We experienced two bronchogenic cysts with severe adhesion to the trachea. Both cysts were located on the right side of the trachea. The surgical procedure was changed to thoracotomy from thoracoscopic surgery due to strong adhesion to the trachea. In case 1, sharp separation of the cystic wall from the trachea led to tracheal wall damage. In case 2, the cystic wall except a portion which was adhered to the trachea was resected. The inner side of the residual cystic wall underwent dull curettage to remove the epithelial component. There were no postoperative complications in either case

Kenzaki, Koichirou

Kondo, Kazuya

Sakiyama, Shoji

Tangoku, Akira

Toba, Hiroaki

Yoshida, Mitsuteru

Tokushima University Institutional Repository

INTRODUCTIONVideo-assisted thoracoscopic surgery (VATS) iscurrently the first choice of treatment for broncho-genic cyst (BC). Problems associated with cyst ex-cision by VATS include adhesion to and peelingfrom the surrounding mediastinal organs, damageto mediastinal organs, incomplete excision of thecyst, recurrence from residual material, and transi-tion to thoracotomy. Among cases encountered atour facility since 1988, we report 2 recent cases ofbronchogenic cyst in which satisfactory results wereobtained by two different methods of treatment forstrong adhesion to the trachea.CASESCase 1 : A 25-year-old female was referred to ourhospital because of an abnormal shadow on chestX-ray. Chest computed tomography (CT) demon-strated a tumor shadow 2 cm in diameter on theright side of the trachea at the upper level, 3 cmfrom the carina. T2-weighted magnetic resonanceimaging (MRI) demonstrated a high-intensity tu-mor on the right side of the trachea (Figure 1a). Thepatient was strongly suspected as having broncho-genic cyst. She presented no symptoms, and labo-ratory studies revealed no notable findings. Surgerywas performed by thoracoscopy through the 7th in-tercostal space at the midaxillary line. This revealeda whitish tumor 2 cm in diameter which was en-wrapped in the mediastinal pleura at the cephalicportion of the azygous vein and posterior to the su-perior vena cava (SVC) (Figure 2a). Two additionalports were inserted and we started to resect thetumor thoracoscopically. The cystic wall was sepa-rated from adjacent organs using sharp and dull de-tachment procedures, the cystic wall was subse-quently damaged, and a yellow-white, viscous fluidwas released. As the cyst was adhered strongly tothe right side of the trachea (Figure 2b), the surgi-cal procedure was changed to a limited thora-cotomy from thoracoscopic surgery. The sharp sepa-CASE REPORTTwo cases of bronchogenic cyst with severe adhesion tothe tracheaMitsuteru Yoshida, Kazuya Kondo, Hiroaki Toba, Koichirou Kenzaki, Shoji Sakiyama,and Akira TangokuDepartment of Oncological and Regenerative Surgery, Institute of Health Biosciences, The Univer-sity of Tokushima Graduate School, Tokushima, JapanAbstract : We experienced two bronchogenic cysts with severe adhesion to the trachea.Both cysts were located on the right side of the trachea. The surgical procedure was changedto thoracotomy from thoracoscopic surgery due to strong adhesion to the trachea. Incase 1, sharp separation of the cystic wall from the trachea led to tracheal wall damage.In case 2, the cystic wall except a portion which was adhered to the trachea was re-sected. The inner side of the residual cystic wall underwent dull curettage to remove theepithelial component. There were no postoperative complications in either case. J. Med.Invest. 54 : 187-190, February, 2007Keywords : bronchogenic cyst, adhesion, VATSReceived for publication November 20, 2006 ; accepted December19, 2006.Address correspondence and reprint requests to MitsuteruYoshida, Department of Oncological and Regenerative Surgery,Institute of Health Biosciences, The University of TokushimaGraduate School, Kuramoto-cho, Tokushima 770-8503, Japanand Fax : +81-88-633-7143/7144.The Journal of Medical Investigation Vol. 54 2007１８７ration of the cystic wall from the tracheal wall ledto tracheal wall damage (Figure 3a). The damagedsite was sutured with an absorbent thread, and wascovered with the fifth intercostal muscle. Histopa-thological findings demonstrated that the cyst wallcontained cartilage tissue, pseudostratified ciliatedcolumnar epithelium, bronchial glands, and smoothmuscle. A diagnosis of bronchogenic cyst was made.Bronchoscopy on the 18th post operative day re-vealed the suture line at the repair site of the tra-chea (Figure 2c). There were no postoperative com-plications.Case 2 : A 28-year-old female was referred to ourhospital because of an abnormal shadow on chestX-ray. CT demonstrated an irregular tumor 4 cm indiameter on the anterior of the trachea at the levelof the carina. T2-weighted MRI demonstrated a high-intensity tumor (Figure 1b). The patient was stronglysuspected of having bronchogenic cyst. She hadno symptoms, and laboratory studies were devoidof notable findings. Three ports were inserted. Awhitish, 4 cm-diameter tumor enwrapped in medi-astinal pleura was located at the cephalic portion ofthe azygous vein and posterior to the SVC. Althoughtumor resection was started thoracoscopically, thecystic wall was adhered strongly to the trachealwall, as in case 1. The surgical procedure was thuschanged to a limited thoracotomy. The cystic wallexcept a portion which was adhered strongly to thetracheal wall was resected. The inner side of theresidual cystic wall which was adhered to the tra-cheal wall, underwent dull curettage to remove theepithelial component (Figure 3b). Microscopically,the cyst wall contained ciliated epithelium, bronchialglands, and cartilage. A diagnosis of bronchogeniccyst was made. There were no postoperative com-plications.Figure 1. Preoperative MRI image of each case.(a) Case 1 : T2-weighted MRI image demonstrating a high-intensity, cystic tumor 2 cm in diameter approximately 3 cm from thecarina on the right and towards the head.(b) Case 2 : T2-weighted MRI image demonstrating a high-intensity, wavy cystic tumor.Figure 2. Perioperative findings in case 1.(a) A tumor was present in the dorsum of the SVC. It was spherical with a smooth surface.(b) The tumor was adhered powerfully to the right side of the trachea.(c) Bronchoscopy on postoperative hospital Day 18 revealed the formation of granulation tissue at the repair site (right side of thecartilaginous portion of the trachea) , but there were no postoperative complications, and progress was good.M. Yoshida, et al. Bronchogenic cyst with severe adhesion to the trachea１８８DISCUSSIONThe respiratory organs and esophagus have theforegut as their primordium. During gestationalweek 7, this duct lengthens vertically, leading tothe separation of the respiratory system anteriorlyand the pharyngoesophageal system posteriorly.Separation of a portion of these structures will resultin a mediastinal bronchogenic cyst or esophagealcyst. According to 1995 statistics of the JapaneseAssociation for Thoracic Surgery, mediastinal cystscomprise 14.1% of all mediastinal tumors, and thefrequency of bronchogenic cysts is the highestamong mediastinal cystic disease. The sites of occur-rence are the paratrachea, tracheal carina, pulmo-nary hilum, and paraesophagus (1) .The first choice of treatment for bronchogeniccysts is surgical resection. Now, because of the re-cent dissemination of the thoracoscopic procedure,thoracoscopic removal or thoracoscopic-assisted re-moval is the method of first choice (1, 2). A thora-coscopic procedure is of course superior for reduc-ing postoperative pain, the number of inpatient days,and for cosmetic reasons. However, surgeons haveexperienced some cases of bronchogenic cyststrongly adherent to various adjacent organs, as wehave presented.We experienced two bronchogenic cyst cases withsevere adhesion to the trachea among 14 cases ofbronchogenic cysts in our hospital. These two bron-chogenic cysts were located in the right paratra-cheal region, and were adhered strongly to the tra-cheal wall. The cystic wall and tracheal wall were“shared”. In both cases, we first selected a thora-coscopic procedure, but had to convert to limitedthoracotomy because of strong cystic adhesion tothe trachea. In the fist case, we could not separatethe cystic wall from the tracheal wall, and subse-quently damaged the latter. The damaged site wassutured with an absorbent thread, and was cov-ered with the fifth intercostal muscle. In the secondcase, the strongly adhesive portion of the cystic wallwas left attached to the tracheal wall so as not todamage the tracheal wall, and the inner side of theresidual cystic wall underwent dull curettage to re-move the epithelial component. Neither case hadpostoperative complications.Kanemitsu, et al. reported one case of incompleteresected cyst wall because of tight adhesion to thetrachea in 11 mediastinal bronchogenic cyst cases(3). Ribet, et al. also reported one case of injury tothe trachea and 2 cases of incomplete resected cystwall in 43 mediastinal bronchogenic cyst cases (4).Recently, Martinod et al. reported thoracoscopic ex-Figure 3. Methods of treatment(a) Case 1The tracheal wall and cystic wall were shared, and the bronchial wall was consequently damaged during separation and was suturedwith an absorbent thread. The site was then covered with the fifth intercostal muscle.(b) Case 2The cystic wall on the tracheal side was spared, and the interior of the cystic wall underwent dull curettage to remove the epithelialcomponent.The Journal of Medical Investigation Vol. 54 February 2007 １８９cision of 20 mediastinal bronchogenic cysts. In 7patients, they had to convert from thoracoscopy tothoracotomy. Eleven (55%) bronchogenic cysts hadstrong adhesions to vital structures, and a portion ofthe cyst wall was left in place in 5 cases. There wereno late complications and no cyst recurrence (5).We suggest two reasons for severe adhesion tothe trachea. One reason is incomplete separationbetween the cystic wall and the trachea during em-bryogenesis. The other reason is that infection iscaused because a fine fistula is present between thetrachea and the cyst. As a result, acute inflamma-tion is caused and the cyst adheres to the trachea.We think that the reason for adhesion to the tra-chea is the former one because our two cases wereasymptomatic.There are various bronchogenic cysts rangingfrom cases with easy resection using thoracoscopyto cases which are difficult to resect because ofstrong cystic adhesion to the neighboring organs.In these difficult cases, including the 2 presentedhere, conversion to a supplementary limited thora-cotomy is strongly advised. Also, a small patch ofthe cystic wall should be left in place after destruc-tion of the mucosal lining to avoid recurrence.REFERENCES1. Fujimoto T, Yamanaka A, Hirai T : Applicationof thoracoscopic surgery handicraft for medi-astinal cysts. Nippon Kokyuu Gekagakkaishi13 (7)(in Japanese) : 15-19, 19992. Urschel J, Horan TA : Mediastinoscopic treat-ment of mediastinal cysts. Ann Thoracic Surg58 : 1698-1701, 19943. Kanemitsu Y, Nakayama H, Asamura H, KondoH, Tsuchiya R, Naruke T : Clinacal features andmanagement of bronchogenic cysts. Report of17cases. Surgery Today Jpn J Surg 29 : 1201-1205, 19994. Michel ER, Marie CC, Bernard G : Broncho-genic cysts of the mediastinu- m. J Thorac Car-diovasc Surg 109 : 1003-1010, 19955. Martinod E, Pons F, Azorin J, Mouroux J,Dahan M, Faillon JM, Dujon A, Lajos PS,Riquet M, Jancovici R : Thoracoscopic exci-sion of mediastinal bronchogenic cysts : Re-sults in 20 Cases. Ann Thorax Surg 69 : 1525-1528, 2000M. Yoshida, et al. Bronchogenic cyst with severe adhesion to the trachea１９０

Two cases of bronchogenic cyst with severe adhesion to the trachea

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Two cases of bronchogenic cyst with severe adhesion to the trachea

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