Barros Romão Cyndi Myrelle da Silva, Santos Júnior Cláudio José Dos, Leite Luiz Arthur Calheiros, Gomes Alves Maria Jordana Rocha, Araújo Nathalia Silva, Castro Anderson F L, Moura Muriel Silva, Gomes Vitória Mikaelly da Silva, Sampaio Batinga Arthur Moacir Costa, Queiroz João Antonio S, Dos Santos Natanael Barbosa
Department of Oncology, State University of Health Sciences of Alagoas, Maceió, Alagoas, Brazil.
Hematology Service, Gaffrée and Guinle Hospital, Federal University of the State of Rio de Janeiro, Rio de Janeiro, Brazil.
Am J Case Rep. 2017 Jun 21;18:692-695. doi: 10.12659/ajcr.903059.
BACKGROUND Blastic plasmacytoid dendritic cell neoplasm (BPDCN) is a rare hematodermic malignancy neoplasm with highly aggressive course and poor prognosis. This disease typically presents with cutaneous involvement as the first manifestation, with subsequent or simultaneous spread to bone marrow and peripheral blood. CASE REPORT Here, we report the case of a 51-year-old woman who presented a violaceus skin lesion on the lateral region of the right thigh, weight loss, fever, and lymphadenopathies. Computed tomography (CT) displayed thoracic and abdominal lymph node and alveolar bleeding. Flow cytometry from circulating blastic cells was compatible with BPDCN (CD4+, CD56+ and CD123+). She underwent 5 cycles of hyper-CVAD alternating with high-dose methotrexate and cytarabine, but the patient died due to alveolar bleeding and sepsis. CONCLUSIONS We report a rare case of BPDCN characterized by an aggressive course, presence of atypical skin lesion, a finding suggestive of pulmonary infiltration, and nonresponse to induction chemotherapy, leading to late diagnosis and therapeutic management. Because of the late recognition of the skin lesion, neoplastic cells infiltrated the dermis and spread as the disease progressed rapidly to a fatal course.
背景 母细胞性浆细胞样树突状细胞肿瘤(BPDCN)是一种罕见的血液系统恶性肿瘤,病程高度侵袭性,预后较差。该疾病通常以皮肤受累为首发表现,随后或同时扩散至骨髓和外周血。 病例报告 在此,我们报告一例51岁女性病例,该患者右大腿外侧出现紫蓝色皮肤病变、体重减轻、发热及淋巴结病。计算机断层扫描(CT)显示胸腹部淋巴结及肺泡出血。循环母细胞的流式细胞术检测结果与BPDCN相符(CD4+、CD56+和CD123+)。她接受了5个周期的Hyper-CVAD方案交替联合大剂量甲氨蝶呤和阿糖胞苷治疗,但患者因肺泡出血和败血症死亡。 结论 我们报告了一例罕见的BPDCN病例,其特点为病程侵袭性、存在非典型皮肤病变、提示肺部浸润的表现以及对诱导化疗无反应,导致诊断和治疗延迟。由于皮肤病变发现较晚,肿瘤细胞浸润真皮,并随着疾病迅速进展至致命病程而扩散。