{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,29]],"date-time":"2025-10-29T03:52:36Z","timestamp":1761709956317,"version":"3.41.2"},"reference-count":10,"publisher":"Oxford University Press (OUP)","issue":"5","license":[{"start":{"date-parts":[[2022,5,24]],"date-time":"2022-05-24T00:00:00Z","timestamp":1653350400000},"content-version":"vor","delay-in-days":23,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,5,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>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.<\/jats:p>","DOI":"10.1093\/omcr\/omac055","type":"journal-article","created":{"date-parts":[[2022,5,24]],"date-time":"2022-05-24T10:13:48Z","timestamp":1653387228000},"source":"Crossref","is-referenced-by-count":2,"title":["A rare case of combined immunodeficiency with cytopenia whose symptoms were controlled by cyclosporine"],"prefix":"10.1093","volume":"2022","author":[{"given":"Bazgir","family":"Narges","sequence":"first","affiliation":[{"name":"Department of Internal Medicine, Shahid Beheshti University of Medical Sciences, Tehran, Iran"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eskandarzadeh","family":"Shabnam","sequence":"additional","affiliation":[{"name":"Department of Allergy and Clinical Immunology, Shahid Beheshti University of Medical Sciences"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eslamian","family":"Golnaz","sequence":"additional","affiliation":[{"name":"Department of Allergy and Clinical Immunology, Shahid Beheshti University of Medical Sciences"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chavoshzadeh","family":"Zahra","sequence":"additional","affiliation":[{"name":"Department of Allergy and Clinical Immunology, Shahid Beheshti University of Medical Sciences"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shah","family":"Jaffer","sequence":"additional","affiliation":[{"name":"Drexel University College of Medicine, Pennsylvania 19104, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4427-5121","authenticated-orcid":false,"given":"Qaderi","family":"Shohra","sequence":"additional","affiliation":[{"name":"Medical Research Center, Kateb University"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2022,5,23]]},"reference":[{"key":"2022052410075166400_ref1","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1186\/s13223-018-0290-5","article-title":"Primary immunodeficiency","volume":"14","author":"McCusker","year":"2018","journal-title":"Allergy Asthma Clin Immunol"},{"key":"2022052410075166400_ref2","doi-asserted-by":"crossref","first-page":"94","DOI":"10.1111\/j.1365-2141.2007.06616.x","article-title":"Rituximab for the treatment of autoimmune cytopenias in children with immune deficiency","volume":"138","author":"Kim","year":"2007","journal-title":"Br J Haematol"},{"key":"2022052410075166400_ref3","doi-asserted-by":"crossref","first-page":"851","DOI":"10.1097\/MOP.0000000000000833","article-title":"Autoimmunity as a continuum in primary immunodeficiency","volume":"31","author":"Walter","year":"2019","journal-title":"Curr Opin Pediatr"},{"key":"2022052410075166400_ref4","doi-asserted-by":"crossref","first-page":"2337","DOI":"10.1182\/blood-2014-06-583260","article-title":"Autoimmune and other cytopenias in primary immunodeficiencies: pathomechanisms, novel differential diagnoses, and treatment","volume":"124","author":"Seidel","year":"2014","journal-title":"Blood"},{"key":"2022052410075166400_ref5","doi-asserted-by":"crossref","first-page":"254","DOI":"10.1128\/CVI.00001-16","article-title":"Flow cytometry, a versatile tool for diagnosis and monitoring of primary immunodeficiencies","volume":"23","author":"Abraham","year":"2016","journal-title":"Clin Vaccine Immunol"},{"key":"2022052410075166400_ref6","doi-asserted-by":"crossref","first-page":"761","DOI":"10.1016\/j.jaip.2018.12.017","article-title":"Mechanism-based precision therapy for the treatment of primary immunodeficiency and primary immunodysregulatory diseases","volume":"7","author":"Leiding","year":"2019","journal-title":"J Allergy Clin Immunol Pract"},{"key":"2022052410075166400_ref7","doi-asserted-by":"crossref","first-page":"3","DOI":"10.3389\/fped.2016.00003","article-title":"Current challenges in immune and other acquired cytopenias of childhood","volume":"4","author":"Ghosh","year":"2016","journal-title":"Front Pediatr"},{"key":"2022052410075166400_ref8","doi-asserted-by":"crossref","first-page":"2608","DOI":"10.3389\/fimmu.2021.652487","article-title":"Common variable immunodeficiency and autoimmune diseases: a retrospective study of 95 adult patients in a single tertiary care center","volume":"12","author":"Mormile","year":"2021","journal-title":"Front Immunol"},{"key":"2022052410075166400_ref9","doi-asserted-by":"crossref","first-page":"9","DOI":"10.3109\/03009749209101478","article-title":"Cyclosporin A. Mode of action and effects on bone and joint tissues","volume":"21","author":"Russell","year":"1992","journal-title":"Scand J Rheumatol"},{"key":"2022052410075166400_ref10","doi-asserted-by":"crossref","first-page":"355","DOI":"10.2169\/internalmedicine.1372-18","article-title":"Cyclosporine therapy in patients with transfusion-independent non-severe aplastic anemia: a retrospective analysis","volume":"58","author":"Matsuda","year":"2019","journal-title":"Intern Med"}],"container-title":["Oxford Medical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/omcr\/article-pdf\/2022\/5\/omac055\/43794505\/omac055.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/omcr\/article-pdf\/2022\/5\/omac055\/43794505\/omac055.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,5,24]],"date-time":"2022-05-24T10:14:02Z","timestamp":1653387242000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/omcr\/article\/doi\/10.1093\/omcr\/omac055\/6590588"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,5,1]]},"references-count":10,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2022,5,1]]}},"URL":"https:\/\/doi.org\/10.1093\/omcr\/omac055","relation":{},"ISSN":["2053-8855"],"issn-type":[{"type":"electronic","value":"2053-8855"}],"subject":[],"published-other":{"date-parts":[[2022,5]]},"published":{"date-parts":[[2022,5,1]]},"article-number":"omac055"}}