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• W2139901663 abstract "In July, 2008, a 55-year-old woman was referred to us with a 2-month history of progressive aphasia after a febrile episode. She had been diagnosed with chronic adult T-cell leukaemia in June, 1999, and had had a nonmyeloablative peripheral-blood stem-cell transplant with conditioning of pentostatin and 2 Gy total body irradiation in March, 2002. The stem cells had been harvested from her brother, an asymptomatic carrier of human T-cell lymphotropic virus type 1 (HTLV-1). After treatment, she had complete remission. Ciclosporin was discontinued 6 months after the transplant ation because of graft-versus-host disease. Our patient had had no bacterial or viral infections. She had no skin lesions, lymphadenopathy, or splenomegaly. Her leucocyte count was 8·74×109/L with 2·5% adult T-cell leukaemia-like cells. The leukaemia was therefore con sidered to be in remission. CD3-positive cells in the peripheral blood were confi rmed as donor derived by XY-fl uorescence insitu hybridisation (XY-FISH). Serum IgG was 13·7 g/L, IgA was 2·82 g/L, and IgM was 1·16 g/L. Our patient was immunocompetent; absolute number of circulating cells positive for CD4 was 1·74×109/L, CD8 0·475×109/L, CD19 0·522×109/L, and CD56 0·326×109/L. Brain MRI showed hyperintense, non-enhancing lesions in the left temporoparietal lobe and right frontal lobe (fi gure A), which suggested encephalitis, progressive multiple leukoencephalopathy, demyelinating disorder, or lymphoma. Serum concentrations of calcium, lactate dehydro genase, soluble interleukin-2 receptor, and β-2 microglobulin were not raised. CSF examination, culture, and PCR, abdominal ultrasonography, endoscopy, and 18F-fl urodeoxyglucose (18F-FDG) PET were normal. Craniotomy and biopsy of the left temporal lobe was done in July, 2008. Histopathology showed perivascular accumulation and cerebral infi ltration by mononuclear tumour cells with large, indented nuclei (fi gure B). Tumour cells were positive for CD3, CD4, CD8, and CD45RO, but negative for B-cell markers (CD20 and CD79a), suggesting cerebral invasion by adult T-cell leukaemia cells (webappendix). XY-FISH showed an XY genotype of the tumour cells (fi gure C), suggesting that they had originated from the donated leucocytes. After whole brain irradiation and localised irradiation of the left temporoparietal lobe, our patient’s aphasia improved remarkably, and repeat MRI showed shrinkage of the brain lesions. In October, 2010, our patient was fully recovered. For patients with adult T-cell leukaemia, asymptomatic carriers of HTLV-1 have been widely considered acceptable as donors in allogeneic stem-cell transplantation. The immune system is thought to have an important role in leukaemia occurrence in HTLV-1-seropositive carriers. The only reported case of donor-derived adult T-cell leukaemia was in 2006, in an immunosuppressed recipient of a peripheral-blood stem-cell transplant. No cases of donorderived adult T-cell leukaemia have been reported in immunocompetent patients. The prognosis tends to be better for these patients than for those with CNS involvement subsequent to systemic adult T-cell leukaemia. Only two cases of isolated CNS involvement have been reported so far. The function of CD4-positive cells was not fully analysed, so there is a possibility of some functional defi cit in immune reconstitution. Our patient’s case suggests that the consent practices in patients who receive HTLV-1-positive cells should be re-evaluated." @default.
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• W2139901663 date "2011-03-01" @default.
• W2139901663 modified "2023-10-18" @default.
• W2139901663 title "Donor-derived adult T-cell leukaemia" @default.
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• W2139901663 doi "https://doi.org/10.1016/s0140-6736(11)60315-2" @default.
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