Hirano Taichi, Higuchi Yusuke, Yuki Hiromichi, Hirata Shinya, Nosaka Kisato, Ishii Norito, Hashimoto Takashi, Mitsuya Hiroaki, Okuno Yutaka
Department of Hematology, Rheumatology, and Infectious Disease, Kumamoto University Graduate School of Medicine.
J Clin Exp Hematop. 2015;55(2):83-8. doi: 10.3960/jslrt.55.83.
A 60-year-old male patient suffered from mild exertional dyspnea, wheezing, and systemic blisters. He was diagnosed with paraneoplastic pemphigus (PNP) with follicular lymphoma in the pancreas head and pelvic cavity. He was first treated with eight cycles of rituximab; his blisters and erosions gradually improved and highly elevated levels of auto-antibodies related to PNP gradually decreased to normal levels. However, obstructive and restrictive respiratory failure still progressed. Computed tomography of the inspiratory and expiratory phases revealed obstructive pulmonary disorder, leading to a diagnosis of bronchiolitis obliterans (BO). The patient underwent plasma exchange and was repeatedly treated with rituximab monotherapy and rituximab-containing chemotherapies, but died 7 months after the diagnosis of BO. Early introduction of rituximab-containing regimens may be necessary to prevent the development of BO accompanying PNP. However, when a diagnosis of PNP-related BO is made, lung transplantation may also be considered for patients in whom rituximab-containing regimens are effective for PNP.
一名60岁男性患者出现轻度劳力性呼吸困难、喘息和全身性水疱。他被诊断为副肿瘤性天疱疮(PNP),同时患有胰腺头部和盆腔的滤泡性淋巴瘤。他首先接受了8个周期的利妥昔单抗治疗;他的水疱和糜烂逐渐改善,与PNP相关的自身抗体高水平逐渐降至正常水平。然而,阻塞性和限制性呼吸衰竭仍在进展。吸气和呼气期的计算机断层扫描显示阻塞性肺疾病,导致闭塞性细支气管炎(BO)的诊断。患者接受了血浆置换,并反复接受利妥昔单抗单药治疗和含利妥昔单抗的化疗,但在诊断为BO后7个月死亡。早期引入含利妥昔单抗的方案可能有必要预防PNP伴发的BO的发生。然而,当诊断为PNP相关的BO时,对于含利妥昔单抗方案对PNP有效的患者,也可考虑肺移植。