Chronic Graft-Versus-Host-Disease-Related Polymyositis: a 17-Months-Old Child with a Rare and Late Complication of Haematopoietic Stem Cell Transplantation

BACKGROUND: Chronic graft versus host disease (cGVHD) occurs in 20–30% of paediatric patients receiving haemopoietic stem cell transplantation (HSCT). Neuromuscular disorders such as polymyositis are considered a rare and distinctive but non-diagnostic manifestation of cGVHD and, in the absence of o...

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Autores principales: Chinello, Matteo, Balter, Rita, De Bortoli, Massimiliano, Vitale, Virginia, Zaccaron, Ada, Bonetti, Elisa, Tonin, Paola, Vattemi, Gaetano, Guglielmi, Valeria, Cesaro, Simone
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Università Cattolica del Sacro Cuore 2020
Materias:
Case Report
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6951354/
https://www.ncbi.nlm.nih.gov/pubmed/31934312
http://dx.doi.org/10.4084/MJHID.2020.002
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author Chinello, Matteo
Balter, Rita
De Bortoli, Massimiliano
Vitale, Virginia
Zaccaron, Ada
Bonetti, Elisa
Tonin, Paola
Vattemi, Gaetano
Guglielmi, Valeria
Cesaro, Simone
author_facet Chinello, Matteo
Balter, Rita
De Bortoli, Massimiliano
Vitale, Virginia
Zaccaron, Ada
Bonetti, Elisa
Tonin, Paola
Vattemi, Gaetano
Guglielmi, Valeria
Cesaro, Simone
author_sort Chinello, Matteo
collection PubMed
description BACKGROUND: Chronic graft versus host disease (cGVHD) occurs in 20–30% of paediatric patients receiving haemopoietic stem cell transplantation (HSCT). Neuromuscular disorders such as polymyositis are considered a rare and distinctive but non-diagnostic manifestation of cGVHD and, in the absence of other characteristic signs and symptoms, biopsy is highly recommended to exclude other causes. CASE REPORT: We report a case of a 17-months-old child affected by hemophagocytic lymphohistiocytosis who underwent a matched unrelated donor haematopoietic stem cell transplantation (HSCT). She developed severe cGVHD-related polymyositis that was successfully treated with high-dose steroid therapy, rituximab and sirolimus. CONCLUSIONS: This is the first case of cGVHD-related-polymyositis described in a pediatric patient which was successfully treated with rituximab.
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spelling pubmed-69513542020-01-13 Chronic Graft-Versus-Host-Disease-Related Polymyositis: a 17-Months-Old Child with a Rare and Late Complication of Haematopoietic Stem Cell Transplantation Chinello, Matteo Balter, Rita De Bortoli, Massimiliano Vitale, Virginia Zaccaron, Ada Bonetti, Elisa Tonin, Paola Vattemi, Gaetano Guglielmi, Valeria Cesaro, Simone Mediterr J Hematol Infect Dis Case Report BACKGROUND: Chronic graft versus host disease (cGVHD) occurs in 20–30% of paediatric patients receiving haemopoietic stem cell transplantation (HSCT). Neuromuscular disorders such as polymyositis are considered a rare and distinctive but non-diagnostic manifestation of cGVHD and, in the absence of other characteristic signs and symptoms, biopsy is highly recommended to exclude other causes. CASE REPORT: We report a case of a 17-months-old child affected by hemophagocytic lymphohistiocytosis who underwent a matched unrelated donor haematopoietic stem cell transplantation (HSCT). She developed severe cGVHD-related polymyositis that was successfully treated with high-dose steroid therapy, rituximab and sirolimus. CONCLUSIONS: This is the first case of cGVHD-related-polymyositis described in a pediatric patient which was successfully treated with rituximab. Università Cattolica del Sacro Cuore 2020-01-01 /pmc/articles/PMC6951354/ /pubmed/31934312 http://dx.doi.org/10.4084/MJHID.2020.002 Text en This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by-nc/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Chinello, Matteo
Balter, Rita
De Bortoli, Massimiliano
Vitale, Virginia
Zaccaron, Ada
Bonetti, Elisa
Tonin, Paola
Vattemi, Gaetano
Guglielmi, Valeria
Cesaro, Simone
Chronic Graft-Versus-Host-Disease-Related Polymyositis: a 17-Months-Old Child with a Rare and Late Complication of Haematopoietic Stem Cell Transplantation
title Chronic Graft-Versus-Host-Disease-Related Polymyositis: a 17-Months-Old Child with a Rare and Late Complication of Haematopoietic Stem Cell Transplantation
title_full Chronic Graft-Versus-Host-Disease-Related Polymyositis: a 17-Months-Old Child with a Rare and Late Complication of Haematopoietic Stem Cell Transplantation
title_fullStr Chronic Graft-Versus-Host-Disease-Related Polymyositis: a 17-Months-Old Child with a Rare and Late Complication of Haematopoietic Stem Cell Transplantation
title_full_unstemmed Chronic Graft-Versus-Host-Disease-Related Polymyositis: a 17-Months-Old Child with a Rare and Late Complication of Haematopoietic Stem Cell Transplantation
title_short Chronic Graft-Versus-Host-Disease-Related Polymyositis: a 17-Months-Old Child with a Rare and Late Complication of Haematopoietic Stem Cell Transplantation
title_sort chronic graft-versus-host-disease-related polymyositis: a 17-months-old child with a rare and late complication of haematopoietic stem cell transplantation
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6951354/
https://www.ncbi.nlm.nih.gov/pubmed/31934312
http://dx.doi.org/10.4084/MJHID.2020.002
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