To characterize mandibular morphology in patients with Parry-Romberg syndrome (PRS).
Design
Retrospective study.
Setting
A craniofacial center.
Patients
Thirty-three patients with PRS affecting mandible.
Interventions
Preoperative computed tomography data were analyzed using Mimics 26.0 (Materialise Inc.). Distances between landmarks—condyle process (Con), coronoid process (Cor), mandibular foramen (IAF), gonion (Go), and mental foramen—were measured to represent the sizes of skeletal units. Positional asymmetry was reflected by angles between lines connecting bilateral landmarks and the occlusal plane.
Main outcome measure(s)
Bilateral differences in unit sizes were compared. Absolute difference (Δ) > 5 mm and relative size (RS) < 80% were set to evaluate the severity of hypoplasia. Angular measurements were compared to 0° using one-sample t tests. Pearson correlation analysis was conducted to examine the relationship between onset ages and both RS and angular measurements.
Results
The affected side's skeletal units were smaller. Severe hypoplasia was more common in the angular unit. Angles between Con-Con’, Cor-Cor’, IAF-IAF’, and the occlusal plane were less than 0°, whereas the angle with Go-Go’ was greater than 0°. Age of onset was positively correlated with the condylar unit size and the Con-Con’ to occlusal plane angle.
Conclusions
Patients with PRS affecting mandible exhibit smaller skeletal units on the affected side, particularly in the angular unit. Most landmarks on affected side tend to cluster toward the occlusal plane. Earlier onset of PRS correlates with more pronounced condylar asymmetry. Evaluating the severity of functional units involvement and implementing appropriate treatment should be considered.
BlaszczykMKrólickiLKrasuMGlinskaOJablonskaS. Progressive facial hemiatrophy: central nervous system involvement and relationship with scleroderma en coup de sabre. J Rheumatol. 2003;30(9):1997‐2004.
SharmaPKSekarAAmirAPPrabhuALR. Parry-Romberg syndrome: a case report and literature review. Radiol Case Rep. 2024;19(6):2230‐2238. doi:https://doi.org/10.1016/j.radcr.2024.02.053
5.
TollefsonMMWitmanPM. En coup de sabre morphea and Parry-Romberg syndrome: a retrospective review of 54 patients. J Am Acad Dermatol. 2007;56(2):257‐263. doi:https://doi.org/10.1016/j.jaad.2006.10.959
6.
AsherSWBergBO. Progressive hemifacial atrophy: report of three cases, including one observed over 43 years, and computed tomographic findings. Arch Neurol. 1982;39(1):44‐46. doi:https://doi.org/10.1001/archneur.1982.00510130046011
7.
PenslerJMMurphyGFMullikenJB. Clinical and ultrastructural studies of Romberg's hemifacial atrophy. Plast Reconstr Surg. 1990;85(5):669‐674. discussion 675-6.
8.
RoddiRRiggioEGilbertPMVaandragerJMvan der MeulenJC. Progressive hemifacial atrophy in a patient with lupus erythematosus. Plast Reconstr Surg. 1994;93(5):1067‐1072. doi:https://doi.org/10.1097/00006534-199404001-00027
9.
UllmanSDanielsenPLFledeliusHCDaugaard-JensenJSerupJ. Scleroderma en Coup de Sabre, Parry-Romberg hemifacial atrophy and associated manifestations of the eye, the oral cavity and the teeth: a Danish follow-up study of 35 patients diagnosed between 1975 and 2015. Dermatology.2021;237(2):204‐212. doi:https://doi.org/10.1159/000507925
10.
SommerAGambichlerTBacharach-BuhlesMvon RothenburgTAltmeyerPKreuterA. Clinical and serological characteristics of progressive facial hemiatrophy: a case series of 12 patients. J Am Acad Dermatol. 2006;54(2):227‐233. doi:https://doi.org/10.1016/j.jaad.2005.10.020
11.
JablonskaSBlaszczykM. Long-lasting follow-up favours a close relationship between progressive facial hemiatrophy and scleroderma en coup de sabre. J Eur Acad Dermatol Venereol. 2005;19(4):403‐404. doi:https://doi.org/10.1111/j.1468-3083.2005.00979.x
GuerrerosantosJGuerrerosantosFOrozcoJ. Classification and treatment of facial tissue atrophy in Parry-Romberg disease. Aesthetic Plast Surg.2007;31(5):424‐434. doi:https://doi.org/10.1007/s00266-006-0215-4
14.
HoehnkeCEderMPapadopulosNAZimmermannABrockmannGBiemerEKovacsL.Minimal invasive reconstruction of posttraumatic hemi facial atrophy by 3-D computer-assisted lipofilling. J Plast Reconstr Aesthet Surg. 2007;60(10):1138‐1144. doi:https://doi.org/10.1016/j.bjps.2007.01.068
15.
HuJYinLTangXGuiLZhangZ. Combined skeletal and soft tissue reconstruction for severe Parry-Romberg syndrome. J Craniofac Surg. 2011;22(3):937‐941. doi:https://doi.org/10.1097/SCS.0b013e31820fe27d
16.
Abi-RafehJJaberiMCattelanLAljerianAGilardinoMS. Soft-tissue reconstruction in progressive hemifacial atrophy: current evidence and future directions. Plast Reconstr Surg.2022;150(3):607‐617. doi:https://doi.org/10.1097/prs.0000000000009423
QiaoJGuiLFuXNiuFLiuJChenYWangMChenJYouY.A novel method of mild to moderate Parry-Romberg syndrome reconstruction: computer-assisted surgery with mandibular outer cortex and fat grafting. J Craniofac Surg. 2017;28(2):359‐365. doi:https://doi.org/10.1097/scs.0000000000003293
19.
CruyssenFMeeusJSchoenaersJPolitisC. Parry Romberg syndrome: a long-term retrospective cohort study of 10 patients. Oral Maxillofac Surg Cases. 2018;4(3):73‐83.
20.
Ramirez-YañezGOStewartAFrankenECamposK. Prevalence of mandibular asymmetries in growing patients. Eur J Orthod. 2011;33(3):236‐242. doi:https://doi.org/10.1093/ejo/cjq057
NairMAjilaVHegdeSBabuGSGhoshR. Clinical and radiographic features of Parry-Romberg syndrome. J Istanb Univ Fac Dent. 2017;51(3):45‐49. doi:https://doi.org/10.17096/jiufd.72612
23.
YimSYangIHBaekSH. Characterization of dental phenotypes and treatment modalities in Korean patients with Parry-Romberg syndrome. Korean J Orthod. 2020;50(6):407‐417. doi:https://doi.org/10.4041/kjod.2020.50.6.407
24.
de Vasconcelos CarvalhoMdo NascimentoGJAndradeEAndradeMSobralAP. Association of aesthetic and orthodontic treatment in Parry-Romberg syndrome. J Craniofac Surg. 2010;21(2):436‐439. doi:https://doi.org/10.1097/SCS.0b013e3181cfe917
WongMPhillipsCDHagiwaraMShatzkesDR. Parry Romberg syndrome: 7 cases and literature review. AJNR Am J Neuroradiol. 2015;36(7):1355‐1361. doi:https://doi.org/10.3174/ajnr.A4297
27.
SmithRJJacksonITXiaoHSharmaR. 3-D CT scan measured bony deficiency of the facial skeleton in progressive hemifacial atrophy. Eur J Plast Surg.1994;17(6):292‐297. doi:https://doi.org/10.1007/BF00181094
28.
LiSMeiLChenQGaoKZhengWLiY. Unilateral injection of botulinum toxin type A into the masseter muscle induces mandibular asymmetry in adolescent rats by suppressing the angular process growth. Am J Orthod Dentofacial Orthop. 2024;165(2):232‐244. doi:https://doi.org/10.1016/j.ajodo.2023.09.009
29.
ParkWKimBCYuHSYiCKLeeSH. Architectural characteristics of the normal and deformity mandible revealed by three-dimensional functional unit analysis. Clin Oral Investig. 2010;14(6):691‐698. doi:https://doi.org/10.1007/s00784-009-0349-2
30.
MunSHParkMLeeJLimHJKimBC. Volumetric characteristics of prognathic mandible revealed by skeletal unit analysis. Ann Anat. 2019;226:3‐9. doi:https://doi.org/10.1016/j.aanat.2019.07.007
31.
YouKHLeeKJLeeSHBaikHS. Three-dimensional computed tomography analysis of mandibular morphology in patients with facial asymmetry and mandibular prognathism. Am J Orthod Dentofacial Orthop. 2010;138(5):540.e1‐8. discussion 540-1. doi:https://doi.org/10.1016/j.ajodo.2010.04.025
32.
DamstraJOosterkampBCJansmaJRenY. Combined 3-dimensional and mirror-image analysis for the diagnosis of asymmetry. Am J Orthod Dentofacial Orthop. 2011;140(6):886‐894. doi:https://doi.org/10.1016/j.ajodo.2010.03.032
33.
ChoiJWKimBHKimHSYuTHKimBCLeeSH. Three-dimensional functional unit analysis of hemifacial microsomia mandible-a preliminary report. Maxillofac Plast Reconstr Surg. 2015;37(1):28. doi:https://doi.org/10.1186/s40902-015-0027-z
34.
Yu-FengLLaiGZhi-YongZ. Combined treatments of facial contour deformities resulting from Parry-Romberg syndrome. J Reconstr Microsurg. 2008;24(5):333‐342. doi:https://doi.org/10.1055/s-2008-1080536
35.
GrippaudoCDeliRGrippaudoFRDi CuiaTParadisiM. Management of craniofacial development in the Parry-Romberg syndrome: report of two patients. Cleft Palate Craniofac J. 2004;41(1):95‐104. doi:https://doi.org/10.1597/02-066
SlackGCTabitCJAllamKAKawamotoHKBradleyJP. Parry-Romberg reconstruction: optimal timing for hard and soft tissue procedures. J Craniofac Surg. 2012;23(7 Suppl 1):1969‐1973. doi:https://doi.org/10.1097/SCS.0b013e318258bd11
38.
HunstadJPShifrinDAKortesisBG. Successful treatment of Parry-Romberg syndrome with autologous fat grafting: 14-year follow-up and review. Ann Plast Surg. 2011;67(4):423‐425. doi:https://doi.org/10.1097/SAP.0b013e31820b3aa8
39.
IsraelJSChenJTFarmerRLSiebertJW. Challenging traditional thinking: early free tissue transfer for active hemifacial atrophy in children. Plast Reconstr Surg. 2020;145(2):483‐492. doi:https://doi.org/10.1097/prs.0000000000006511
40.
MaXHuangJXueHWangHWangTChenYLongXWangX.Damage evaluation of craniofacial localized scleroderma using magnetic resonance imaging. Quant Imaging Med Surg.2024;14(2):1891‐1903. doi:https://doi.org/10.21037/qims-23-980
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