Literature DB >> 7639279

Immune mediated agranulocytosis and anemia associated with thymoma.

K Postiglione1, R Ferris, J P Jaffe, D Stroncek.   

Abstract

Thymoma has been associated with a variety of autoimmune disorders. We report a case of agranulocytosis and anemia in a 68-year-old woman with a spindle cell thymoma. She was unresponsive to treatment with antibiotics, granulocyte-colony stimulating factor (G-CSF), prednisone, and high-dose intravenous immunoglobulin. Serial bone marrow examinations on this therapy showed progression from a cellular marrow with mild myeloid and erythroid hyperplasia and lymphocytosis, to granulocyte aplasia and severe erythroid hypoplasia. Her serum contained granulocyte-specific antibodies and inhibited the growth in culture of her own marrow cells and marrow cells from a normal donor. An IgG fraction from her serum also inhibited the growth of marrow cells. Although the patient's spindle cell thymoma was surgically removed, she remained neutropenic. She was treated with six plasma exchanges followed by 1,000 milligrams of intravenous cyclophosphamide 2 days after the final plasma exchange and daily G-CSF. Three weeks later her peripheral blood showed marked leukocytosis with pronounced neutrophilia and a left shift. Although her agranulocytosis resolved, she died of fungal sepsis. This case demonstrates that aggressive plasma exchange and immunosuppressive therapy may benefit patients with agranulocytosis associated with thymoma.

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Year:  1995        PMID: 7639279     DOI: 10.1002/ajh.2830490412

Source DB:  PubMed          Journal:  Am J Hematol        ISSN: 0361-8609            Impact factor:   10.047


  2 in total

Review 1.  Good's syndrome and pure white cell aplasia complicated by cryptococcus infection: A case report and review of the literature.

Authors:  K Akinosoglou; M Melachrinou; D Siagris; E Koletsis; M Marangos; C A Gogos; E E Solomou
Journal:  J Clin Immunol       Date:  2014-03-14       Impact factor: 8.317

Review 2.  [Polycythemia vera with Good's syndrome and agranulocytosis: report of a case and literatures review].

Authors:  W W Tian; D P Liu; S C Bian; L M Ma; T Wang; Y X Xie; J P Zhao; T Z Zhao
Journal:  Zhonghua Xue Ye Xue Za Zhi       Date:  2016-06-14
  2 in total

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