This study aimed to analyze the morphology of the hypoplasic mandible and its evolution during the growth period to better understand how it differs from the pediatric healthy mandible.
Method:
Three-dimensional mandibular models of hypoplasic and healthy children aged from 39 gestational weeks to 7 years old were analyzed with a morphometric method including data clustering. Morphological distinctions between pathological and healthy mandibles were highlighted. Bilateral and unilateral mandibular hypoplasia were distinguished.
Results:
The study sample was composed of 31 hypoplasic children and as many sex- and age-matched healthy children. Morphological distinctions between pathological and healthy mandibles were highlighted only from the first year of life. In bilateral hypoplasia, the overall mandibular dimensions were reduced while there was only a ramus asymmetry in unilateral mandibular hypoplasia (mean ± SD of the difference between the Grp03c and Grp03b subgroups: 6.80 ± 6.37 – P value = 1.64e–3 for the height of the left ramus versus 0.18 ± 4.18 – P value = .82 for the height of the right ramus). Supervised classification trees were built to identify the pathology and discriminate unilateral from bilateral mandibular hypoplasia (prediction rates = 81% and 84%, respectively).
Conclusions:
Based on a morphometric analysis, we demonstrated that mandibular hypoplasia significantly impacts the mandibular morphology only from the first year of life, with a distinction between bilateral and unilateral hypoplasia.
BreugemCCEvansKNPoetsCFSuriSPicardAFilipCPaesECMehendaleFVSaalHMBasartH. Best practices for the diagnosis and evaluation of infants with Robin sequence: a clinical consensus report. JAMA Pediatr. 2016;170(9):894–902.
2.
CaptierGFaureJ-MBaeumlerMCanovasFDematteiCDauresJ-P. Prenatal assessment of the antero-posterior jaw relationship in human fetuses: from anatomical to ultrasound cephalometric analysis. Cleft Palate Craniofac J. 2011;48(4):465–472.
3.
ChengATLCorkeMLoughran-FowldsABirmanCHaywardPWatersKA. Distraction osteogenesis and glossopexy for Robin sequence with airway obstruction. ANZ J Surg. 2011;81(5):320–325.
4.
ChengMTLeviBHyunJSLoDDMontoroDTLisieckiJBradleyJPBuchmanSRLongakerMTWanDC. Pierre Robin sequence and Treacher Collins hypoplastic mandible comparison using three-dimensional morphometric analysis. J Craniofac Surg. 2012;23(7 suppl 1):1959–1963.
5.
CohenMM.Robin sequences and complexes: causal heterogeneity and pathogenetic/phenotypic variability. Am J Med Genet. 1999; 84(4):311–315.
6.
CohenNCohenEGaieroAZeccaSFicheraGBaldiFGiodanettoJFMercierJMCohenA. Maxillofacial features and systemic malformations in expanded spectrum Hemifacial Microsomia. Am J Med Genet A. 2017;173(5):1208–1218.
7.
CoutierLGuyonAReixPFrancoP. Impact of prone positioning in infants with Pierre Robin sequence: a polysomnography study. Sleep Med. 2019;54:257–261.
8.
de Buys RoessinghASHerzogGHohlfeldJ. Respiratory distress in Pierre Robin: successful use of pharyngeal tube. J Pediatr Surg. 2007;42(9):1495–1499.
9.
Di PasquoEAmielJRothPMalanVLindKChalouhiCSoupreVGordonCTLyonnetSSalomonLJ, et al.Efficiency of prenatal diagnosis in Pierre Robin sequence. Prenat Diagn. 2017;37(11):1169–1175.
10.
EriksenJHermannNVDarvannTAKreiborgS. Early postnatal development of the mandible in children with isolated cleft palate and children with nonsyndromic Robin sequence. Cleft Palate Craniofac J. 2006;43(2):160–167.
11.
EvansKNSieKCHopperRAGlassRPHingAVCunninghamML. Robin sequence: from diagnosis to development of an effective management plan. Pediatrics. 2011;127(5):936–948.
12.
GiudiceABaroneSBelhousKMoriceASoupreVBennardoFBoddaertNVazquezM-PAbadieVPicardA.Pierre Robin sequence: a comprehensive narrative review of the literature over time. J Stomatol Oral Maxillofac Surg. 2018;119(5):419–428.
13.
HermannNVKreiborgSDarvannTAJensenBLDahlEBolundS.Craniofacial morphology and growth comparisons in children with Robin sequence, isolated cleft palate, and unilateral complete cleft lip and palate. Cleft Palate Craniofac J. 2003a;40(4):373–396.
14.
HermannNVKreiborgSDarvannTAJensenBLDahlEBolundS. Early craniofacial morphology and growth in children with nonsyndromic Robin sequence. Cleft Palate Craniofac J. 2003b;40(2):131–143.
15.
KatzESMitchellRBD’AmbrosioCM. Obstructive sleep apnea in infants. Am J Respir Crit Care Med. 2012;185(8):805–816.
16.
KaufmanLRousseeuwPJ. Finding Groups in Data an Introduction to Cluster Analysis. 1st ed. NJ Wiley; 2005.
17.
KimBCBertinHKimH-JKangS-HMercierJPerrinJ-PCorrePLeeS-H. Structural comparison of hemifacial microsomia mandible in different age groups by three-dimensional skeletal unit analysis. J Craniomaxillofac Surg. 2018;46(11):1875–1882.
18.
KirschnerRELowDWRandallPBartlettSPMcDonald-McGinnDMSchultzPJZackaiEHLaRossaD.Surgical airway management in Pierre Robin sequence: is there a role for tongue-lip adhesion?Cleft Palate Craniofac J. 2003;40(1):13–18.
19.
KlingenbergCP. Size, shape, and form: concepts of allometry in geometric morphometrics. Dev Genes Evol. 2016;226(3):113–137.
20.
LaitinenSRantaR. Cephalometric measurements in patients with Pierre Robin syndrome and isolated cleft-palate. Scand J Plast Reconstr Surg Hand Surg. 1992;26(2):177–183.
21.
MosimannJE.Size allometry: size and shape variables with characterizations of the lognormal and generalized gamma distributions. J Am Stat Assoc. 1970;65(330):930–945.
22.
MutoTYamazakiATakedaS. A cephalometric evaluation of the pharyngeal airway space in patients with mandibular retrognathia and prognathia, and normal subjects. Int J Oral Maxillofac Surg. 2008;37(3):228–231.
23.
NemecUNemecSFBruggerPCWeberMBartschBBettelheimDGrebuerMPrayerD. Normal mandibular growth and diagnosis of micrognathia at prenatal MRI. Prenat Diagn. 2015;35(2):108–116.
PaesECvan NunenDPFSpelemanLMuradinMSMSmariusBKonMMink van derABvan der MolenABNiersTLEMVeldhoenES, et al.A pragmatic approach to infants with Robin sequence: a retrospective cohort study and presence of a treatment algorithm. Clin Oral Investig. 2015;19(8):2101–2114.
26.
PoetsCFMaasCBuchenauWArandJVierzigABraumannBMüller-HagedornS. Multicenter study on the effectiveness of the pre-epiglottic baton plate for airway obstruction and feeding problems in Robin sequence. Orphanet J Rare Dis. 2017;12(1):46.
27.
R Core Team: R. A Language and Environment for Statistical Computing: Austria R Foundation for Statistical Computing; 2016.
28.
RahbarRRobsonCDMullikenJBSchwartzLDicanzioJKennaMAMcGillTJHealyGB. Craniofacial, temporal bone, and audiologic abnormalities in the spectrum of hemifacial microsomia. Arch Otolaryngol Head Neck Surg. 2001;127(3):265–271.
29.
RajeswaranRChandrasekharanAJosephSVenkata SaiPMDevBReddyS. Ultrasound versus MRI in the diagnosis of fetal head and trunk anomalies. J Matern Fetal Neonatal Med. 2009;22(2):115–123.
30.
RemyFGodio-RaboutetYCaptierGBurgartPThollonLGuyotL.Morphometric characterization of the very young child mandibular growth pattern: what happen before and after the deciduous dentition development?Am J Phys Anthropol. 2019;170(4):496–506.
31.
RemyFGodio-RaboutetYVernaEGorincourGBonnaurePAdalianPGuyotLThollonL. Characterization of the perinatal mandible growth pattern: preliminary results. Surg Radiol Anat. 2018;40(6):667–679.
32.
RogersGLimAATMullikenJBPadwaBL. Effect of a syndromic diagnosis on mandibular size and sagittal position in Robin sequence. J Oral Maxillofac Surg. 2009;67(11):2323–2331.
33.
RottenDLevaillantJMMartinezHLe pointeHDVicautE. The fetal mandible: a 2D and 3D sonographic approach to the diagnosis of retrognathia and micrognathia. Ultrasound Obstet Gynecol. 2002;19(2):122–130.
34.
SchaeferRBStadlerJAGosainAK. To distract or not to distract: an algorithm for airway management in isolated Pierre Robin sequence. Plast Reconstr Surg. 2004;113(4):1113–1125.
StroblCMalleyJTutzG.An introduction to recursive partitioning: rationale, application and characteristics of classification and regression trees, bagging and random forests. Psychol Methods. 2009;14(4):323–348.
37.
SullivanCBerthonjonesMIssaFEvesL. Reversal of obstructive sleep-apnea by continuous positive airway pressure applied through the nares. Lancet. 1981;1(8225):862–865.
38.
SuriSRossRBTompsonBD. Craniofacial morphology and adolescent facial growth in Pierre Robin sequence. Am J Orthod Dentofacial Orthop. 2010;137(6):763–774.
39.
TibesarRJScottARMcNamaraCSampsonDLanderTASidmanJD. Distraction osteogenesis of the mandible for airway obstruction in children: long-term results. Otolaryngol Head Neck Surg. 2010;143(1):90–96.
40.
TomaskiSZalzalGSaalH. Airway obstruction in the Pierre Robin sequence. Laryngoscope. 1995;105(2):111–114.
41.
van LieshoutMJSJoostenKFMMathijssenIMJKoudstaalMJHoeveHLJvan der SchroeffMPWolviusEB. Robin sequence: a European survey on current practice patterns. J Craniomaxillofac Surg. 2015;43(8):1626–1631.
42.
VatlachSMaasCPoetsCF. Birth prevalence and initial treatment of Robin sequence in Germany: a prospective epidemiologic study. Orphanet J Rare Dis. 2014;9(1):9.
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