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

Copyright © 2020 Elsevier Inc. All rights reserved.

Détails bibliographiques
Publié dans:Clinical immunology (Orlando, Fla.). - 1999. - 213(2020) vom: 15. Apr., Seite 108365
Auteur principal: Essa, Mohammed F (Auteur)
Autres auteurs: Elbashir, Enas, Alroqi, Fayhan, Mohammed, Reem, Alsultan, Abdulrahman
Format: Article en ligne
Langue:English
Publié: 2020
Accès à la collection:Clinical immunology (Orlando, Fla.)
Sujets:Case Reports Journal Article FERMT3 protein, human Membrane Proteins Neoplasm Proteins
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100 1 |a Essa, Mohammed F  |e verfasserin  |4 aut 
245 1 0 |a Successful hematopoietic stem cell transplant in leukocyte adhesion deficiency type III presenting primarily as malignant infantile osteopetrosis 
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520 |a 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 
650 4 |a Case Reports 
650 4 |a Journal Article 
650 7 |a FERMT3 protein, human  |2 NLM 
650 7 |a Membrane Proteins  |2 NLM 
650 7 |a Neoplasm Proteins  |2 NLM 
700 1 |a Elbashir, Enas  |e verfasserin  |4 aut 
700 1 |a Alroqi, Fayhan  |e verfasserin  |4 aut 
700 1 |a Mohammed, Reem  |e verfasserin  |4 aut 
700 1 |a Alsultan, Abdulrahman  |e verfasserin  |4 aut 
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