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一名进行性骨化性肌炎患儿因1-羟基-1,1-二膦酸乙烷(EHDP)导致慢性中毒。

Chronic intoxication by ethane-1-hydroxy-1,1-diphosphonate (EHDP) in a child with myositis ossificans progressiva.

作者信息

Pazzaglia U E, Beluffi G, Ravelli A, Zatti G, Martini A

机构信息

Clinica Ortopedica, Ospedale F. Del Ponte, Varese, Italy.

出版信息

Pediatr Radiol. 1993;23(6):459-62. doi: 10.1007/BF02012453.

Abstract

A child with myositis ossificans progressiva was treated for 8 years with ethane-1-hydroxy-1,1-diphosphonate (EHDP) 30-40 mg/kg per day. Latterly he complained of severe, progressive bone and joint pain which made standing and walking almost impossible. A radiographic skeletal survey showed diffuse ricket-like lesions. Withdrawal of EHDP therapy produced substantial improvement in his general condition as well as in the radiographic appearance of the bones. Multiple exostoses were observed in this case and, particularly those around the knees, presented a peculiar morphology. This supports the theory that exostoses originate from a defect of metaphyseal modelling.

摘要

一名进行性骨化性肌炎患儿接受了8年的1-羟基-1,1-二膦酸乙烷(EHDP)治疗,剂量为每日30 - 40毫克/千克。后来他抱怨严重的进行性骨和关节疼痛,几乎无法站立和行走。骨骼X线检查显示弥漫性佝偻病样病变。停用EHDP治疗后,他的一般状况以及骨骼的X线表现都有了显著改善。该病例中观察到多发外生骨疣,尤其是膝盖周围的外生骨疣呈现出特殊的形态。这支持了外生骨疣起源于干骺端塑形缺陷的理论。

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