Leković Danijela, Gotić Mirjana, Mitrović Olivera, Radojković Milica, Bila Jelena, Dencic-Fekete Marija, Kraguljac-Kurtović Nada, Peruničić-Jovanović Maja, Cokić Vladan
J Med Case Rep. 2013 Sep 11;7:224. doi: 10.1186/1752-1947-7-224.
The emergence of multiple myeloma as a second malignancy in patients with essential thrombocythemia is extremely rare. Several cases have been published so far, pointing out the impact of a cytotoxic effect during treatment of essential thrombocythemia on the development of multiple myeloma.
We report the case of a 52-year-old Caucasian man who presented to our hospital because of leukocytosis, a slightly decreased hemoglobin level and thrombocytosis. After a complete hematological work-up, essential thrombocythemia was diagnosed. The patient was included in a multicenter clinical study, treated with anagrelide and his platelet counts were maintained in the normal range for more than 3 years. A sudden drop in his hemoglobin level with normal leukocyte and platelet count occurred at the same time as a back pain. Magnetic resonance imaging of his spine revealed the existence of a pathological fracture of Th4, the collapse of the upper edge of Th7 and osteolytic lesions of multiple thoracic vertebrae. Repeated hematological examinations, including bone biopsy with immunohistochemistry, disclosed diagnosis of multiple myeloma of the non-secretory type.
To the best of our knowledge this is the first published case in which multiple myeloma developed during the treatment of essential thrombocythemia with the non-cytotoxic drug anagrelide. Our attempts to find a common origin for the coexistence of multiple myeloma and essential thrombocythemia have not confirmed the genetic basis of their appearance. Further studies are needed to determine the biological impact of this coexistence.
真性红细胞增多症患者发生多发性骨髓瘤作为第二恶性肿瘤极为罕见。迄今为止已发表了几例病例，指出真性红细胞增多症治疗期间的细胞毒性作用对多发性骨髓瘤发生的影响。
我们报告一例52岁白种男性患者，因白细胞增多、血红蛋白水平略有下降和血小板增多前来我院就诊。经过全面的血液学检查，诊断为真性红细胞增多症。该患者被纳入一项多中心临床研究，接受阿那格雷治疗，其血小板计数维持在正常范围超过3年。在出现背痛的同时，他的血红蛋白水平突然下降，白细胞和血小板计数正常。脊柱磁共振成像显示胸4病理性骨折、胸7上缘塌陷以及多个胸椎溶骨性病变。反复血液学检查，包括骨活检及免疫组化，确诊为非分泌型多发性骨髓瘤。
据我们所知，这是首例在用非细胞毒性药物阿那格雷治疗真性红细胞增多症期间发生多发性骨髓瘤的报道病例。我们试图寻找多发性骨髓瘤与真性红细胞增多症共存的共同起源，但未证实它们出现的遗传基础。需要进一步研究以确定这种共存的生物学影响。
