Cointin M, Sommelet-Olive D, Cuny J F, Bretagne M C
Centre de Convalescence Pédiatrique St Lambert-Lioux, Gordes.
Ann Pediatr (Paris). 1990 Sep;37(7):458-60.
Clinical, roentgenographic and biologic features of etretinate bone toxicity in a 13-year-old girl with pachyonychia congenita syndrome are reported. Etretinate is a synthetic derivative of vitamin A that infrequently induces bone and joint abnormalities in children. The following manifestations can be observed: cortical hyperostosis, pain, calcification of tendons, thinning of long bones, demineralization, premature closure of epiphyses, or abnormal remodelling. Onset of these anomalies is often delayed since etretinate has a long half-life. Mechanisms are unknown. We advocate use of the minimum effective dosage and regular monitoring of patients.
报告了一名患有先天性厚甲综合征的13岁女孩使用依曲替酯后出现骨毒性的临床、放射学和生物学特征。依曲替酯是维生素A的合成衍生物，在儿童中很少引起骨骼和关节异常。可观察到以下表现：皮质骨增生、疼痛、肌腱钙化、长骨变薄、骨质减少、骨骺过早闭合或异常重塑。由于依曲替酯半衰期长，这些异常的发作往往延迟。其机制尚不清楚。我们主张使用最低有效剂量并对患者进行定期监测。
