Di Rocco M, Callea F, Pollice B, Faraci M, Campiani F, Borrone C
Divisione Pediatria II, Istituto G. Gaslini, Genova, Italy.
Eur J Pediatr. 1995 Oct;154(10):835-9. doi: 10.1007/BF01959793.
We report on five patients from three families with neurogenic arthrogryposis, cholestasis and tubular renal dysfunction. Despite a similar clinical picture the liver histology showed a broad pathological spectrum, ranging from pigment storage to parenchymal giant cell transformation and ductopenia. The findings are compared with those of other cases from the literature in search of a correct nosology of the syndrome characterized by arthrogryposis, renal and liver disease. CONCLUSION. We propose to consider the picture of arthrogryposis, renal tubular dysfunction and cholestasis as a single syndrome.
我们报告了来自三个家庭的五名患有神经源性关节挛缩、胆汁淤积和肾小管功能障碍的患者。尽管临床表现相似,但肝脏组织学显示出广泛的病理谱,从色素沉着到实质巨细胞转化和胆管减少。将这些发现与文献中其他病例的发现进行比较,以寻找以关节挛缩、肾脏和肝脏疾病为特征的综合征的正确分类学。结论。我们建议将关节挛缩、肾小管功能障碍和胆汁淤积的情况视为一种单一综合征。