Ikematsu Yuki, Izumi Miiru, Katahira Katsuyuki, Ueno Tsuyoshi, Moriuchi Yuki, Ose Mizuko, Noda Naotaka, Hara Makiko, Otsuka Junji, Wakamatsu Kentaro, Kawasaki Masayuki
Department of Respiratory Medicine, National Hospital Organization Omuta National Hospital, Fukuoka, Japan.
Department of Respiratory Medicine, National Hospital Organization Fukuoka National Hospital, Fukuoka, Japan.
Respir Med Case Rep. 2022 Mar 24;37:101642. doi: 10.1016/j.rmcr.2022.101642. eCollection 2022.
Werner syndrome (WS) is a rare progressive disorder that is characterized by premature aging of all organs. Malignancy is a frequent complication of WS, however, lung cancer patients with WS are much rare. In patients with WS, the treatment for malignancy is often limited due to other complications of severe skin ulcer, diabetes mellitus and cardiovascular disease. Currently, immune-checkpoint inhibitors (ICIs) are standard therapy for several cancer patients and the combination of nivolumab plus ipilimumab has also been approved for the treatment of non-small cell lung cancer (NSCLC). Recent studies have also reported that serious immune-related adverse events (irAEs) induced by ICIs may correlate with elderly or more vulnerable patients. However, the efficacy and safety of ICIs in NSCLC patients with WS remain unclear. To the best of our knowledge, this is the first case describing a NSCLC patient with WS receiving the combination immunotherapy of nivolumab and ipilimumab. Our case showed objective response to ICIs, however, several immune-related adverse events (irAEs) including hypothyroidism, adrenal insufficiency, hard rash and interstitial lung disease occurred, thus resulted in early treatment discontinuation. Our case suggests that immunotherapy for NSCLC patients with WS could be effective, but physicians may be aware of the possibility of multiple irAEs undergoing immunotherapy for NSCLC patients with WS.
沃纳综合征(WS)是一种罕见的进行性疾病,其特征是所有器官过早衰老。恶性肿瘤是WS的常见并发症,然而,患有WS的肺癌患者非常罕见。在WS患者中,由于严重皮肤溃疡、糖尿病和心血管疾病等其他并发症,恶性肿瘤的治疗往往受到限制。目前,免疫检查点抑制剂(ICIs)是几种癌症患者的标准治疗方法,纳武单抗加伊匹单抗的联合用药也已被批准用于治疗非小细胞肺癌(NSCLC)。最近的研究还报告称,ICIs诱导的严重免疫相关不良事件(irAEs)可能与老年或更脆弱的患者相关。然而,ICIs在患有WS的NSCLC患者中的疗效和安全性仍不清楚。据我们所知,这是第一例描述患有WS的NSCLC患者接受纳武单抗和伊匹单抗联合免疫治疗的病例。我们的病例显示对ICIs有客观反应,然而,发生了包括甲状腺功能减退、肾上腺功能不全、硬皮疹和间质性肺病在内的几种免疫相关不良事件(irAEs),从而导致早期治疗中断。我们的病例表明,对患有WS的NSCLC患者进行免疫治疗可能有效,但医生可能需要意识到患有WS的NSCLC患者接受免疫治疗时发生多种irAEs的可能性。