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A rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine

Combined Immunodeficiency (CID) is a group of inborn error of Immunity (IEI) that may present with both infectious and non-infectious complications. Autoimmunity is an unusual presentation of CID and can be presented as cytopenia. The initial management of cytopenia is corticosteroids and IVIG. The...

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Autores principales: Narges, Bazgir, Shabnam, Eskandarzadeh, Golnaz, Eslamian, Zahra, Chavoshzadeh, Jaffer, Shah, Shohra, Qaderi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9127943/
https://www.ncbi.nlm.nih.gov/pubmed/35619687
http://dx.doi.org/10.1093/omcr/omac055
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author Narges, Bazgir
Shabnam, Eskandarzadeh
Golnaz, Eslamian
Zahra, Chavoshzadeh
Jaffer, Shah
Shohra, Qaderi
author_facet Narges, Bazgir
Shabnam, Eskandarzadeh
Golnaz, Eslamian
Zahra, Chavoshzadeh
Jaffer, Shah
Shohra, Qaderi
author_sort Narges, Bazgir
collection PubMed
description Combined Immunodeficiency (CID) is a group of inborn error of Immunity (IEI) that may present with both infectious and non-infectious complications. Autoimmunity is an unusual presentation of CID and can be presented as cytopenia. The initial management of cytopenia is corticosteroids and IVIG. The role of other cytotoxic and immunosuppressive drugs in management of cytopenia is not fully understood. The objective of this clinical case report is to highlight the possibly beneficial role of cyclosporine in controlling cytopenia in CID patients. A 26-month-old child with generalized ecchymosis was referred to Mofid Children's Hospital in Tehran, Iran. Physical examination revealed no substantial findings other than ecchymosis, and complete blood count (CBC) revealed thrombocytopenia. Diagnosis of CID and cytopenia followed. The patient was treated by 5 times prednisolone and 4 times Rituximab. Finally, his ecchymosis was controlled by Cellcept, which was then tempered and substituted by cyclosporine.
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spelling pubmed-91279432022-05-25 A rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine Narges, Bazgir Shabnam, Eskandarzadeh Golnaz, Eslamian Zahra, Chavoshzadeh Jaffer, Shah Shohra, Qaderi Oxf Med Case Reports Case Report Combined Immunodeficiency (CID) is a group of inborn error of Immunity (IEI) that may present with both infectious and non-infectious complications. Autoimmunity is an unusual presentation of CID and can be presented as cytopenia. The initial management of cytopenia is corticosteroids and IVIG. The role of other cytotoxic and immunosuppressive drugs in management of cytopenia is not fully understood. The objective of this clinical case report is to highlight the possibly beneficial role of cyclosporine in controlling cytopenia in CID patients. A 26-month-old child with generalized ecchymosis was referred to Mofid Children's Hospital in Tehran, Iran. Physical examination revealed no substantial findings other than ecchymosis, and complete blood count (CBC) revealed thrombocytopenia. Diagnosis of CID and cytopenia followed. The patient was treated by 5 times prednisolone and 4 times Rituximab. Finally, his ecchymosis was controlled by Cellcept, which was then tempered and substituted by cyclosporine. Oxford University Press 2022-05-23 /pmc/articles/PMC9127943/ /pubmed/35619687 http://dx.doi.org/10.1093/omcr/omac055 Text en © The Author(s) 2022. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com https://creativecommons.org/licenses/by-nc/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (https://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Case Report
Narges, Bazgir
Shabnam, Eskandarzadeh
Golnaz, Eslamian
Zahra, Chavoshzadeh
Jaffer, Shah
Shohra, Qaderi
A rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine
title A rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine
title_full A rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine
title_fullStr A rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine
title_full_unstemmed A rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine
title_short A rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine
title_sort rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9127943/
https://www.ncbi.nlm.nih.gov/pubmed/35619687
http://dx.doi.org/10.1093/omcr/omac055
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