Abstract: Pulmonary sclerosing pneumocytoma (PSP) was first reported by Liebow and Hubbell (1956). It was initially called pulmonary sclerosing hemangioma (PSH) because it was thought to be derived from endothelial cells with rich vascularity. However, the 2015 World Health Organization Classification of Lung Tumors removed the term “sclerosing hemangioma,” since these tumors lack vascular origin (Travis et al., 2015), and the renamed tumor was considered a rare and benign lung tumor. However, several reports have described PSP with lymph node metastasis (Soo et al., 2017). Thus, PSP is not always benign and has the potential to metastasize. PSP shows varying levels of ¹⁸F-fluorodeoxy-glucose (¹⁸FDG) accumulation, with the maximum standardized uptake value (SUV_max) ranging from the background value to 6.4 (Chen et al., 2012; Jiang et al., 2018). Therefore, it is easily misdiagnosed as other malignant tumors and is often removed surgically or treated improperly. Here, we present a rare case of PSP in a young woman with lymph node metastasis, a high ¹⁸FDG uptake on positron emission tomography/computed tomography (PET/CT) scan, and an SUV_max of 6.1.

肺硬化性肺细胞瘤伴淋巴结转移及PET/CT ¹⁸FDG高摄取：一例罕见病例报告及文献综述

[1]ArumugamVG, JosephLD, ThangavelP, et al., 2017. Sclerosing pneumocytoma of the lung: a case report. J Clin Diagn Res, 11(2):ED12-ED14.

[2]BaeK, SongDH, JeonKN, et al., 2019. Pulmonary sclerosing pneumocytoma presenting a peritumoral halo and an intervening lucent zone on computed tomography: radiology–pathology correlation. Thorac Cancer, 10(5):1295-1296.

[3]ChenQY, WuLJ, HuHZ, et al., 2012. A case of pulmonary sclerosing hemangioma with low ¹⁸FDG uptake in PET. Oncol Lett, 3(3):646-648.

[4]CherianSV, Estrada-Y-MartinRM, IraniAD, et al., 2017. A 33-year-old woman with a fluorodeoxyglucose-avid left lower lobe mass. Chest, 151(5):e119-e122.

[5]ChienNC, LinCW, TzengJE, 2009. Sclerosing haemangioma with lymph node metastasis. Respirology, 14(4):614-616.

[6]ChoHJ, LeeJH, LeeGK, et al., 2017. Case of sclerosing pneumocytoma combined with a typical carcinoid and pulmonary adenocarcinoma in different lobes. Thorac Cancer, 8(4):372-375.

[7]DavidN, ChandyST, BenjaminSR, et al., 2022. Pulmonary sclerosing pneumocytoma—a case series. Indian J Thorac Cardiovasc Surg, 38(2):167-172.

[8]de LucaG, MartucciN, SetolaS, et al., 2015. Sclerosing hemangioma of the lung mimicking pulmonary metastasis. Lung, 193(3):447-448.

[9]Devouassoux-ShisheboranM, HayashiT, LinnoilaRI, et al., 2000. A clinicopathologic study of 100 cases of pulmonary sclerosing hemangioma with immunohistochemical studies: TTF-1 is expressed in both round and surface cells, suggesting an origin from primitive respiratory epithelium. Am J Surg Pathol, 24(7):906-916.

[10]JiangL, HuangY, TangQY, et al., 2018. ¹⁸F‑FDG PET/CT characteristics of pulmonary sclerosing hemangioma vs. pulmonary hamartoma. Oncol Lett, 16(1):660-665.

[11]KatakuraH, SatoM, TanakaF, et al., 2005. Pulmonary sclerosing hemangioma with metastasis to the mediastinal lymph node. Ann Thorac Surg, 80(6):2351-2353.

[12]KhannaA, AlshabaniK, MukhopadhyayS, et al., 2019. Sclerosing pneumocytoma: case report of a rare endobronchial presentation. Medicine, 98(15):e15038.

[13]KhooACH, HamzahF, OngCK,2017. Incidental sclerosing pneumocytoma detected on bone scintigraphy. Clin Nucl Med, 42(1):e77-e79.

[14]KimKH, SulHJ, KangDY, 2003. Sclerosing hemangioma with lymph node metastasis. Yonsei Med J, 44(1):150-154.

[15]KimMK, JangSJ, KimYH, et al., 2015. Bone metastasis in pulmonary sclerosing hemangioma. Korean J Intern Med, 30(6):928-930.

[16]KocamanG, YenigünMB, ErsözCC, et al., 2021. Pulmonary sclerosing pneumocytoma with mediastinal lymph node metastasis: a case report. Gen Thorac Cardiovasc Surg, 69(1):142-146.

[17]LeeK, SasakiM, KakimotoT, et al., 2019. Pulmonary sclerosing pneumocytoma demonstrating intratumoral hematoma. Respir Med Case Rep, 28:100870.

[18]LiebowAA, HubbellDS, 1956. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer, 9(1):53-75.

[19]LimJH, LeeN, ChoiDW, et al., 2016. Pulmonary sclerosing pneumocytoma mimicking lung cancer: case report and review of the literature. Thorac Cancer, 7(4):508-511.

[20]MaedaH, KanzakiM, IsakaT, et al., 2015. Video-assisted thoracoscopic enucleation after congenital cardiac surgery. J Surg Case Rep, 2015(9):rjv123.

[21]MasudaM, EndoS, NatsugoeS, et al., 2018. Thoracic and cardiovascular surgery in Japan during 2015. Gen Thorac Cardiovasc Surg, 66(10):581-615.

[22]Miyagawa-HayashinoA, TazelaarHD, LangelDJ, et al., 2003. Pulmonary sclerosing hemangioma with lymph node metastases: report of 4 cases. Arch Pathol Lab Med, 127(3):321-325.

[23]MorikawaK, MisumiS, FukudaT, et al., 2019. Pulmonary sclerosing pneumocytoma presenting as slow-growing multiple nodules over a long period. Radiol Case Rep, 14(5):602-607.

[24]NguyenXC, LeeWW, ChungJH, et al., 2007. FDG uptake, glucose transporter type 1, and Ki-67 expressions in non-small-cell lung cancer: correlations and prognostic values. Eur J Radiol, 62(2):214-219.

[25]PokharelS, DhillonSS, YlaganL, et al., 2016. Sclerosing pneumocytoma with lymph node metastasis. J Thorac Oncol, 11(10):1802-1804.

[26]SatohY, TsuchiyaE, WengSY, et al., 1989. Pulmonary sclerosing hemangioma of the lung. A type II pneumocytoma by immunohistochemical and immunoelectron microscopic studies. Cancer, 64(6):1310-1317.

[27]SakaiT, MiyoshiT, UmemuraS, et al., 2019. Large pulmonary sclerosing pneumocytoma with massive necrosis and vascular invasion: a case report. Oxf Med Case Reports, 2019(7):omz066.

[28]SchiergensTS, KhalilPN, MayrD, et al., 2011. Pulmonary sclerosing hemangioma in a 21-year-old male with metastatic hereditary non-polyposis colorectal cancer: report of a case. World J Surg Oncol, 9:62.

[29]ShiinaY, SakairiY, WadaH, et al., 2018. Sclerosing pneumocytoma diagnosed by preoperative endobronchial ultrasound-guided transbronchial needle aspiration (EBUS-TBNA). Surg Case Rep, 4:20.

[30]SooIX, SittampalamK, LimCH, 2017. Pulmonary sclerosing pneumocytoma with mediastinal lymph node metastasis. Asian Cardiovasc Thorac Ann, 25(7-8):547-549.

[31]SunY, ZhouLX, ZhaoM, et al., 2012. Histogenesis of pulmonary sclerosing hemangioma. Chin J Pathol, 41(4):239-242 (in Chinese).

[32]TanakaI, InoueM, MatsuiY, et al., 1986. A case of pneumocytoma (so-called sclerosing hemangioma) with lymph node metastasis. Jpn J Clin Oncol, 16(1):77-86.

[33]TravisWD, BrambillaE, NicholsonAG, et al., 2015. The 2015 World Health Organization classification of lung tumors: impact of genetic, clinical and radiologic advances since the 2004 classification. J Thorac Oncol, 10(9):1243-1260.

[34]XuJ, DongYW, YinGT, et al., 2019. ¹⁸F-FDG PET/CT imaging: a supplementary understanding of pulmonary sclerosing pneumocytoma. Thorac Cancer, 10(7):1552-1560.

[35]YanoM, YamakawaY, KiriyamaM, et al., 2002. Sclerosing hemangioma with metastases to multiple nodal stations. Ann Thorac Surg, 73(3):981-983.