Crifasi P A, Patterson M C, Bonde D, Michels V V
Department of Medical Genetics, Mayo Clinic, Rochester, Minnesota 55905, USA.
Am J Med Genet. 1997 Jun 13;70(3):261-6.
Hajdu-Cheney syndrome is an autosomal dominant disorder of acroosteolysis, skull deformities, characteristic facial abnormalities, osteoporosis, joint laxity, early loss of teeth, hearing loss, and a hoarse voice. We report on an 8 1/2-year-old boy with Hajdu-Cheney syndrome and cystic kidney disease, congenital heart disease, hydrocephalus, cleft lip and palate, hydrosyringomyelia, club feet, splenomegaly, hypospadias, vertebral anomalies, and upper airway obstruction. A review of 44 patients did not uncover any other patients with all of these manifestations, nor any patient with upper airway obstruction. Hajdu-Cheney syndrome appears to encompass a broader phenotype than previously recognized. The documentation of these additional anomalies is valuable because the findings of acroosteolysis and osteoporosis can present later in the course.
哈伊杜-切尼综合征是一种常染色体显性疾病,表现为肢端骨质溶解、颅骨畸形、特征性面部异常、骨质疏松、关节松弛、牙齿早失、听力丧失和声音嘶哑。我们报告了一名8岁半患有哈伊杜-切尼综合征的男孩,他还患有多囊肾病、先天性心脏病、脑积水、唇腭裂、脊髓空洞症、马蹄内翻足、脾肿大、尿道下裂、脊柱异常和上呼吸道梗阻。对44例患者的回顾未发现有其他患者具备所有这些表现,也没有患者出现上呼吸道梗阻。哈伊杜-切尼综合征似乎包含比以往认识到的更广泛的表型。记录这些额外的异常情况很有价值,因为肢端骨质溶解和骨质疏松的表现可能在病程后期才出现。