Literature DB >> 23624969

A neonate with Coombs-negative hemolytic jaundice with spherocytes but normal erythrocyte indices: a rare case of autosomal-recessive hereditary spherocytosis due to alpha-spectrin deficiency.

H M Yaish1, R D Christensen, A Agarwal.   

Abstract

The diagnosis of hereditary spherocytosis (HS) in a newborn infant is generally made on the basis of a positive family history, spherocytes on blood film and Coombs-negative hemolytic jaundice of variable severity with an elevated mean corpuscular hemoglobin concentration (MCHC) and a low mean corpuscular volume (MCV). In general, sodium dodecyl sulfate polyacrylamide gel electrophoresis (SDS-PAGE) quantification of erythrocyte membrane proteins is not needed to make the clinical diagnosis of HS. However, we observed that a neonate with no family history of HS, but with abundant spherocytosis on repeated blood films, Coombs-negative hemolytic jaundice and normal MCHC and MCV measurements, where SDS-PAGE revealed alpha-spectrin deficiency, a rare autosomal-recessive variety of HS that generally has a severe clinical phenotype.

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Year:  2013        PMID: 23624969     DOI: 10.1038/jp.2012.67

Source DB:  PubMed          Journal:  J Perinatol        ISSN: 0743-8346            Impact factor:   2.521


  4 in total

1.  Evaluating eosin-5-maleimide binding as a diagnostic test for hereditary spherocytosis in newborn infants.

Authors:  R D Christensen; A M Agarwal; R H Nussenzveig; N Heikal; M A Liew; H M Yaish
Journal:  J Perinatol       Date:  2014-11-06       Impact factor: 2.521

2.  Mean corpuscular volume of control red blood cells determines the interpretation of eosin-5'-maleimide (EMA) test result in infants aged less than 6 months.

Authors:  Olga Ciepiela; Anna Adamowicz-Salach; Weronika Bystrzycka; Jan Łukasik; Iwona Kotuła
Journal:  Ann Hematol       Date:  2015-04-25       Impact factor: 3.673

3.  Nonimmune hydrops fetalis due to autosomal recessive hereditary spherocytosis.

Authors:  Dawn M Hannah; Terry B Tressler; Claudia D Taboada
Journal:  Case Rep Womens Health       Date:  2017-10-02

4.  Novel compound heterozygous mutations in the SPTA1 gene, causing hereditary spherocytosis in a neonate with Coombs‑negative hemolytic jaundice.

Authors:  Xiong Wang; Aiguo Liu; Yanjun Lu; Qun Hu
Journal:  Mol Med Rep       Date:  2019-02-08       Impact factor: 2.952

  4 in total

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