Hemophagocytic bone marrow aplasia with plasma cells in a RAG2-deficient SCID case after a nonconditioned transplantation from a fully matched sibling
We report a RAG2-deficient patient with severe combined immunodeficiency and hemophagocytic bone marrow aplasia with plasma cells after a nonconditioned transplantation from a fully matched sibling. After engraftment, disseminated BCGosis appeared because of graft versus host disease prophylaxis. On...
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| Veröffentlicht in: | Journal of pediatric hematology/oncology 2013-11, Vol.35 (8), p.e335-e337 |
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| container_issue | 8 |
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| container_title | Journal of pediatric hematology/oncology |
| container_volume | 35 |
| creator | Yildiran, Alisan Fişgin, Tunc Güner, Sukru N Kiliç, Mehtap Sancak, Recep Özyürek, Emel Albayrak, Davut Duru, Feride |
| description | We report a RAG2-deficient patient with severe combined immunodeficiency and hemophagocytic bone marrow aplasia with plasma cells after a nonconditioned transplantation from a fully matched sibling. After engraftment, disseminated BCGosis appeared because of graft versus host disease prophylaxis. On the 55th day, eosinophilia, neutropenia, and thrombocytopenia developed. Aplasia, hemophagocytic histiocytes, and plasma cells were found on his bone marrow with very high level of serum immunoglobulin E. We could not discriminate exactly whether BCGosis or alloimmune response is the cause of hemophagocytic aplasia with plasma cells. Despite the second hematopoietic stem cell transplantation with a reduced intensity conditioning regime, his marrow aplasia did not recover and he died. This case suggests that BCGosis might be associated with hemophagocytic marrow aplasia with plasma cells in an alloimmune reaction. |
| doi_str_mv | 10.1097/MPH.0b013e31827e8448 |
| format | Article |
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After engraftment, disseminated BCGosis appeared because of graft versus host disease prophylaxis. On the 55th day, eosinophilia, neutropenia, and thrombocytopenia developed. Aplasia, hemophagocytic histiocytes, and plasma cells were found on his bone marrow with very high level of serum immunoglobulin E. We could not discriminate exactly whether BCGosis or alloimmune response is the cause of hemophagocytic aplasia with plasma cells. Despite the second hematopoietic stem cell transplantation with a reduced intensity conditioning regime, his marrow aplasia did not recover and he died. This case suggests that BCGosis might be associated with hemophagocytic marrow aplasia with plasma cells in an alloimmune reaction.</description><identifier>ISSN: 1077-4114</identifier><identifier>EISSN: 1536-3678</identifier><identifier>DOI: 10.1097/MPH.0b013e31827e8448</identifier><identifier>PMID: 23389499</identifier><language>eng</language><publisher>United States</publisher><subject>BCG Vaccine - adverse effects ; BCG Vaccine - immunology ; Bone Marrow Diseases - etiology ; Bone Marrow Diseases - pathology ; DNA-Binding Proteins - genetics ; Hematopoietic Stem Cell Transplantation - adverse effects ; Humans ; Infant ; Lymphohistiocytosis, Hemophagocytic - etiology ; Lymphohistiocytosis, Hemophagocytic - pathology ; Male ; Nuclear Proteins - genetics ; Plasma Cells - pathology ; Severe Combined Immunodeficiency - genetics ; Severe Combined Immunodeficiency - surgery ; Transplantation Conditioning ; Transplantation, Homologous - adverse effects</subject><ispartof>Journal of pediatric hematology/oncology, 2013-11, Vol.35 (8), p.e335-e337</ispartof><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c307t-893dea7ed206d2af543b7451d853ea52b8c11f3a26de1fb0114c7653e796d50a3</citedby><cites>FETCH-LOGICAL-c307t-893dea7ed206d2af543b7451d853ea52b8c11f3a26de1fb0114c7653e796d50a3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/23389499$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Yildiran, Alisan</creatorcontrib><creatorcontrib>Fişgin, Tunc</creatorcontrib><creatorcontrib>Güner, Sukru N</creatorcontrib><creatorcontrib>Kiliç, Mehtap</creatorcontrib><creatorcontrib>Sancak, Recep</creatorcontrib><creatorcontrib>Özyürek, Emel</creatorcontrib><creatorcontrib>Albayrak, Davut</creatorcontrib><creatorcontrib>Duru, Feride</creatorcontrib><title>Hemophagocytic bone marrow aplasia with plasma cells in a RAG2-deficient SCID case after a nonconditioned transplantation from a fully matched sibling</title><title>Journal of pediatric hematology/oncology</title><addtitle>J Pediatr Hematol Oncol</addtitle><description>We report a RAG2-deficient patient with severe combined immunodeficiency and hemophagocytic bone marrow aplasia with plasma cells after a nonconditioned transplantation from a fully matched sibling. After engraftment, disseminated BCGosis appeared because of graft versus host disease prophylaxis. On the 55th day, eosinophilia, neutropenia, and thrombocytopenia developed. Aplasia, hemophagocytic histiocytes, and plasma cells were found on his bone marrow with very high level of serum immunoglobulin E. We could not discriminate exactly whether BCGosis or alloimmune response is the cause of hemophagocytic aplasia with plasma cells. Despite the second hematopoietic stem cell transplantation with a reduced intensity conditioning regime, his marrow aplasia did not recover and he died. This case suggests that BCGosis might be associated with hemophagocytic marrow aplasia with plasma cells in an alloimmune reaction.</description><subject>BCG Vaccine - adverse effects</subject><subject>BCG Vaccine - immunology</subject><subject>Bone Marrow Diseases - etiology</subject><subject>Bone Marrow Diseases - pathology</subject><subject>DNA-Binding Proteins - genetics</subject><subject>Hematopoietic Stem Cell Transplantation - adverse effects</subject><subject>Humans</subject><subject>Infant</subject><subject>Lymphohistiocytosis, Hemophagocytic - etiology</subject><subject>Lymphohistiocytosis, Hemophagocytic - pathology</subject><subject>Male</subject><subject>Nuclear Proteins - genetics</subject><subject>Plasma Cells - pathology</subject><subject>Severe Combined Immunodeficiency - genetics</subject><subject>Severe Combined Immunodeficiency - surgery</subject><subject>Transplantation Conditioning</subject><subject>Transplantation, Homologous - adverse effects</subject><issn>1077-4114</issn><issn>1536-3678</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2013</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpdkc9O3DAQxq0KVCj0DRDykUuoHTtxckTLn0WiKmrpOZrYY9YosRfbK7QvwvPiFdBDTzMa_-Yb-fsIOeHsnLNe_fh5vzxnI-MCBe9qhZ2U3RdyyBvRVqJV3V7pmVKV5FwekG8pPTHGlZD1V3JQC9H1su8PyesS57BewWPQ2-w0HYNHOkOM4YXCeoLkgL64vKK7fgaqcZoSdZ4C_X1xU1cGrdMOfaZ_FreXVENCCjZjLIAPXgdvXHZF1NAcwaci4zPsJtTGMBfKbqZpW05mvSpQcuPk_OMx2bcwJfz-UY_I3-urh8Wyuvt1c7u4uKu0YCpXXS8MgkJTs9bUYBspRiUbbrpGIDT12GnOrYC6NchtMYtLrdrypvrWNAzEETl7113H8LzBlIfZpd0fwWPYpIHL4mrTs5YXVL6jOoaUItphHV1xajtwNuwSGUoiw_-JlLXTjwubcUbzb-kzAvEGrPGJ8g</recordid><startdate>20131101</startdate><enddate>20131101</enddate><creator>Yildiran, Alisan</creator><creator>Fişgin, Tunc</creator><creator>Güner, Sukru N</creator><creator>Kiliç, Mehtap</creator><creator>Sancak, Recep</creator><creator>Özyürek, Emel</creator><creator>Albayrak, Davut</creator><creator>Duru, Feride</creator><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope></search><sort><creationdate>20131101</creationdate><title>Hemophagocytic bone marrow aplasia with plasma cells in a RAG2-deficient SCID case after a nonconditioned transplantation from a fully matched sibling</title><author>Yildiran, Alisan ; 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After engraftment, disseminated BCGosis appeared because of graft versus host disease prophylaxis. On the 55th day, eosinophilia, neutropenia, and thrombocytopenia developed. Aplasia, hemophagocytic histiocytes, and plasma cells were found on his bone marrow with very high level of serum immunoglobulin E. We could not discriminate exactly whether BCGosis or alloimmune response is the cause of hemophagocytic aplasia with plasma cells. Despite the second hematopoietic stem cell transplantation with a reduced intensity conditioning regime, his marrow aplasia did not recover and he died. This case suggests that BCGosis might be associated with hemophagocytic marrow aplasia with plasma cells in an alloimmune reaction.</abstract><cop>United States</cop><pmid>23389499</pmid><doi>10.1097/MPH.0b013e31827e8448</doi></addata></record> |
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| ispartof | Journal of pediatric hematology/oncology, 2013-11, Vol.35 (8), p.e335-e337 |
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| language | eng |
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| source | MEDLINE; Journals@Ovid Complete |
| subjects | BCG Vaccine - adverse effects BCG Vaccine - immunology Bone Marrow Diseases - etiology Bone Marrow Diseases - pathology DNA-Binding Proteins - genetics Hematopoietic Stem Cell Transplantation - adverse effects Humans Infant Lymphohistiocytosis, Hemophagocytic - etiology Lymphohistiocytosis, Hemophagocytic - pathology Male Nuclear Proteins - genetics Plasma Cells - pathology Severe Combined Immunodeficiency - genetics Severe Combined Immunodeficiency - surgery Transplantation Conditioning Transplantation, Homologous - adverse effects |
| title | Hemophagocytic bone marrow aplasia with plasma cells in a RAG2-deficient SCID case after a nonconditioned transplantation from a fully matched sibling |
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