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The Recurrent Mediastinal Bronchogenic Cyst: a Benign Tumour with an Invasive Character

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  • University of Tunis El Manar. Faculty of medicine of Tunis

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Background: Bronchogenic cyst (BC) is a congenital cyst, which is frequently observed in the mediastinum. Its recurrence after surgical resection is very rare and mainly caused by the persistence of a part of the primary BC's wall. Objectives: Our aim was to present two cases of recurrent BC that were diagnosed in our Department. Besides, we targeted to perform a mini-review of similar cases reported in the English literature. Results: We describe 2 cases of mediastinal BC which recurred after 14 and 33 years. Both patients presented respiratory symptoms and the diagnosis of recurrent BC was suspected by the radiologic findings and confirmed by the microscopic examination. This phenomenon is very rare and has been initially reported by Craig Miller et al. in 1978. The cyst caused recurrent bronchial obstruction and compression of the superior vena cava and pulmonary artery. Since then, only ten cases have been reported so far (seven in the English literature, two in the Japanese literature and one in the French literature). Conclusion: Both cases reported highlights that despite the invasive nature of the recurrent BC, complete surgical resection must be always tried.
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Current Respiratory Medicine Reviews, 2016, 12, 000-000 1
1573-398X/16 $58.00+.00 © 2016 Bentham Science Publishers
The Recurrent Mediastinal Bronchogenic Cyst: A Benign Tumour with an
Invasive Character: Case Report
Mohamed Sadok Boudaya*,1, Rim Zaimi1, Mouna Mlika2, Adel Maghli1 and Tarek Kilani1
1Department of Thoracic Surgery. Abderrahman Mami Hospital, Ariana, Tunisia
2Department of Pathology. Abderrahman Mami Hospital, Ariana, Tunisia
Abstract: Background: Bronchogenic cyst (BC) is a congenital cyst, which is frequently observed in the mediastinum. Its
recurrence after surgical resection is very rare and mainly caused by the persistence of a part of the primary BC's wall.
Objectives: Our aim was to present two cases of recurrent BC that were diagnosed in our Department. Besides, we
targeted to perform a mini-review of similar cases reported in the English literature.
Results: We describe 2 cases of mediastinal BC which recurred after 14 and 33 years. Both patients presented respiratory
symptoms and the diagnosis of recurrent BC was suspected by the radiologic findings and confirmed by the microscopic
examination. This phenomenon is very rare and has been initially reported by Craig Miller et al. in 1978. The cyst caused
recurrent bronchial obstruction and compression of the superior vena cava and pulmonary artery. Since then, only ten
cases have been reported so far (seven in the English literature, two in the Japanese literature and one in the French
literature).
Conclusion: Both cases reported highlights that despite the invasive nature of the recurrent BC, complete surgical
resection must be always tried.
Keywords: Bronchogenic cyst, recurrence, surgery, diagnosis.
INTRODUCTION
Bronchogenic cyst (BC) is a congenital cyst, which is
frequently observed in the mediastinum. Its recurrence after
surgical resection is very rare and mainly caused by the
persistence of a part of the primary BC's wall.
Our aim was to present two cases of BC that were
diagnosed in our Department and recurred after respectively
14 and 33 years. Besides, we performed a mini-review of
similar cases reported in the English literature.
CASE 1
A 38-year-old patient, who was admitted 14 years ago in
our Department for a sub carinal BC with compression of the
pericardium and pericardial, presented chest pain. The
surgical resection that was performed 14 years ago consisted
in a complete cystectomy extended to a part of the
pericardium and the right vagus nerve via a posterolateral
thoracotomy in the 5th intercostal space. Fig. (1) illustrates
the radiological findings of the first cyst.
The radiological exploration that was performed to assess
the present complaint, showed a cystic formation of
65x35x30 mm in the sub carinal region (Fig. 2). It had a
close contact with the left atrium and the pulmonary artery.
Its appearance on MRI was consistent with a bronchogenic
*Address correspondence to this author at the Department of Thoracic
Surgery. Abderrahman Mami Hospital, Ariana, Tunisia; Tel: 98914532;
Fax: 71710780; E-mail: mohamedsadok.boudaya@gmail.com
Fig. (1). Case #1: CT presentation before the first surgery.
cyst. Surgical resection was performed through a right
postero-lateral thoracotomy. The cyst was intra-pericardial in
its location. It was adherent to the carina, the left stem
bronchus and the right pulmonary artery with no plan of
cleavage. Facing the possibility of a cardiac or a large vessel
wound, circulatory assistance via the femoral vessels was
established. The cyst was embedded in the atrial wall
causing posterior myocardial wounds that were repaired. All
the cystic wall was resected. The pathological examination
confirmed the recurrence of the BC. The patient was
discharged after 9 days and a 4-day-thoracic drainage.
2 Current Respiratory Medicine Reviews, 2016, Vol. 12, No. 2 Boudaya et al.
Fig. (2). Case #1: CT presentation before the second surgery.
CASE 2
An 80-year-old woman with a past medical history of
chronic bronchitis, smoking, hypertension and aortic disease
was admitted in our Department for a recurrent BC. She
presented, 33 years ago, a subcarinal BC which was
incompletely resected through a median sternotomy because
of its adherence to the posterior cardiac wall.
The present symptoms consisted in dyspnea, dysphagia,
vomiting and weight loss.
Ultrasound trans-cardiac chest examination revealed a
cystic mass compressing the left atrium and a pulmonary
arterial hypertension achieving 55 mmHg. CT-scan and MRI
examination features were in favour of a BC measuring 9 x 7
cm (Fig. 3). Gastric-fibroscopy showed a compression of the
esophagus and a 3-centimeter diverticulum of the middle
third of the esophagus. An incomplete surgical resection was
initially performed through a mediastinoscopy. Microscopic
examination concluded to a BC.
Fig. (3). Case #2: CT presentation before the second surgery.
One month later, a second surgical resection via a
posterolateral thoracotomy was performed (Fig. 4). The
remaining cystic wall was incompletely resected because of
the adherence of the cyst to the trunk of the pulmonary
artery, the left atrium and the ascending aorta. The patient
was discharged after 11 days and died after a 27-day-period
because of a massive upper gastrointestinal haemorrhage.
Fig. (4). Case #2: Intraoperative aspect of the recurrent bronchogenic
cyst.
DISCUSSION
Recurrence of BC is mainly due to the persistence of part
of the epithelium lining. This phenomenon is very rare and
has been initially reported by Craig Miller et al. in 1978 [2].
The cyst caused recurrent bronchial obstruction and
compression of the superior vena cava and pulmonary artery.
Since then, only ten cases have been reported so far (seven in
the English literature, two in the Japanese literature and one
in the French literature) [3, 4]. Recurrences were mainly
observed after a follow up period varying between 3 and 25
years. In our second case, the recurrence was discovered
after a 33-year-follow up period which seems to be the
longest period reported in the literature.
The main sites of recurrence are latero-tracheal,
esophageal and para-hiliary [5-7]. The difference in the
recurrence rate between the different sites may be attributed
to the difficult access. Highest risk is observed in intra-
pericardial location. In our two cases, the cyst was directly
adherent to the heart, the atrium and the pulmonary artery
justifying an extra-corporeal circulation. Such a procedure
was reported in one case of the literature because of atrial
adherence [8].
Recurrent Mediastinal Bronchogenic Cyst Current Respiratory Medicine Reviews, 2016, Vol. 12, No. 2 3
The treatment of the BC is still controversial especially
when it is intimately correlated to infection or bleeding (with
or without bronchial fistula) but the mainstay treatment is
surgical resection. A minimally invasive approach may be
proposed either by regular monitoring, or by one or several
fine-needle aspiration. Some authors advocate the abstention
in asymptomatic cysts. According to other authors, complete
surgical resection of BC must be the rule even in incidental
cysts [1, 4, 6]. Surgical resection may be performed through
postero-lateral thoracotomy, standard median sternotomy,
video-thoracoscopy or even mediastinoscopy. In
uncomplicated cases, thoracoscopy has yielded good results
[9]. In case of recurrences, we are frequently facing
extension in the posterior mediastinum, pleural symphysis
tight and fibrosis around the cyst. In these circumstances, the
thoracotomy is the standard way that gives the best exposure.
In conclusion, the invasive nature of the recurrent BC
induces a challenging complete resection that must be
always tried.
CONFLICT OF INTEREST
The authors confirm that this article content has no
conflict of interest.
ACKNOWLEDGEMENTS
Declared none.
REFERENCES
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Presentation and management of bronchogenic cysts in the adult.
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mediastinal bronchogenic cyst. Cause of bronchial obstruction and
compression of superior vena cava and pulmonary artery. Chest
1978; 74: 21820.
[3] Benslimane A, Bellorini M, Funck F, Guillard N, Lefevre T.
Recurrent bronchogenic cyst with rupture into the pericardium. A
case report. Arch mal coeur vaiss 1998; 91: 1187-91.
[4] Hasegawa T, Murayama F, Endo S, Sohara Y. Recurrent
bronchogenic cyst 15 years after incomplete excision. Interact
CardioVasc Thorac Surg 2003; 2: 685-7.
[5] Maier HC. Bronchogenic cysts of the mediastinum. Ann Surg
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bronchogenic cyst-a case report. Nippon Kyobu Geka Gakkai
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[8] Chamberlain MH, Wells FC. Recurrent bronchogenic cyst in a
Jehovah's witness. J Cardiovasc Surg (Torino) 2000; 41: 785-6.
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cysts in adults: a single-center experience. Ann Thorac Surg 2004;
78: 98791.
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Received: January 13, 2016 Revised: February 15, 2016 Accepted: February 24, 2016
ResearchGate has not been able to resolve any citations for this publication.
Article
The recurrence of a benign mediastinal bronchogenic cyst 20 years after partial excision precipitated potentially serious vascular and pulmonary complications. Aggressive total surgical excision should be feasible in the majority of cases. An approach via a median sternotomy offers distinct advantages in certain cases and should be considered. Computerized axial tomographic scanning promises to provide improved definition of mediastinal anatomic features and should be a valuable noninvasive diagnostic method in selected cases.
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Mediastinal bronchogenic cyst is a benign tumor and occupied about 5% of mediastinal tumor in Japan. As a benign tumor, recurrence of bronchogenic cyst is very rare, and we found only 2 cases were reported. We experienced a recurrent bronchogenic cyst found 14 years after first operation. The patient was 56-year-old male, and admitted to our hospital for further evaluation of his abnormal shadow on chest roentogenogram. He had no symptoms. He was operated an bronchogenic cyst for 14 years ago. Chest X-ray, CT, and MRI demonstrated a paratracheal cystic mass which was suspected of a recurrent bronchogenic cyst. An operation was done, and the tumor was resected. A histopathological study showed that the tumor had thin wall and had a ciliated epithelium, then it was diagnosed bronchogenic cyst. The most likely explanation for the recurrence in our patient is an incomplete resection during the initial operation. It is though that only a very small amount of epithelial tissue need remain for recurrence. For prevention of recurrence, complete resection of cyst is necessary, and long-term follow-up is indicated to detect recurrence.
Article
Bronchogenic cysts are congenital anomalies of the bronchial tree that are often asymptomatic at presentation in adults. Management of asymptomatic bronchogenic cyst in this population remains controversial. Eighteen patients with bronchogenic cysts were treated at our institution since 1975. At initial presentation, 10 patients (56 percent) were asymptomatic and 8 (44 percent) were symptomatic. Cough and pain were the most frequent symptoms. Two patients presented with potentially serious complications, one with respiratory distress from airway compression and the other with infection and airway fistulae. Chest radiographs were abnormal but nondiagnostic in 17 out of 18 (94 percent) patients. Chest computerized tomography (CT) scans were abnormal in eight of eight (100 percent) patients, but they confirmed the benign cystic nature in only five of eight (62.5 percent). Overall, considering the use of all imaging modalities and clinical suspicion, bronchogenic cyst was considered in the preoperative differential diagnosis in only 11 of 18 (61 percent) patients. Fifteen of 18 cysts were resected initially. Three of the asymptomatic patients who were followed up initially ultimately required resection because of the development of symptoms. A trend toward increased postoperative complications was noted in patients who were symptomatic at the time of surgery (27 percent vs 14 percent). In conclusion, adult patients with asymptomatic bronchogenic cyst may develop symptoms over time. Symptoms in adults can sometimes be potentially serious. Since a confident preoperative diagnosis is not always possible and because surgical complications may be more common in the symptomatic patient, we recommend surgical resection of all suspected bronchogenic cysts in operable candidates.
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A case of tamponnade due to intrapericardial rupture of a recurrent bronchogenic cyst, presenting as pericarditis, is described. This case is unique because it demonstrates the possibility of rupture of a bronchogenic cyst into the pericardium and by the unusual mode of presentation. It also shows that bronchogenic cysts may recur many years after incomplete ablation. Bronchogenic cysts are benign dysembrioplasic formations characterised by their respiratory epithelial lining. The usual presentation in the adult is by haemorrhage or infection, but our case shows that recurrent pericarditis without an obvious cause may be due to bronchogenic cyst, which should be systematically excluded. The diagnosis suspected after medical imaging (chest X-ray, scanner, magnetic resonance imaging) is confirmed by histology. Total surgical ablation is the treatment of choice and may be curative.
Article
A 26-year-old female practising Jehovah's witness presented with a history of recurrent respiratory infections since childhood and more recently, recurrent cardiac dysrhythmias. A large bronchogenic cyst arising beneath the carina and compressing the bronchi was felt to be responsible. The cyst was adherent to the roof of the left atrium. She initially underwent a partial excision through the standard approach of left thoracotomy. Complete resection via this route was hazardous with real danger of laceration of the atrial wall. Repeat surgery was performed on cardiopulmonary bypass 3 years later for recurrence of symptoms. Complete excision was achieved.
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A case of a 50-year-old male who developed left recurrent laryngeal nerve palsy due to a bronchogenic cyst is presented. The bronchogenic cyst recurred following incomplete excision and multiple attempts at percutaneous aspiration. Recurrent laryngeal nerve palsy is an unusual complication of bronchogenic cysts. This case highlights the need for complete excision of these cysts and the lack of efficacy of cyst aspiration.
Article
Mediastinal bronchogenic cysts are rarely diagnosed in adults, hence surgical experience is limited particularly with regard to video-assisted thoracoscopic surgery. In support of the thoracoscopic approach we report our single-center experience in this rare entity. Between June 1995 and December 2002, a nonselected series of 12 consecutive patients presenting with mediastinal bronchogenic cysts underwent video-assisted thoracoscopic surgery. Six cysts (50%) had been diagnosed 2 to 22 years prior, only three of which became symptomatic. In asymptomatic patients (n = 7) surgery was performed because of increasing cyst size (n = 3), patient's request (n = 3), or suspected metastasis (n = 1). Mediastinal bronchogenic cysts were correctly diagnosed by computed tomography in 83% (10/12) and by magnetic resonance imaging in 100% (9/9). Using a three-trocar technique thoracoscopic surgery was successfully performed in 11 of 12 cases (92%). We noted no signs of acute cyst infection. No serious postoperative complications were observed. In 1 patient conversion to open thoracotomy was necessary due to extensive pleural adhesions. In another case thoracoscopic excision of the cyst wall was incomplete. Patients with thoracoscopic excision were discharged after a median of 5.5 days (range 4 to 14 days). No recurrences or complications were observed during a mean follow-up of 40.5 months. Considering the low conversion and complication rate in our series, video-assisted thoracoscopic surgery should be the primary therapeutic choice among adults with symptomatic mediastinal bronchogenic cysts. Surgical intervention in patients with asymptomatic and uncomplicated cysts appears optional.
Bronchogenic cysts of the mediastinum are rare congenital anomalies that arise early in gestation from abnormal budding of the developing respiratory system. Recommended treatment is surgical resection and incomplete resection may lead to local recurrence more than 10 years later with or without various symptoms. We reported a case and reviewed literatures. A case of a recurrent bronchogenic cyst in a 42-year-old man 15 years after first resection is presented. The patient had a persistent high fever resistant to antibiotic therapy. Magnetic resonance imaging and subsequent thoracotomy revealed a recurrent bronchogenic cyst. This case illustrates that incompletely resected mediastinal bronchogenic cyst may recur with later symptoms. Recommended treatment for bronchogenic cysts is complete surgical excision.
Presentation and management of bronchogenic cysts in the adult Recurrent mediastinal bronchogenic cyst. Cause of bronchial obstruction and compression of superior vena cava and pulmonary artery Recurrent bronchogenic cyst with rupture into the pericardium. A case report
  • Sr Patel
  • Dp Meeker
  • Cv Biscotti
  • Tj Kirby
  • Tw Rice
  • Dc Miller
  • Jp Walter
  • Df Guthaner
  • Jb Mark
  • M Bellorini
  • F Funck
  • N Guillard
  • T Lefevre
  • T Hasegawa
  • F Murayama
  • S Endo
  • Y Sohara
[1] Patel SR, Meeker DP, Biscotti CV, Kirby TJ, Rice TW. Presentation and management of bronchogenic cysts in the adult. Chest 1994; 106: 79-85. [2] Miller DC, Walter JP, Guthaner DF, Mark JB. Recurrent mediastinal bronchogenic cyst. Cause of bronchial obstruction and compression of superior vena cava and pulmonary artery. Chest 1978; 74: 218-20. [3] Benslimane A, Bellorini M, Funck F, Guillard N, Lefevre T. Recurrent bronchogenic cyst with rupture into the pericardium. A case report. Arch mal coeur vaiss 1998; 91: 1187-91. [4] Hasegawa T, Murayama F, Endo S, Sohara Y. Recurrent bronchogenic cyst 15 years after incomplete excision. Interact CardioVasc Thorac Surg 2003; 2: 685-7. [5]