| Literature DB >> 265092 |
P C Huijgens, E A van der Veen, S Meijer, O G Muntinghe.
Abstract
The syndrome of Shwachman is characterized by pancreatic insufficiency and bone marrow dysfunction, usually manifesting itself as neutropenia. The pancreas shows replacement of the exocrine glands by adipose tissue; sweat electrolytes are normal. A 23-year-old male who was known to suffer from neutropenia and pancreatic dysfunction from early childhood, presented with fever, acquired Pelger-Huet anomaly (of the polymorphonuclear granulocytes) and sideroblastic anaemia, a combination of symptoms suggestive of preleukaemia. A few months later he died of acute myeloblastic leukaemia and autopsy showed a dystrophic pancreas. Considering this case history it seems possible that the haematological anomalies of Shwachman's syndrome are signs of preleukaemia. Careful follow-up of patients suffering from Shwachman's syndrome seems warranted.Entities:
Mesh:
Year: 1977 PMID: 265092 DOI: 10.1111/j.1600-0609.1977.tb01473.x
Source DB: PubMed Journal: Scand J Haematol ISSN: 0036-553X