Moore C A, Weaver D D
Department of Medical Genetics, Indiana University School of Medicine, Indianopolis 46202.
Am J Med Genet. 1989 Jun;33(2):231-7. doi: 10.1002/ajmg.1320330218.
We report on 5 relatives in 3 generations with an apparent new type of distal arthrogryposis. These individuals have manifestations of type I distal arthrogryposis, but in addition, have craniofacial anomalies that include facial asymmetry, hypertelorism, downslanting palpebral fissures, high nasal bridge, malar hypoplasia, micrognathia, highly arched palate, notched chin, and posteriorly angulated ears. Their intelligence is normal. Although these manifestations preclude us from placing this family in the type I (isolated) distal arthrogryposis category, we also are unable to place them in any of the recognized subtypes of type II distal arthrogryposis. Thus, we think this family may have a previously undescribed form of autosomal dominant type II distal arthrogryposis.
我们报告了一个三代家族中的5名亲属,他们患有一种明显的新型远端关节挛缩症。这些个体具有I型远端关节挛缩症的表现,但此外,还存在颅面异常,包括面部不对称、眼距过宽、睑裂向下倾斜、鼻梁高、颧骨发育不全、小颌畸形、高拱腭、下巴有凹口以及耳朵向后成角。他们的智力正常。尽管这些表现使我们无法将这个家族归类为I型(孤立型)远端关节挛缩症,但我们也无法将他们归入II型远端关节挛缩症的任何一种已确认的亚型。因此,我们认为这个家族可能患有一种以前未描述过的常染色体显性II型远端关节挛缩症。