Literature DB >> 3878294

Neonatal hepatitis with obstructive jaundice in an SZ heterozygous alpha 1-antitrypsin-deficient boy and destructive lung disease in his SZ mother. A review of the literature.

Y Vandenplas, J Franckx, I Liebaers, P Ketelbant, L Sacre.   

Abstract

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.

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Year:  1985        PMID: 3878294     DOI: 10.1007/bf00441785

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  31 in total

Review 1.  alpha1-antitrypsin deficiency (first of two parts).

Authors:  J O Morse
Journal:  N Engl J Med       Date:  1978-11-09       Impact factor: 91.245

2.  Cirrhosis associated with alpha-1-antitrypsin deficiency: a previously unrecognized inherited disorder.

Authors:  H L Sharp; R A Bridges; W Krivit; E F Freier
Journal:  J Lab Clin Med       Date:  1969-06

3.  alpha1-Antitrypsin deficiency in nonsmokers.

Authors:  L F Black; F Kueppers
Journal:  Am Rev Respir Dis       Date:  1978-03

4.  Prenatal diagnosis of inborn errors of metabolism. Present status and new approaches.

Authors:  E Vamos; I Liebaers
Journal:  Enzyme       Date:  1984

5.  High-level production of biologically active human alpha 1-antitrypsin in Escherichia coli.

Authors:  M Courtney; A Buchwalder; L H Tessier; M Jaye; A Benavente; A Balland; V Kohli; R Lathe; P Tolstoshev; J P Lecocq
Journal:  Proc Natl Acad Sci U S A       Date:  1984-02       Impact factor: 11.205

6.  Expression of human alpha 1-antitrypsin in Escherichia coli.

Authors:  A Bollen; R Loriau; A Herzog; P Hérion
Journal:  FEBS Lett       Date:  1984-01-23       Impact factor: 4.124

7.  Prenatal diagnosis of alpha 1-antitrypsin deficiency by direct analysis of the mutation site in the gene.

Authors:  V J Kidd; M S Golbus; R B Wallace; K Itakura; S L Woo
Journal:  N Engl J Med       Date:  1984-03-08       Impact factor: 91.245

8.  Transport of proteases across neonatal intestine and development of liver disease in infants with alpha-1 antitrypsin deficiency.

Authors:  J N Udall; K J Bloch; W A Walker
Journal:  Lancet       Date:  1982-06-26       Impact factor: 79.321

9.  Mass screening of newborn Swedish infants for alpha antitrypsin deficiency.

Authors:  C B Laurell; T Sveger
Journal:  Am J Hum Genet       Date:  1975-03       Impact factor: 11.025

10.  Histopathologic study of the liver in the early cholestatic phase of alpha-1-antitrypsin deficiency.

Authors:  M Hadchouel; M Gautier
Journal:  J Pediatr       Date:  1976-08       Impact factor: 4.406

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