Tuberc Respir Dis.  2014 Sep;77(3):136-140. 10.4046/trd.2014.77.3.136.

Mycobacterium abscessus Lung Disease in a Patient with Kartagener Syndrome

• Kim JH
• Song WJ
• Jun JE
• Ryu DH
• Lee JE
• Jeong HJ
• Jeong SH
• Kang HK
• Kim JS
• Lee H
• Chon HR
• Jeon K
• Kim D
• Kim J
• Koh WJ

• KMID: 2320560
• DOI: http://doi.org/10.4046/trd.2014.77.3.136

Abstract

Primary ciliary dyskinesia (PCD) is characterized by the congenital impairment of mucociliary clearance. When accompanied by situs inversus, chronic sinusitis and bronchiectasis, PCD is known as Kartagener syndrome. The main consequence of impaired ciliary function is a reduced mucus clearance from the lungs, and susceptibility to chronic respiratory infections due to opportunistic pathogens, including nontuberculous mycobacteria (NTM). There has been no report of NTM lung disease combined with Kartagener syndrome in Korea. Here, we report an adult patient with Kartagener syndrome complicated with Mycobacterium abscessus lung disease. A 37-year-old female presented to our hospital with chronic cough and sputum. She was ultimately diagnosed with M. abscessus lung disease and Kartagener syndrome. M. abscessus was repeatedly isolated from sputum specimens collected from the patient, despite prolonged antibiotic treatment. The patient's condition improved and negative sputum culture conversion was achieved after sequential bilateral pulmonary resection.

Keyword

Kartagener Syndrome; Primary Ciliary Dyskinesia; Bronchiectasis; Nontuberculous Mycobacteria; Mycobacterium Infections, Nontuberculous

MeSH Terms

Adult
Bronchiectasis
Cough
Female
Humans
Kartagener Syndrome*
Korea
Lung
Lung Diseases*
Mucociliary Clearance
Mucus
Mycobacterium Infections, Nontuberculous
Mycobacterium*
Nontuberculous Mycobacteria
Respiratory Tract Infections
Sinusitis
Situs Inversus
Sputum

Figure

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  Figure 1 (A) Chest radiography showed dextrocardia and situs inversus. Note the cavitary lesions in the right upper lobe and nodulostreaky opacity, suggesting bronchiectasis in the left middle lung zones. (B) Paranasal sinus radiography showed total opacification involving the bilateral ethmoid sinus and bilateral maxillary sinus.

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  Figure 2 (A, C, E, G) Chest high-resolution computed tomography showed multiple cavities in both upper lobes. Note the severe bronchiectasis in the left middle lobe and the lingular segment of the right upper lobe. (B, D, F, H) Although the cavitary lesion in the right upper lobe improved after 20 months of antibiotic treatment, the size of the multiple cavities in the left upper lobe increased.

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  Figure 3 (A, C, E, G) At 9 months after the left-sided surgery, chest high-resolution computed tomography showed an aggravation of the cavitary lesion in the right upper lobe. (B, D, F, H) At 24 months of the right-sided surgery, follow-up chest high-resolution computed tomography scans indicate no new lesions.

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