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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| Format: | Article |
| Language: | English |
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Frontiers Media S.A.
2022-10-01
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| Series: | Frontiers in Immunology |
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fimmu.2022.1020362/full |
| _version_ | 1797990850958983168 |
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| author | Motoi Yamashita Shiori Eguchi Dan Tomomasa Takahiro Kamiya Daiki Niizato Noriko Mitsuiki Takeshi Isoda Hanako Funakoshi Yuki Mizuno Kentaro Okamoto Tuan Minh Nguyen Hidetoshi Takada Masatoshi Takagi Kohsuke Imai Kohsuke Imai Tomohiro Morio Hirokazu Kanegane |
| author_facet | Motoi Yamashita Shiori Eguchi Dan Tomomasa Takahiro Kamiya Daiki Niizato Noriko Mitsuiki Takeshi Isoda Hanako Funakoshi Yuki Mizuno Kentaro Okamoto Tuan Minh Nguyen Hidetoshi Takada Masatoshi Takagi Kohsuke Imai Kohsuke Imai Tomohiro Morio Hirokazu Kanegane |
| author_sort | Motoi Yamashita |
| collection | DOAJ |
| 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. |
| first_indexed | 2024-04-11T08:43:07Z |
| format | Article |
| id | doaj.art-662fd5bf24564482853392d7c4f0b1bc |
| institution | Directory Open Access Journal |
| issn | 1664-3224 |
| language | English |
| last_indexed | 2024-04-11T08:43:07Z |
| publishDate | 2022-10-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Immunology |
| spelling | doaj.art-662fd5bf24564482853392d7c4f0b1bc2022-12-22T04:34:05ZengFrontiers Media S.A.Frontiers in Immunology1664-32242022-10-011310.3389/fimmu.2022.10203621020362Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type IMotoi Yamashita0Shiori Eguchi1Dan Tomomasa2Takahiro Kamiya3Daiki Niizato4Noriko Mitsuiki5Takeshi Isoda6Hanako Funakoshi7Yuki Mizuno8Kentaro Okamoto9Tuan Minh Nguyen10Hidetoshi Takada11Masatoshi Takagi12Kohsuke Imai13Kohsuke Imai14Tomohiro Morio15Hirokazu Kanegane16Department of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, JapanDepartment of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, JapanDepartment of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, JapanDivision of Clinical Management, Clinical Research Center, Tokyo Medical and Dental University (TMDU), Tokyo, JapanDepartment of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, JapanDepartment of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, JapanDepartment of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, JapanDivision of Infectious Diseases, Department of Pediatrics, Tokyo Metropolitan Children’s Medical Center, Tokyo, JapanDepartment of Pediatric Surgery, Tokyo Medical and Dental University Hospital, Tokyo, JapanDepartment of Pediatric Surgery, Tokyo Medical and Dental University Hospital, Tokyo, JapanDepartment of Hematology, Children’s Hospital 1, HCM, VietnamDepartment of Child Health, Faculty of Medicine, University of Tsukuba, Tsukuba, JapanDepartment of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, JapanDepartment of Community Pediatrics, Perinatal and Maternal Medicine, Tokyo Medical and Dental University (TUDU), Tokyo, JapanDepartment of Pediatrics, National Defense Medical College, Tokorozawa, JapanDepartment of Pediatrics and Developmental Biology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, JapanDepartment of Child Health and Development, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University (TMDU), Tokyo, JapanLeukocyte 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.https://www.frontiersin.org/articles/10.3389/fimmu.2022.1020362/fullleukocyte adhesion deficiencyposttransplant cyclophosphamidehematopoietic cell transplantationgraft-versus-host diseaseinborn errors of immunity |
| spellingShingle | Motoi Yamashita Shiori Eguchi Dan Tomomasa Takahiro Kamiya Daiki Niizato Noriko Mitsuiki Takeshi Isoda Hanako Funakoshi Yuki Mizuno Kentaro Okamoto Tuan Minh Nguyen Hidetoshi Takada Masatoshi Takagi Kohsuke Imai Kohsuke Imai Tomohiro Morio Hirokazu Kanegane Case report: HLA-haploidentical hematopoietic cell transplant with posttransplant cyclophosphamide in a patient with leukocyte adhesion deficiency type I Frontiers in Immunology leukocyte adhesion deficiency posttransplant cyclophosphamide hematopoietic cell transplantation graft-versus-host disease inborn errors of immunity |
| 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 | leukocyte adhesion deficiency posttransplant cyclophosphamide hematopoietic cell transplantation graft-versus-host disease inborn errors of immunity |
| url | https://www.frontiersin.org/articles/10.3389/fimmu.2022.1020362/full |
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