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Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome

BACKGROUND: Kartagener syndrome, an autosomal recessive disorder with a triad of chronic sinusitis, bronchiectasis, and situs inversus, is characterized by recurrent respiratory tract infections and chronic inflammation of the lung. Information on comorbidities other than infections in patients with...

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Autores principales: Nagai, Yoshiaki, Koyama, Nobuyuki, Iwai, Yuki, Tsubochi, Hiroyoshi, Hiruta, Masahiro, Mizushina, Yoshiko, Koyama, Shinichiro, Ishikawa, Yuichi, Hagiwara, Koichi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7161191/
https://www.ncbi.nlm.nih.gov/pubmed/32299405
http://dx.doi.org/10.1186/s12890-020-1133-y
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author Nagai, Yoshiaki
Koyama, Nobuyuki
Iwai, Yuki
Tsubochi, Hiroyoshi
Hiruta, Masahiro
Mizushina, Yoshiko
Koyama, Shinichiro
Ishikawa, Yuichi
Hagiwara, Koichi
author_facet Nagai, Yoshiaki
Koyama, Nobuyuki
Iwai, Yuki
Tsubochi, Hiroyoshi
Hiruta, Masahiro
Mizushina, Yoshiko
Koyama, Shinichiro
Ishikawa, Yuichi
Hagiwara, Koichi
author_sort Nagai, Yoshiaki
collection PubMed
description BACKGROUND: Kartagener syndrome, an autosomal recessive disorder with a triad of chronic sinusitis, bronchiectasis, and situs inversus, is characterized by recurrent respiratory tract infections and chronic inflammation of the lung. Information on comorbidities other than infections in patients with Kartagener syndrome is currently limited. CASE PRESENTATION: A 39-year-old, non-smoking female was diagnosed with Kartagener syndrome and admitted to Saitama Medical Center, Jichi Medical University, Japan. Computed tomography revealed an endobronchial massive shadow at the ostial site of the right upper lobe bronchus with atelectasis of the right upper lobe. The mass was surgically resected and pathologically diagnosed as mucoepidermoid carcinoma. The lesion had no vascular invasions and no metastases to the lungs or lymph nodes. The surgical margin was negative for carcinoma. Following surgery, the patient has been in good condition. CONCLUSIONS: The present case showed different clinicopathological characteristics from those previously reported in cases of Kartagener syndrome complicated by carcinoma. To the best of our knowledge, this is the first reported case of a young, non-smoking female with comorbid Kartagener syndrome and pulmonary mucoepidermoid carcinoma. This case report may provide a new perspective on the complications of Kartagener syndrome.
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spelling pubmed-71611912020-04-22 Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome Nagai, Yoshiaki Koyama, Nobuyuki Iwai, Yuki Tsubochi, Hiroyoshi Hiruta, Masahiro Mizushina, Yoshiko Koyama, Shinichiro Ishikawa, Yuichi Hagiwara, Koichi BMC Pulm Med Case Report BACKGROUND: Kartagener syndrome, an autosomal recessive disorder with a triad of chronic sinusitis, bronchiectasis, and situs inversus, is characterized by recurrent respiratory tract infections and chronic inflammation of the lung. Information on comorbidities other than infections in patients with Kartagener syndrome is currently limited. CASE PRESENTATION: A 39-year-old, non-smoking female was diagnosed with Kartagener syndrome and admitted to Saitama Medical Center, Jichi Medical University, Japan. Computed tomography revealed an endobronchial massive shadow at the ostial site of the right upper lobe bronchus with atelectasis of the right upper lobe. The mass was surgically resected and pathologically diagnosed as mucoepidermoid carcinoma. The lesion had no vascular invasions and no metastases to the lungs or lymph nodes. The surgical margin was negative for carcinoma. Following surgery, the patient has been in good condition. CONCLUSIONS: The present case showed different clinicopathological characteristics from those previously reported in cases of Kartagener syndrome complicated by carcinoma. To the best of our knowledge, this is the first reported case of a young, non-smoking female with comorbid Kartagener syndrome and pulmonary mucoepidermoid carcinoma. This case report may provide a new perspective on the complications of Kartagener syndrome. BioMed Central 2020-04-16 /pmc/articles/PMC7161191/ /pubmed/32299405 http://dx.doi.org/10.1186/s12890-020-1133-y Text en © The Author(s). 2020 Open AccessThis 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/. The Creative Commons Public Domain Dedication waiver (http://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
Nagai, Yoshiaki
Koyama, Nobuyuki
Iwai, Yuki
Tsubochi, Hiroyoshi
Hiruta, Masahiro
Mizushina, Yoshiko
Koyama, Shinichiro
Ishikawa, Yuichi
Hagiwara, Koichi
Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome
title Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome
title_full Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome
title_fullStr Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome
title_full_unstemmed Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome
title_short Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome
title_sort pulmonary mucoepidermoid carcinoma arising in a patient with kartagener syndrome
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7161191/
https://www.ncbi.nlm.nih.gov/pubmed/32299405
http://dx.doi.org/10.1186/s12890-020-1133-y
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