Mégarbané André, Ghanem Ismat, Waked Naji, Dagher Fernand
Unité de Génétique Médicale, Faculté de Médecine, Université Saint-Joseph, Beirut, Lebanon.
Am J Med Genet A. 2006 Jul 15;140(14):1491-6. doi: 10.1002/ajmg.a.31316.
This report describes a young girl and her cousin presenting with postnatal short stature, strabismus, photophobia, retinitis pigmentosa, short neck, rhizomelic shortening of the long bones, short and slightly bowed humeri with prominent deltoid tuberosities, short and wide ribs and clavicles, dorso-lumbar scoliosis, biconcave vertebral bodies of the thoraco-lumbar spine, and narrowed lumbar canal. In addition, in the girl there were amelogenesis imperfecta of the hypomaturation type, and the radiographs showed short distal ulnae, sloping epiphyses of the radii, short femoral necks, and slightly flat uncovered femoral heads. The children's parents are first cousins. Differential diagnoses are discussed and the possibility of a newly recognized oculo-skeletal syndrome is raised.
本报告描述了一名年轻女孩及其表弟,他们表现出出生后身材矮小、斜视、畏光、色素性视网膜炎、短颈、长骨近端缩短、肱骨短且略弯曲并伴有明显的三角肌粗隆、肋骨和锁骨短而宽、背腰段脊柱侧弯、胸腰段脊柱椎体双凹形以及腰椎管狭窄。此外,该女孩存在低成熟型釉质发育不全,X线片显示尺骨远端短、桡骨骨骺倾斜、股骨颈短以及股骨头略扁平且未覆盖。患儿的父母是近亲。文中讨论了鉴别诊断,并提出了一种新认识的眼骨骼综合征的可能性。