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Case Reporthttp://mjiri.iums.ac.ir Medical Journal of the Islamic Republic of Iran (MJIRI)
Iran University of Medical Sciences
_______________________________________________________________________________________________________1. MD, Assistant Professor of Surgery, Department of Surgery Medicine, 22 Bahman Hospital, Mashhad Islamic Azad University, Mashhad,Iran. [email protected]. MD, Pathologist, Department of Pathology, Razavi Hospital, Mashhad, Iran. [email protected]. (Corresponding author) Medical Student, Student Research Committee, Faculty of Medicine, Mashhad Islamic Azad University, Mashhad,Iran. [email protected]
4. Medical Student, Student Research Committee, Faculty of Medicine, Mashhad Islamic Azad University, Mashhad, Iran. [email protected]
Retroperitoneal bronchogenic cyst: a case report
Ali Mirsadeghi1, Farid Farrokhi2, Azadeh Fazli-Shahri3, Bahareh Gholipour4
Received: 4 August 2013 Accepted: 7 December 2013 Published: 13 July 2014
AbstractBronchogenic cysts are among developmental disorders of the primitive foregut which are typically found abovethe diaphragm. Bronchial cysts discovered in the abdominal cavity or retroperitoneum are extremely rare. Wepresent a rare case of a retroperitoneal bronchogenic cyst which was incidentally detected after a wrestling inju-ry in a 23-year-old man who had a negative medical history. Although initial imaging studies suggested an ad-renal tumor, histopathological analysis provided a definite diagnosis of bronchogenic cyst. Though rare, bron-chogenic cysts must be considered in the differential diagnosis of retroperitoneal cystic lesions. This is the firstcase of a retroperitoneal bronchogenic cyst reported in Iran.
Keywords: bronchogenic cyst, retroperitoneum, adrenal.
Cite this article as: Mirsadeghi A, Farrokhi F, Fazli-Shahri A, Gholipour B. Retroperitoneal bronchogenic cyst: a case report.Med J IslamRepub Iran 2014 (13 July). Vol. 28:56.
IntroductionBronchogenic cysts are benign cystic
congenital aberrations resulting from anabnormal budding of the tracheobronchialtree between 26 and 40 days of embryo-genesis. They are usually discovered in thethorax, especially in the mediastinum.Rarely, they can develop below the dia-phragm, and a retroperitoneal position isexceptionally unusual (1-3). To ourknowledge, only 66 cases have been in-dexed in the Pubmed among the Englishliterature (1-4, 7, 8), and there has been noreport in Iran.
Herein, we present a case of bronchogeniccyst occurred in the retroperitoneum in a23-year-old man with a review of relevantliterature.
Case reportA 23-year-old man, who had been injured
during a wrestling competition, was re-ferred to our hospital with an abdominalpain. He had no significant past medicalhistory. On physical examination, he was athin young patient with stable vital signs.He had pain and mild tenderness in left up-per quadrant region. Laboratory tests in-cluding complete blood counts, liver func-tion test (LFT) and blood chemistry werenormal. Anti-hydatic cyst antibody test wasnegative. Erythrocyte sedimentation rate(ESR) was high (62 mm/h) and culture forenterococcus was positive. Abdominalcomputed tomography scan revealed acomplicated cystic lesion with dimensionsof 118 x 130 mm between spleen and theleft kidney and a compression effect intothe kidney. The mass had septa and calcifi-cation in its inferior part. Using contrastenhancement, no abnormal vessel or en-hancement was noted (Fig. 1). Thus the pa-
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tient underwent a semi-elective operation.During the operation, a huge cystic struc-ture was discovered filled with thick,brownish secretions in retroperitoneal area.The cyst was about 20 x 20 x 20 cm in di-ameter and adjacent to spleen, adrenalgland and kidney. Postoperative course wasuneventful; the patient was discharged onday 3, and had remained asymptomatic dur-ing the follow-up period of 4 years.
On gross examination, the specimen con-sisted of an opened creamy brown ovoidcystic lesion, measuring 10 x 8 x 7 cm withirregular surface. On opening internal sur-face was brownish and irregular, wallthickness from 0.2 cm to 1.5 cm. The cystwas submitted for microscopic examina-tion.
Histological sections showed fragmentsof cystic wall, lined by bronchial typepseudostratified cylindrical epithelium rest-ing on fibrovascular connective tissue con-taining extensive foci of mononuclear in-flammatory cells aggregates and presenceof smooth muscle fascicles. There were fo-ci of non-neoplastic adrenal tissue attachedto external surface of the cystic lesion. Asubsequent cytological examination werenegative for malignancy and acute inflam-matory fluid. These findings were con-sistent with a bronchogenic cyst (Fig. 2).
DiscussionBronchogenic cysts are rare primitive
foregut-derived developmental anomalieswith bronchial type pseudostratified cylin-drical epithelium which are usually discov-ered above the diaphragm. Subdiaphrag-matic cysts are extremely rare. Studies haveshown that retroperitoneal bronchogeniccysts usually occur in both sexes in equalratio and a wide age range (1, 3, 4, 8).
Although the exact pathogenesis is stillunknown, in 1985, this hypothesis was putforward by Sumiyoshi et al. (1, 3, 8) that aretroperitoneal bronchial cyst can be result-ed from pinching off and trapping of theabnormal buds of the tracheobronchial treeafter migration in to the abdomen beforefusion of the diaphragm components. Thereis another alternative theory which seemsless reliable and described intraabdominalaberrant budding from the primitive foregut(4).
In most instances, retroperitoneal bron-chogenic cysts have occurred in the left ad-renal gland or the superior body of the pan-creas region; and they were asymptomaticand were discovered incidentally during animaging of the chest (1, 3, 8). There is anincreasing tendency between size of thecyst and age of the patient (5).
There are some differential diagnosis fora retroperitoneal cyst lined by pseudostrati-fied ciliated cylindrical epithelium, such asintra-abdominal cystic teratoma, bron-chopulmonary sequestration, cyst of uro-genital and mullerian origin, and other
Fig. 1. Computed tomographic scan demonstrating asharply marginated mass with septa and calcificationbetween spleen and the left kidney with compressioneffect into the kidney.
Fig. 2. Microscopic section of the bronchogeniccyst showing cystic cavities lined by pseudostrat-ified cylindrical epithelium on a thick fibrouswall containing cartilage, seromucous glands,smooth muscle fascicles and extensive foci ofmononuclear inflammatory cells aggregates(H&E, x 40).
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foregut cysts, in addition to a bronchogeniccyst. Present of secretory respiratory liningepithelium (cuboid or cylindrical ciliatedepithelium) surrounding by smooth musclesimilar to those in normal bronchi, as inthis case, along with presence of cartilage,elastic tissues and seromucous glands, clar-ifies a definite pathological diagnosis ofbronchogenic cyst (1, 4, 8).
Regardless of being disputable, surgicalresection even in asymptomatic cases isrecommended to obtain definitive histolog-ical diagnosis and avoid future probabledevelopment of symptoms and complica-tions, such as infection, hemorrhage, or ne-oplasia within the cyst (1, 6, 7, 8).
Despite the fact that the occurrence ofbronchogenic cysts in the retroperitoneumis extremely rare, and also their preopera-tive diagnosis is so difficult, they must beconsidered in the differential diagnosis ofretroperitoneal cystic lesions.
References1. Govaerts K, Eyken PV, Verswijvel G, Speeten
KV. A bronchogenic cyst, presenting as a retroperitone-al cystic mass. Rare Tumors. 2012 January 2; 4(1): e13.
2. Chu P, Hwang TI, Teng T, Lee C. A retroperitonealbronchogenic cyst successfully treated by laparoscopicsurgery. Ann Saudi Med 2007; 27: 199-200.
3. Sohn K, Kim K, Maeng E. RetroperitonealBronchogenic Cyst: A Case Report. J Korean RadiolSoc 2007; 57: 451-453.
4. Hsieh S, Tseng H, Huang J. RetroperitonealBronchogenic Cyst: A Case Report. Chin Med J(Taipei) 1997; 59: 311-4.
5. Kim KH, Kim JI, Ahn CH, Kim JS, Ku YM,Shin OR, Lee EJ, Lim KW. The First Case of Intra-peritoneal Bronchogenic Cyst in Korea Mimicking aGallbladder Tumor. J Korean Med Sci 2004 June;19(3): 470–473.
6. Jo W, Shin JS, Lee IS. SupradiaphragmaticBronchogenic Cyst Extending Into the Retroperito-neum. Ann Thorac Surg 2006; 81: 369-370.
7. Manz M, Schmeer T, Horcic M, Meier R,Maurer CA. Bronchogenic cyst: a rare cause of aretroperitoneal mass. Zentralbl Chir 2009 Dec;134(6): 570-2.
8. Haddadin WJ, Reid R, Jindal RM. A retroperi-toneal bronchogenic cyst: a rare cause of a mass inthe adrenal region. J Clin Pathol 2001 October;54(10): 801–802.
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