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在6个家族中发现了三种不同类型的X连锁先天性多发性关节挛缩症。

Three distinct types of X-linked arthrogryposis seen in 6 families.

作者信息

Hall J G, Reed S D, Scott C I, Rogers J G, Jones K L, Camarano A

出版信息

Clin Genet. 1982 Feb;21(2):81-97. doi: 10.1111/j.1399-0004.1982.tb00742.x.

Abstract

Six families with arthrogryposis (congenital contractures) inherited in an X-linked recessive manner are reported. Family histories from a study of over 350 patients with congenital contractures of the joints (arthrogryposis) were reviewed and of these, three probands had family histories consistent with X-linked recessive inheritance. Three other families were recognized through correspondence. Three forms of X-linked recessively inherited arthrogryposis are described: (1) Severe lethal X-linked arthrogryposis with severe contractures scoliosis deformities, hypotonia, and death due to respiratory insufficiency within 3 months of birth (1 family); (2) Moderately severe X-linked arthrogryposis with severe contractures, ptosis, microphallus, cryptorchidism, inguinal hernias, and normal intelligence (2 families); and (3) Resolving X-linked arthrogryposis with mild to moderate contractures at birth which improve dramatically with time (2 families and 1 sporadic case).

摘要

本文报告了6个以X连锁隐性方式遗传的先天性关节挛缩症(关节弯曲)家系。回顾了对350多名先天性关节挛缩症(关节弯曲)患者的研究家族史,其中3名先证者的家族史符合X连锁隐性遗传。另外3个家系是通过信件往来确认的。本文描述了X连锁隐性遗传的3种先天性关节挛缩症:(1)严重致死性X连锁先天性关节挛缩症,伴有严重挛缩、脊柱侧弯畸形、肌张力减退,出生后3个月内因呼吸功能不全死亡(1个家系);(2)中度严重X连锁先天性关节挛缩症,伴有严重挛缩、上睑下垂、小阴茎、隐睾、腹股沟疝,智力正常(2个家系);(3)可缓解的X连锁先天性关节挛缩症,出生时伴有轻至中度挛缩,随时间推移显著改善(2个家系和1例散发病例)。

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