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II型马耶夫斯基骨发育异常原发性侏儒症(MOPD II):自然病史及临床发现

Majewski osteodysplastic primordial dwarfism type II (MOPD II): natural history and clinical findings.

作者信息

Hall Judith G, Flora Christina, Scott Charles I, Pauli Richard M, Tanaka Kimi I

机构信息

Department of Medical Genetics, UBC and Children's and Women's Health Centre of British Columbia Vancouver, British Columbia, Canada.

出版信息

Am J Med Genet A. 2004 Sep 15;130A(1):55-72. doi: 10.1002/ajmg.a.30203.

Abstract

A description of the clinical features of Majewski osteodysplastic primordial dwarfism type II (MOPD II) is presented based on 58 affected individuals (27 from the literature and 31 previously unreported cases). The remarkable features of MOPD II are: severe intrauterine growth retardation (IUGR), severe postnatal growth retardation; relatively proportionate head size at birth which progresses to true and disproportionate microcephaly; progressive disproportion of the short stature secondary to shortening of the distal and middle segments of the limbs; a progressive bony dysplasia with metaphyseal changes in the limbs; epiphyseal delay; progressive loose-jointedness with occasional dislocation or subluxation of the knees, radial heads, and hips; unusual facial features including a prominent nose, eyes which appear prominent in infancy and early childhood, ears which are proportionate, mildly dysplastic and usually missing the lobule; a high squeaky voice; abnormally, small, and often dysplastic or missing dentition; a pleasant, outgoing, sociable personality; and autosomal recessive inheritance. Far-sightedness, scoliosis, unusual pigmentation, and truncal obesity often develop with time. Some individuals seem to have increased susceptibility to infections. A number of affected individuals have developed dilation of the CNS arteries variously described as aneurysms and Moya Moya disease. These vascular changes can be life threatening, even in early years because of rupture, CNS hemorrhage, and strokes. There is variability between affected individuals even within the same family.

摘要

基于58例受累个体(27例来自文献,31例为既往未报道病例),对II型Majewski骨发育不良性原始侏儒症(MOPD II)的临床特征进行了描述。MOPD II的显著特征包括:严重的宫内生长迟缓(IUGR)、严重的出生后生长迟缓;出生时头部大小相对成比例,随后发展为真性且不成比例的小头畸形;由于四肢远端和中段缩短,身材矮小逐渐出现不成比例;一种进行性骨发育异常,伴有四肢干骺端改变;骨骺延迟;关节逐渐松弛,偶尔出现膝盖、桡骨头和髋关节脱位或半脱位;特殊的面部特征,包括突出的鼻子、婴儿期和幼儿期看起来突出的眼睛、大小比例合适但轻度发育异常且通常没有耳垂的耳朵;高音调的声音;异常小且常发育异常或缺失的牙列;性格开朗、外向、善于社交;以及常染色体隐性遗传。随着时间的推移,常出现远视、脊柱侧弯、异常色素沉着和躯干肥胖。一些个体似乎对感染的易感性增加。许多受累个体出现了中枢神经系统动脉扩张,有各种描述,如动脉瘤和烟雾病。这些血管变化可能危及生命,即使在早年也可能因破裂、中枢神经系统出血和中风而导致危险。即使在同一家族中,受累个体之间也存在差异。

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