Nasal Bifidity: An Unusual Pediatric Congenital Anomaly and Review of the Literature

被引:0
|
作者
Chen, Li Hsuen N. [1 ]
Carro, Marina A. [2 ]
Castiglione, Charles L. [3 ]
Hughes, Christopher D. [3 ]
机构
[1] Univ Connecticut, Sch Dent Med, Div Oral & Maxillofacial Surg, Farmington, CT USA
[2] Quinnipiac Univ, Frank H Netter Sch Med, North Haven, CT USA
[3] Connecticut Childrens, Div Plast & Craniofacial Surg, 282 Washington St,Suite 2L, Hartford, CT 06106 USA
关键词
Early rhinoseptoplasty; isolated bifid nose; isolated bifid septum; FACIAL REGION; EMBRYOLOGY; FACE;
D O I
10.1097/SCS.0000000000009304
中图分类号
R61 [外科手术学];
学科分类号
摘要
Congenital nasal anomalies are rare, with an estimated incidence of 1/20,000 to 40,000 live births. Hyperplasia and duplication anomalies are the most uncommon, comprising about 1% of reported cases. The authors present the case of a 6-year-old girl who presented to our institution with an isolated congenital bifid nasal septum. Parents reported a visibly abnormal nose since birth, and it had been continuously monitored by the parents and pediatrician. She demonstrated no history of difficulty breathing or other nasal concerns and was otherwise growing and developing normally. On physical examination, she was breathing comfortably through her bilateral nasal airways. Her nasal examination revealed a widened mid-vault with deep dorsal grooving and a bifid tip. Magnetic resonance imaging demonstrated an isolated bifid nasal septum without other facial malformation or intracranial extension. She underwent an open septorhinoplasty. Intraoperatively, the authors identified an anomalous dorsal nasal bone extension with a resultant bifidity in the body and caudal portions of the septum. Ostectomy and cartilaginous repositioning allowed for an autogenous reconstruction without the need for grafting. She subsequently recovered well without postoperative complications and continues to have improved nasal appearance with maintenance of function. A review of recent literatures revealed 2 other cases that are similar in presentation. The authors proposed that embryologically there might have been a change in expression of bone morphogenetic protein in the frontonasal area leading to caudal extension of the nasal bone. This in turn interferes with the fusion of nasal septum resulting in the bifid septum and dual dome morphology.
引用
收藏
页码:1235 / 1237
页数:3
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