Abdullah M A, Al-Hasnan Z, Okamoto E, Abomelha A M
Endocrinology & Metabolism Unit, Deparment of Pediatrics, Box 9, Security Forces Hospital, PO Box 3643, Riyadh 11481, Kingdom of Saudi Arabia.
Saudi Med J. 2000 Mar;21(3):297-9.
We report for the first time from the Arabian Gulf area 3 patients with arthrogryposis multiplex congenita, cholestasis and renal tubular dysfunction from a Saudi family with 2 other siblings and 3 cousins who possibly died with a similar clinical picture. We also document for the second time in literature other findings in this syndrome including cerebral abnormalities (hypoplastic corpus callosum), congenital heart disease and nerve deafness. We suggest that some of these cases might benefit from ursodeoxycholic acid therapy. We believe that this autosomal recessive disorder is possibly under-diagnosed in this region with a high consanguineous marriage rate.
我们首次报道了来自阿拉伯湾地区的3例患有先天性多发性关节挛缩症、胆汁淤积和肾小管功能障碍的患者,他们来自一个沙特家庭,该家庭还有另外2名兄弟姐妹和3名表亲,后者可能死于类似的临床症状。我们还在文献中第二次记录了该综合征的其他发现,包括脑部异常(胼胝体发育不全)、先天性心脏病和神经性耳聋。我们建议其中一些病例可能受益于熊去氧胆酸治疗。我们认为,在这个近亲结婚率很高的地区,这种常染色体隐性疾病可能未得到充分诊断。