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Correspondence

Adult T-Cell Leukemia with HTLV-I–Associated Myelopathy after Complete Remission of Acute Myelogenous Leukemia

N Engl J Med 1998; 338:333January 29, 1998

Article

To the Editor:

Human T-cell lymphotropic virus type I (HTLV-I) is the causative agent of adult T-cell leukemia–lymphoma (ATL) and HTLV-I–associated myelopathy–tropical spastic paraparesis (HAM–TSP), a neurologic disorder. The virus is transmitted through sexual contact, breast-feeding, and blood transfusion. We describe a patient with both ATL and HAM–TSP, which developed during a complete remission of acute myelogenous leukemia.

A 70-year-old heterosexual man, who had been born and lived in an area of Japan where HTLV-I was not endemic, was admitted to our hospital in June 1985. Acute myelogenous leukemia was diagnosed, and he received numerous blood transfusions for two years during chemotherapy. Starting in October 1987, his peripheral lymphocyte count gradually increased to 17,800 per cubic millimeter, but myeloblasts were absent. The lymphocytes had morphologic abnormalities, including flower forms and notched nuclei, and were positive for CD2, CD3, CD4, CD5, CD25, and HLA-DR on flow-cytometric analysis. His serum contained anti–HTLV-I antibodies (detected by an indirect immunofluorescence assay, a gelatin-particle–agglutination test, and an enzyme immunoassay), and the proviral genome was found in circulating lymphocytes (by Southern blot analysis).

A gait disturbance with hyperreflexia, muscle weakness, and atrophy of the legs developed in June 1988. No abnormalities in the central nervous system were detected by magnetic resonance imaging. The cerebrospinal fluid contained lymphocytes, oligoclonal immunoglobulins (detected by agarose-gel electrophoresis), and anti–HTLV-I antibodies (detected by Western blot analysis). These findings met the diagnostic criteria for HAM–TSP. 1 Preserved serum samples obtained from the patient (at the time of his first admission, in June 1985, and at discharge in November 1985) and his wife and three daughters were negative for anti–HTLV-I antibodies. These serologic findings suggested that HTLV-I had been transmitted through blood transfusion, but we could not obtain serologic data on HTLV-I in the blood donors.

The risk of HTLV-I infection is increased in patients with leukemia who require blood transfusions.2 In healthy carriers of HTLV-I, the latent period before the development of ATL is more than 20 years.3 In our patient, the latent period preceding the development of HAM–TSP was 28 months. In patients with hematologic cancers who have received blood transfusions, the latent period may be shortened by immunosuppression due to chemotherapy or radiotherapy.4,5 Nationwide screening for anti–HTLV-I antibodies in donated blood will make such cases very rare in the future.

Masatoshi Kanno, M.D.
Shinobu Nakamura, M.D.
Tamotsu Matsuda, M.D.
Kanazawa University School of Medicine, Kanazawa 920, Japan

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    Fujio Umehara, Takaaki Hagiwara, Michiyoshi Yoshimura, Keiko Higashi, Kimiyoshi Arimura. (2008) Enlarged, multifocal upper limb neuropathy with HTLV-I associated myelopathy in a patient with chronic adult T-cell leukemia. Journal of the Neurological Sciences 266:1-2, 167-170
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    Lawrence T. Goodnough. (2005) Risks of Blood Transfusion. Anesthesiology Clinics of North America 23:2, 241-252
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    Eduardo Gotuzzo, César Arango, Abelardo de Queiroz-Campos, Raúl E. Istúriz. (2000) HUMAN T-CELL LYMPHOTROPIC VIRUS-I IN LATIN AMERICA. Infectious Disease Clinics of North America 14:1, 211-239
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    Goodnough, Lawrence T., Brecher, Mark E., Kanter, Michael H., AuBuchon, James P., . (1999) Transfusion Medicine — Blood Transfusion. New England Journal of Medicine 340:6, 438-447
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