Literature DB >> 32092470

Successful hematopoietic stem cell transplant in leukocyte adhesion deficiency type III presenting primarily as malignant infantile osteopetrosis.

Mohammed F Essa1, Enas Elbashir2, Fayhan Alroqi3, Reem Mohammed4, Abdulrahman Alsultan5.   

Abstract

Leukocyte adhesion deficiency type III (LAD-III) is caused by mutations in FERMT3 that encodes Kindlin-3 which regulates integrins activation. LAD-III predisposes to infections and bleeding. Osteopetrosis was reported in some cases. We report three patients who presented as malignant infantile osteopetrosis. One had recurrent infections and none had bleeding. Exome sequencing revealed a novel homozygous mutation in FERMT3 c.1555C > T (p.Gln519Ter). Two patients underwent successful hematopoietic stem cell transplant (HSCT) from matched siblings with resolution of osteopetrosis. The third patient died secondary to sepsis prior to HSCT. Our results support early HSCT in LAD-III prior to the occurrence of life-threatening complications.
Copyright © 2020 Elsevier Inc. All rights reserved.

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Year:  2020        PMID: 32092470     DOI: 10.1016/j.clim.2020.108365

Source DB:  PubMed          Journal:  Clin Immunol        ISSN: 1521-6616            Impact factor:   3.969


  4 in total

1.  Allogeneic hematopoietic stem cell transplantation in leukocyte adhesion deficiency type I and III.

Authors:  Shahrzad Bakhtiar; Emilia Salzmann-Manrique; Henric-Jan Blok; Dirk-Jan Eikema; Sheree Hazelaar; Mouhab Ayas; Amos Toren; Gal Goldstein; Despina Moshous; Franco Locatelli; Pietro Merli; Gerard Michel; Gülyüz Öztürk; Ansgar Schulz; Carsten Heilmann; Marianne Ifversen; Rob F Wynn; Olga Aleinikova; Yves Bertrand; Abdelghani Tbakhi; Paul Veys; Musa Karakukcu; Alphan Kupesiz; Ardeshir Ghavamzadeh; Rupert Handgretinger; Emel Unal; Antonio Perez-Martinez; Muge Gokce; Fulvio Porta; Tekin Aksu; Gülsün Karasu; Isabel Badell; Per Ljungman; Elena Skorobogatova; Akif Yesilipek; Tsila Zuckerman; Robbert R G Bredius; Polina Stepensky; Bella Shadur; Mary Slatter; Andrew R Gennery; Michael H Albert; Peter Bader; Arjan Lankester
Journal:  Blood Adv       Date:  2021-01-12

Review 2.  Disease Presentation, Treatment Options, and Outcomes for Myeloid Immunodeficiencies.

Authors:  Elizabeth M Kang
Journal:  Curr Allergy Asthma Rep       Date:  2021-03-05       Impact factor: 4.806

Review 3.  Actin Remodeling Defects Leading to Autoinflammation and Immune Dysregulation.

Authors:  Riccardo Papa; Federica Penco; Stefano Volpi; Marco Gattorno
Journal:  Front Immunol       Date:  2021-01-07       Impact factor: 7.561

Review 4.  Understanding the Role of LFA-1 in Leukocyte Adhesion Deficiency Type I (LAD I): Moving towards Inflammation?

Authors:  Julia Fekadu; Ute Modlich; Peter Bader; Shahrzad Bakhtiar
Journal:  Int J Mol Sci       Date:  2022-03-25       Impact factor: 5.923

  4 in total

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