Vandenplas Y, Franckx J, Liebaers I, Ketelbant P, Sacre L
Eur J Pediatr. 1985 Nov;144(4):391-4. doi: 10.1007/BF00441785.
SZ-Alpha 1-antitrypsin deficiency, leading to severe transient neonatal cholestasis with mild hepatitis is rare. In our patient, intrahepatic bile duct hypoplasia was suspected. Since cholecystography and hepatic scintigraphy failed to reveal intrahepatic bile ducts, a diagnostic surgical liver biopsy was performed. Typical intrahepatocytic PAS-positive granules and a low serum alpha 1-antitrypsin concentration allowed a correct diagnosis of the SZ phenotype. The administration of an elementary diet quickly improved the clinical condition of our patient. Follow-up studies of this case and others will allow a better establishment of prognostic criteria and help in genetic counselling.
伴有轻度肝炎的严重短暂性新生儿胆汁淤积症的SZ-α1抗胰蛋白酶缺乏症很罕见。在我们的患者中,怀疑存在肝内胆管发育不全。由于胆囊造影和肝脏闪烁扫描未能显示肝内胆管,因此进行了诊断性手术肝活检。典型的肝内PAS阳性颗粒和低血清α1抗胰蛋白酶浓度使得能够正确诊断SZ表型。给予要素饮食迅速改善了我们患者的临床状况。对该病例和其他病例的随访研究将有助于更好地确立预后标准并有助于遗传咨询。