Schweitzer V G, Kemink J L, Graham M D
Am J Otol. 1984 Jul;5(5):387-91.
A rare congenital autosomal dominant syndrome of thickened bilateral lop auricles, conductive hearing loss, ossicular anomalies, and micrognathia is reported. The anomaly is presumably a defect in the first and second branchial arch development during the sixth and seventh weeks of gestation with an auricle abnormality (first and second arch), abnormal incus and malleus (first and second arch), abnormal stapes (second arch), and micrognathia (first arch). Recognition of low-set or malformed auricles with a unilateral or bilateral conductive hearing loss should alert the otolaryngologist to possible middle ear abnormalities and other associated branchial cleft anomalies. Surgical correction of the congenital conductive hearing loss may include prosthetic ossicular reconstruction and otoplasty. The possibility of associated congenital anomalies of other systems (for example, vestibular, cardiac, genitourinary, reproductive, and so on) should be evaluated. An accurate pedigree and family medical and genetic history should be obtained to screen for other involved family members and for assessment of genetic passage of the trait.
报告了一种罕见的先天性常染色体显性综合征,其特征为双侧耳廓增厚、传导性听力损失、听小骨异常和小颌畸形。这种异常可能是妊娠第六和第七周期间第一和第二鳃弓发育缺陷所致,伴有耳廓异常(第一和第二弓)、砧骨和锤骨异常(第一和第二弓)、镫骨异常(第二弓)以及小颌畸形(第一弓)。认识到低耳位或畸形耳廓伴有单侧或双侧传导性听力损失,应提醒耳鼻喉科医生注意可能存在的中耳异常及其他相关鳃裂异常。先天性传导性听力损失的手术矫正可能包括听小骨假体重建和耳廓成形术。应评估其他系统(如前庭、心脏、泌尿生殖、生殖等)相关先天性异常的可能性。应获取准确的家系以及家族医学和遗传病史,以筛查其他受累家庭成员,并评估该性状的遗传传递情况。