Revisiting Pulmonary Sclerosing Pneumocytoma
<p>Axial (<b>A</b>) and coronal (<b>B</b>) CT scan images of a nodule (arrow) in the left upper lobe corresponding to a histologically confirmed pulmonary adenocarcinoma (<b>C</b>) (hematoxylin-eosin, original magnification, ×100).</p> "> Figure 2
<p>(<b>A</b>) Axial CT scan image of a left lower nodule close to the costophrenic angle. (<b>B</b>) Histological panoramic view of a well-delimited solid tumor growing beneath the pleural surface (hematoxylin-eosin, original magnification ×1.5).</p> "> Figure 3
<p>Histological patterns of sclerosing pneumocytoma, including sclerotic (<b>A</b>) and solid (<b>B</b>) (hematoxylin-eosin, original magnification ×100).</p> "> Figure 4
<p>Histological detail of sclerosing pneumocytoma showing stromal and surface cells, and foamy macrophages (<b>A</b>) (hematoxylin-eosin, original magnification, ×240). Immunohistochemical study showing positivity with TTF-1 (<b>B</b>), FLI-1 (<b>C</b>), vimentin (<b>D</b>), and AE1/AE3 cytokeratin (<b>E</b>).</p> "> Figure 5
<p>Low-power view of the neuroendocrine proliferation beneath the bronchial epithelium and the infiltrative. component close to pre-existing blood vessels (<b>A</b>) (hematoxylin-eosin, original magnification, ×40). High-power view of tumor cells showing neuroendocrine features (<b>B</b>) (hematoxylin-eosin, original magnification, ×400) and intense chromogranin immunostaining (<b>C</b>).</p> ">
Abstract
:1. Introduction
2. Material and Methods
3. Clinical Features
4. Histopathological Diversity
5. Immunohistochemical and Molecular Findings
6. Diagnostic Walks on the Tight Rope
7. Prognostic Features of Concern
8. Tumor Associations
9. A Clinical Paradigmatic Example
10. Concluding Remarks
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Conflicts of Interest
References
- Liebow, A.A.; Hubbell, D.S. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer 1956, 9, 53–75. [Google Scholar] [CrossRef] [PubMed]
- Devouassoux-Shisheboran, M.; Hayashi, T.; Linnoila, R.I.; Koss, M.N.; Travis, W.D. 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, 906–916. [Google Scholar] [CrossRef] [PubMed]
- Chen, B.; Gao, J.; Chen, H.; Cao, Y.; He, X.; Zhan, W.; Luo, M.; Zhang, S.; Li, W. Pulmonary sclerosing hemangioma: A unique epithelial neoplasm of the lung (report of 26 cases). World J. Surg. Oncol. 2013, 11, 85. [Google Scholar] [CrossRef] [PubMed]
- Gao, Q.; Zhou, J.; Zheng, Y.; Cui, J.; Teng, X. Clinical and histopathological features of pulmonary sclerosing pneumocytoma with dense spindle stromal cells and lymph node metastasis. Histopathology 2020, 77, 718–727. [Google Scholar] [CrossRef] [PubMed]
- Wang, X.; Ng, C.S.; Shi, X.; Yin, W. Characteristics of metastatic and nonmetastatic pulmonary sclerosing pneumocytomas: A clinicopathological study of 68 cases and 15 reported metastatic cases. Lab. Investig. 2023, 103, 100135. [Google Scholar] [CrossRef] [PubMed]
- Liang, J.; Du, Q.; Ye, X.; Huang, W. Pulmonary sclerosing pneumocytoma containing spindle cells with sarcomatoid features: A case report with literature review. Diagn. Pathol. 2023, 18, 16. [Google Scholar] [CrossRef] [PubMed]
- Shang, Z.; Han, Y.; Shao, J.; Zhu, L.; Teng, H.; Zhang, J. Challenging of frozen diagnoses of small sclerosing pneumocytoma. J. Clin. Pathol. 2021, 74, 730–734. [Google Scholar] [CrossRef] [PubMed]
- Yang, C.H.; Lee, L.Y. Pulmonary sclerosing pneumocytoma remains a diagnostic challenge using frozen sections: A clinicopathological analysis of 59 cases. Histopathology 2018, 72, 500–508. [Google Scholar] [CrossRef] [PubMed]
- Lizarraga Madrigal, D.; Cabrera Charleston, M.; Khan, A.; Eapen, G.; Kalhor, N.; Gan, Q.; Grosu, H. Pulmonary sclerosing pneumocytomas mimicking lung cancer. Cureus 2023, 15, e37395. [Google Scholar] [CrossRef]
- 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. [Google Scholar] [CrossRef]
- Fan, X.; Lin, L.; Wang, J.; Wang, Y.; Feng, A.; Nie, L.; Wu, H.; Meng, F.; Xu, H. Genome profile in an extremely rare case of pulmonary sclerosing pneumocytoma presenting with diffusely-scattered nodules in the right lung. Cancer Biol. Ther. 2018, 19, 13–19. [Google Scholar] [CrossRef] [PubMed]
- Benito-Martinez, E.; Galeano-Valle, F.; González, A.; Edgar, M.A.; Oprea-Ilies, G.; Ioachimescu, A.G.; Pasquel, F.J. Ectopic ACTH syndrome with association of multiple sclerosing pneumocytomas and multiple carcinoid tumorlets. J. Endocr. Soc. 2019, 3, 937–942. [Google Scholar] [CrossRef] [PubMed]
- Pal, P.; Chetty, R. Multiple sclerosing pneumocytomas: A review. J. Clin. Pathol. 2020, 73, 531–534. [Google Scholar] [CrossRef] [PubMed]
- Zhang, W.D.; Shi, Z.E.; Zhang, M.Y.; Yin, Y.H.; Li, H.; Qu, Y.Q. Giant pulmonary sclerosing pneumocytoma with potentially malignant biological behavior: A case report and literature review. Ann. Transl. Med. 2023, 11, 365. [Google Scholar] [CrossRef] [PubMed]
- Iyoda, A.; Hiroshima, K.; Shiba, M.; Haga, Y.; Moriya, Y.; Sekine, Y.; Shibuya, K.; Iizasa, T.; Fujisawa, T. Clinicopathological analysis of pulmonary sclerosing hemangioma. Ann. Thorac. Surg. 2004, 78, 1928–1931. [Google Scholar] [CrossRef] [PubMed]
- Wei, S.; Tian, J.; Song, X.; Chen, Y. Recurrence of pulmonary sclerosing hemangioma. Thorac. Cardiovasc. Surg. 2008, 56, 120–122. [Google Scholar] [CrossRef] [PubMed]
- Kim, M.K.; Jang, S.J.; Kim, Y.H.; Kim, S.W. Bone metastasis in pulmonary sclerosing hemangioma. Korean J. Intern. Med. 2015, 30, 928–930. [Google Scholar] [CrossRef] [PubMed]
- Xu, H.M.; Li, W.H.; Hou, N.; Zhang, S.G.; Li, H.F.; Wang, S.Q.; Yu, Z.Y.; Li, Z.J.; Zeng, M.Y.; Zhu, G.M. Neuroendocrine differentiation in 32 cases of so-called sclerosing hemangioma of the lung: Identified by immunohistochemical and ultrastructural study. Am. J. Surg. Pathol. 1997, 21, 1013–1022. [Google Scholar] [CrossRef]
- Wang, Z.; Yang, M.Q.; Huang, W.J.; Zhang, D.; Xu, H.T. Sclerosing pneumocytoma mixed with a typical carcinoid tumor. A case report and review of literature. Medicine 2019, 98, e14315. [Google Scholar] [CrossRef]
- Cho, H.J.; Lee, J.H.; Lee, G.K.; Hong, E.K.; Kim, H.Y. Case of sclerosing pneumocytoma combined with a typical carcinoid and pulmonary adenocarcinoma in different lobes. Thorac. Cancer 2017, 8, 372–375. [Google Scholar] [CrossRef]
- Cai, L.; Wang, T.; Jiang, L.; Yang, P.; Hu, J.; Jiang, J. Sclerosing pneumocytoma mixed with columnar clear cell adenoma and a typical carcinoid: Case report and review of literature. Int. J. Clin. Exp. Pathol. 2020, 13, 2599–2607. [Google Scholar] [PubMed]
- Mayer, N.; Carboni, G.L.; Thielken, A.; Wagnetz, D. Sclerosing pneumocytoma. A host for a typical carcinoid with pleural metastasis. A wolf in sheep’s clothing. Chest 2021, 159, e1–e5. [Google Scholar] [CrossRef] [PubMed]
- Mlika, M.; Rais, A.; Abdelkbir, A.; Ben Saad, S.; Abdennadher, M.; Baccouche, I.; El Mezni, F. Collision lung tumor associating typical carcinoid tumor to sclerosing hemangioma. Clin. Case Rep. 2022, 10, e06237. [Google Scholar] [CrossRef] [PubMed]
- Boland, J.M.; Lee, H.E.; Barr Fritcher, E.G.; Voss, J.S.; Jessen, E.; Davila, J.I.; Kipp, B.R.; Graham, R.P.; Maleszewski, J.J.; Yi, E.S. Molecular genetic landscape of sclerosing pneumocytomas. Evidence for aberrant mTOR pathway signaling and lack of recurrent translocations. Am. J. Clin. Pathol. 2021, 155, 397–404. [Google Scholar] [CrossRef] [PubMed]
- Yeh, Y.C.; Chu, P.Y.; Lin, S.Y.; Wang, S.Y.; Ho, H.L.; Wang, Y.C. Comprehensive genomic and transcriptomic analysis of sclerosing pneumocytoma. Mod. Pathol. 2024, 37, 100354. [Google Scholar] [CrossRef] [PubMed]
- Zheng, Q.; Zhou, J.; Li, G.; Man, S.; Lin, Z.; Wang, T.; Chen, B.; Lin, F. Pulmonary sclerosing pneumocytoma: Clinical features and prognosis. World J. Surg. Oncol. 2022, 30, 140. [Google Scholar] [CrossRef] [PubMed]
- Zhang, Y.; Ran, C.; Li, W. Central and peripheral pulmonary sclerosing pneumocytomas: Multi-phase CT study and comparison with Ki-67. Radiol. Oncol. 2023, 57, 310–316. [Google Scholar] [CrossRef]
- Shin, S.Y.; Kim, M.Y.; Oh, S.Y.; Lee, H.J.; Hong, S.A.; Jang, S.J.; Kim, S.S. Pulmonary sclerosing pneumocytoma of the lung: CT characteristics in a large series of a tertiary referral center. Medicine 2015, 94, e498. [Google Scholar] [CrossRef] [PubMed]
- Liu, W.; Tian, X.Y.; 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. [Google Scholar] [CrossRef]
- Goto, T.; Maeshima, A.; Kato, R. Microscopic sclerosing hemangioma diagnosed by histopathological examination after lung cancer surgery. Ann. Thorac. Cardiovasc. Surg. 2011, 17, 507–510. [Google Scholar] [CrossRef]
- Lulu, X.; Jian, S. Concomitance of pulmonary spindle cell carcinoma and sclerosing pneumocytoma in a woman. A case report. Medicine 2019, 98, e18416. [Google Scholar] [CrossRef] [PubMed]
- Kay, S.; Still, W.J.; Borochovitz, D. Sclerosing hemangioma of the lung: An endothelial or epithelial neoplasm? Hum. Pathol. 1977, 8, 468–474. [Google Scholar] [CrossRef] [PubMed]
- Katzenstein, A.L.; Weise, D.L.; Fulling, K.; Battifora, H. So-called sclerosing hemangioma of the lung. Evidence for mesothelial origin. Am. J. Surg. Pathol. 1983, 7, 3–14. [Google Scholar] [CrossRef] [PubMed]
- Huszar, M.; Suster, S.; Herczeg, E.; Geiger, B. Sclerosing hemangioma of the lung. Immunohistochemical demonstration of mesenchymal origin using antibodies to tissue-specific intermediate filaments. Cancer 1986, 58, 2422–2427. [Google Scholar] [CrossRef]
- Wang, E.; Lin, D.; Wang, Y.; Wu, G.; Yuan, X. Immunohistochemical and Ultrastructural Markers Suggest Different Origins for Cuboidal and Polygonal Cells in Pulmonary Sclerosing Hemangioma. Hum. Pathol. 2004, 35, 503–508. [Google Scholar] [CrossRef] [PubMed]
- Nagata, N.; Dairaku, M.; Sueishi, K.; Tanaka, K. Sclerosing hemangioma of the lung. An epithelial tumor composed of immunohistochemically heterogenous cells. Am. J. Clin. Pathol. 1987, 88, 552–559. [Google Scholar] [CrossRef] [PubMed]
- Wang, Y.; Wang, E.; Wu, G.; Zhang, Z.; Lin, D. Immunohistochemical and ultrastructural study of so-called sclerosing hemangioma of the lung suggests different origins of cells. Zhongguo Fei Ai Za Zhi 2003, 6, 92–96. [Google Scholar] [CrossRef] [PubMed]
- Baratella, E.; Cernic, S.; Minelli, P.; Furlan, G.; Crimì, F.; Rocco, S.; Ruaro, B.; Cova, M.A. Accuracy of CT-guided core-needle biopsy in diagnosis of thoracic lesions suspicious for primitive malignancy of the lung: A five-year retrospective analysis. Tomography 2022, 8, 2828–2838. [Google Scholar] [CrossRef]
- Tamrazi, A.; Sundaresan, S.; Parvizi, A.; Eller, A.; Fujii, J.; Shaheen, Z.; Campbell, A. Clinical value of CT-guided biopsy of small (1.5 cm) suspicious lung nodules: Diagnostic accuracy, molecular characterization and long-term clinical outcomes. Cancer Treat. Res. Commun. 2022, 33, 100626. [Google Scholar] [CrossRef]
- Li, Y.; Fang, Y.; Huang, Y.Y.; Wei, C. Comparison between computed tomography-guided core and fine needle lung biopsy. A meta-analysis. Medicine 2022, 101, e29016. [Google Scholar] [CrossRef]
- Morikawa, K.; Misumi, S.; Fukuda, T.; Ojiri, H.; Matsudaira, H.; Sato, S. Pulmonary sclerosing pneumocytoma presenting as slow-growing multiple nodules over a long period. Radiol. Case Rep. 2019, 14, 602–607. [Google Scholar] [CrossRef]
- Lee, K.; Sasaki, M.; Kakimoto, T.; Morinaga, S.; Yamamoto, T.; Iwamaru, A.; Ogata, K.; Minematsu, N. Pulmonary sclerosing pneumocytoma demonstrating intratumoral hematoma. Respir. Med. Case Rep. 2019, 28, 100870. [Google Scholar] [CrossRef] [PubMed]
- Le, H.Y.; Pham, D.P.; Nguyen, K.T.; Hoang, V.A.; Trinh, T.S.; Do, Q. Pulmonary sclerosing pneumocytoma in an 18-year-old male patient: A case report and literature review. Medicine 2020, 99, e20869. [Google Scholar] [CrossRef] [PubMed]
- Saripalli, K.R.; Wang, M.Q.W.; Chow, C.Y.; Chew, S.Y. Pulmonary sclerosing pneumocytoma. Approaching a solitary pulmonary nodule and the limitations of risk prediction models. BMJ Case Rep. 2023, 16, e257208. [Google Scholar] [CrossRef]
- Saha, K.; Sit, N.K.; Jash, D.; Maji, A. Diagnosis of sclerosing hemangioma of the lung: Don’t rely on fine-needle aspiration cytology diagnosis alone. J. Cancer Res. Ther. 2013, 9, 748–750. [Google Scholar] [CrossRef] [PubMed]
- Dettrick, A.; Meikle, A.; Fong, K.M. Fine-needle aspiration diagnosis of sclerosing hemangioma (pneumocytoma): Report of a case and review of the literature. Diagn. Cytopathol. 2014, 42, 242–246. [Google Scholar] [CrossRef] [PubMed]
- Onorati, M.; Nicola, M.; Bianchi, C.L.; Bini, F.; Bellaviti, N.; Di Nuovo, F. Pitfalls and key features of a case of sclerosing pneumocytoma: A cytological challenge? Acta Cytol. 2016, 60, 85–88. [Google Scholar] [CrossRef] [PubMed]
- Hissong, E.; Rao, R. Pneumocytoma (sclerosing hemangioma), a potential pitfall. Diagn. Cytopathol. 2017, 45, 744–749. [Google Scholar] [CrossRef]
- Kosmas, K.; Vamvakaris, I.; Psychogiou, E.; Megas, P. Pulmonary sclerosing pneumocytoma mimicking malignancy in endobronchial ultrasound-guided transbronchial needle aspiration: A case report. Cytology 2021, 32, 356–359. [Google Scholar] [CrossRef]
- Chan, A.C.L.; Chan, J.K.C. Can pulmonary sclerosing haemangioma be accurately diagnosed by intra-operative frozen section? Histopathology 2002, 41, 392–403. [Google Scholar] [CrossRef]
- Teng, X.; Teng, X. First report of pulmonary sclerosing pneumocytoma with malignant transformation in both cuboidal surface cells and stromal round cells: A case report. BMC Cancer 2019, 19, 1154. [Google Scholar] [CrossRef]
- Wang, Q.; Lu, C.; Jiang, M.; Li, M.; Yang, X.; Zhang, L.; He, Y.; Mao, C.; Fu, P.; Yang, Y.; et al. Case report and literature review: Pulmonary sclerosing pneumocytoma with multiple metastases harboring AKT1 E17K somatic mutation and TP53 C176Y germline mutation. Front. Med. 2021, 8, 655574. [Google Scholar] [CrossRef]
- Suzuki, H.; Saitoh, Y.; Koh, E.; Hoshino, H.; Kase, D.; Kasei, Y.; Azuhata, Y.; Kishi, H.; Hiroshima, K.; Sekine, Y. Pulmonary sclerosing hemangioma with pleural dissemination: Report of a case. Surg. Today 2011, 41, 258–261. [Google Scholar] [CrossRef]
- Zhang, W.; Liu, Y.; Chai, Y.; Shi, K.; Chen, J.; Shi, D.; Wu, X. Case report: Rare pulmonary sclerosing pneumocytoma: Large, multiple, metastatic, and fatal. Front. Med. 2021, 8, 661032. [Google Scholar] [CrossRef] [PubMed]
- Nassar, A.A.; Jaroszewski, D.E.; Helmers, R.A.; Colby, T.V.; Patel, B.M.; Mookadam, F. Diffuse idiopathic pulmonary neuroendocrine cell hyperplasia. A systematic overview. Am. J. Respir. Crit. Care Med. 2011, 184, 8–16. [Google Scholar] [CrossRef] [PubMed]
Architecture | Solid Sclerotic
Hemorrhagic
|
Cell types | Stromal cells (round/polygonal) Cuboidal cells (surface)
|
Immunoprofile | Positive
|
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Manini, C.; Vezzini, S.; Conte, A.; Sciacca, G.; Infantino, A.; Santos-Pereira, P.; López, J.I. Revisiting Pulmonary Sclerosing Pneumocytoma. Clin. Pract. 2024, 14, 1440-1450. https://doi.org/10.3390/clinpract14040116
Manini C, Vezzini S, Conte A, Sciacca G, Infantino A, Santos-Pereira P, López JI. Revisiting Pulmonary Sclerosing Pneumocytoma. Clinics and Practice. 2024; 14(4):1440-1450. https://doi.org/10.3390/clinpract14040116
Chicago/Turabian StyleManini, Claudia, Simone Vezzini, Antonella Conte, Giuseppe Sciacca, Alessandro Infantino, Poliana Santos-Pereira, and José I. López. 2024. "Revisiting Pulmonary Sclerosing Pneumocytoma" Clinics and Practice 14, no. 4: 1440-1450. https://doi.org/10.3390/clinpract14040116