Epstein S, Hamersma H, Beighton P
S Afr Med J. 1979 Jun 30;55(27):1105-10.
Sclerosteosis is a rare autosomal recessive condition which is characterized by excessive skeletal overgrowth, distortion of the facies, cranial nerve abnormalities and raised intracranial pressure. Syndactyly and digital malformation are associated features. Radiological examination reveals thickened sclerotic bone maximally involving the skull, including the pituitary fossa. Sclerosis and hyperostosis are present throughout the skeleton. Biochemical and endocrine tests were carred out on 3 patients with sclerosteosis in an attempt to detect any dysfunction of calcium regulation of the pituitary. Results revealed no abnormality of basal parathyroid or calcitonin secretion. Histological examination revealed quantitatively increased bone resorption in comparison with normal subjects, although the pattern resembled osteosclerosis. Regulation of growth hormone, adrenocorticotrophin, gonadotrophin and thyrotrophin function were intact. We conclude that pituitary function and calcium 'homeostasis' are normal in this disorder.
硬化性骨病是一种罕见的常染色体隐性疾病,其特征为骨骼过度生长、面部畸形、颅神经异常及颅内压升高。并指畸形和手指畸形是相关特征。放射学检查显示硬化骨增厚,主要累及颅骨,包括垂体窝。全身骨骼均有硬化和骨质增生。对3例硬化性骨病患者进行了生化和内分泌检查,试图检测垂体钙调节功能是否存在任何异常。结果显示基础甲状旁腺或降钙素分泌无异常。组织学检查显示,与正常受试者相比,骨吸收在数量上有所增加,尽管其模式类似骨硬化。生长激素、促肾上腺皮质激素、促性腺激素和促甲状腺激素功能的调节均正常。我们得出结论,该疾病中垂体功能和钙稳态是正常的。