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Bronchial oncocytic carcinoma in an adult: a case report and literature review

BACKGROUND: Lung salivary-type tumors originating from bronchial submucosal glands are rare, only four types of salivary gland-type tumors are listed in 2015 WHO classification of lung tumors. Here, we report a rare case of oncocytic carcinoma (OC) in the right main bronchus. CASE PRESENTATION: A 34...

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Autores principales: Shen, Yi-Fan, Wang, Cheng-Long, Chu, Zhi-Gang, Cao, You-De, Luo, Yan, Zhang, Yang-Li, Cao, Yi-Jia
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10559420/
https://www.ncbi.nlm.nih.gov/pubmed/37803309
http://dx.doi.org/10.1186/s12890-023-02669-0
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author Shen, Yi-Fan
Wang, Cheng-Long
Chu, Zhi-Gang
Cao, You-De
Luo, Yan
Zhang, Yang-Li
Cao, Yi-Jia
author_facet Shen, Yi-Fan
Wang, Cheng-Long
Chu, Zhi-Gang
Cao, You-De
Luo, Yan
Zhang, Yang-Li
Cao, Yi-Jia
author_sort Shen, Yi-Fan
collection PubMed
description BACKGROUND: Lung salivary-type tumors originating from bronchial submucosal glands are rare, only four types of salivary gland-type tumors are listed in 2015 WHO classification of lung tumors. Here, we report a rare case of oncocytic carcinoma (OC) in the right main bronchus. CASE PRESENTATION: A 34-year-old man presented to our hospital with a two-month history of recurrent hemoptysis and with one month of inspiratory dyspnea. Pulmonary function tests showed mild restrictive ventilatory dysfunction and severe diffusion dysfunction. Furthermore, the flow volume loop showed a variable extra-thoracic obstruction. Computed tomography (CT) of the chest revealed that a polypiform nodule of 13 mm in diameter was at the proximal right main bronchus. Testing for purified protein derivative was positive (category 2). The nodule was resected under bronchoscopy. The bronchial aspirate was negative for mycobacterium tuberculosis and tumor cells. The biopsy sample showed a solid and acinar predominant pattern with abundant eosinophilic cytoplasm. The bronchial mucosa was destroyed and replaced by tumor cells. The loose edematous stromal reaction could be seen in a local area. Immunohistochemically, tumor cells were positive for CK, EMA, Vimentin, CD117, CK7, S100, Mammaglobin and SOX10. Only scattered tumor cells were stained by basal cell markers, including CK5/6, P40 and P63. Electron microscopy revealed numerous swelling mitochondria with lacking mitochondrial cristae in tumor cells. Fluorescence in situ hybridization (FISH) testing for MAML2 and ETV6 rearrangement were negative. Next-generation sequencing analysis of 520 genes in the tissue biopsy specimen showed no somatic mutation. The diagnosis of OC was made. Subsequently, the patient underwent a right upper lobectomy with sleeve resection of the main bronchus and lymph dissection. No recurrent evidence was seen during two years of chest CT follow-up. CONCLUSIONS: To our knowledge, this is the first case of primary OC in the bronchus. This patient has no recurrence during two years of follow-up, indicating that primary OC in the bronchus has the same favorable prognosis as in salivary glands. Moreover, complete excision and thorough sampling to know the invasive growth pattern is important to reach the correct diagnosis. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12890-023-02669-0.
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spelling pubmed-105594202023-10-08 Bronchial oncocytic carcinoma in an adult: a case report and literature review Shen, Yi-Fan Wang, Cheng-Long Chu, Zhi-Gang Cao, You-De Luo, Yan Zhang, Yang-Li Cao, Yi-Jia BMC Pulm Med Case Report BACKGROUND: Lung salivary-type tumors originating from bronchial submucosal glands are rare, only four types of salivary gland-type tumors are listed in 2015 WHO classification of lung tumors. Here, we report a rare case of oncocytic carcinoma (OC) in the right main bronchus. CASE PRESENTATION: A 34-year-old man presented to our hospital with a two-month history of recurrent hemoptysis and with one month of inspiratory dyspnea. Pulmonary function tests showed mild restrictive ventilatory dysfunction and severe diffusion dysfunction. Furthermore, the flow volume loop showed a variable extra-thoracic obstruction. Computed tomography (CT) of the chest revealed that a polypiform nodule of 13 mm in diameter was at the proximal right main bronchus. Testing for purified protein derivative was positive (category 2). The nodule was resected under bronchoscopy. The bronchial aspirate was negative for mycobacterium tuberculosis and tumor cells. The biopsy sample showed a solid and acinar predominant pattern with abundant eosinophilic cytoplasm. The bronchial mucosa was destroyed and replaced by tumor cells. The loose edematous stromal reaction could be seen in a local area. Immunohistochemically, tumor cells were positive for CK, EMA, Vimentin, CD117, CK7, S100, Mammaglobin and SOX10. Only scattered tumor cells were stained by basal cell markers, including CK5/6, P40 and P63. Electron microscopy revealed numerous swelling mitochondria with lacking mitochondrial cristae in tumor cells. Fluorescence in situ hybridization (FISH) testing for MAML2 and ETV6 rearrangement were negative. Next-generation sequencing analysis of 520 genes in the tissue biopsy specimen showed no somatic mutation. The diagnosis of OC was made. Subsequently, the patient underwent a right upper lobectomy with sleeve resection of the main bronchus and lymph dissection. No recurrent evidence was seen during two years of chest CT follow-up. CONCLUSIONS: To our knowledge, this is the first case of primary OC in the bronchus. This patient has no recurrence during two years of follow-up, indicating that primary OC in the bronchus has the same favorable prognosis as in salivary glands. Moreover, complete excision and thorough sampling to know the invasive growth pattern is important to reach the correct diagnosis. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12890-023-02669-0. BioMed Central 2023-10-06 /pmc/articles/PMC10559420/ /pubmed/37803309 http://dx.doi.org/10.1186/s12890-023-02669-0 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Shen, Yi-Fan
Wang, Cheng-Long
Chu, Zhi-Gang
Cao, You-De
Luo, Yan
Zhang, Yang-Li
Cao, Yi-Jia
Bronchial oncocytic carcinoma in an adult: a case report and literature review
title Bronchial oncocytic carcinoma in an adult: a case report and literature review
title_full Bronchial oncocytic carcinoma in an adult: a case report and literature review
title_fullStr Bronchial oncocytic carcinoma in an adult: a case report and literature review
title_full_unstemmed Bronchial oncocytic carcinoma in an adult: a case report and literature review
title_short Bronchial oncocytic carcinoma in an adult: a case report and literature review
title_sort bronchial oncocytic carcinoma in an adult: a case report and literature review
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10559420/
https://www.ncbi.nlm.nih.gov/pubmed/37803309
http://dx.doi.org/10.1186/s12890-023-02669-0
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