Villavisanis Dillan F, Blum Jessica D, Taylor Jesse A
Division of Plastic and Reconstructive Surgery, Children's Hospital of Philadelphia, Philadelphia, PA.
J Craniofac Surg. 2023;34(1):e25-e28. doi: 10.1097/SCS.0000000000008890. Epub 2022 Aug 11.
Birk-Barel intellectual disability dimorphism syndrome, also referred to as KCNK9 imprinting syndrome, is an exceedingly rare condition described in under 20 cases that presents with intellectual disability, hypotonia, scoliosis, dysphonia, dysphagia, and craniofacial dysmorphic features. The condition follows an autosomal dominant pattern of inheritance in the maternally expressed KCNK9 gene on chromosome 8. Due to the complexity of presentation, patients with Birk-Barel syndrome are optimally managed by a multidisciplinary team including a craniofacial surgeon. Previously described craniofacial dysmorphic features include micrognathia, cleft palate, dolichocephaly, broad nasal tip, and broad philtrum, among others. Here the authors describe a genetically confirmed case that has been managed in our institution's multidisciplinary cleft and craniofacial clinic. The authors aim to discuss Birk-Barel syndrome for a surgical and craniofacial audience with considerations for operative management in the context of a multidisciplinary team.
伯克-巴雷尔智力残疾二态性综合征,也称为KCNK9印记综合征,是一种极为罕见的疾病,报道病例不足20例,表现为智力残疾、肌张力减退、脊柱侧弯、发音障碍、吞咽困难和颅面部畸形特征。该病遵循位于8号染色体上的母系表达的KCNK9基因的常染色体显性遗传模式。由于临床表现复杂,伯克-巴雷尔综合征患者最好由包括颅面外科医生在内的多学科团队进行管理。先前描述的颅面部畸形特征包括小颌畸形、腭裂、长头畸形、鼻尖宽大、人中宽等。本文作者描述了一例在本机构多学科腭裂和颅面诊所接受治疗的基因确诊病例。作者旨在为外科和颅面领域的读者讨论伯克-巴雷尔综合征,并在多学科团队的背景下考虑手术管理。