Bilateral Multiple Pulmonary Sclerosing Pneumocytomain A Young Male: Case Report

Bilateral Multiple Pulmonary Sclerosing Pneumocytomain A Young Male: Case Report | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case report Bilateral Multiple Pulmonary Sclerosing Pneumocytomain A Young Male: Case Report Xianshuai Li, Shuqian He, xianguo chen This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-686381/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Background: Pulmonary sclerosing pneumocytoma (PSP) is a relatively rare benign tumor, and multiple pulmonary sclerosing pneumocytomas are even rarer. PSP is usually seen in adults over 50 years old, featuring a female to male ratio of 5:1. We report an extremely rare case of a 20-year-old male patient with bilateral multiple pulmonary sclerosing pneumocytoma. This rare benign disease also presents diagnostic challenges throughout the treatment process. Case presentation: A 20-year-old male was diagnosed with bilateral multiple pulmonary nodules on chest CT. The patient underwent regular chest CT reexamination over the course of six months, which indicated a tendency of slow enlargement of pulmonary nodules. After complete examination and exclusion of tumors in other parts of the body, he underwent two separate surgeries, and bilateral multiple nodules were removed. Intraoperative frozen section analysis suggested that some nodules were adenocarcinoma and the remaining nodules were considered benign tumors. Postoperative immunohistochemistry indicated that all nodules were pulmonary sclerosing pneumocytomas. Conclusions: Bilateral multiple pulmonary sclerosing pneumocytoma are rare in young men. In clinical practice, the differentiation of this disease from malignancy is challenging, and surgical treatment is a safe option for pulmonary nodules with a tendency to become larger. Surgery Oncology Pulmonary nodule Pulmonary sclerosing pneumocytoma Lung adenocarcinoma Lung surgery Multiple Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Background Pulmonary sclerosing pneumocytoma (PSP) is a rare benign tumor. Clinically, PSP of the lung is most frequently encountered in middle-aged women, with a peak incidence at 50 years of age( 1 ). Typically, PSP presents with a single nodule, and multiple nodules are very rare. In addition, the diagnosis of sclerosing alveolar cell tumor remains challenging, both radiologically and histologically. We report an extremely rare case of a 20-year-old male patient with bilateral multiple pulmonary sclerosing pneumocytoma. This case also presents diagnostic challenges throughout the treatment process. Case Presentation The patient, a 20-year-old male, was admitted to the hospital due to multiple nodules in both lungs. Prior to hospitalization, the patient underwent regular chest CT reexamination over the course of six months, which indicated a tendency of slow enlargement of pulmonary nodules. The patient's physical examination showed no obvious abnormalities, and there was no previous family history of tumor. No significant increase in tumor markers was observed in blood samples. Enhanced chest CT showed multiple high-density nodules in both lungs with clear boundaries (Figs. 1 , 2 ). The largest nodule was located in the right lower lobe, measuring approximately 2.8 cm * 2.3 cm. Enhanced chest CT showed obvious homogeneous enhancement of the nodules. No enlarged lymph nodes were found in the mediastinum or hilum. PET-CT showed multiple nodules in both lungs, increased uptake of the imaging agent, and SUVmax: 4.8-5, no obvious metabolic abnormality was found in other parts of the body (Fig. 3 ). A fine-needle aspiration (FNA) cytology of the right lower pulmonary nodule revealed a large number of epithelioid cells with small cell atypia, and malignancy could not be ruled out. Considering the young age of the patient, the tendency of pulmonary nodules to become larger, and the fact that the current examination could not rule out malignant tumor, we decided to remove the pulmonary nodules surgically. We performed thoracoscopic right middle lung lobectomy + right upper lung wedge resection + right lower lung wedge resection under general anesthesia(Fig. 4 ). intraoperative frozen sections showed that there were microinvasive adenocarcinomas in the right upper lung (0.4 cm in diameter), two nodules in the right middle lung (both 0.9 cm in diameter), and two nodules in the right lower lung (2.1 cm in diameter). Postoperative immunohistochemistry indicated that all nodules were pulmonary sclerosing pneumocytomas(Fig. 5 ), and the hilar and mediastinal lymph node samples were negative. The patient recovered smoothly and was discharged from the hospital. One month later, he returned to the hospital for left lung surgery. He underwent thoracoscopic wedge resection of the left upper lung and left lower lung. Postoperative immunohistochemistry showed all nodules were pulmonary sclerosing pneumocytomas: CK-Pan (+), EMA (+), TTF-1 (+), VIM (+), P40 (-). The patients recovered and was discharged smoothly. Discussion Pulmonary sclerosing pneumocytoma (PSP), formerly known as pulmonary sclerosing hemangioma, is a rare pulmonary tumor initially described by Liebow et al( 2 ). in 1956, as a tumor with marked sclerosis and vascularization. PSP is usually seen in female over 50 years old, and most of them are single nodules. In the 2015, the World Health Organization (WHO) classification “miscellaneous tumors” was changed to “adenomas”( 3 ). The essential feature of PSP is the presence of cuboidal surface cells and round stromal cells, which consist of four major histological patterns; hemangiomatous, papillary, sclerotic, and solid( 3 ). The immunohistochemical staining pattern is positive for TTF-1 and EMA( 4 ). Our case is a 20-year-old young man with bilateral multiple PSP, which has rarely been reported. Over the last years, many studies have discussed the origin, differential diagnosis, potential malignancy and best treatment option for PSP. For many years, in fact, this tumor was presumed to be of vascular origin. Instead, immunohistochemical results, in particular the TTF-1 expression, confirm its epithelial origin from the respiratory epithelium( 4 ). Because of the unspecific radiological features mimicking malignancies and its histological heterogeneity, the differential diagnosis with adenocarcinoma and carcinoid tumors is still challenging. Patients are usually asymptomatic and it is detected incidentally. Chest CT scan usually reveals a round, solitary, well-circumscribed, homogeneous nodule and FDG PET/CT scan usually reveals a low to moderate uptake in PSPs( 5 ). Our patient also had no clinical symptoms, and chest CT and PET-CT findings were similar to those reported in the literature. From the imaging inferences, the pulmonary nodules of the patient were considered benign first. We also performed fine-needle aspiration (FNA) cytology on the largest nodules, but no evidence of malignant tumor was found. Therefore, we followed the patient for 6 months, and found that the pulmonary nodules had a tendency to slowly grow larger. In view of this situation, we decided to perform surgical treatment for the patient. Pre-operative diagnosis, when feasible, is helpful to plan the best therapeutic strategy. Gal et al. was the first to report that the cytologic diagnosis of PSP requires the identification of its dual cell population, made up of abundant stromal cells and fewer surface cells( 6 ). The problem is that fine-needle aspiration (FNA) cytology is poorly diagnostic, both due to the rarely distinct separation of the two tumor cell types in the specimen and to the disease rarity and hence potential unfamiliarity with its cytologic features of most pathologists. Preoperative cytologic and histological findings of PSP are limited to few case reports, in which computed tomography (CT)-guided FNA, EBUS-TBNA or intraoperative frozen sections (FS) often led to misdiagnosis( 7 ). In our case, intraoperative frozen section suggested PSP coexisting with adenocarcinoma, but immunohistochemical results revealed that all nodules were sclerosing alveolar cell tumor. PSP combined with lung adenocarcinoma has indeed been reported in very few reports( 8 , 9 ). PSP can also be combined and admixed with other lung tumors, such as mucinous adenomatous hyperplasia( 10 ), glandular papilloma (GP)( 11 ), pulmonary spindle cell carcinoma (PSCC)( 12 ), extensive neuroendocrine lesions (including pulmonary neuroendocrine cell hyperplasia, multiple carcinoid tumorlets and typical carcinoid tumors within one pulmonary lobe)( 13 ). FNA cytology is poorly diagnostic, and the misdiagnosis rate of intraoperative frozen section is also high. Shang Z et al. reports the rate of intraoperative misdiagnosis of PSP smaller than 1 cm was 11.1%( 14 ). The pathological diagnosis of PSP is challenging, and obtaining sufficient samples and immunohistochemical examination is the key to improve the diagnosis rate. PSP can be associated with other tumors at different sites, such as hereditary nonpolyposis colorectal cancer (HNPCC, Lynch syndrome)( 15 ), familial adenomatous polyposis( 16 ), retroperitoneal liposarcoma and renal carcinoma( 17 ), multiple uterine fibromas, cysts of the thyroid and kidney( 18 ), and endometrial stromal sarcoma( 19 ). This indicates that other malignant tumors can coexist with multiple solid pulmonary nodules and be misdiagnosed at times as multiple pulmonary metastases, which would affect the clinical treatment decision; therefore, these masses need to be carefully identified. It has been reported that PSP may present with hilar and mediastinal lymph node metastasis and pleural dissemination( 20 – 22 ). In the present case, the hilar and mediastinal lymph nodes were not significantly enlarged, and the pathological results from the hilar lymph nodes were negative. Even with the help of enhanced CT, PET-CT, puncture biopsy or intraoperative rapid pathological reports, PSP may be misdiagnosed. Surgical resection is the treatment of choice for PSP because there have been no reports of recurrence in the literature. Lymph node dissection should be considered for huge tumors because lymph node metastases correlate with a larger tumor size(23). The chest CT of this patient revealed 7 nodules, 3 in the left lobe and 4 right. We resected all the nodules through two surgeries, and the patient recovered smoothly each time. We will perform a long follow-up to assess recurrence and metastasis for this patient. Conclusions Bilateral multiple PSPs are very rare in young men. The clinical diagnosis of sclerosing alveolar cell tumor is challenging, and a knowledge of this rare type of neoplasm is fundamental, for both surgeons and pathologists. Surgery is an important method for diagnosis and treatment of sclerosing alveolar cell tumor. List Of Abbreviations Pulmonary sclerosing pneumocytoma (PSP) glandular papilloma (GP) pulmonary spindle cell carcinoma (PSCC) hereditary nonpolyposis colorectal cancer (HNPCC, Lynch syndrome) Declarations Ethics approval and consent to participate This study has been approval and consented by the patient. This study was approved by the Medical Ethics Committee of Jinhua Central Hospital. Consent for publication The work described was original research that has not been published previously, and not under consideration for publication elsewhere, in whole or in part. All the authors listed have approved the manuscript for publication and copyright transfer of this paper. Written informed consent for publication was obtained from all participants. Availability of data and materials Not applicable Competing interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article. Funding The present study was supported by the Jinhua Science and Technology Bureau (2020-3-041-1).The funder subsidized the English language editing servicesand publishing expenses of this paper. Author Contributions LXS was responsible for patient management and was a major contributor in writing the manuscript. HSQ contributed to data collection, data analysis and bibliography retrieval. CXG contributed to case conception, revising for important intellectual content and final approval ofreport, supervising surgeon. All authors read and approved the final manuscript. Acknowledgements Not applicable References Kuo KT, Hsu WH, Wu YC, Huang MH, Li WY. Sclerosing hemangioma of the lung: an analysis of 44 cases. J Chin Med Assoc. 2003;66(1):33–8. Liebow AA, Hubbell DS. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer. 1956;9:53–75. Travis WD, Brambilla E, Nicholson AG, Yatabe Y, Austin JHM, Beasley MB, Chirieac LR, Dacic S, Duhig E, Flieder DB, et al. The 2015 World Health Organization Classification of Lung Tumors: Impact of Genetic, Clinical and Radiologic Advances Since the 2004 Classification. J Thorac Oncol. 2015;10:1243–60. Devouassoux-Shisheboran M, Hayashi T, Linnoila RI, Koss MN, Travis WD. 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. 2000;24(7):906–16. Shin SY, Kim MY, Oh SY, Lee HJ, Hong SA, Jang SJ, et al. Pulmonary sclerosing pneumocytoma of the lung: CT characteristics in a large series of a tertiary referral center. Med (Baltim). 2015;94(4):e498. Gal AA, Nassar VH, Miller JI. Cytopathologic diagnosis of pulmonary sclerosing hemangioma. Diagn Cytopathol. 2002;26:163–6. Trabucco SMR, Brascia D, Cazzato G, De Iaco G, Colagrande A, Signore F, Ingravallo G, Resta L, Marulli G. Pulmonary Sclerosing Pneumocytoma: A Pre and Intraoperative Diagnostic Challenge. Report of Two Cases and Review of the Literature. Medicina (Kaunas). 2021 May 23;57(6):524. Schiergens TS, Khalil PN, Mayr D, et al. Pulmonary sclerosing hemangioma in a 21-year-old male with metastatic hereditary non-polyposis colorectal cancer: report of a case. World J Surg Oncol. 2011;9:62. doi: 10.1186/1477-7819-9-62 . Published 2011 Jun 6. Hosaka N, Sasaki T, Adachi K, et al. Pulmonary sclerosing hemangioma associated with familial adenomatous polyposis. Hum Pathol. 2004;35(6):764–8. doi: 10.1016/j.humpath.2004.02.003 . Longo R, Carillio G, Torrisi A, et al. An unusual case of three synchronous tumors in a young woman. Tumori. 2005;91(3):267–9. Rizzo S, Pandolfi U, Villani L, et al. Pulmonary sclerosing hemangioma in a woman with multiple uterine fibromas, cysts to thyroid and kidney. Anticancer Res. 1996;16(3A):1297–9. Aubry MC, Myers JL, Colby TV, Leslie KO, Tazelaar HD. Endometrial stromal sarcoma metastatic to the lung: a detailed analysis of 16 patients. Am J Surg Pathol. 2002;26(4):440–449. 13. Wang Y, He Q, Shi W, Wang J, Ji H. A mixture of carcinoid tumors, extensive neuroendocrine proliferation, and multiple pulmonary sclerosing hemangiomas. World J Surg Oncol. 2014;12:209. Published 2014 Jul 15. Shang Z, Han Y, Shao J, Zhu L, Teng H, Zhang J. Challenging of frozen diagnoses of small sclerosing pneumocytoma. J Clin Pathol. 2021 Mar;29:jclinpath–2020. Liu W, Tian XY, Li Y, Zhao Y, Li B, Li Z. Coexistence of pulmonary sclerosing hemangioma and primary adenocarcinoma in the same nodule of lung. Diagn Pathol. 2011;6:41. Published 2011 May 20. Suzuki K, Shiono S, Kato H, Yanagawa N, Sato T. Kyobu Geka. Small sclerosing hemangioma combined with primary lung cancer; report of a case. Thorac Cardiovasc Surg. 2006;59(7):590–3. Choi KH, Baek HA, Park HS, et al. Sclerosing hemangioma, presenting as a pneumonic pattern with mucinous adenomatous hyperplasia of the lung. Pathol Int. 2008;58(11):735–40. Kitawaki Y, Fujishima F, Taniuchi S, et al. Coexistence of glandular papilloma and sclerosing pneumocytoma in the bronchiole. Pathol Int. 2018;68(7):425–30. LuLu X, Jian S. Concomitance of pulmonary spindle cell carcinoma and sclerosing pneumocytoma in a woman: A case report. Med (Baltim). 2019;98(51):e18416. Wang X, Zhang L, Wang Y, Jia X, Wang J, Zhang H. Sclerosing pneumocytoma with metastasis to the mediastinal and regional lymph nodes. Indian J Pathol Microbiol. 2018;61(3):407–9. Suzuki H, Saitoh Y, Koh E, et al. Pulmonary sclerosing hemangioma with pleural dissemination: report of a case. Surg Today. 2011;41(2):258–61. Yano M, Yamakawa Y, Kiriyama M, Hara M, Murase T. Sclerosing hemangioma with metastases to multiple nodal stations. Ann Thorac Surg. 2002;73(3):981–3. Adachi Y, Tsuta K, Hirano R, Tanaka J, Minamino K, Shimo T, et al. Pulmonary sclerosing hemangioma with lymph nodemetastasis: a case report and literature review. Oncol Lett. 2014;7:997–1000. Cite Share Download PDF Status: Posted Version 1 posted You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-686381","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case report","associatedPublications":[],"authors":[{"id":43391673,"identity":"7e39a8d3-0eac-4653-99c5-a26419b0bbc6","order_by":0,"name":"Xianshuai Li","email":"","orcid":"","institution":"Jinhua Hospital of Zhejiang University: Jinhua Municipal Central Hospital","correspondingAuthor":false,"prefix":"","firstName":"Xianshuai","middleName":"","lastName":"Li","suffix":""},{"id":43391674,"identity":"5934b3ae-1cc2-4e9d-8e01-44fb9708ad15","order_by":1,"name":"Shuqian He","email":"","orcid":"","institution":"Jinhua Hospital of Zhejiang 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14:41:17","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":1322191,"visible":true,"origin":"","legend":"Location of right pulmonary nodules in the second operation(The arrows point to the nodules)","description":"","filename":"2.png","url":"https://assets-eu.researchsquare.com/files/rs-686381/v1/29f623c7a2dafe824730bfa7.png"},{"id":12107565,"identity":"147ee5df-48c4-416c-8e18-25ae141a4631","added_by":"auto","created_at":"2021-08-04 14:41:17","extension":"jpg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":127548,"visible":true,"origin":"","legend":"PET-CT images show multiple nodules in both lungs, increased uptake of imaging agent, and SUVmax: 4.8-5(The arrows point to the nodules)","description":"","filename":"3.jpg","url":"https://assets-eu.researchsquare.com/files/rs-686381/v1/27041d14507f396a0a155c44.jpg"},{"id":12107563,"identity":"68ca2222-0cce-4a48-9d18-29cdb31cfd7e","added_by":"auto","created_at":"2021-08-04 14:41:17","extension":"png","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":1234209,"visible":true,"origin":"","legend":"Photographs of surgical specimens of PSP","description":"","filename":"4.png","url":"https://assets-eu.researchsquare.com/files/rs-686381/v1/811769d61864e8f7ff5fad8a.png"},{"id":12107568,"identity":"2f859661-67a9-42e6-9201-11c32a6dee44","added_by":"auto","created_at":"2021-08-04 14:41:17","extension":"png","order_by":5,"title":"Figure 5","display":"","copyAsset":false,"role":"figure","size":2694791,"visible":true,"origin":"","legend":"Postoperative immunohistochemistry of PSP,CK-Pan (+), EMA (+), TTF-1 (+), VIM (+)","description":"","filename":"5.png","url":"https://assets-eu.researchsquare.com/files/rs-686381/v1/1c021530362b79fe9759c86f.png"},{"id":13707410,"identity":"ffbd6a01-cf05-4d38-bb97-676d1ee09e9f","added_by":"auto","created_at":"2021-09-17 14:02:37","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":6295021,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-686381/v1/16307a79-2569-41db-9fc9-5467f3410032.pdf"}],"financialInterests":"","formattedTitle":"\u003cp\u003eBilateral Multiple Pulmonary Sclerosing Pneumocytomain A Young Male: Case Report\u003c/p\u003e","fulltext":[{"header":"Background","content":"\u003cp\u003ePulmonary sclerosing pneumocytoma (PSP) is a rare benign tumor. Clinically, PSP of the lung is most frequently encountered in middle-aged women, with a peak incidence at 50 years of age(\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e). Typically, PSP presents with a single nodule, and multiple nodules are very rare. In addition, the diagnosis of sclerosing alveolar cell tumor remains challenging, both radiologically and histologically. We report an extremely rare case of a 20-year-old male patient with bilateral multiple pulmonary sclerosing pneumocytoma. This case also presents diagnostic challenges throughout the treatment process.\u003c/p\u003e"},{"header":"Case Presentation","content":"\u003cp\u003eThe patient, a 20-year-old male, was admitted to the hospital due to multiple nodules in both lungs. Prior to hospitalization, the patient underwent regular chest CT reexamination over the course of six months, which indicated a tendency of slow enlargement of pulmonary nodules. The patient's physical examination showed no obvious abnormalities, and there was no previous family history of tumor. No significant increase in tumor markers was observed in blood samples. Enhanced chest CT showed multiple high-density nodules in both lungs with clear boundaries (Figs.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e, \u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). The largest nodule was located in the right lower lobe, measuring approximately 2.8 cm * 2.3 cm. Enhanced chest CT showed obvious homogeneous enhancement of the nodules. No enlarged lymph nodes were found in the mediastinum or hilum. PET-CT showed multiple nodules in both lungs, increased uptake of the imaging agent, and SUVmax: 4.8-5, no obvious metabolic abnormality was found in other parts of the body (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e). A fine-needle aspiration (FNA) cytology of the right lower pulmonary nodule revealed a large number of epithelioid cells with small cell atypia, and malignancy could not be ruled out.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eConsidering the young age of the patient, the tendency of pulmonary nodules to become larger, and the fact that the current examination could not rule out malignant tumor, we decided to remove the pulmonary nodules surgically. We performed thoracoscopic right middle lung lobectomy\u0026thinsp;+\u0026thinsp;right upper lung wedge resection\u0026thinsp;+\u0026thinsp;right lower lung wedge resection under general anesthesia(Fig.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e4\u003c/span\u003e). intraoperative frozen sections showed that there were microinvasive adenocarcinomas in the right upper lung (0.4 cm in diameter), two nodules in the right middle lung (both 0.9 cm in diameter), and two nodules in the right lower lung (2.1 cm in diameter). Postoperative immunohistochemistry indicated that all nodules were pulmonary sclerosing pneumocytomas(Fig.\u0026nbsp;\u003cspan refid=\"Fig5\" class=\"InternalRef\"\u003e5\u003c/span\u003e), and the hilar and mediastinal lymph node samples were negative. The patient recovered smoothly and was discharged from the hospital. One month later, he returned to the hospital for left lung surgery. He underwent thoracoscopic wedge resection of the left upper lung and left lower lung. Postoperative immunohistochemistry showed all nodules were pulmonary sclerosing pneumocytomas: CK-Pan (+), EMA (+), TTF-1 (+), VIM (+), P40 (-). The patients recovered and was discharged smoothly.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003ePulmonary sclerosing pneumocytoma (PSP), formerly known as pulmonary sclerosing hemangioma, is a rare pulmonary tumor initially described by Liebow et al(\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e). in 1956, as a tumor with marked sclerosis and vascularization. PSP is usually seen in female over 50 years old, and most of them are single nodules. In the 2015, the World Health Organization (WHO) classification \u0026ldquo;miscellaneous tumors\u0026rdquo; was changed to \u0026ldquo;adenomas\u0026rdquo;(\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e). The essential feature of PSP is the presence of cuboidal surface cells and round stromal cells, which consist of four major histological patterns; hemangiomatous, papillary, sclerotic, and solid(\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e). The immunohistochemical staining pattern is positive for TTF-1 and EMA(\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e). Our case is a 20-year-old young man with bilateral multiple PSP, which has rarely been reported.\u003c/p\u003e \u003cp\u003eOver the last years, many studies have discussed the origin, differential diagnosis, potential malignancy and best treatment option for PSP. For many years, in fact, this tumor was presumed to be of vascular origin. Instead, immunohistochemical results, in particular the TTF-1 expression, confirm its epithelial origin from the respiratory epithelium(\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e). Because of the unspecific radiological features mimicking malignancies and its histological heterogeneity, the differential diagnosis with adenocarcinoma and carcinoid tumors is still challenging. Patients are usually asymptomatic and it is detected incidentally. Chest CT scan usually reveals a round, solitary, well-circumscribed, homogeneous nodule and FDG PET/CT scan usually reveals a low to moderate uptake in PSPs(\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e). Our patient also had no clinical symptoms, and chest CT and PET-CT findings were similar to those reported in the literature. From the imaging inferences, the pulmonary nodules of the patient were considered benign first. We also performed fine-needle aspiration (FNA) cytology on the largest nodules, but no evidence of malignant tumor was found. Therefore, we followed the patient for 6 months, and found that the pulmonary nodules had a tendency to slowly grow larger. In view of this situation, we decided to perform surgical treatment for the patient. Pre-operative diagnosis, when feasible, is helpful to plan the best therapeutic strategy. Gal et al. was the first to report that the cytologic diagnosis of PSP requires the identification of its dual cell population, made up of abundant stromal cells and fewer surface cells(\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e). The problem is that fine-needle aspiration (FNA) cytology is poorly diagnostic, both due to the rarely distinct separation of the two tumor cell types in the specimen and to the disease rarity and hence potential unfamiliarity with its cytologic features of most pathologists. Preoperative cytologic and histological findings of PSP are limited to few case reports, in which computed tomography (CT)-guided FNA, EBUS-TBNA or intraoperative frozen sections (FS) often led to misdiagnosis(\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e). In our case, intraoperative frozen section suggested PSP coexisting with adenocarcinoma, but immunohistochemical results revealed that all nodules were sclerosing alveolar cell tumor. PSP combined with lung adenocarcinoma has indeed been reported in very few reports(\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e, \u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e). PSP can also be combined and admixed with other lung tumors, such as mucinous adenomatous hyperplasia(\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e), glandular papilloma (GP)(\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e), pulmonary spindle cell carcinoma (PSCC)(\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e), extensive neuroendocrine lesions (including pulmonary neuroendocrine cell hyperplasia, multiple carcinoid tumorlets and typical carcinoid tumors within one pulmonary lobe)(\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e). FNA cytology is poorly diagnostic, and the misdiagnosis rate of intraoperative frozen section is also high. Shang Z et al. reports the rate of intraoperative misdiagnosis of PSP smaller than 1 cm was 11.1%(\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e). The pathological diagnosis of PSP is challenging, and obtaining sufficient samples and immunohistochemical examination is the key to improve the diagnosis rate.\u003c/p\u003e \u003cp\u003ePSP can be associated with other tumors at different sites, such as hereditary nonpolyposis colorectal cancer (HNPCC, Lynch syndrome)(\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e), familial adenomatous polyposis(\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e), retroperitoneal liposarcoma and renal carcinoma(\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e), multiple uterine fibromas, cysts of the thyroid and kidney(\u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e), and endometrial stromal sarcoma(\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e). This indicates that other malignant tumors can coexist with multiple solid pulmonary nodules and be misdiagnosed at times as multiple pulmonary metastases, which would affect the clinical treatment decision; therefore, these masses need to be carefully identified. It has been reported that PSP may present with hilar and mediastinal lymph node metastasis and pleural dissemination(\u003cspan additionalcitationids=\"CR21\" citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e). In the present case, the hilar and mediastinal lymph nodes were not significantly enlarged, and the pathological results from the hilar lymph nodes were negative.\u003c/p\u003e \u003cp\u003eEven with the help of enhanced CT, PET-CT, puncture biopsy or intraoperative rapid pathological reports, PSP may be misdiagnosed. Surgical resection is the treatment of choice for PSP because there have been no reports of recurrence in the literature. Lymph node dissection should be considered for huge tumors because lymph node metastases correlate with a larger tumor size(23). The chest CT of this patient revealed 7 nodules, 3 in the left lobe and 4 right. We resected all the nodules through two surgeries, and the patient recovered smoothly each time. We will perform a long follow-up to assess recurrence and metastasis for this patient.\u003c/p\u003e"},{"header":"Conclusions","content":"\u003cp\u003eBilateral multiple PSPs are very rare in young men. The clinical diagnosis of sclerosing alveolar cell tumor is challenging, and a knowledge of this rare type of neoplasm is fundamental, for both surgeons and pathologists. Surgery is an important method for diagnosis and treatment of sclerosing alveolar cell tumor.\u003c/p\u003e"},{"header":"List Of Abbreviations","content":" \u003cp\u003ePulmonary sclerosing pneumocytoma (PSP)\u003c/p\u003e \u003cp\u003eglandular papilloma (GP)\u003c/p\u003e \u003cp\u003epulmonary spindle cell carcinoma (PSCC)\u003c/p\u003e \u003cp\u003ehereditary nonpolyposis colorectal cancer (HNPCC, Lynch syndrome)\u003c/p\u003e \u003c/div\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eEthics approval and consent to participate\u0026nbsp;\u003c/strong\u003eThis study has been approval and consented by the patient. This study was approved by the Medical Ethics Committee of Jinhua Central Hospital.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u003c/strong\u003e \u0026nbsp;The work described was original research that has not been published previously, and not under consideration for publication elsewhere, in whole or in part.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eAll the authors listed have approved the manuscript for publication and copyright transfer of this paper.\u003c/p\u003e\n\u003cp\u003eWritten informed consent for publication was obtained from all participants.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAvailability of data and materials \u003c/strong\u003eNot applicable\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCompeting interests\u003c/strong\u003e\u0026nbsp; The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding \u003c/strong\u003eThe present study was supported by the Jinhua Science and Technology Bureau (2020-3-041-1).The funder subsidized the English language editing servicesand publishing expenses of this paper.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthor Contributions\u003c/strong\u003eLXS was responsible for patient management and was a major contributor in writing the manuscript. HSQ contributed to data collection, data analysis and bibliography retrieval. CXG contributed to case conception, revising for important intellectual content and final approval ofreport, supervising surgeon. All authors read and approved the final manuscript.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgements \u0026nbsp;\u003c/strong\u003eNot applicable\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e \u003cli\u003e\u003cspan\u003eKuo KT, Hsu WH, Wu YC, Huang MH, Li WY. Sclerosing hemangioma of the lung: an analysis of 44 cases. J Chin Med Assoc. 2003;66(1):33\u0026ndash;8.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLiebow AA, Hubbell DS. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer. 1956;9:53\u0026ndash;75.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eTravis WD, Brambilla E, Nicholson AG, Yatabe Y, Austin JHM, Beasley MB, Chirieac LR, Dacic S, Duhig E, Flieder DB, et al. The 2015 World Health Organization Classification of Lung Tumors: Impact of Genetic, Clinical and Radiologic Advances Since the 2004 Classification. J Thorac Oncol. 2015;10:1243\u0026ndash;60.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDevouassoux-Shisheboran M, Hayashi T, Linnoila RI, Koss MN, Travis WD. 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. 2000;24(7):906\u0026ndash;16.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eShin SY, Kim MY, Oh SY, Lee HJ, Hong SA, Jang SJ, et al. Pulmonary sclerosing pneumocytoma of the lung: CT characteristics in a large series of a tertiary referral center. Med (Baltim). 2015;94(4):e498.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGal AA, Nassar VH, Miller JI. Cytopathologic diagnosis of pulmonary sclerosing hemangioma. Diagn Cytopathol. 2002;26:163\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eTrabucco SMR, Brascia D, Cazzato G, De Iaco G, Colagrande A, Signore F, Ingravallo G, Resta L, Marulli G. Pulmonary Sclerosing Pneumocytoma: A Pre and Intraoperative Diagnostic Challenge. Report of Two Cases and Review of the Literature. Medicina (Kaunas). 2021 May 23;57(6):524.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSchiergens TS, Khalil PN, Mayr D, et al. Pulmonary sclerosing hemangioma in a 21-year-old male with metastatic hereditary non-polyposis colorectal cancer: report of a case. World J Surg Oncol. 2011;9:62. doi:\u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.1186/1477-7819-9-62\u003c/span\u003e\u003c/span\u003e. Published 2011 Jun 6.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eHosaka N, Sasaki T, Adachi K, et al. Pulmonary sclerosing hemangioma associated with familial adenomatous polyposis. Hum Pathol. 2004;35(6):764\u0026ndash;8. doi:\u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.1016/j.humpath.2004.02.003\u003c/span\u003e\u003c/span\u003e.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLongo R, Carillio G, Torrisi A, et al. An unusual case of three synchronous tumors in a young woman. Tumori. 2005;91(3):267\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eRizzo S, Pandolfi U, Villani L, et al. Pulmonary sclerosing hemangioma in a woman with multiple uterine fibromas, cysts to thyroid and kidney. Anticancer Res. 1996;16(3A):1297\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eAubry MC, Myers JL, Colby TV, Leslie KO, Tazelaar HD. Endometrial stromal sarcoma metastatic to the lung: a detailed analysis of 16 patients. Am J Surg Pathol. 2002;26(4):440\u0026ndash;449. 13. Wang Y, He Q, Shi W, Wang J, Ji H. A mixture of carcinoid tumors, extensive neuroendocrine proliferation, and multiple pulmonary sclerosing hemangiomas. World J Surg Oncol. 2014;12:209. Published 2014 Jul 15.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eShang Z, Han Y, Shao J, Zhu L, Teng H, Zhang J. Challenging of frozen diagnoses of small sclerosing pneumocytoma. J Clin Pathol. 2021 Mar;29:jclinpath\u0026ndash;2020.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLiu W, Tian XY, Li Y, Zhao Y, Li B, Li Z. Coexistence of pulmonary sclerosing hemangioma and primary adenocarcinoma in the same nodule of lung. Diagn Pathol. 2011;6:41. Published 2011 May 20.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSuzuki K, Shiono S, Kato H, Yanagawa N, Sato T. Kyobu Geka. Small sclerosing hemangioma combined with primary lung cancer; report of a case. Thorac Cardiovasc Surg. 2006;59(7):590\u0026ndash;3.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eChoi KH, Baek HA, Park HS, et al. Sclerosing hemangioma, presenting as a pneumonic pattern with mucinous adenomatous hyperplasia of the lung. Pathol Int. 2008;58(11):735\u0026ndash;40.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eKitawaki Y, Fujishima F, Taniuchi S, et al. Coexistence of glandular papilloma and sclerosing pneumocytoma in the bronchiole. Pathol Int. 2018;68(7):425\u0026ndash;30.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLuLu X, Jian S. Concomitance of pulmonary spindle cell carcinoma and sclerosing pneumocytoma in a woman: A case report. Med (Baltim). 2019;98(51):e18416.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eWang X, Zhang L, Wang Y, Jia X, Wang J, Zhang H. Sclerosing pneumocytoma with metastasis to the mediastinal and regional lymph nodes. Indian J Pathol Microbiol. 2018;61(3):407\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSuzuki H, Saitoh Y, Koh E, et al. Pulmonary sclerosing hemangioma with pleural dissemination: report of a case. Surg Today. 2011;41(2):258\u0026ndash;61.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eYano M, Yamakawa Y, Kiriyama M, Hara M, Murase T. Sclerosing hemangioma with metastases to multiple nodal stations. Ann Thorac Surg. 2002;73(3):981\u0026ndash;3.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eAdachi Y, Tsuta K, Hirano R, Tanaka J, Minamino K, Shimo T, et al. Pulmonary sclerosing hemangioma with lymph nodemetastasis: a case report and literature review. Oncol Lett. 2014;7:997\u0026ndash;1000.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"Pulmonary nodule, Pulmonary sclerosing pneumocytoma, Lung adenocarcinoma, Lung surgery, Multiple","lastPublishedDoi":"10.21203/rs.3.rs-686381/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-686381/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eBackground:\u003c/strong\u003e Pulmonary sclerosing pneumocytoma (PSP) is a relatively rare benign tumor, and multiple pulmonary sclerosing pneumocytomas are even rarer.\u0026nbsp;PSP is usually seen in adults over 50 years old, featuring a female to male ratio of 5:1. We report an extremely rare case of a 20-year-old male patient with bilateral multiple pulmonary sclerosing pneumocytoma. This rare benign disease also presents diagnostic challenges throughout the treatment process.\u003c/p\u003e\u003cp\u003e\u003cstrong\u003eCase presentation:\u003c/strong\u003e A 20-year-old male was diagnosed with bilateral multiple pulmonary nodules on chest CT. The patient underwent regular chest CT reexamination over the course of six months, which indicated a tendency of slow enlargement of pulmonary nodules. After complete examination and exclusion of tumors in other parts of the body, he underwent two separate surgeries, and bilateral multiple nodules were removed. Intraoperative frozen section analysis suggested that some nodules were adenocarcinoma and the remaining nodules were considered benign tumors. Postoperative immunohistochemistry indicated that all nodules were pulmonary sclerosing pneumocytomas. \u003c/p\u003e\u003cp\u003e\u003cstrong\u003eConclusions: \u003c/strong\u003eBilateral multiple pulmonary sclerosing pneumocytoma are rare in young men. In clinical practice, the differentiation of this disease from malignancy is challenging, and surgical treatment is a safe option for pulmonary nodules with a tendency to become larger.\u003c/p\u003e","manuscriptTitle":"Bilateral Multiple Pulmonary Sclerosing Pneumocytomain A Young Male: Case Report","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2021-08-04 14:41:15","doi":"10.21203/rs.3.rs-686381/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"fbe5f49d-f816-4cd9-af4d-0f627923973c","owner":[],"postedDate":"August 4th, 2021","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[{"id":6201880,"name":"Surgery"},{"id":6201881,"name":"Oncology"}],"tags":[],"updatedAt":"2021-08-04T14:41:16+00:00","versionOfRecord":[],"versionCreatedAt":"2021-08-04 14:41:15","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-686381","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-686381","identity":"rs-686381","version":["v1"]},"buildId":"_2-kVJe1T_tPrBINL-cwx","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}