Li Qiangsheng, Zhang Jing, Gong Youjie, Wan Xudong, Zhang Song, Li Zhongwang, Liu Jun
Department of Cardiothoracic Surgery, Bengbu Third People's Hospital Affiliated to Bengbu Medical University, Bengbu, Anhui 233000, P.R. China.
Department of Pathology, Bengbu Third People's Hospital Affiliated to Bengbu Medical University, Bengbu, Anhui 233000, P.R. China.
Oncol Lett. 2025 May 2;30(1):321. doi: 10.3892/ol.2025.15067. eCollection 2025 Jul.
Pulmonary pleomorphic carcinoma (PPC), classified as the predominant subtype of pulmonary sarcomatoid carcinoma under the current World Health Organization (WHO) criteria, accounts for 0.1-0.4% of all non-small cell lung carcinoma cases and typically manifests radiologically as solid masses with peripheral infiltration. In the present report, a novel clinicopathological manifestation of PPC presenting as a primary solitary cystic airspace with recurrent spontaneous pneumothorax (SP), challenging conventional diagnostic paradigms, is described. A 66-year-old man with recurrent SP was initially misdiagnosed with pulmonary bullae based on the peripheral cystic airspaces observed on computed tomography. Persistent air leakage prompted video-assisted thoracoscopic wedge resection, which revealed biphasic histology: Malignant spindle cell proliferations (vimentin-positive) mixed with conventional adenocarcinoma components (transcription termination factor 1-positive/napsin A-positive), consistent with the WHO 2021 diagnostic criteria for PPC. The patient reached sustained remission without adjuvant therapy, and disease-free survival was maintained for 29 months. The present case highlights three critical implications: First, primary cystic airspaces represent a rare but clinically significant radiological phenotype of PPC that mimic benign bullous lesions, particularly when obscured by pneumothorax; second, recurrent SP may serve as the initial manifestation of occult pulmonary malignancy, necessitating rigorous evaluation of cystic lung lesions; third, early surgical intervention offers dual diagnostic and therapeutic value, even in patients with compromised pulmonary function. These findings expand the recognized spectrum of the imaging heterogeneity of PPC and underscore the need for heightened clinical suspicion of cystic lung cancer in high-risk populations.
肺多形性癌（PPC），根据当前世界卫生组织（WHO）标准被归类为肺肉瘤样癌的主要亚型，占所有非小细胞肺癌病例的0.1 - 0.4%，典型的影像学表现为伴有外周浸润的实性肿块。在本报告中，描述了一种PPC的新型临床病理表现，其表现为原发性孤立性囊状气腔并伴有复发性自发性气胸（SP），这对传统诊断模式提出了挑战。一名患有复发性SP的66岁男性最初基于计算机断层扫描观察到的外周囊状气腔被误诊为肺大疱。持续漏气促使进行电视辅助胸腔镜楔形切除术，结果显示为双相组织学：恶性梭形细胞增殖（波形蛋白阳性）与传统腺癌成分（转录终止因子1阳性/ napsin A阳性）混合，符合WHO 2021年PPC的诊断标准。该患者未经辅助治疗即实现持续缓解，无病生存期维持了29个月。本病例突出了三个关键意义：第一，原发性囊状气腔是PPC一种罕见但具有临床意义的影像学表型，可模仿良性大疱性病变，尤其是当被气胸掩盖时；第二，复发性SP可能是隐匿性肺恶性肿瘤的初始表现，需要对肺部囊性病变进行严格评估；第三，早期手术干预具有诊断和治疗双重价值，即使对于肺功能受损的患者也是如此。这些发现扩展了已认识到的PPC影像学异质性范围，并强调在高危人群中需要提高对肺部囊性癌的临床怀疑。
