Antley-Bixler syndrome is a rare form of craniosynostosis characterized by unique clinical features that present particular challenges to long-term treatment. Typical craniofacial features include midface hypoplasia, trapezoidocephaly, frontal bossing, ocular proptosis, low-set protrusive ears, and choanal atresia. A staged surgical approach including fronto-orbital and midface advancements is required for intracranial pressure reduction, globe protection, airway patency, and malocclusion. In this case report, the combined, longitudinal surgical and orthodontic treatment strategies employed to correct severe facial deformity in a long-term 21-year survivor of Antley-Bixler syndrome are reviewed.
AllamK.A., WanD.C., KhwanngernK., KawamotoH.K., TannaN., PerryA.Treatment of apert syndrome: a long-term follow-up study.Plast Reconstr Surg.2011; 127: 1601–1611.
2.
AntleyR., BixlerD.Trapezoidocephaly, midfacial hypoplasia and cartilage abnormalities with multiple synostoses and skeletal fractures.Birth Defects Orig Artic Ser.1975; 11: 397–401.
3.
BradleyJ.P., GabbayJ.S., TaubP.J., HellerJ.B., O'HaraC.M., BenhaimP.Monobloc advancement by distraction osteogenesis decreases morbidity and relapse.Plast Reconstr Surg.2006; 118: 1585–1597.
4.
BradleyJ.P., KawamotoH.K., TaubP., WexlerA., CahanL.Antley-Bixler syndrome: correction of facial deformities and long-term survival.Plast Reconstr Surg.2003; 111: 1454–1460.
5.
ChabchoubA., Siala-GaigiS., MarrakchiZ., JebnounS., AyachiR., KhroufN.The Antley-Bixler syndrome. a new case without radiohumeral synostosis.Genet Couns.1998; 9: 113–118.
6.
ChunK., Siegel-BarteltJ., ChitayatD., PhillipsJ., RayP.N.FGFR2 mutation associated with clinical manifestations consistent with Antley-Bixler syndrome.Am J Med Genet.1998; 77: 219–224.
7.
CragunD., HopkinR.J.Gene Reviews.Seattle, WA: University of Washington; 1993.
8.
CrisponiG., PorcuC., PiuM.E.Antley-Bixler syndrome: case report and review of the literature.Clin Dysmorphol.1997; 6: 61–68.
EscobarL.F., BixlerD., SadoveM., BullM.J.Antley-Bixler syndrome from a prognostic perspective: report of a case and review of the literature.Am J Med Genet.1988; 29: 829–836.
12.
FearonJ.A.Midterm follow-up of midface distraction.Plast Reconstr Surg2008; 122: 674–675.
13.
HassellS., ButlerM.G.Antley-Bixler syndrome: report of a patient and review of literature.Clin Genet.1994; 46: 372–376.
NoutE., KoudstaalM.J., WolviusE.B., Van der WalK.G.Additional orthognathic surgery following Le Fort III and monobloc advancement.Int J Oral Maxillofac Surg.2011; 40: 679–684.
SchinzelA., SavoldelliG., BrinerJ., SiggP., MassiniC.Antley-Bixler syndrome in sisters: a term newborn and a prenatally diagnosed fetus.Am J Med Genet.1983; 14: 139–147.
19.
ShetyeP.R., BoutrosS., GraysonB.H., McCarthyJ.G.Midterm follow-up of midface distraction for syndromic craniosynostosis: a clinical and cephalometric study.Plast Reconstr Surg.2007; 120: 1621–1632.
20.
SuzukiK., KandaY., SugiyamaK., KatohT., WadaY., YasuiY.Antley-Bixler syndrome in a sister and brother.Jpn J Hum Genet.1987; 32: 247–252.
