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Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type I

Leukocyte adhesion deficiency type I (LAD-I) is a rare autosomal recessive inborn error of immunity (IEI) caused by the defects in CD18, encoded by the ITGB2 gene. LAD-I is characterized by defective leukocyte adhesion to the vascular endothelium and impaired migration of leukocytes. Allogeneic hema...

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Autores principales: Yamashita, Motoi, Eguchi, Shiori, Tomomasa, Dan, Kamiya, Takahiro, Niizato, Daiki, Mitsuiki, Noriko, Isoda, Takeshi, Funakoshi, Hanako, Mizuno, Yuki, Okamoto, Kentaro, Nguyen, Tuan Minh, Takada, Hidetoshi, Takagi, Masatoshi, Imai, Kohsuke, Morio, Tomohiro, Kanegane, Hirokazu
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9638093/
https://www.ncbi.nlm.nih.gov/pubmed/36353617
http://dx.doi.org/10.3389/fimmu.2022.1020362
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author Yamashita, Motoi
Eguchi, Shiori
Tomomasa, Dan
Kamiya, Takahiro
Niizato, Daiki
Mitsuiki, Noriko
Isoda, Takeshi
Funakoshi, Hanako
Mizuno, Yuki
Okamoto, Kentaro
Nguyen, Tuan Minh
Takada, Hidetoshi
Takagi, Masatoshi
Imai, Kohsuke
Morio, Tomohiro
Kanegane, Hirokazu
author_facet Yamashita, Motoi
Eguchi, Shiori
Tomomasa, Dan
Kamiya, Takahiro
Niizato, Daiki
Mitsuiki, Noriko
Isoda, Takeshi
Funakoshi, Hanako
Mizuno, Yuki
Okamoto, Kentaro
Nguyen, Tuan Minh
Takada, Hidetoshi
Takagi, Masatoshi
Imai, Kohsuke
Morio, Tomohiro
Kanegane, Hirokazu
author_sort Yamashita, Motoi
collection PubMed
description Leukocyte adhesion deficiency type I (LAD-I) is a rare autosomal recessive inborn error of immunity (IEI) caused by the defects in CD18, encoded by the ITGB2 gene. LAD-I is characterized by defective leukocyte adhesion to the vascular endothelium and impaired migration of leukocytes. Allogeneic hematopoietic cell transplant (HCT) is the only curative treatment for LAD-I. In an absence of ideal donor for HCT, human leukocyte antigen (HLA)-haploidentical HCT is performed. Posttransplant cyclophosphamide (PT-CY) is a relatively new graft-versus-host disease (GVHD) prophylactic measure and has been increasingly used in HLA-haploidentical HCT for malignant and nonmalignant diseases. However, experience in using PT-CY for rare IEIs, such as LAD-I, is very limited. We report a case of LAD-I successfully treated with HLA-haploidentical HCT with PT-CY. Complete chimerism was achieved, and the patient was cured. Her transplant course was complicated by mild GVHD, cytomegalovirus reactivation and veno-occlusive disease/sinusoidal obstruction syndrome, which were successfully treated. HLA-haploidentical HCT with PT-CY is a safe and effective option for patients with LAD-I when HLA-matched donors are unavailable.
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spelling pubmed-96380932022-11-08 Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type I Yamashita, Motoi Eguchi, Shiori Tomomasa, Dan Kamiya, Takahiro Niizato, Daiki Mitsuiki, Noriko Isoda, Takeshi Funakoshi, Hanako Mizuno, Yuki Okamoto, Kentaro Nguyen, Tuan Minh Takada, Hidetoshi Takagi, Masatoshi Imai, Kohsuke Morio, Tomohiro Kanegane, Hirokazu Front Immunol Immunology Leukocyte adhesion deficiency type I (LAD-I) is a rare autosomal recessive inborn error of immunity (IEI) caused by the defects in CD18, encoded by the ITGB2 gene. LAD-I is characterized by defective leukocyte adhesion to the vascular endothelium and impaired migration of leukocytes. Allogeneic hematopoietic cell transplant (HCT) is the only curative treatment for LAD-I. In an absence of ideal donor for HCT, human leukocyte antigen (HLA)-haploidentical HCT is performed. Posttransplant cyclophosphamide (PT-CY) is a relatively new graft-versus-host disease (GVHD) prophylactic measure and has been increasingly used in HLA-haploidentical HCT for malignant and nonmalignant diseases. However, experience in using PT-CY for rare IEIs, such as LAD-I, is very limited. We report a case of LAD-I successfully treated with HLA-haploidentical HCT with PT-CY. Complete chimerism was achieved, and the patient was cured. Her transplant course was complicated by mild GVHD, cytomegalovirus reactivation and veno-occlusive disease/sinusoidal obstruction syndrome, which were successfully treated. HLA-haploidentical HCT with PT-CY is a safe and effective option for patients with LAD-I when HLA-matched donors are unavailable. Frontiers Media S.A. 2022-10-24 /pmc/articles/PMC9638093/ /pubmed/36353617 http://dx.doi.org/10.3389/fimmu.2022.1020362 Text en Copyright © 2022 Yamashita, Eguchi, Tomomasa, Kamiya, Niizato, Mitsuiki, Isoda, Funakoshi, Mizuno, Okamoto, Nguyen, Takada, Takagi, Imai, Morio and Kanegane https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Immunology
Yamashita, Motoi
Eguchi, Shiori
Tomomasa, Dan
Kamiya, Takahiro
Niizato, Daiki
Mitsuiki, Noriko
Isoda, Takeshi
Funakoshi, Hanako
Mizuno, Yuki
Okamoto, Kentaro
Nguyen, Tuan Minh
Takada, Hidetoshi
Takagi, Masatoshi
Imai, Kohsuke
Morio, Tomohiro
Kanegane, Hirokazu
Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type I
title Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type I
title_full Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type I
title_fullStr Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type I
title_full_unstemmed Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type I
title_short Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type I
title_sort case report: hla-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type i
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9638093/
https://www.ncbi.nlm.nih.gov/pubmed/36353617
http://dx.doi.org/10.3389/fimmu.2022.1020362
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