Respircase Cilt: 4 - Sayı: 3 Yıl: 2015
Respiratory Case Reports KAYNAKLAR 1. Gaerte SC, Meyer CA, Winer-Muram HT, Tarver RD, Conces DJ Jr. Fat-containing lesions of the chest. Radiographics 2002; 22:61–78. 2. Epler GR, McLoud TC, Munn CS, Colby TV. Pleural lipoma. Diagnosis by computed tomography. Chest 1986; 90:265-8. [CrossRef] 3. Zidane A, Atoini F, Arsalane A, Traibi A, Hammoumi M, Ouariachi F, et al. Parietal pleura lipoma: a rare intrathoracic tumor. Gen Thorac Cardiovasc Surg 2011; 59:363–6. [CrossRef] 4. Politis J, Funahashi A, Gehlsen JA, DeCock D, Stengel BF, Choi H. Intrathoracic lipomas. Report of three cases and review of the literature with emphasis on endobronchial lipoma. J Thorac Cardiovasc Surg 1979; 77:550–6. 5. Granville L, Laga AC, Allen TC, Dishop M, Roggli VL, Churg A, et al. Review and update of uncommon primary pleural pumors: a practical approach to diagnosis. Arch Pathol Lab Med 2005; 129:1428–43. 6. Tateishi U, Gladish GW, Kusumoto M, Hasegawa T, Yokoyama R, Tsuchiya R, et al. Chest wall tumours: radiologic findings and pathologic correlation Part 1. Benign tumours. Radiographics 2003; 23:1477–90. 7. Chen M, Yang J, Zhu L, Zhao H. Intrathoracic giant pleural lipoma: case report and review of the literature. J Cardiothorac Surg 2013; 8:196. [CrossRef] 8. Solsona-Norbon B, Sanchez-Paris O, Bernal-Sprekelsen JC, Puchades-Gil F, Garcia-Vilanova A, Monsalve-Perez J. Hourglass thoracic lipoma of infancy: case report and review of the literature. J Pediatric Surg 1997; 32:785–6. [CrossRef] 9. Müller NL. Imaging of the pleura. Radiology 1993; 186: 297–309. [CrossRef] 10. Sakurai H, Kaji M, Yamazaki K, Suemasu K. Intrathoracic lipomas: their clinicopathological behaviors are not as straightforward as expected. Ann Thorac Surg 2008; 86:261–5. [CrossRef] Cilt - Vol. 4 Sayı - No. 3 194
Respir Case Rep 2015;4(3):195-199 DOI: 10.5505/respircase.2015.43265 OLGU SUNUMU CASE REPORT Ayşe Baççıoğlu 1 , Eylem Yıldırım 1 , Füsun Kalpaklıoğlu 1 , Yasemin Karadeniz Bilgili 2 RESPIRATORY CASE REPORTS Mounier-Kuhn Sendromu (MKS), trakea ve bronşların genişlemesi, tekrarlayan solunum yolu enfeksiyonları ve bronşektazi ile karakterizedir. Burada MKS saptanan iki olgu nadir görülmesi nedeniyle sunuldu. Olguların ortak özellikleri yıllardır zor astım/KOAH tanılarıyla takip edilmeleri, radyolojik incelemelerinde bronşektazi varlığı ve bronş sekresyonlarında nadir görülen bakterilerin üremesiydi. Birinci olgu 43 ve ikinci olgu 63 yaşında kadın olup toraks radyolojisinde trakea ve ana bronş çaplarının ileri derecede geniş olması ile tanı aldılar. Tedavi olarak birinci olguda trakeaya stent konuldu ve semptomları düzelmekle beraber her seferinde stent yerinden çıkıp hemoptiziye yol açınca vazgeçildi. İkinci olguda ise hipoksemisi olmamasına rağmen polistemisi vardı. Her iki olguya tedavi olarak bronkodilatör, mukolitik ve enfeksiyon kontrolü için proflaktik antibiyoterapi ve aşılarla immünizasyon uygulandı. Sonuç olarak MKS, tedaviye dirençli bronkospazmda ayırt edici tanı olarak akla getirilmelidir. Anahtar Sözcükler: Bronşektazi, Mounier-Kuhn Sendromu, trakeabronkomegali. Mounier-Kuhn syndrome (MKS) is a syndrome characterized by the expansion of the trachea-bronchus, recurrent respiratory tract infections, and bronchiectasis. Herein, two cases were presented with MKS, as a rare disease. For years, cases had been followed up as asthma/chronic-obstructive-pulmonary-disease. In the radiological examinations of cases, there were bronchiectasic areas, and the growth of rare bacteria in bronchial secretions. The two women were diagnosed as MKS when they were 43 and 63-yrs-old respectively, with severe enlargement of the tracheal and main bronchus diameter in the thoracic radiology. A tracheal stent was placed in case-1, and although her symptoms were relieved, we stopped trying the procedure because of the recurrent displacement of the stent caused by hemoptysis. Case-2 had polycythemia with no hypoxemia. Both cases were given supportive therapy including bronchodilator, mucolytic, prophylactic antibiotic to control infection and vaccine immunization. As a result MKS should be kept in mind in the distinctive diagnosis of treatment resistant bronchospasm. Key words: Bronchiectasis, Mounier-Kuhn Syndrome, tracheobronchomegaly. 1 Kırıkkale Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları Anabilim Dalı, İmmünoloji ve Allerji Hastalıkları Bilim Dalı, Kırıkkale 2 Kırıkkale Üniversitesi Tıp Fakültesi, Radyoloji Anabilim Dalı, Kırıkkale 1 Department of Chest Diseases, Division of Immunology and Allergic Diseases, Kırıkkale University Faculty of Medicine, Kırıkkale, Turkey 2 Department of Radiology, Kırıkkale University Faculty of Medicine, Kırıkkale, Turkey Başvuru tarihi (Submitted): 11.11.2014 Kabul tarihi (Accepted): 27.01.2015 İletişim (Correspondence): Ayşe Baççıoğlu, Kırıkkale Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları Anabilim Dalı, İmmünoloji ve Allerji Hastalıkları Bilim Dalı, Kırıkkale e-mail: aysebaccioglu@gmail.com 195
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Respir Case Rep <strong>2015</strong>;4(3):195-199 DOI: 10.5505/respircase.<strong>2015</strong>.43265<br />
OLGU SUNUMU<br />
CASE REPORT<br />
Ayşe Baççıoğlu 1 , Eylem <strong>Yıl</strong>dırım 1 , Füsun Kalpaklıoğlu 1 , Yasemin Karadeniz Bilgili 2<br />
RESPIRATORY CASE REPORTS<br />
Mounier-Kuhn Sendromu (MKS), trakea ve bronşların<br />
genişlemesi, tekrarlayan solunum yolu enfeksiyonları<br />
ve bronşektazi ile karakterizedir. Burada MKS saptanan<br />
iki olgu nadir görülmesi nedeniyle sunuldu. Olguların<br />
ortak özellikleri yıllardır zor astım/KOAH<br />
tanılarıyla takip edilmeleri, radyolojik incelemelerinde<br />
bronşektazi varlığı ve bronş sekresyonlarında nadir<br />
görülen bakterilerin üremesiydi. Birinci olgu 43 ve<br />
ikinci olgu 63 yaşında kadın olup toraks radyolojisinde<br />
trakea ve ana bronş çaplarının ileri derecede<br />
geniş olması ile tanı aldılar. Tedavi olarak birinci<br />
olguda trakeaya stent konuldu ve semptomları düzelmekle<br />
beraber her seferinde stent yerinden çıkıp<br />
hemoptiziye yol açınca vazgeçildi. İkinci olguda ise<br />
hipoksemisi olmamasına rağmen polistemisi vardı.<br />
Her iki olguya tedavi olarak bronkodilatör, mukolitik<br />
ve enfeksiyon kontrolü için proflaktik antibiyoterapi ve<br />
aşılarla immünizasyon uygulandı. Sonuç olarak MKS,<br />
tedaviye dirençli bronkospazmda ayırt edici tanı olarak<br />
akla getirilmelidir.<br />
Anahtar Sözcükler: Bronşektazi, Mounier-Kuhn Sendromu,<br />
trakeabronkomegali.<br />
Mounier-Kuhn syndrome (MKS) is a syndrome characterized<br />
by the expansion of the trachea-bronchus,<br />
recurrent respiratory tract infections, and bronchiectasis.<br />
Herein, two cases were presented with MKS, as a<br />
rare disease. For years, cases had been followed up<br />
as asthma/chronic-obstructive-pulmonary-disease. In<br />
the radiological examinations of cases, there were<br />
bronchiectasic areas, and the growth of rare bacteria<br />
in bronchial secretions. The two women were diagnosed<br />
as MKS when they were 43 and 63-yrs-old<br />
respectively, with severe enlargement of the tracheal<br />
and main bronchus diameter in the thoracic radiology.<br />
A tracheal stent was placed in case-1, and although<br />
her symptoms were relieved, we stopped trying<br />
the procedure because of the recurrent displacement<br />
of the stent caused by hemoptysis. Case-2 had polycythemia<br />
with no hypoxemia. Both cases were given<br />
supportive therapy including bronchodilator, mucolytic,<br />
prophylactic antibiotic to control infection and<br />
vaccine immunization. As a result MKS should be<br />
kept in mind in the distinctive diagnosis of treatment<br />
resistant bronchospasm.<br />
Key words: Bronchiectasis, Mounier-Kuhn Syndrome,<br />
tracheobronchomegaly.<br />
1 Kırıkkale Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları Anabilim<br />
Dalı, İmmünoloji ve Allerji Hastalıkları Bilim Dalı, Kırıkkale<br />
2 Kırıkkale Üniversitesi Tıp Fakültesi, Radyoloji Anabilim Dalı, Kırıkkale<br />
1 Department of Chest Diseases, Division of Immunology and<br />
Allergic Diseases, Kırıkkale University Faculty of Medicine,<br />
Kırıkkale, Turkey<br />
2 Department of Radiology, Kırıkkale University Faculty of<br />
Medicine, Kırıkkale, Turkey<br />
Başvuru tarihi (Submitted): 11.11.2014 Kabul tarihi (Accepted): 27.01.<strong>2015</strong><br />
İletişim (Correspondence): Ayşe Baççıoğlu, Kırıkkale Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları Anabilim<br />
Dalı, İmmünoloji ve Allerji Hastalıkları Bilim Dalı, Kırıkkale<br />
e-mail: aysebaccioglu@gmail.com<br />
195
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