Literature DB >> 265092

Syndrome of Shwachman and leukaemia.

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


  6 in total

1.  Shwachman-Diamond syndrome with exocrine pancreatic dysfunction and bone marrow failure maps to the centromeric region of chromosome 7.

Authors:  S Goobie; M Popovic; J Morrison; L Ellis; H Ginzberg; G R Boocock; N Ehtesham; C Bétard; C G Brewer; N M Roslin; T J Hudson; K Morgan; T M Fujiwara; P R Durie; J M Rommens
Journal:  Am J Hum Genet       Date:  2001-03-15       Impact factor: 11.025

2.  Response to comments from Drs. Alter and Rosenberg.

Authors:  Yigal Dror; Michaela Cada
Journal:  Haematologica       Date:  2015-09       Impact factor: 9.941

3.  Shwachman's syndrome and leukaemia.

Authors:  J Caselitz; G Klöppel; G Delling; R Grüttner; U Holdhoff; M Stern
Journal:  Virchows Arch A Pathol Anat Histol       Date:  1979-12

4.  A case of Shwachman syndrome with increased spontaneous chromosome breakage.

Authors:  H Tada; T Ri; H Yoshida; K Ishimoto; M Kaneko; Y Yamashiro; T Shinohara
Journal:  Hum Genet       Date:  1987-11       Impact factor: 4.132

5.  The impact of category, cytopathology and cytogenetics on development and progression of clonal and malignant myeloid transformation in inherited bone marrow failure syndromes.

Authors:  Michaela Cada; Catherin I Segbefia; Robert Klaassen; Conrad V Fernandez; Rochelle A Yanofsky; John Wu; Yves Pastore; Mariana Silva; Jeffrey H Lipton; Josee Brossard; Bruno Michon; Sharon Abish; MacGregor Steele; Roona Sinha; Mark Belletrutti; Vicky Breakey; Lawrence Jardine; Lisa Goodyear; Lillian Sung; Mary Shago; Joseph Beyene; Preeti Sharma; Bozana Zlateska; Yigal Dror
Journal:  Haematologica       Date:  2015-02-14       Impact factor: 9.941

Review 6.  Congenital neutropenia: diagnosis, molecular bases and patient management.

Authors:  Jean Donadieu; Odile Fenneteau; Blandine Beaupain; Nizar Mahlaoui; Christine Bellanné Chantelot
Journal:  Orphanet J Rare Dis       Date:  2011-05-19       Impact factor: 4.123

  6 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.