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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...
Autores principales: | , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Oxford University Press
2022
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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. |
format | Online Article Text |
id | pubmed-9127943 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Oxford University Press |
record_format | MEDLINE/PubMed |
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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