Literature DB >> 1832935

Ankyrin deficiency in dominant hereditary spherocytosis: report of three cases.

A Iolascon1, E Miraglia del Giudice, C Camaschella, L Pinto, B Nobili, S Perrotta, S Cutillo.   

Abstract

We describe three Italian subjects from two unrelated families affected with isolated hereditary spherocytosis (HS) without other clinical abnormalities, associated with partial spectrin and ankyrin deficiency. In both families the propositus has normal biological parents, and thus appears to be the result of a new mutation; in one of them the disease is further transmitted in an autosomal dominant fashion. Cytogenetic analysis of the latter family excluded abnormalities of the short arm of chromosome 8. We speculate that in both kindreds ankyrin deficiency is the primary defect related to ankyrin gene mutation. Several pieces of evidence suggest that ankyrin deficiency is probably the most common molecular defect in HS. It is inherited in a, dominant manner and its clinical and biochemical expression is heterogenous.

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Year:  1991        PMID: 1832935     DOI: 10.1111/j.1365-2141.1991.tb04487.x

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


  4 in total

1.  Severe Ankyrin-R deficiency results in impaired surface retention and lysosomal degradation of RhAG in human erythroblasts.

Authors:  Timothy J Satchwell; Amanda J Bell; Bethan R Hawley; Stephanie Pellegrin; Kathryn E Mordue; Cees Th B M van Deursen; Nicole Heitink-Ter Braak; Gerwin Huls; Mathie P G Leers; Eline Overwater; Rienk Y J Tamminga; Bert van der Zwaag; Elisa Fermo; Paola Bianchi; Richard van Wijk; Ashley M Toye
Journal:  Haematologica       Date:  2016-05-31       Impact factor: 9.941

2.  Molecular basis of spectrin deficiency in beta spectrin Durham. A deletion within beta spectrin adjacent to the ankyrin-binding site precludes spectrin attachment to the membrane in hereditary spherocytosis.

Authors:  H Hassoun; J N Vassiliadis; J Murray; S J Yi; M Hanspal; R E Ware; S S Winter; S S Chiou; J Palek
Journal:  J Clin Invest       Date:  1995-12       Impact factor: 14.808

3.  An 11-amino acid beta-hairpin loop in the cytoplasmic domain of band 3 is responsible for ankyrin binding in mouse erythrocytes.

Authors:  Marko Stefanovic; Nicholas O Markham; Erin M Parry; Lisa J Garrett-Beal; Amanda P Cline; Patrick G Gallagher; Philip S Low; David M Bodine
Journal:  Proc Natl Acad Sci U S A       Date:  2007-08-22       Impact factor: 11.205

4.  Combined ankyrin and spectrin deficiency in hereditary spherocytosis.

Authors:  A Pekrun; S W Eber; A Kuhlmey; W Schröter
Journal:  Ann Hematol       Date:  1993-08       Impact factor: 3.673

  4 in total

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