Chronic Renal Failure, Aplastic Anemia, Myelopathy, Uveitis,. Sjogren's Syndrome and Panniculitis. Motomi Osato, Kazunari Yamaguchi*, Sadahiro Tamiya, ...
CASE REPORT
A HumanT-Cell Lymphotropic Virus Type-I Carrier with
Chronic Renal Failure, Aplastic Anemia, Myelopathy, Uveitis, Sjogren's
Syndrome and Panniculitis
Motomi Osato, Kazunari Yamaguchi*, Sadahiro Tamiya, Hiroshi Yamasaki, Toshiya Okubo, Hitoshi Suzushima, Norio Asou, Kenmei Sakata*, Makoto Kawakita and Kiyoshi Takatsuki A 53-year-old female infected with human T lymphotropic virus type-I (HTLV-I) suffered from chronic renal failure, aplastic anemia, myelopathy, uveitis, Sjogren's syndromeand WeberChristian disease. Although HTLV-Iantibody was negative in cerebrospinal fluid, she was
diagnosed as HTLV-Iassociated myelopathy/tropical
spastic paraparesis (HAM/TSP)based on
clinical and histological findings. Though to date there is no direct evidence, other complications have also been reported to be HTLV-Irelated diseases. This case provided the unique opportunity to observe various HTLV-Irelated diseases. (Internal Medicine 35: 742-745, 1996)
Key words: human T lymphotropic virus type-I associated myelopathy/tropical spastic paraparesis (HAM/TSP), Weber-Christian disease, human T lymphotropic virus type-I (HTLV-I) related disease
Introduction HumanT-cell lymphotropic virus type-I (HTLV-I) is well known to cause adult T cell leukemia/lymphoma (ATL). Recent studies have revealed that HTLV-Iinfection is also related
to(HAM/TSP), HTLV-Iassociated myelopathy/tropical spastic paraparesis HTLV-I uveitis, Sjogren's syndrome, chronic
bronchiectasis and retinodetachment. No blood transfusions were given before admission. Her parents died of renal failure. She was diagnosed as having IgA nephropathy in 1979 and hemodialysis was started because of chronic renal failure since August 1990. On admission, physical examinations showed purpura and pale skin, with neither lymphadenopathy nor hepatosplenomegaly. Neurological examinations revealed
hyperreflexia associated with a positive Babinski's reflex in the lower limbs. A mild impairment of the sense was also noted in both lower limbs. Pancytopenia (hemoglobin 4.0 g/dl, white blood cell count 1.0 x 109//, platelet count 7.0 x 109//) and diseases. hypocellular bone marrow (no erythroblasts, 17% myeloid cells, 65% lymphocytes and 18% plasma cells) were evident. No abnormal cells including lymphocytes possessing tabulated For editorial comment, see p 677. nuclei were found in the peripheral blood or bone marrow. Renal dysfunction (blood urea nitrogen 41.7 mg/dl, creatinine 1.0 mg/dl) occurred during hemodialysis. An immunological Case Report study showed that her anti-nuclear antibody was positive, antiA 53-year-old Japanese womanwas admitted to our hospital DNAantibody negative, anti-SS-A and SS-B antibodies negain September 1990 because of bleeding tendency due to pancy- tive, anti-HTLV-I antibody positive in serum (particle agglutitopenia that occurred one month before. She was born in nation assay x512) and negative in cerebrospinal fluid (CSF). KumamotoPrefecture, Kyushu island, an endemic area of Humanleukocyte antigen (HLA) typing was A2 Al l Bw46
lung disease, arthropathy, infective dermatitis , diabetes mellitus,
thyroiditis, myositis, congenital hydrocephalus and strongyloidiasis (1). These conditions are referred to as HTLV-Irelated diseases. Wepresent here an HTLV-Icarrier with various
HTLV-Iinfection.
In the past, she suffered from acute nephritis,
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From the Second Department of Internal Medicine and *the Blood Transfusion Service, KumamotoUniversity School of Medicine, Kumamoto Received for publication January 18, 1996; Accepted for publication July 1, 1996 Reprint requests should be addressed to Dr. Motomi Osato, the Second Department of Internal Medicine, KumamotoUniversity School of Medicine, 1 - 1 - 1 Honjo, Kumamoto860 742
Internal
Medicine Vol. 35, No. 9 (September 1996)
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