McPherson E, Clemens M
University of Pittsburgh, Pennsylvania, USA.
Am J Med Genet. 1997 May 2;70(1):28-31.
We describe a patient who was born with flexion contractures and pterygia at the elbows, clubfeet, torticollis, and several rib fractures. During infancy and childhood, multiple fractures of the lower limbs occurred with minimal trauma and led to disabling deformities. When evaluated at age 19 years, he was normally intelligent, but extremely short, with severe kyphoscoliosis compromising his pulmonary function. Pterygia limited elbow extension to 90 degrees, and severe lower limb deformities prevented ambulation. He did not have blue sclerae, dentinogenesis imperfecta, or hearing loss. X-ray studies showed demineralized bones, severe deformity and cystic change at old fracture sites, and vertebral wedging. Collagen studies on skin fibroblasts were normal.
我们描述了一名患者,其出生时肘部存在屈曲挛缩和翼状胬肉、马蹄内翻足、斜颈以及多处肋骨骨折。在婴儿期和儿童期,下肢多次发生轻微创伤性骨折,并导致致残性畸形。19岁接受评估时,他智力正常,但身材极矮,严重的脊柱后凸侧弯损害了他的肺功能。翼状胬肉使肘部伸展受限至90度,严重的下肢畸形导致无法行走。他没有蓝色巩膜、牙本质发育不全或听力丧失。X线检查显示骨质脱矿、陈旧骨折部位严重畸形和囊性改变以及椎体楔形改变。对皮肤成纤维细胞的胶原研究结果正常。