Literature DB >> 32355479

Deletion in the A4GALT Gene Associated with Rare "P null" Phenotype: The First Report from India.

Shamee Shastry1, Kapaettu Satyamoorthy2, Kiran V Acharya3, Vijay Ram Reddy1, Ganesh Mohan1, Chenna Deepika1, Dinesh Reghunathan2, Manjunath B Joshi2.   

Abstract

BACKGROUND: The present report illustrates a case with rare "P null" phenotype due to a large deletion in chromosome 22q13.2 and with clinically significant anti-PP1P<sup>k</sup> antibody. Patient blood management in such cases is challenging. CASE REPORT: The transfusion center supporting the tertiary care referral center in the southern part of India received a blood sample from a trauma case for pre-transfusion testing. An antibody to a high-frequency blood group antigen was initially suspected. Following extensive immune-hematological workup, the patient was diagnosed to have naturally occurring anti-PP1P<sup>k</sup> antibody and a rare "P null" phenotype. The genomic DNA of the patient was analyzed by exome sequencing followed by Sanger's sequencing. Molecular diagnostics revealed a large 21-bp deletion in chromosome 22q13.2 which encodes the A4GALT gene, resulting in truncation of seven amino acids I245-251P and resulted in rare "P null" phenotype. Patient blood management strategies were adopted to manage the patient conservatively without blood transfusion.
CONCLUSION: A large deletion in chromosome 22q13.2 had resulted in a rare "P null" phenotype in the present case. The patient was a victim of a road traffic accident, required emergency hospitalization, as well as surgical intervention, and his plasma had antibodies to high-frequency antigens. A rare donor registry plays a major role in providing transfusion support to such cases.
Copyright © 2019 by S. Karger AG, Basel.

Entities:  

Keywords:  A4GALT gene; Anti-PP1Pk; Blood transfusion; Large deletion; Null phenotype; Patient blood management; Rare blood group

Year:  2019        PMID: 32355479      PMCID: PMC7184825          DOI: 10.1159/000501916

Source DB:  PubMed          Journal:  Transfus Med Hemother        ISSN: 1660-3796            Impact factor:   3.747


  15 in total

1.  Identification of anti-PP1P(k) in a blood donor and her family: a case report following her pregnancy and review.

Authors:  Georgette R Benidt; Elizabeth A Jaben; Jeffrey L Winters; James R Stubbs
Journal:  Transfus Apher Sci       Date:  2010-10-27       Impact factor: 1.764

2.  Expression of a novel missense mutation found in the A4GALT gene of Amish individuals with the p phenotype.

Authors:  Asa Hellberg; Anne-Christine Schmidt-Melbye; Marion E Reid; Martin L Olsson
Journal:  Transfusion       Date:  2007-12-07       Impact factor: 3.157

3.  Acute renal failure due to haemoglobinuria secondary to P antigen.

Authors:  Clara Gómez-Pérez; Ane Mujika-López; Teresa Visus-Fernández-de Manzanos; Eva Álvarez-Quintana; Francisco J Gainza-de Los Ríos; Nerea Gómez-Larrambe
Journal:  Nefrologia       Date:  2014       Impact factor: 2.033

4.  A novel mutation in A4GALT was identified in a Chinese individual with p phenotype.

Authors:  Xiaofei Li; Xueqin Diao; Xiaoye Xia; Xiaozhen Hong; Faming Zhu
Journal:  Transfusion       Date:  2016-09-09       Impact factor: 3.157

5.  Large deletions involving the regulatory upstream regions of A4GALT give rise to principally novel P1PK-null alleles.

Authors:  Julia S Westman; Asa Hellberg; Thierry Peyrard; Britt Thuresson; Martin L Olsson
Journal:  Transfusion       Date:  2014-01-14       Impact factor: 3.157

6.  Unexpected Non-Maternally Derived Anti-PP1Pk in an 11-Week-Old Patient.

Authors:  Hollie M Reeves; Victoria Cary; Mary Ann Mino; Claire McGrath; James A Westra; Connie Piccone; Katharine A Downes
Journal:  J Pediatr       Date:  2016-11-14       Impact factor: 4.406

7.  [A rare p phenotype caused by a 26-bp deletion in α 1,4-galactosyltransferase gene].

Authors:  Xianguo Xu; Xiaozhen Hong; Kairong Ma; Xiaofei Lan; Shu Chen; Ying Liu; Yanling Ying; Faming Zhu; Hangjun Lv
Journal:  Zhonghua Yi Xue Yi Chuan Xue Za Zhi       Date:  2013-06

8.  Hemolytic disease of the newborn due to anti-PP1 P k (anti-Tj a).

Authors:  C Levene; R Sela; Y Rudolphson; I Nathan; M Karplus; A Dvilansky
Journal:  Transfusion       Date:  1977 Nov-Dec       Impact factor: 3.157

9.  A profile of rare bloods in India and its impact in blood transfusion service.

Authors:  Sanmukh R Joshi; K Vasantha
Journal:  Asian J Transfus Sci       Date:  2012-01

10.  Evaluation of an amino acid residue critical for the specificity and activity of human Gb3/CD77 synthase.

Authors:  Radoslaw Kaczmarek; Katarzyna Mikolajewicz; Katarzyna Szymczak; Maria Duk; Edyta Majorczyk; Anna Krop-Watorek; Anna Buczkowska; Marcin Czerwinski
Journal:  Glycoconj J       Date:  2016-08-18       Impact factor: 2.916

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  2 in total

1.  P-Null Phenotype Due to a Rare Frame-Shift Mutation and with Allo-Anti-PP1Pk Causing a Severe Hemolytic Transfusion Reaction: A Case Report with Clinical Management.

Authors:  Ashish N Kanani; Snehal B Senjaliya; Manisha M Rajapara; Judith Aeschlimann; Connie M Westhoff; Sanmukh R Joshi
Journal:  Transfus Med Hemother       Date:  2021-02-25       Impact factor: 3.747

2.  Oration - Dr Sanmukh Joshi.

Authors: 
Journal:  Asian J Transfus Sci       Date:  2021-04
  2 in total

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