Whyte M P, Murphy W A, Fallon M D, Sly W S, Teitelbaum S L, McAlister W H, Avioli L V
Am J Med. 1980 Jul;69(1):64-74. doi: 10.1016/0002-9343(80)90501-x.
Three adult sisters with osteopetrosis in infancy had spontaneous resolution of bone modeling defects and osteosclerosis. During adolescence, basal ganglia calcification developed in two. Renal tubular acidosis (type I) was diagnosed in each during early adulthood. The disorder was transmitted apparently as a recessive trait--the same mode of inheritance as for the "malignant" form of osteopetrosis which is usually fatal during childhood. Electron microscopy of bone suggested that osteoclasts failed to form "ruffled membranes" characteristic of active bone resorbing cells. Chronic systemic acidosis may have ameliorated the skeletal manifestations of this new syndrome.
三名在婴儿期患骨硬化症的成年姐妹,其骨骼塑形缺陷和骨硬化症出现了自发缓解。在青春期,其中两人出现了基底节钙化。在成年早期,三人都被诊断出患有Ⅰ型肾小管性酸中毒。这种疾病显然以隐性性状遗传,与通常在儿童期致命的“恶性”骨硬化症的遗传方式相同。对骨骼的电子显微镜检查表明,破骨细胞未能形成活跃骨吸收细胞特有的“皱襞膜”。慢性全身性酸中毒可能改善了这种新综合征的骨骼表现。