Bohring A, Oppermann H C
Department of Pediatrics, University of Kiel, Germany.
Am J Med Genet. 1997 Aug 8;71(2):194-6.
We report on a newborn boy with congenital asymmetrically hypoplastic fibulae, lateral oligodactyly, and mild left ectrodactyly. The patient's grandfather had a short femoral shaft with a slightly smaller collodiaphyseal angle on the left as compared to the right side, probably a proximal focal femoral deficiency (PFFD). The upper limbs were not affected in either patient. PFFD in the grandfather and hypoplastic fibulae with lateral ray defects in the grandson raise the possibility of genetic transmission, specifically autosomal-dominant inheritance with variable penetrance and expressivity. This case gives further support to the fibular developmental field concept postulated by Lewin und Opitz [1986: Am J Med Genet (Suppl) 2:215-238].
我们报告了一名患有先天性不对称性腓骨发育不全、外侧多指畸形和轻度左侧裂手畸形的男婴。患者的祖父左侧股骨干短,与右侧相比,股骨干骺端角略小,可能为近端局灶性股骨缺损(PFFD)。两名患者的上肢均未受影响。祖父的PFFD以及孙子的腓骨发育不全伴外侧射线缺陷增加了遗传传递的可能性,特别是具有可变外显率和表达性的常染色体显性遗传。该病例进一步支持了Lewin和Opitz [1986:《美国医学遗传学杂志》(增刊)2:215 - 238]提出的腓骨发育场概念。