Hatayama Yasuki, Sugiyama Harutoshi, Murakami Daisuke, Oura Hirotaka, Shima Yukiko, Shirato Miho, Nishino Takayoshi, Nakazawa Tadao, Suehiro Kenichi, Arai Makoto
Department of Gastroenterology, Tokyo Women's Medical University Yachiyo Medical Center, Chiba 276-8523, Japan.
Department of Pathology, Tokyo Women's Medical University Yachiyo Medical Center, Chiba 276-8523, Japan.
J Med Cases. 2023 Aug;14(8):282-288. doi: 10.14740/jmc4135. Epub 2023 Aug 28.
Prior reports described cases of lymphoproliferative diseases occurring after methotrexate (MTX) administration, which are called methotrexate-associated lymphoproliferative disorders (MTX-LPDs). It has become clear that these lymphoproliferative diseases also occur following treatment with other immunosuppressive drugs, and they have been termed as other iatrogenic immunodeficiency-associated lymphoproliferative disorders (OIIA-LPDs). In most of these cases, the duration of immunosuppressive drugs is very long, on the order of years. In the present study, we evaluated the development of lymphoproliferative disease despite the short duration of immunosuppressive treatment and determined the tumor doubling time. A 71-year-old woman was diagnosed with adult-onset Still's disease. The patient was administered prednisone 30 mg per day starting on February 25, 2022 and MTX 6 mg per week starting 2 weeks later. Because she was a hepatitis B virus (HBV) carrier, nucleic acid analog therapy was also started to prevent HBV activation. Eight weeks later, biweekly tocilizumab was started. After 5 months of MTX administration, a solitary liver tumor measuring 37 × 32 mm was detected. Three months later, repeat computed tomography revealed that the liver tumor had grown rapidly to 7 cm in diameter. We considered the possibility of OIIA-LPDs and stopped MTX therapy. Biopsy specimens of the liver tumor exhibited lymphocyte proliferation, which was consistent with OIIA-LPDs. The doubling time for tumor growth was 33 days. Despite withdrawing MTX for 6 weeks, the tumor continued to grow, and thus, the patient was referred to the hematology unit. In previously reported cases of MTX-LPDs of hepatic origin, the average duration of MTX administration was 7.3 (2 - 13) years. This report describes a primary hepatic OIIA-LPDs-associated tumor that rapidly increased in size after an extremely short period of MTX administration.
先前的报告描述了甲氨蝶呤（MTX）给药后发生的淋巴增殖性疾病病例，这些疾病被称为甲氨蝶呤相关淋巴增殖性疾病（MTX-LPDs）。现已明确，这些淋巴增殖性疾病也会在使用其他免疫抑制药物治疗后发生，它们被称为其他医源性免疫缺陷相关淋巴增殖性疾病（OIIA-LPDs）。在大多数此类病例中，免疫抑制药物的使用时间非常长，长达数年。在本研究中，我们评估了尽管免疫抑制治疗时间较短但淋巴增殖性疾病的发生情况，并确定了肿瘤倍增时间。一名71岁女性被诊断为成人斯蒂尔病。患者自2022年2月25日起每天服用30毫克泼尼松，2周后开始每周服用6毫克MTX。由于她是乙肝病毒（HBV）携带者，还开始了核酸类似物治疗以预防HBV激活。8周后，开始每两周使用一次托珠单抗。MTX给药5个月后，检测到一个直径为37×32毫米的孤立性肝肿瘤。3个月后，重复计算机断层扫描显示肝肿瘤迅速增大至直径7厘米。我们考虑了OIIA-LPDs的可能性并停止了MTX治疗。肝肿瘤的活检标本显示淋巴细胞增殖，这与OIIA-LPDs一致。肿瘤生长的倍增时间为33天。尽管停用MTX 6周，但肿瘤仍继续生长，因此，该患者被转诊至血液科。在先前报道的肝源性MTX-LPDs病例中，MTX给药的平均时间为7.3（2 - 13）年。本报告描述了一例原发性肝OIIA-LPDs相关肿瘤，在极短时间的MTX给药后迅速增大。
