Billon Emilien, Stoppa Anne-Marie, Mescam Lena, Bocci Massimo, Monneur Audrey, Perrot Delphine, Bertucci François
1INSERM UMR1068, CNRS UMR725, Department of Medical Oncology, Centre de Recherche en Cancérologie de Marseille (CRCM), Institut Paoli-Calmettes, 232 Bd de Sainte-Marguerite, 13009 Marseille, France.
2Department of Hematology, Institut Paoli-Calmettes, Marseille, France.
Clin Sarcoma Res. 2018 Jun 11;8:11. doi: 10.1186/s13569-018-0097-7. eCollection 2018.
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.
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.
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.
卡波西肉瘤是一种低度间质性血管增殖性肿瘤，最常见于免疫功能低下的个体，如感染艾滋病毒的患者。医源性卡波西肉瘤发生在接受免疫抑制治疗的患者中。利妥昔单抗是一种针对全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细胞产生免疫抑制作用，可能会使艾滋病毒阳性患者的卡波西肉瘤恶化。此前仅在两名艾滋病毒阴性患者中报道过与利妥昔单抗相关的卡波西肉瘤，并接受了手术治疗。
在此，我们报告一例在接受滤泡性淋巴瘤治疗的55岁艾滋病毒阴性患者中，在利妥昔单抗治疗期间发生的卡波西肉瘤。该病变在利妥昔单抗维持治疗期间于直肠部位出现，表现为明显的溃疡性结肠炎，无任何皮肤病变。利妥昔单抗的提前停用导致卡波西肉瘤完全消退，无需任何额外的特异性治疗。
据我们所知，这是第三例在艾滋病毒阴性患者中诊断出的与利妥昔单抗相关的卡波西肉瘤病例，第一例发生在直肠部位，第一例在利妥昔单抗停用后完全消退。该病例提高了对接受利妥昔单抗治疗的艾滋病毒阴性患者医源性卡波西肉瘤的认识，并进一步凸显了免疫抑制在疾病病理生理学中的重要性。
