Neonatal liver failure and haemophagocytic lymphohistiocytosis caused by a new perforin mutation

Acta Paediatr. 2010 May;99(5):778-780. doi: 10.1111/j.1651-2227.2009.01647.x. Epub 2009 Jan 5.

Abstract

Acute liver failure is a rare heterogeneous syndrome in neonates. We report of a newborn with haemophagocytic lymphohistiocytosis presenting as acute liver failure. Pancytopenia and multi-organ failure occurred later in the course. He carried two mutations of the perforin gene (PRF-1), one of which not previously described, causing a complete loss of perforin expression and natural killer cell function.

Conclusion: Perforin expression and function should be promptly assessed in neonatal/infantile acute liver failure, as haemophagocytic lymphohistiocytosis requires specific treatment and represents a contra-indication to liver transplant.

Publication types

  • Case Reports

MeSH terms

  • Blood Coagulation Disorders / etiology
  • Fatal Outcome
  • Fever / etiology
  • Humans
  • Infant, Newborn
  • Jaundice, Neonatal / etiology
  • Killer Cells, Natural
  • Liver Failure, Acute / etiology*
  • Lymphohistiocytosis, Hemophagocytic / complications
  • Lymphohistiocytosis, Hemophagocytic / diagnosis*
  • Lymphohistiocytosis, Hemophagocytic / genetics
  • Male
  • Mutation*
  • Perforin / genetics*

Substances

  • Perforin