可逆利妥昔单抗诱导的直肠卡波西肉瘤在hiv阴性滤泡性淋巴瘤患者中误诊为溃疡性结肠炎。

Clinical Sarcoma Research Pub Date : 2018-06-11 eCollection Date: 2018-01-01 DOI:10.1186/s13569-018-0097-7
Emilien Billon, Anne-Marie Stoppa, Lena Mescam, Massimo Bocci, Audrey Monneur, Delphine Perrot, François Bertucci
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引用次数: 6

摘要

背景:卡波西肉瘤是一种低级别间充质血管增生性肿瘤,最常见于免疫功能低下的个体,如hiv感染患者。医源性卡波西肉瘤发生在接受免疫抑制治疗的患者中。利妥昔单抗是一种靶向泛B细胞标志物CD20的嵌合单克隆抗体。由于其通过减少成熟b细胞的免疫抑制作用,可能使hiv阳性患者的卡波西氏肉瘤恶化。利妥昔单抗相关的卡波西氏肉瘤以前仅在两名hiv阴性患者中报道过,并接受了手术治疗。病例介绍:在这里,我们报告了一名55岁的hiv阴性滤泡性淋巴瘤患者在美罗华单抗治疗下发生的卡波西肉瘤。在维持利妥昔单抗治疗期间,病变在直肠水平发展,表现为明显的溃疡性结肠炎,无任何皮肤病变。过早停用利妥昔单抗导致卡波西氏肉瘤完全消退,无需任何额外的特异性治疗。结论:据我们所知,这是hiv阴性患者在利妥昔单抗下诊断出的卡波西氏肉瘤的第三例,是直肠水平的第一例,也是停药后完全消退的第一例。本病例提高了接受利妥昔单抗治疗的hiv阴性患者对医源性卡波西氏肉瘤的认识,并进一步强调了免疫抑制在疾病病理生理学中的重要性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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Reversible rituximab-induced rectal Kaposi's sarcoma misdiagnosed as ulcerative colitis in a patient with HIV-negative follicular lymphoma.

Background: Kaposi's sarcoma is a low-grade mesenchymal angioproliferative tumor, most commonly observed in immunocompromised individuals, such as HIV-infected patients. Iatrogenic Kaposi's sarcoma occurs in patients undergoing immunosuppressive therapies. Rituximab is a chimeric monoclonal antibody targeted against the pan B cell marker CD20. Because of its immunosuppressive effects through reduction of mature B-cells, it may exacerbate Kaposi's sarcoma in HIV-positive patients. Rituximab-related Kaposi's sarcomas have been previously reported in only two HIV-negative patients and were treated surgically.

Case presentation: Here, we report on a Kaposi's sarcoma that developed under rituximab treatment in a HIV-negative 55-year-old patient treated for follicular lymphoma. The lesion developed during the maintenance rituximab therapy at the rectal level with an aspect of apparent ulcerative colitis, without any cutaneous lesion. The premature stop of rituximab led to the complete regression of Kaposi's sarcoma, without any additional specific treatment.

Conclusions: To our knowledge, this is the third case of Kaposi's sarcoma diagnosed under rituximab in a HIV-negative patient, the first one at the rectal level and the first one that completely regresses after stop of rituximab. This case raises awareness of iatrogenic Kaposi's sarcoma in HIV-negative patients treated with rituximab, and further highlights the importance of immunosuppression in the pathophysiology of disease.

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期刊介绍: Clinical Sarcoma Research considers for publication articles related to research on sarcomas, including both soft tissue and bone. The journal publishes original articles and review articles on the diagnosis and treatment of sarcomas along with new insights in sarcoma research, which may be of immediate or future interest for diagnosis and treatment. The journal also considers negative results, especially those from studies on new agents, as it is vital for the medical community to learn whether new agents have been proven effective or ineffective within subtypes of sarcomas. The journal also aims to offer a forum for active discussion on topics of major interest for the sarcoma community, which may be related to both research results and methodological topics.
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