J. 2003;40:373–96.CrossRefPubMed
8.
Mao Z, Ye L. Effects of mandibular distraction osteogenesis on three-dimensional upper airway anatomy in newborns affected by isolated Pierre Robin sequence. J Craniofac Surg. 2021;32:1459–63.CrossRefPubMed
9.
Ramieri V, Basile E, Bosco G, Caresta E, Papoff P, Cascone P. Three-dimensional airways volumetric analysis before and after fast and early mandibular osteodistraction. J Craniomaxillofac Surg. 2017;45:377–80. CrossRefPubMed
10.
Abramson ZR, Susarla SM, Lawler ME, Peacock ZS, Troulis MJ, Kaban LB. Effects of mandibular distraction osteogenesis on three-dimensional airway anatomy in children with congenital micrognathia. J Oral Maxillofac Surg. 2013;71:90–7.CrossRefPubMed
11.
Breugem CC, Logjes RJH, Nolte JW, Flores RL. Advantages and disadvantages of mandibular distraction in Robin sequence. Semin Fetal Neonatal Med. 2021;26:101283.CrossRefPubMed
12.
van der Haven I, Mulder JW, van der Wal KG, Hage JJ, de Lange-de Klerk ES, Haumann TJ. The jaw index: new guide defining micrognathia in newborns. Cleft Palate Craniofac J. 1997;34:240–1.CrossRefPubMed
13.
Wiechers C, Buchenau W, Arand J, Oertel AF, Peters K, Muller-Hagedorn S, et al. Mandibular growth in infants with Robin sequence treated with the Tubingen palatal plate. Head Face Med. 2019;15:17.CrossRefPubMedPubMedCentral
14.
Wiechers C, Arand J, Koos B, Poets CF. Evidence and practical aspects of treatment with the Tubingen palatal plate. Semin Fetal Neonatal Med. 2021;26:101281.CrossRefPubMed
15.
16.
Hermann NV, Kreiborg S, Darvann TA, Jensen BL, Dahl E, Bolund S. Early craniofacial morphology and growth in children with nonsyndromic Robin Sequence. Cleft Palate Craniofac J. 2003;40:131–43.CrossRefPubMed
17.
Suri S, Ross RB, Tompson BD. Craniofacial morphology and adolescent facial growth in Pierre Robin sequence. Am J Orthod Dentofacial Orthop. 2010;137:763–74.CrossRefPubMed
18.
Matsuda A, Suda N, Motohashi N, Tsuji M, Ohyama K. Skeletal characteristics and treatment outcome of five patients with Robin sequence. Angle Orthod. 2006;76:898–908. CrossRefPubMed
19.
Laitinen SH, Ranta RE. Cephalometric measurements in patients with Pierre Robin syndrome and isolated cleft palate. Scand J Plast Reconstr Surg Hand Surg. 1992;26:177–83.CrossRefPubMed
20.
Daskalogiannakis J, Ross RB, Tompson BD. The mandibular catch-up growth controversy in Pierre Robin sequence. Am J Orthod Dentofacial Orthop. 2001;120:280–5. CrossRefPubMed
21.
Figueroa AA, Glupker TJ, Fitz MG, BeGole EA. Mandible, tongue, and airway in Pierre Robin sequence: a longitudinal cephalometric study. Cleft Palate Craniofac J. 1991;28:425–34.CrossRefPubMed
22.
Vegter F, Hage JJ, Mulder JW. Pierre Robin syndrome: mandibular growth during the first year of life. Ann Plast Surg. 1999;42:154–7.PubMed
23.
Varadarajan S, Balaji TM, Raj AT, Gupta AA, Patil S, Alhazmi TH, et al. Genetic mutations associated with pierre robin syndrome/sequence: a systematic review. Mol Syndromol. 2021;12:69–86. CrossRefPubMedPubMedCentral
24.
25.
Enlow DH, Harris DB. A study of the postnatal growth of the human mandible. Am J Orthod. 1964;50:25–50.CrossRef
26.
Cozza P, Baccetti T, Franchi L, De Toffol L, McNamara JA Jr. Mandibular changes produced by functional appliances in Class II malocclusion: a systematic review. Am J Orthod Dentofacial Orthop. 2006;129:599.e1-12 (Discussion e1-6).CrossRefPubMed
27.
Limbrock GJ, Castillo-Morales R, Hoyer H, Stover B, Onufer CN. The Castillo-Morales approach to orofacial pathology in Down syndrome. Int J Orofacial Myol. 1993;19:30–7.CrossRef
28.
von Lukowicz M, Herzog N, Ruthardt S, Quante M, Iven G, Poets CF. Effect of a 1-week intense myofunctional training on obstructive sleep apnoea in children with Down syndrome. Arch Dis Child. 2019;104:275–9. CrossRef
29.
Camacho M, Certal V, Abdullatif J, Zaghi S, Ruoff CM, Capasso R, et al. Myofunctional therapy to treat obstructive sleep apnea: a systematic review and meta-analysis. Sleep. 2015;38:669–75. CrossRefPubMedPubMedCentral
30.
Li J, Liu Y, Yuan X, Ding X, Guo Y, Qiu L. Dynamical changes of mandible and upper airway after mandibular distraction osteogenesis in pierre robin sequence. J Craniofac Surg. 2020;31:513–6. CrossRefPubMed
31.
Pfaff MJ, Metzler P, Kim Y, Steinbacher DM. Mandibular volumetric increase following distraction osteogenesis. J Plast Reconstr Aesthet Surg. 2014;67:1209–14. CrossRefPubMed
32.
Roy S, Munson PD, Zhao L, Holinger LD, Patel PK. CT analysis after distraction osteogenesis in Pierre Robin Sequence. Laryngoscope. 2009;119:380–6. CrossRefPubMed
33.
Volk AS, Davis MJ, Narawane AM, Abu-Ghname A, Dempsey RF, Lambert EM, et al. Quantification of mandibular morphology in Pierre Robin sequence to optimize mandibular distraction osteogenesis. Cleft Palate Craniofac J. 2020;57:1032–40. CrossRefPubMed
34.
Almajed A, Viezel-Mathieu A, Gilardino MS, Flores RL, Tholpady SS, Côté A. Outcome following surgical interventions for micrognathia in infants with pierre robin sequence: a systematic review of the literature. Cleft Palate Craniofac J. 2017;54:32–42. CrossRefPubMed
35.
Murage KP, Costa MA, Friel MT, Havlik RJ, Tholpady SS, Flores RL. Complications associated with neonatal mandibular distraction osteogenesis in the treatment of Robin sequence. J Craniofac Surg. 2014;25:383–7.  CrossRefPubMed
Metadata
Title
Prospective cohort study on facial profile changes in infants with Robin sequence and healthy controls
Authors
Cornelia Wiechers
Julian Sowula
Karen Kreutzer
Christoph E. Schwarz
Christina Weismann
Michael Krimmel
Christian F. Poets
Bernd Koos
Publication date
05-04-2024
Publisher
Springer Nature Singapore
Published in
World Journal of Pediatrics
Print ISSN: 1708-8569
Electronic ISSN: 1867-0687
DOI
https://doi.org/10.1007/s12519-024-00797-z