Adult T Cell Leukemia/Lymphoma Diagnosed from Mucocutaneous Candidiasis.
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生来健康であった59歳の女性が適切な治療にもかかわらず11ヵ月にわたり両手指爪, 外陰部, 鼠径部, 口腔内に難治性カンジダ症を発症した。経過中に躯幹, 四肢に2週間で自然軽快する浸潤性紅斑を認め, 病理組織学的にPautrier’s microabscessが認められた。また血清抗human T-cell lymphotropic virus type I(HTLV-I)抗体陽性, DNA診断(southern blot analysis)で血液中, 組織中にHTLV-Iのmonoclonalな取り込みが認められ, ATL(adult T cell leukemia/lymphoma)と診断した。ATLは発症に細胞性免疫の低下が強く関与しているといわれている。今回我々は難治性汎発性カンジダ症および出没する皮疹からATLを疑い臨床的, 免疫学的に細胞性免疫の低下が認められ病理組織学的, 血液学的にATLと確認された症例を経験したので報告した。Keywords:
Mucocutaneous zone
Adult T-cell leukemia/lymphoma
A T-cell line (KH-2) from a Japanese T-cell leukemia patient with hypercalcemia has been characterized and shown to be similar in its properties to Human T-cell leukemia virus (HTLV)-positive T-cell lines from T-cell leukemia patients from different parts of the world. The HTLV isolated from the Japanese T-cell leukemia patient with hypercalcemia belongs to the HTLV family and has properties similar to HTLV from T-cell leukemia patients from the United States, Israel, and the Caribbean.
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Adult T-cell leukemia/lymphoma
Etiology
Peripheral T-cell lymphoma
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Sera and peripheral blood lymphocytes of 40 adult T-cell leukemia (ATL) patients in non-ATL-endemic Hokkaido were examined for the prevalence of human T-cell leukemia virus type 1 (HTLV-I). All patients had HTLV-I-specific antibodies. When the peripheral lymphocytes were assessed after short-term cultivation, HTLV-I antigens and virus particles were detected. The seroprevalence in 96 cases of non-T-cell leukemias and lymphomas and in 30,056 healthy individuals in Hokkaido were 3.1% and 0.7%, respectively. HTLV-I seropositive inhabitants of Hokkaido can be estimated at about 40,000, and one out of every few thousand HTLV-I carriers is likely to develop ATL.
Seroprevalence
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Pathognomonic
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80 歳台男性。全身に広がる血疱を伴う紅斑と紫斑で当科を受診した。血液検査でヒト T 細胞白血病ウイルス 1 型（Human T-cell leukemia virus type 1，HTLV-1）抗体陽性で，成人 T 細胞性白血病・リンパ腫（Adult T-cell Leukemia/Lymphoma，ATL）様細胞 6360/μl，可溶性 IL-2 レセプター（sIL-2R）22,545 U/ml と高値であり，CT 検査で多発リンパ節腫大と脾腫があった。皮膚生検で真皮血管周囲に異型リンパ球浸潤があり，免疫組織化学的に浸潤細胞は CD3，CD4，CD7，CD25，CCR4 陽性であった。左鼠径リンパ節生検でも異型細胞浸潤があった。急速に呼吸状態が悪化し，FiO2 0.6 でSpO2 94％と呼吸不全となった。CT 検査で小葉中心性陰影やスリガラス影があり，胸水細胞診で ATL 細胞を多数確認し，ATL 急性型と診断した。年齢や既往，全身状態から，多剤併用化学療法ではなく，メチルプレドニゾロン 40 mg/day とエトポシド 25 mg/day の内服を開始したが効果は不十分であった。モガムリズマブの投与追加により，呼吸状態は改善し，皮疹と末梢血中 ATL 様細胞は消失し，sIL-2R も 346 U/ml と低下した。ATL は近年，骨髄移植に適さない高齢発症例が増えており，治療戦略に苦慮するが，経口化学療法とモガムリズマブによる治療を組み合わせることで良好な治療結果を得られた 1 例を経験したため報告する。
Adult T-cell leukemia/lymphoma
Blisters
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Adult T-cell leukemia/lymphoma
Neoplasm
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Spontaneous T-cell leukemia similar to human adult T-cell leukemia (ATL) was found in an African green monkey naturally infected with simian retrovirus closely related to human T-cell leukemia virus type I (HTLV-I). Monoclonal integration of the simian retrovirus was detected in the primary leukemic cells, suggesting an association of the retrovirus with ATL-like leukemia in the monkey.
Simian
African Green Monkey
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Adult T-cell leukemia/lymphoma
Peripheral T-cell lymphoma
Etiology
T-Cell Lymphoma
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We describe five patients with adult T-cell leukemia/lymphoma (ATL) with neither integration of human T-cell leukemia virus type I (HTLV-I) into their leukemia cells nor anti-HTLV-I antibody in their sera. These findings indicate that HTLV-I may not have been involved in leukemogenesis in these patients. The clinicohematological, cytopathological, and immunological features of HTLV-I-negative ATL were exactly the same as those of HTLV-I-associated ATL. Leukemia cells with pleomorphic nuclei, generalized lymphadenopathy, hepatosplenomegaly, skin lesions, hypercalcemia, and elevated lactate dehydrogenase levels, all of which are characteristic features of typical ATL, were also seen in these patients with HTLV-I-negative ATL. Leukemia cells expressed T3, T4, and pan-T-cell antigens in three cases, and T3 and pan-T-cell antigens in two. All five patients had lived in ATL-nonendemic areas. The finding of HTLV-I-negative ATL suggests that factor(s) other than HTLV-I infection may be involved in ATL leukemogenesis.
Hepatosplenomegaly
Adult T-cell leukemia/lymphoma
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Human T-cell leukemia virus type I (HTLV-I) is etiologically associated with adult T-cell leukemia/lymphoma (ATL). We cloned and sequenced host DNA adjacent to the long terminal repeats of HTLV-I from uncultured leukemic cells of 4 ATL patients. The region flanking the provirus was generally A/T-rich (60–64% A/T), and a nucleotide composition bias was noticed when sequences within 25 bp on both sides of the integration target site were analyzed. In the 6-bp direct repeat, both end positions are preferentially occupied by G/C, whereas the middle positions are preferentially occupied by A/T. Furthermore, AA or TT dinucleotides are frequently present on each side adjacent to the center of the direct repeat. Our finding suggests preferential integration target sites of HTLV-I in the host genome. Further study is warranted to determine whether each of the target sequence preference is a general property of HTLV-I integration or may be associated with the leukemogenesis of ATL. © 1996 Wiley-Liss, Inc.
Provirus
Adult T-cell leukemia/lymphoma
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