Department of Plastic Surgery, Baylor College of Medicine, Baylor College of Medicine, 6621 Fannin Street, Houston, TX 77030, USA.
Am J Med Genet A. 2009 Sep;149A(9):1910-5. doi: 10.1002/ajmg.a.32836.
Ankyloblepharon-ectodermal defects-cleft lip/palate (AEC) Syndrome is a rare disorder characterized by ectodermal dysplasia, along with other malformations such as cleft lip and palate, and various secondary issues such as chronic sinusitis, otitis media, and conductive hearing loss (CHL). The International Research Symposium for AEC Syndrome convened at Baylor College of Medicine in Houston, Texas. Patients with a suspected diagnosis of AEC syndrome attended, and members of the dental, dermatology, plastic surgery, otolaryngology, and audiology services examined each patient. Eighteen patients with a diagnosis of AEC were evaluated. Mean age was 7.5 years (range: 4 months-30 years). Fourteen of the 15 subjects tested (93.33%) demonstrated CHL, with seven showing moderate to severe hearing deficits (41-90 dB). Nine of 13 respondents reported hoarseness or voice problems; 8 were noted to display this on examination. Fourteen of 16 subjects reported speech was below average for age; 8 were in speech therapy. All 18 subjects reported a history of otitis externa or otitis media. Eleven of the subjects (61.11%) required myringotomy and pressure equalizing (PE) tubes. All patients demonstrated cleft palate defects. Of these, 16 (94.11%) presented with clefting of the soft palate, and 10 (58.82%) showed hard palate defects. Three subjects (16.67%) were noted to have submucous clefts. Our experience leads us to propose that while the oroauditory problems in those with AEC syndrome is likely multifactorial, many issues may stem from palatal clefting. Despite this, some abnormalities persist following surgical cleft closure, which indicates other complicating factors are also involved.
先天性无睑-外胚层发育不良-唇腭裂(AEC)综合征是一种罕见疾病,其特征为外胚层发育不良,同时伴有唇腭裂等其他畸形,以及慢性鼻窦炎、中耳炎和传导性听力损失(CHL)等各种继发性问题。AEC 综合征国际研究研讨会在德克萨斯州休斯顿的贝勒医学院举行。有疑似 AEC 综合征诊断的患者参加了会议,牙科、皮肤科、整形外科、耳鼻喉科和听力学服务的成员检查了每位患者。对 18 例 AEC 诊断患者进行了评估。平均年龄为 7.5 岁(范围:4 个月-30 岁)。15 名接受测试的受试者中有 14 名(93.33%)存在 CHL,其中 7 名存在中度至重度听力障碍(41-90dB)。9 名受访者中有 9 名(61.11%)报告声音嘶哑或声音问题;8 名在检查中发现了这一问题。14 名受访者中有 13 名(81.25%)报告其言语能力低于同龄水平;8 名正在接受言语治疗。所有 18 名患者均报告有外耳炎或中耳炎病史。11 名受试者(61.11%)需要鼓膜切开术和压力平衡(PE)管。所有患者均表现出腭裂缺陷。其中,16 名(94.11%)有软腭裂,10 名(58.82%)有硬腭裂。3 名患者(16.67%)有黏膜下腭裂。我们的经验表明,虽然 AEC 综合征患者的口耳问题可能是多因素的,但许多问题可能源于腭裂。尽管如此,一些异常在腭裂手术后仍然存在,这表明还有其他复杂因素也参与其中。