Literature DB >> 11442486

Two further British families with the 'cryohydrocytosis' form of hereditary stomatocytosis.

P G Haines1, H G Jarvis, S King, F H Noormohamed, M C Chetty, J Fisher, P Hill, A Nicolaou, G W Stewart.   

Abstract

We describe two families with the 'cryohydrocytosis' form of stomatocytosis. Both show a mild stomatocytic anaemia with Hb levels of 12-16 g/dl and reticulocyte counts of 4.3-24%, with very marked autohaemolysis at refrigerator temperatures and pseudohyperkalaemia as a result of loss of K from red cells on storage at room temperature. The ouabain + bumetanide-insensitive 'passive leak' K influx showed a 'U'-shaped temperature dependence, with a minimum at 23 degrees C. In one family, there was consistent variation in haematological severity within the pedigree. In the other, the parents of the proposita were normal, but all three of her children were affected, consistent with a new mutation of a dominant condition. Cold storage of the red cells led to a very marked increase in osmotic fragility and macrospherocytosis, explaining why a diagnosis of 'hereditary spherocytosis' can easily be reached in these pedigrees.

Entities:  

Mesh:

Substances:

Year:  2001        PMID: 11442486     DOI: 10.1046/j.1365-2141.2001.02792.x

Source DB:  PubMed          Journal:  Br J Haematol        ISSN: 0007-1048            Impact factor:   6.998


  2 in total

1.  The GPA-dependent, spherostomatocytosis mutant AE1 E758K induces GPA-independent, endogenous cation transport in amphibian oocytes.

Authors:  Andrew K Stewart; David H Vandorpe; John F Heneghan; Fouad Chebib; Kathleen Stolpe; Arash Akhavein; E Jennifer Edelman; Yelena Maksimova; Patrick G Gallagher; Seth L Alper
Journal:  Am J Physiol Cell Physiol       Date:  2009-11-11       Impact factor: 4.249

Review 2.  The Molecular Basis for Altered Cation Permeability in Hereditary Stomatocytic Human Red Blood Cells.

Authors:  Joanna F Flatt; Lesley J Bruce
Journal:  Front Physiol       Date:  2018-04-16       Impact factor: 4.566

  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.