Schotland H M, Eldridge R, Sommer S S, Malawar M
Department of Medicine, New York University Medical Center, New York.
Am J Med Genet. 1992 Jul 15;43(5):815-22. doi: 10.1002/ajmg.1320430513.
We report on the cosegregation of neurofibromatosis 1 (NF 1) and osseous fibrous dysplasia in a family. The father and 3 children by 2 women are affected. A fourth child had neither NF 1 nor osseous fibrous dysplasia. All 4 affected individuals had NF 1, i.e., café-au-lait spots in 4, neurofibromata in 4, Lisch nodules in 3, macrocrania in 3, scoliosis in 2, and curvature of the long bones in 2. Each demonstrated various fibroosseous lesions of the skeleton including non-ossifying fibromas in 3 and both non-ossifying fibromas and fibrous dysplasia in one. This pattern suggests that the fibrous bony lesions are a component of NF 1 in this family. Alternatively, a mutant gene resulting in the fibrous changes in bone could be linked to the gene for NF 1. Another possibility is the coincidence of the 2 non-linked traits segregating in the same family.
我们报告了一个家族中神经纤维瘤病1型(NF 1)与骨纤维发育不良的共分离现象。父亲以及与两名女性所生的3个孩子均患病。第四个孩子既没有NF 1也没有骨纤维发育不良。所有4名患病个体都患有NF 1,即4人有咖啡斑,4人有神经纤维瘤,3人有Lisch结节,3人有巨头畸形,2人有脊柱侧弯,2人有长骨弯曲。每个人都表现出骨骼的各种纤维性骨病变,包括3人有非骨化性纤维瘤,1人既有非骨化性纤维瘤又有纤维发育不良。这种模式表明,在这个家族中纤维性骨病变是NF 1的一个组成部分。或者,导致骨骼纤维性改变的突变基因可能与NF 1基因连锁。另一种可能性是这两种不连锁的性状在同一家族中同时出现。