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Published in: BMC Pulmonary Medicine 1/2020

01-12-2020 | Lung Cancer | Case report

Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome

Authors: Yoshiaki Nagai, Nobuyuki Koyama, Yuki Iwai, Hiroyoshi Tsubochi, Masahiro Hiruta, Yoshiko Mizushina, Shinichiro Koyama, Yuichi Ishikawa, Koichi Hagiwara

Published in: BMC Pulmonary Medicine | Issue 1/2020

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Abstract

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.
Literature
1.
go back to reference Greenstone M, Rutman A, Dewar A, Mackay I, Cole PJ. Primary ciliary dyskinesia: cytological and clinical features. Q J Med. 1988;67(253):405–23.PubMed Greenstone M, Rutman A, Dewar A, Mackay I, Cole PJ. Primary ciliary dyskinesia: cytological and clinical features. Q J Med. 1988;67(253):405–23.PubMed
2.
go back to reference Rott HD. Kartagener's syndrome and the syndrome of immotile cilia. Hum Genet. 1979;46(3):249–61.CrossRef Rott HD. Kartagener's syndrome and the syndrome of immotile cilia. Hum Genet. 1979;46(3):249–61.CrossRef
3.
go back to reference Afzelius BA. A human syndrome caused by immotile cilia. Science. 1976;193(4250):317–9.CrossRef Afzelius BA. A human syndrome caused by immotile cilia. Science. 1976;193(4250):317–9.CrossRef
5.
go back to reference Leonardi HK, Jung-Legg Y, Legg MA, Neptune WB. Tracheobronchial mucoepidermoid carcinoma. Clinicopathological features and results of treatment. J Thorac Cardiovasc Surg. 1978;76(4):431–8.CrossRef Leonardi HK, Jung-Legg Y, Legg MA, Neptune WB. Tracheobronchial mucoepidermoid carcinoma. Clinicopathological features and results of treatment. J Thorac Cardiovasc Surg. 1978;76(4):431–8.CrossRef
6.
go back to reference Yousem SA, Hochholzer L. Mucoepidermoid tumors of the lung. Cancer. 1987;60(6):1346–52.CrossRef Yousem SA, Hochholzer L. Mucoepidermoid tumors of the lung. Cancer. 1987;60(6):1346–52.CrossRef
7.
go back to reference Behboudi A, Enlund F, Winnes M, Andren Y, Nordkvist A, Leivo I, Flaberg E, Szekely L, Makitie A, Grenman R, et al. Molecular classification of mucoepidermoid carcinomas-prognostic significance of the MECT1-MAML2 fusion oncogene. Genes Chromosomes Cancer. 2006;45(5):470–81.CrossRef Behboudi A, Enlund F, Winnes M, Andren Y, Nordkvist A, Leivo I, Flaberg E, Szekely L, Makitie A, Grenman R, et al. Molecular classification of mucoepidermoid carcinomas-prognostic significance of the MECT1-MAML2 fusion oncogene. Genes Chromosomes Cancer. 2006;45(5):470–81.CrossRef
8.
go back to reference Baruah BD, Chari MV. Kartagener's syndrome with bronchogenic carcinoma. J Indian Med Assoc. 1952;21(10):438–9.PubMed Baruah BD, Chari MV. Kartagener's syndrome with bronchogenic carcinoma. J Indian Med Assoc. 1952;21(10):438–9.PubMed
9.
go back to reference Kawanishi M, Matsushima T, Yagi S, Y N, Soejima R. A case report of epidermoid carcinoma of the lung considered recurrent pneumonia in Kartagener's Syndrome. Kawasaki Med J. 1980;6:247–51. Kawanishi M, Matsushima T, Yagi S, Y N, Soejima R. A case report of epidermoid carcinoma of the lung considered recurrent pneumonia in Kartagener's Syndrome. Kawasaki Med J. 1980;6:247–51.
10.
go back to reference Hachiya T, Koizumi T, Hayasaka M, Honda T, Kubo K, Sekiguchi M, Mochizuki I. A case of Kartagener's syndrome with lung Cancer. J Jpn Soc Resp Endoscopy. 1993;15:179–84. Hachiya T, Koizumi T, Hayasaka M, Honda T, Kubo K, Sekiguchi M, Mochizuki I. A case of Kartagener's syndrome with lung Cancer. J Jpn Soc Resp Endoscopy. 1993;15:179–84.
11.
go back to reference Higashi Y, Sanuki K, Agari M, Nasuno Y, Yokoyama T, Gondou T. A case of small cell lung Cancer accompaning SIADH and Kartagener syndrome. Jpn J Chest Dis. 1994;53:45–8. Higashi Y, Sanuki K, Agari M, Nasuno Y, Yokoyama T, Gondou T. A case of small cell lung Cancer accompaning SIADH and Kartagener syndrome. Jpn J Chest Dis. 1994;53:45–8.
12.
go back to reference Horie M, Arai H, Noguchi S, Suzuki M, Sakamoto Y, Oka T. Kartagener syndrome with lung cancer and mediastinal tumor. Nihon Kokyuki Gakkai Zasshi. 2010;48(5):375–8.PubMed Horie M, Arai H, Noguchi S, Suzuki M, Sakamoto Y, Oka T. Kartagener syndrome with lung cancer and mediastinal tumor. Nihon Kokyuki Gakkai Zasshi. 2010;48(5):375–8.PubMed
13.
go back to reference Inoue Y, Suga A, Sekido Y, Yamada S, Iwazaki M. A case of surgically resected lung cancer in a patient with Kartagener's syndrome. Tokai J Exp Clin Med. 2011;36(2):21–4.PubMed Inoue Y, Suga A, Sekido Y, Yamada S, Iwazaki M. A case of surgically resected lung cancer in a patient with Kartagener's syndrome. Tokai J Exp Clin Med. 2011;36(2):21–4.PubMed
14.
go back to reference Papi A, Casoni G, Caramori G, Guzzinati I, Boschetto P, Ravenna F, Calia N, Petruzzelli S, Corbetta L, Cavallesco G, et al. COPD increases the risk of squamous histological subtype in smokers who develop non-small cell lung carcinoma. Thorax. 2004;59(8):679–81.CrossRef Papi A, Casoni G, Caramori G, Guzzinati I, Boschetto P, Ravenna F, Calia N, Petruzzelli S, Corbetta L, Cavallesco G, et al. COPD increases the risk of squamous histological subtype in smokers who develop non-small cell lung carcinoma. Thorax. 2004;59(8):679–81.CrossRef
15.
go back to reference Pandya H, Matthews S. Case report: Mucoepidermoid carcinoma in a patient with congenital agenesis of the left upper lobe. Br J Radiol. 2003;76(905):339–42.CrossRef Pandya H, Matthews S. Case report: Mucoepidermoid carcinoma in a patient with congenital agenesis of the left upper lobe. Br J Radiol. 2003;76(905):339–42.CrossRef
16.
go back to reference Harini N, Chakravarthy R, Archana L. Congenital pulmonary airway malformation with mucoepidermoid carcinoma: a case report and review of literature. Indian J Pathol Microbiol. 2012;55(4):540–2.CrossRef Harini N, Chakravarthy R, Archana L. Congenital pulmonary airway malformation with mucoepidermoid carcinoma: a case report and review of literature. Indian J Pathol Microbiol. 2012;55(4):540–2.CrossRef
17.
go back to reference Vakkila J, Lotze MT. Inflammation and necrosis promote tumour growth. Nat Rev Immunol. 2004;4(8):641–8.CrossRef Vakkila J, Lotze MT. Inflammation and necrosis promote tumour growth. Nat Rev Immunol. 2004;4(8):641–8.CrossRef
18.
go back to reference Raviv S, Hawkins KA, DeCamp MM Jr, Kalhan R. Lung cancer in chronic obstructive pulmonary disease: enhancing surgical options and outcomes. Am J Respir Crit Care Med. 2011;183(9):1138–46.CrossRef Raviv S, Hawkins KA, DeCamp MM Jr, Kalhan R. Lung cancer in chronic obstructive pulmonary disease: enhancing surgical options and outcomes. Am J Respir Crit Care Med. 2011;183(9):1138–46.CrossRef
19.
go back to reference Chung WS, Lin CL, Hsu WH, Kao CH. Increased risk of lung cancer among patients with bronchiectasis: a nationwide cohort study. QJM. 2016;109(1):17–25.CrossRef Chung WS, Lin CL, Hsu WH, Kao CH. Increased risk of lung cancer among patients with bronchiectasis: a nationwide cohort study. QJM. 2016;109(1):17–25.CrossRef
20.
go back to reference Kim YW, Jin KN, Heo EY, Park SS, Chung HS, Kim DK. The association between combined non-cystic fibrosis bronchiectasis and lung cancer in patients with chronic obstructive lung disease. Int J Chron Obstruct Pulmon Dis. 2015;10:873–9.CrossRef Kim YW, Jin KN, Heo EY, Park SS, Chung HS, Kim DK. The association between combined non-cystic fibrosis bronchiectasis and lung cancer in patients with chronic obstructive lung disease. Int J Chron Obstruct Pulmon Dis. 2015;10:873–9.CrossRef
21.
go back to reference Kim YW, Lee CH, Jin KN, Lee JK, Heo EY, Park SS, Chung HS, Kim DK. The regional association between bronchiectasis and lung cancer in chest CT. BMC Pulm Med. 2016;16(1):151.CrossRef Kim YW, Lee CH, Jin KN, Lee JK, Heo EY, Park SS, Chung HS, Kim DK. The regional association between bronchiectasis and lung cancer in chest CT. BMC Pulm Med. 2016;16(1):151.CrossRef
22.
go back to reference Mak JC, Ho SP, Leung RY, Ho PL, Ooi C, Tipoe GL, Yan C, Ip MS, Lam WK, Tsang KW. Elevated levels of transforming growth factor-beta (1) in serum of patients with stable bronchiectasis. Respir Med. 2005;99(10):1223–8.CrossRef Mak JC, Ho SP, Leung RY, Ho PL, Ooi C, Tipoe GL, Yan C, Ip MS, Lam WK, Tsang KW. Elevated levels of transforming growth factor-beta (1) in serum of patients with stable bronchiectasis. Respir Med. 2005;99(10):1223–8.CrossRef
23.
go back to reference Li Y, Sun Z, Wu Y, Babovic-Vuksanovic D, Li Y, Cunningham JM, Pankratz VS, Yang P. Cystic fibrosis transmembrane conductance regulator gene mutation and lung cancer risk. Lung Cancer. 2010;70(1):14–21.CrossRef Li Y, Sun Z, Wu Y, Babovic-Vuksanovic D, Li Y, Cunningham JM, Pankratz VS, Yang P. Cystic fibrosis transmembrane conductance regulator gene mutation and lung cancer risk. Lung Cancer. 2010;70(1):14–21.CrossRef
Metadata
Title
Pulmonary mucoepidermoid carcinoma arising in a patient with Kartagener syndrome
Authors
Yoshiaki Nagai
Nobuyuki Koyama
Yuki Iwai
Hiroyoshi Tsubochi
Masahiro Hiruta
Yoshiko Mizushina
Shinichiro Koyama
Yuichi Ishikawa
Koichi Hagiwara
Publication date
01-12-2020
Publisher
BioMed Central
Published in
BMC Pulmonary Medicine / Issue 1/2020
Electronic ISSN: 1471-2466
DOI
https://doi.org/10.1186/s12890-020-1133-y

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