Transient appearance of GPI-deficient population in a patient with azathioprine-associated bone marrow aplasia

Riyami, Arwa Z. Al; Jacobson, Kevan; Ford, Jason C.; Dalal, Bakul I.

doi:10.1007/s00277-012-1449-7

Ann Hematol

2012

DOI: 10.1007/s00277-012-1449-7

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Transient appearance of GPI-deficient population in a patient with azathioprine-associated bone marrow aplasia

Arwa Z. Al Riyami

Kevan Jacobson

Jason C. Ford

et al.

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Cited by 2 publications

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Azathioprine

2012

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S AzathioprineBone marrow aplasia in a child: case report An 8-year-old boy, who had Crohn's disease, developed bone marrow aplasia during treatment with azathioprine [route not stated].The boy presented with fever and neutropenia after receiving azathioprine at increasing doses from 12.5 to 50 mg/day for 3 months. Laboratory tests included the following results: WBC count 0.8 × 10 9 /L, neutrophils 0.43 × 10 9 /L, haemoglobin 88 g/L, platelet count 131 × 10 9 /L, reticulocyte count 3 × 10 9 /L, and a positive sugar water test. There was a partial absence of CD55, CD59, and fluorescent aerolysin (FLAER) on granulocytes, and of CD59 on RBCs, based on flow cytometric analysis for paroxysmal nocturnal haemoglobinuria (PNH). FLAER analysis of neutrophils revealed a 67% glycosylphosphatidylinositol (GPI)-deficient clone size. He had a 6-thioguanine nucleotide metabolite (TGN) level (on azathioprine 50 mg/day) of 327 pmol/8 × 10 8 , and an RBC level of 6-methyl mercaptopurine ribonucleotide metabolite of 807 pmol/8 × 10 8 RBC. Aplasia with a cellularity of 5% was evident on bone marrow aspirate and biopsy.The boy was rehydrated, and treated with anti-infectives and steroids. Azathioprine was withdrawn, and his symptoms resolved within 3 weeks, with recovery of his blood counts. The GPI-deficient population had completely disappeared on a repeat flow cytometric testing for PNH at 6 months.

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Azathioprine

2012

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Excellent response of severe aplastic anemia to treatment of gut inflammation: A case report and review of the literature

Zhao¹,

Zhao²,

Sun³

et al. 2020

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Transient appearance of GPI-deficient population in a patient with azathioprine-associated bone marrow aplasia

Cited by 2 publications

References 8 publications

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Excellent response of severe aplastic anemia to treatment of gut inflammation: A case report and review of the literature

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