Should asymptomatic bronchogenic cysts in adults be treated conservatively or with surgery?

Kirmani, Bilal; Kirmani, Basitt; Sogliani, Franco

doi:10.1510/icvts.2010.233114

Abstract

A best evidence topic in cardiac surgery was written according to a structured protocol. The question addressed was whether asymptomatic bronchogenic cysts in adults require surgery or whether they can be adequately managed with conservative treatment or observation only. Altogether more than 310 papers were found using the reported search of which 23 represented the best evidence to answer the clinical question. The authors, journal, date and country of publication, patient group studied, study type, relevant outcomes and results of these papers are tabulated. The papers identified included 683 adult patients with bronchogenic cysts. There was a substantial variation between the papers in the proportion of patients presenting with symptoms (6–79%), and all patients with symptoms were managed surgically. The majority of asymptomatic patients underwent empirical surgery to prevent the development of symptoms, to confirm the diagnosis and to rule out malignant transformation. A total of 74 asymptomatic patients were treated conservatively or had definitive diagnosis or treatment delayed. The longest period of observation was 22 years. In total, 33 (45%) of asymptomatic patients eventually developed symptoms requiring surgery. There was no evidence to suggest that surgery following a cyst-related complication increased the postoperative morbidity or mortality, although it was noted to increase the technical difficulty of the procedure. There were no descriptions of misdiagnosis of malignancy as bronchogenic cyst, but 5 (0.7%) of the 683 cysts studied were found to be associated with malignant cells in the cyst wall. The figures cited, however, represent only symptomatic or incidental presentations. As the prevalence of these otherwise benign entities is not known, the rates of progression to symptoms and associated malignancy may be lower than those described. We would advocate informing asymptomatic patients diagnosed with bronchogenic cyst of the 20% morbidity of surgery whether immediate or delayed, the 45% risk of developing symptoms, some of which may be serious, and the 0.7% risk of malignancy. Should patients opt for conservative management, this can be offered only if close long-term follow-up can be guaranteed.

Review, Bronchogenic cysts, Asymptomatic, Surgery

1. Introduction

A best evidence topic was constructed according to a structured protocol. This is fully described in the ICVTS [1].

2. Three-part question

In [adult patients with asymptomatic bronchogenic cysts] does [surgery] improve [symptom-free survival]?

3. Clinical scenario

An otherwise well patient is found to have a well-circumscribed lesion on routine chest X-ray and a computed tomography (CT)-scan is arranged. The report states that there are characteristic features of a bronchogenic cyst and you decide to discuss the management with your colleagues. You suggest that as the patient is asymptomatic, the patient could potentially be treated conservatively with regular follow-up, but others feel that excision is appropriate. Opinion appears to be divided in the department so you seek evidence to determine best practice.

4. Search strategy

Medline 1950 to May 2010 using PubMed interface.

‘Bronchogenic Cyst’[Mesh] AND (‘Surgical Procedures, Operative’[Mesh] OR ‘surgery’[Subheading] OR ‘Thoracic Surgery, Video-Assisted’[Mesh] OR ‘Thoracic Surgery’[Mesh] OR ‘Video-Assisted Surgery’[Mesh]).

The search was limited to human studies involving adults.

5. Search outcome

Three hundred and ten papers were found using the reported search. From these 23 papers were identified that provided the best evidence to answer the question. These are presented in Table 1 .

Table 1.

Summary of the 20 papers derived from the search strategy

Author, date and country	Patient group	Outcomes	Key results	Comments
Study type
(level of evidence)
Laberge et al.,	Review of literature	Presenting complaint	Dysphagia, dyspnoea,	Highly heterogenous
Semin Pediatr Surg,	regarding asymptomatic		infection, haemoptysis	group including all adult
2005, Canada, [2]	congenital lung		and haemothorax (no	and paediatric congenital
	malformations,		figures of incidence)	lung malformations
Review (2a)	including bronchogenic
	cysts, in a paediatric	Postoperative	Adult postoperative	Two cases of
	population	complications	complications not	malignancy in
			discussed	bronchogenic cysts
	Includes papers on adult			noted in adult patients
	congenital lung	Complications of	One adult patient with
	malformations	conservative	abnormal chest X-ray	Conclusions based on
		management	eventually presented	management of
			10 years later with	paediatric malformations
			dyspnoea and was found	but recommend early
			to have	excision of bronchogenic
			bronchioloalveolar	cysts due to progression
			carcinoma associated	to symptoms, malignant
			with bronchogenic cyst.	potential and
			Another patient had ‘long	complications of
			standing’ history of cyst	symptomatic cysts
			infections and later found
			to have associated
			mesenchymal malignancy

Sarper et al., Tex	22 patients from one	Presenting complaint	45% presented with	Recommended surgical
Heart Inst J, 2003,	centre, over 15 years		severe haemoptysis,	resection of all suspected
Turkey, [3]			pneumothorax and	bronchogenic cysts in
	5.2-year median		pleuritis, oesophageal	operable candidates due
Retrospective	postoperative follow-up		compression, infected	to difficulties in
cohort study (2b)			cyst, or postobstructive	establishing definitive
			pneumonia.	diagnoses and frequency
			82% presented with	of complications
			symptoms of cough,
			pain, dyspnoea,
			dysphagia or infection

		Postoperative	5% (one patient)
		complications	persistent air leak
			No recurrence
			No late sequelae

		Complications of	No patients
		conservative	conservatively managed
		management

Kanemitsu et al.,	17 patients (16 adults	Presenting complaint	29% symptomatic (9%	It is noted that the
Surg Today, 1999,	and one paediatric) from		of mediastinal cysts, 67%	advanced age of some
Japan, [4]	one centre, over		of intrapulmonary cysts)	patients at presentation
	30 years		with cough, sputum,	suggests that these cysts
Retrospective			fever, pain or weight loss	can remain
cohort study (2b)	30-month median			asymptomatic forever.
	postoperative follow-up	Preoperative	69% diagnosed with CT	The authors state that the
		diagnosis	100% diagnosed with	most appropriate
			MRI	treatment for
				asymptomatic cysts is
		Operative findings	41% adhesions, 6%	controversial but due to
			complicated	diagnostic limitations and
				the possibility of
		Postoperative	No postoperative	symptoms arising or
		complications	complications.	malignant transformation
			No recurrence	that surgery is advocated

		Complications of	None in one patient
		incomplete resection	where residual tissue
			ablated with
			electro-cautery

		Complications of	No patients
		conservative	conservatively managed
		management
Cioffi et al., Chest,	27 adults of whom	Presenting complaint	50% chest pain, 13%	Conclude that all patients
1998, Italy, [5]	16 with bronchogenic		cough, 6% epigastric	should have surgical
	cyst from one centre,		pain.	resection for definitive
Retrospective	over 20 years		44% asymptomatic	diagnosis and to
cohort study (2b)			(incidental finding)	minimise complications
	Four-year median			that might arise from
	postoperative follow-up	Preoperative	100% preoperatively	symptomatic cysts
		diagnosis	diagnosed with CT, EUS

		Postoperative	No postoperative
		complications	complications

		Complications of	No patients
		conservative	conservatively managed
		management

Aktogu et al., Eur	31 patients (30 adults	Presenting complaint	19% superior vena cava	Thirteen patients
Respir J, 1996,	and one paediatric) from		syndrome, tracheal	symptomatic at the time
Turkey, [6]	one centre, over		compression,	of surgery had complex
	19 years		pneumothorax, pleurisy	peri-cystic adhesions or
Retrospective			or pneumonia.	fistulisation.
cohort study (2b)	Follow-up 2–10 years		81% cough, infection,	Surgical treatment of
	postoperatively		pain, dyspnoea, anorexia/	asymptomatic cysts is
			weight loss, haemoptysis	recommended to avoid
				potentially
		Preoperative	19% asymptomatic	life-threatening
		diagnosis	(incidental finding).	complications and for
			71% undiagnosed with	definitive diagnosis
			CT

		Postoperative	No recurrence
		complications

		Complications of	Two of six initially
		conservative	asymptomatic patients
		management	either had cyst
			enlargement or became
			symptomatic

Ribet et al., Ann	41 patients from one	Presenting complaint	80% symptomatic	Uncertain what
Thorac Surg, 1996,	centre, including 20		(cough, pain, purulent	proportion of
France, [7]	paediatric cases, over		sputum, haemoptysis and	bronchogenic cysts
	25 years		dyspnoea) in adult life	remain asymptomatic
Retrospective				and long-term prognosis
cohort study (2b)	2.6-year mean	Preoperative	45% undiagnosed	unpredictable.
	postoperative follow-up	diagnosis		Preventative surgery
				recommended
		Postoperative	5% (one patient)
		complications	bronchial fistula.
			No recurrence in 88%
			(remainder lost to
			follow-up).
			No deaths

		Complications of	15% (three patients)
		conservative	followed for 11 months,
		management	five years and unknown
			length of time since
			diagnosis developed no
			symptoms but were
			operated on.
			15% (three patients)
			initially with cough,
			recurrent bronchitis and
			no symptoms
			(respectively) refused
			surgery but lost to
			follow-up


Cuypers et al., Eur	20 adult patients from	Presenting complaint	30% symptomatic	The authors found the
J Cardiothorac	one centre, over		(pneumonia, abscess,	risk of malignancy and
Surg, 1996,	18 years		dysphagia and cough)	cyst-related
Belgium, [8]			70% asymptomatic	complications to be
	No long-term follow-up			justification for operative
Retrospective		Preoperative	25% undiagnosed after	treatment in all cases of
cohort study (2b)		diagnosis	CT, bronchoscopy,	bronchogenic cysts
			barium swallow or echo
			(all cases had CT with
			dense fluid)

		Postoperative	No postoperative
		complications	morbidity; one case
			histologically associated
			with squamous cell
			carcinoma

		Complications of	No patients
		conservative	conservatively managed
		management

Ribet et al., J	69 patients from one	Presenting complaint	63.7% symptomatic	Due to the frequency of
Thorac Cardiovasc	centre, including 24		pain, respiratory tract	late complications with
Surg, 1995, France,	paediatric cases, over		infection, cough,	asymptomatic cysts and
[9]	25 years		dyspnoea, dysphagia,	the unpredictable
			heartburn)	prognosis, preventative
Retrospective	4.2-year mean			surgery was
cohort study (2b)	postoperative follow-up	Preoperative	22% initially	recommended
		diagnosis	misdiagnosed (11% not
			as bronchogenic cyst;
			11% as bronchogenic
			cyst when actually cystic
			neurogenic tumour,
			benign lymphoma and
			haemolymphangioma)

		Postoperative	13.4% postoperative
		complications	morbidity (infection,
			chylothorax and phrenic
			paresis).
			12% symptoms of pain
			or dyspnoea
			postoperative (7%
			symptomatic
			preoperative, 5%
			asymptomatic)

		Complications of	11% refused operation,
		conservative	only 4% followed up
		management	(two patients): one
			remained asymptomatic,
			one died of generalised
			malignancy of unknown
			origin.
			7% had incomplete
			operations but cyst
			remained stable or had no
			recurrence

Patel et al., Chest,	18 adult patients from	Presenting complaint	44% symptomatic	There was no statistical
1994, USA, [10]	one centre, over		(cough, pain) of which	difference in the
	19-year period		11% serious (dyspnea,	frequency of
Retrospective			infection).	intraoperative difficulties
cohort study (2b)	Follow-up 12		56% asymptomatic	or postoperative
	months to 11 years			complications between
	(mean not given)	Preoperative	37.5% undiagnosed with	asymptomatic and
		diagnosis	CT, angio, USS, FNA,	symptomatic patients.
			barium swallow,	The authors concluded,
			bronchoscopy or	however, that surgery is
			mediastinoscopy	advocated in
		Operative difficulties	No difference in	asymptomatic cysts due
			frequency of operative	to the potential for
			difficulties between	complications, incorrect
			symptomatic and	diagnosis or progression
			asymptomatic patients	to symptoms
			(P=0.0656)

		Postoperative	No difference (P=0.596)
		complications	in complication rate
			between preoperatively
			asymptomatic (14%) or
			symptomatic (27%)
			patients. Complications
			included phrenic nerve
			paresis.
			11% delayed
			complications
			(oesophageal stricture
			and recurrence)

		Complications of	Three out of seven
		conservative	asymptomatic patients
		management	followed up developed
			symptoms; four patients
			lost to follow-up

Suen et al., Ann	42 patients from one	Presenting complaint	50% symptomatic (pain,	Complete excision
Thorac Surg,	centre, over 30 years		cough, fever, dysphagia,	recommended in most
1993, USA, [11]			purulent sputum,	cases to relieve
			haemoptysis, dyspnoea).	symptoms, prevent
Retrospective			26% complicated	complications and
cohort study (2b)			(dysphagia, haemorrhage,	confirm diagnosis
			infection and one patient
			with adenocarcinoma in
			cyst)

		Preoperative	59% no preoperative
		diagnosis	diagnosis (more recent
			cases, better success
			rate)

		Postoperative	5% postoperative
		complications	complications (wound
			infection and C. Diff
			colitis).
			No recurrence.
			No deaths

		Complications of	Two patients treated
		conservative	conservatively (one
		management	follow-up only and one
			drainage) with no
			complications

St-Georges et al.,	86 patients from one	Presenting complaint	72% patients	35/86 (41%) of patients
Ann Thorac Surg,	centre, over 20 years		symptomatic by time of	operated on had major
1991, Canada, [12]			surgery (57%	operative difficulties: all
			progressive symptoms	these were patients with
Retrospective			and 15% acute).	symptoms
cohort study (2b)			53% patients with >1	preoperatively
			symptom (chest pain,
			cough, dyspnoea, fever,	Resection recommended
			sputum, anorexia/weight	as majority of patients
			loss, dysphagia,	eventually develop
			haemoptysis).	symptoms or
			38% with complications	complications
			(fistula with airway,
			ulceration cyst wall,
			haemorrhage, infection,
			bronchial atresia)
		Preoperative	57% patients presumed
		diagnosis	diagnosis at surgery after
			CT and angiography.
			Positive diagnosis never
			made preoperatively

		Operative findings	41% had complicated
			cysts (fistula, ulcer,
			haemorrhage, infection,
			atresia) at operation

		Postoperative	11% intraoperative
		complications	complications (vagal
			trunk division, segmental
			bronchus laceration,
			oesophageal mucosal
			laceration).
			9% postoperative
			complications
			(atelectasis, pleural
			effusion, wound
			infection, transient
			Horner's syndrome,
			respiratory failure
			requiring tracheostomy
			and haemothorax) with
			major postoperative
			complications in
			symptomatic patients

		Complications of	37 patients were
		conservative	followed up
		management	conservatively, and
			13 (35%) were still
			asymptomatic at time of
			operation

Cartmill and	20 patients from one	Presenting complaint	75% symptomatic (chest	Advocate surgery for
Hughes, Aust N Z J	centre, over 10 years.		pain, cough,	symptom relief,
Surg, 1989,	Included some		haemoptysis, dysphagia).	exclusion of malignancy
Australia, [13]	paediatric patients (not		5% serious complications	and prevention of
	known how many)			catastrophic
Retrospective		Preoperative	45% diagnosed with CT,	complications
cohort study (2b)	Mean postoperative	diagnosis	barium swallow and
	follow-up 67 months		aortography

		Postoperative	5% (one patient) multiple
		complications	PE.
			No recurrence.
			No death

		Complications of	5% (one patient) declined
		conservative	surgery initially but
		management	returned for elective
			treatment after six years.
			Reason not given

Coselli et al., Ann	Eight patients from one	Presenting complaint	75% symptomatic	Recommend excision to
Thorac Surg, 1987,	centre, over 11 years		(dysphagia, epigastric	establish diagnosis,
USA, [14]			pain, respiratory distress,	alleviate symptoms and
			dyspnoea, chest pain)	prevent complications
Case series (4)
		Preoperative	With use of CT; operated
		diagnosis	on to confirm diagnosis

		Operative findings	Previously infected cysts
			more difficult and
			hazardous to excise


Ge et al., Chin Med	22 patients from one	Presenting complaint	91% symptomatic (chest	Recommend that
Sci J, 1995, China	centre, over 20 years.		pain, dyspnoea, cough,	asymptomatic
[15]	Included paediatric		fever, infection,	bronchogenic cysts
	patients, but not		dysphagia, haemoptysis)	should be excised
Retrospective	specified how many			because of the high risk
cohort study (2b)		Preoperative	36.4% diagnosed with	of complications,
	Mean postoperative	diagnosis	CT	although this conclusion
	follow-up seven years			was not tested in the
		Postoperative	No recurrence.	study
		complications	No complications.
			No mortality

Takeda et al.,	105 patients (with any	Presenting complaint	40% symptomatic (chest	Heterogenous population.
Chest, 2003, USA	mediastinal cyst) from		pain, dyspnoea, cough,	Data extracted for
[16]	one centre including		fever, sputum,	bronchogenic cysts only.
	45 adults and six		dysphagia, haemoptysis)	Three patients had
Retropective cohort	paediatric patients with			complicated surgery due
study (2b)	bronchogenic cysts,	Preoperative	Could not extract data	to peri-cystic adhesions.
	over 50-year period	diagnosis	but improved diagnostic	Preventative resection
			capabilities of MRI	preferred because of
			acknowledged	unpredictable clinical
				behaviour
		Postoperative	Could not extract data
		complications	but described as
			‘acceptable’ by authors

		Complications of	Two patients refused
		conservative	treatment: outcome not
		management	described

Gursoy et al., Saudi	28 adult patients with	Presenting complaint	71% symptomatic	Six additional patients
Med J, 2009,	preoperative diagnosis		(dyspnoea, chest pain,	excluded as preoperative
Turkey, [17]	of bronchogenic cyst		cough, fever)	diagnosis of
	from one centre over			bronchogenic cyst not
Retrospective	seven years	Preoperative	82.4% correctly	confirmed histologically
cohort study (2b)		diagnosis	diagnosed with CT
	36-month mean			Surgical resection
	postoperative	Postoperative	11% early complication	recommended in
	follow-up	complications	rate (wound infection,	asymptomatic patients
			prolonged air leak,	because of the possibility
			pneumoperitoneum).	of malignant
			7% late complication	transformation and
			(dyspnea,	anatomic complications
			pneumothorax).	of delayed surgery
			No mortality

		Complications of	No patients
		conservative	conservatively managed
		management

Liu et al., Chin Med	50 adult patients with	Presenting complaint	66% symptomatic	Recommend surgical
Sci J, 2009, China,	histopathologically		(cough, chest pain,	resection to confirm
[18]	proven bronchogenic		hemoptysis, dyspnoea,	diagnosis, avoid
	cyst from one centre of		fever, dysphagia,	development of
Retrospective	24 years		paralysis, hoarseness).	symptoms or malignant
cohort study (2b)			26% serious	change. Conclusions
	6.5-year mean		complications	drawn upon findings of
	follow-up			other studies
		Preoperative	40% diagnosed
		diagnosis	preoperatively.
			14% misdiagnosed after
			all investigations

		Postoperative	4% early complication
		complications	(persistent air leak,
			hoarseness).
			No late complications.
			No mortality


		Complications of	No patients
		conservative	conservatively managed
		management

Kosar et al., Heart	29 patients from one	Presenting complaint	86% symptomatic	The authors suggest
Lung Circ, 2009	centre, including 13		(cough, sputum, pain,	there is a ‘general
Turkey, [19]	paediatric patients,		breathlessness,	consensus’ that all
	over 15 years treated		haemoptysis, fever)	bronchogenic cysts
Retrospective	with either resection			should be operated on to
cohort study (2b)	or de-epithelialisation	Preoperative	89.7% diagnosed	avoid development of
		diagnosis	preoperatively using	symptoms or
			predominantly CT	complications

		Postoperative	17% early complication
		complications	rate (pneumonia, wound
			infections, prolonged air
			leak); higher in
			complicated cysts.
			No late complications.
			No mortality.
			Recurrence in
			de-epithelialised group

		Complications of	No patients
		conservative	conservatively managed
		management

Limaïauiem et al.,	33 patients from one	Presenting complaint	94% symptomatic (chest	Management of all
Lung, 2008,	centre over six years		pain, cough,	bronchogenic cysts
Tunisia, [20]			haemoptysis, dyspnoea,	based on complete
	Follow-up between		fever, dysphagia)	surgical excision.
Retrospective	one and 51 months			Conclude that definitive
cohort study (2b)		Preoperative	Correct diagnosis in	diagnosis is by histology
		diagnosis	33.3%	only and that complete
				surgical excision is
		Postoperative	14% Early complications	mandatory, although
		complications	(pneumothorax,	conclusions based on
			haemorrhage, pleural	findings of other studies
			effusion, seizure).
			No late complications.
			No mortality

		Complications of	No patients
		conservative	conservatively managed
		management

Granato et al.,	30 adult patients treated	Presenting complaint	30% symptomatic	Two symptomatic cysts
Asian Cardiovasc	for bronchogenic cysts		(cough, sputum, pain,	complicated
Thorac Ann, 2009,	from one centre, over		fever, weakness)	intraoperatively by severe
Italy, [21]	32 years			adhesions
		Preoperative	Correct diagnosis in
Retrospective		diagnosis	100% (CT or CT and	One case of large-cell
cohort study (2b)			MRI)	anaplastic carcinoma in
				wall of cyst
		Postoperative	10% intraoperative
		complications	complications.	Excision of
			10% postoperative	asymptomatic cysts
			complications	advocated to avoid
				complex surgery and
		Complications of	No patients	complications, and also
		conservative	conservatively managed	to reduce malignant
		management		potential

De Giacomo et al.,	30 adult patients from	Presenting complaint	37% symptomatic	Authors feel that patients
Eur J Cardiothorac Surg	one centre over		(cough, pain, dysphagia)	cannot be completely
2009, Italy, [22]	12 years			assured about
		Postoperative	None	conservative
Retrospective	Follow-up	complications		management but
cohort study (2b)	3–120 months			acknowledge the

		Complications of	Asymptomatic patients	management is
		conservative	requested surgery	controversial
		management	because of enlarging
			cysts, risk of
			complication or fear of
			malignancy

Costa Júnior Ada	60 patients with	Presenting complaint	92% symptomatic	Heterogenous group of
et al., J Bras	pulmonary		(cough, dyspnoea, pain,	patients and disorders.
Pneumol, 2008,	malformations		infection)	Difficult to extract
Brazil, [23]	(including 27 with			bronchogenic cyst data
	bronchogenic cyst)	Preoperative	‘Frequent’ misdiagnosis
Retrospective	from one centre	diagnosis		Prognosis noted to be
cohort study (2b)	over 35 years,			unpredictable. One
	including 40 paediatric	Postoperative	23% (pneumonia,	patient found to have
	patients	complications	atelectasis, empyema,	adenocarcinoma in wall
			sepsis).	of cyst
			3.3% mortality

		Complications of	Diagnosis/treatment
		conservative	delayed in three patients
		management	up to 36 months (mean
			15): outcome in these
			patients not specified

Weber et al.,	12 patients from a	Presenting complaint	42% symptomatic	Agree that management
Ann Thorac Surg,	single centre		(cough, pain, pneumonia)	of asymptomatic cysts is
2004, Switzerland,	undergoing			controversial but that
[24]	video-assisted	Preoperative	100% correct diagnosis	there appears to be no
	thoracoscopic surgery	diagnosis	with CT with or without	need for urgent surgery
Retrospective	for bronchogenic		MRI. MRI noted to be	in these cases provided
cohort study (2b)	cysts, over seven years		superior	that a simple cyst has
				been clearly diagnosed
	40.5-month mean	Postoperative	None
	follow-up	complications

		Complications of	Six patients observed for
		conservative	between two and
		management	22 years without
			complications.Three
			developed mild
			symptoms.
			Three patients eventually
			requested surgery
			because of fear of
			malignancy/complications
			or enlarging cyst

Author, date and country	Patient group	Outcomes	Key results	Comments
Study type
(level of evidence)
Laberge et al.,	Review of literature	Presenting complaint	Dysphagia, dyspnoea,	Highly heterogenous
Semin Pediatr Surg,	regarding asymptomatic		infection, haemoptysis	group including all adult
2005, Canada, [2]	congenital lung		and haemothorax (no	and paediatric congenital
	malformations,		figures of incidence)	lung malformations
Review (2a)	including bronchogenic
	cysts, in a paediatric	Postoperative	Adult postoperative	Two cases of
	population	complications	complications not	malignancy in
			discussed	bronchogenic cysts
	Includes papers on adult			noted in adult patients
	congenital lung	Complications of	One adult patient with
	malformations	conservative	abnormal chest X-ray	Conclusions based on
		management	eventually presented	management of
			10 years later with	paediatric malformations
			dyspnoea and was found	but recommend early
			to have	excision of bronchogenic
			bronchioloalveolar	cysts due to progression
			carcinoma associated	to symptoms, malignant
			with bronchogenic cyst.	potential and
			Another patient had ‘long	complications of
			standing’ history of cyst	symptomatic cysts
			infections and later found
			to have associated
			mesenchymal malignancy

Sarper et al., Tex	22 patients from one	Presenting complaint	45% presented with	Recommended surgical
Heart Inst J, 2003,	centre, over 15 years		severe haemoptysis,	resection of all suspected
Turkey, [3]			pneumothorax and	bronchogenic cysts in
	5.2-year median		pleuritis, oesophageal	operable candidates due
Retrospective	postoperative follow-up		compression, infected	to difficulties in
cohort study (2b)			cyst, or postobstructive	establishing definitive
			pneumonia.	diagnoses and frequency
			82% presented with	of complications
			symptoms of cough,
			pain, dyspnoea,
			dysphagia or infection

		Postoperative	5% (one patient)
		complications	persistent air leak
			No recurrence
			No late sequelae

		Complications of	No patients
		conservative	conservatively managed
		management

Kanemitsu et al.,	17 patients (16 adults	Presenting complaint	29% symptomatic (9%	It is noted that the
Surg Today, 1999,	and one paediatric) from		of mediastinal cysts, 67%	advanced age of some
Japan, [4]	one centre, over		of intrapulmonary cysts)	patients at presentation
	30 years		with cough, sputum,	suggests that these cysts
Retrospective			fever, pain or weight loss	can remain
cohort study (2b)	30-month median			asymptomatic forever.
	postoperative follow-up	Preoperative	69% diagnosed with CT	The authors state that the
		diagnosis	100% diagnosed with	most appropriate
			MRI	treatment for
				asymptomatic cysts is
		Operative findings	41% adhesions, 6%	controversial but due to
			complicated	diagnostic limitations and
				the possibility of
		Postoperative	No postoperative	symptoms arising or
		complications	complications.	malignant transformation
			No recurrence	that surgery is advocated

		Complications of	None in one patient
		incomplete resection	where residual tissue
			ablated with
			electro-cautery

		Complications of	No patients
		conservative	conservatively managed
		management
Cioffi et al., Chest,	27 adults of whom	Presenting complaint	50% chest pain, 13%	Conclude that all patients
1998, Italy, [5]	16 with bronchogenic		cough, 6% epigastric	should have surgical
	cyst from one centre,		pain.	resection for definitive
Retrospective	over 20 years		44% asymptomatic	diagnosis and to
cohort study (2b)			(incidental finding)	minimise complications
	Four-year median			that might arise from
	postoperative follow-up	Preoperative	100% preoperatively	symptomatic cysts
		diagnosis	diagnosed with CT, EUS

		Postoperative	No postoperative
		complications	complications

		Complications of	No patients
		conservative	conservatively managed
		management

Aktogu et al., Eur	31 patients (30 adults	Presenting complaint	19% superior vena cava	Thirteen patients
Respir J, 1996,	and one paediatric) from		syndrome, tracheal	symptomatic at the time
Turkey, [6]	one centre, over		compression,	of surgery had complex
	19 years		pneumothorax, pleurisy	peri-cystic adhesions or
Retrospective			or pneumonia.	fistulisation.
cohort study (2b)	Follow-up 2–10 years		81% cough, infection,	Surgical treatment of
	postoperatively		pain, dyspnoea, anorexia/	asymptomatic cysts is
			weight loss, haemoptysis	recommended to avoid
				potentially
		Preoperative	19% asymptomatic	life-threatening
		diagnosis	(incidental finding).	complications and for
			71% undiagnosed with	definitive diagnosis
			CT

		Postoperative	No recurrence
		complications

		Complications of	Two of six initially
		conservative	asymptomatic patients
		management	either had cyst
			enlargement or became
			symptomatic

Ribet et al., Ann	41 patients from one	Presenting complaint	80% symptomatic	Uncertain what
Thorac Surg, 1996,	centre, including 20		(cough, pain, purulent	proportion of
France, [7]	paediatric cases, over		sputum, haemoptysis and	bronchogenic cysts
	25 years		dyspnoea) in adult life	remain asymptomatic
Retrospective				and long-term prognosis
cohort study (2b)	2.6-year mean	Preoperative	45% undiagnosed	unpredictable.
	postoperative follow-up	diagnosis		Preventative surgery
				recommended
		Postoperative	5% (one patient)
		complications	bronchial fistula.
			No recurrence in 88%
			(remainder lost to
			follow-up).
			No deaths

		Complications of	15% (three patients)
		conservative	followed for 11 months,
		management	five years and unknown
			length of time since
			diagnosis developed no
			symptoms but were
			operated on.
			15% (three patients)
			initially with cough,
			recurrent bronchitis and
			no symptoms
			(respectively) refused
			surgery but lost to
			follow-up


Cuypers et al., Eur	20 adult patients from	Presenting complaint	30% symptomatic	The authors found the
J Cardiothorac	one centre, over		(pneumonia, abscess,	risk of malignancy and
Surg, 1996,	18 years		dysphagia and cough)	cyst-related
Belgium, [8]			70% asymptomatic	complications to be
	No long-term follow-up			justification for operative
Retrospective		Preoperative	25% undiagnosed after	treatment in all cases of
cohort study (2b)		diagnosis	CT, bronchoscopy,	bronchogenic cysts
			barium swallow or echo
			(all cases had CT with
			dense fluid)

		Postoperative	No postoperative
		complications	morbidity; one case
			histologically associated
			with squamous cell
			carcinoma

		Complications of	No patients
		conservative	conservatively managed
		management

Ribet et al., J	69 patients from one	Presenting complaint	63.7% symptomatic	Due to the frequency of
Thorac Cardiovasc	centre, including 24		pain, respiratory tract	late complications with
Surg, 1995, France,	paediatric cases, over		infection, cough,	asymptomatic cysts and
[9]	25 years		dyspnoea, dysphagia,	the unpredictable
			heartburn)	prognosis, preventative
Retrospective	4.2-year mean			surgery was
cohort study (2b)	postoperative follow-up	Preoperative	22% initially	recommended
		diagnosis	misdiagnosed (11% not
			as bronchogenic cyst;
			11% as bronchogenic
			cyst when actually cystic
			neurogenic tumour,
			benign lymphoma and
			haemolymphangioma)

		Postoperative	13.4% postoperative
		complications	morbidity (infection,
			chylothorax and phrenic
			paresis).
			12% symptoms of pain
			or dyspnoea
			postoperative (7%
			symptomatic
			preoperative, 5%
			asymptomatic)

		Complications of	11% refused operation,
		conservative	only 4% followed up
		management	(two patients): one
			remained asymptomatic,
			one died of generalised
			malignancy of unknown
			origin.
			7% had incomplete
			operations but cyst
			remained stable or had no
			recurrence

Patel et al., Chest,	18 adult patients from	Presenting complaint	44% symptomatic	There was no statistical
1994, USA, [10]	one centre, over		(cough, pain) of which	difference in the
	19-year period		11% serious (dyspnea,	frequency of
Retrospective			infection).	intraoperative difficulties
cohort study (2b)	Follow-up 12		56% asymptomatic	or postoperative
	months to 11 years			complications between
	(mean not given)	Preoperative	37.5% undiagnosed with	asymptomatic and
		diagnosis	CT, angio, USS, FNA,	symptomatic patients.
			barium swallow,	The authors concluded,
			bronchoscopy or	however, that surgery is
			mediastinoscopy	advocated in
		Operative difficulties	No difference in	asymptomatic cysts due
			frequency of operative	to the potential for
			difficulties between	complications, incorrect
			symptomatic and	diagnosis or progression
			asymptomatic patients	to symptoms
			(P=0.0656)

		Postoperative	No difference (P=0.596)
		complications	in complication rate
			between preoperatively
			asymptomatic (14%) or
			symptomatic (27%)
			patients. Complications
			included phrenic nerve
			paresis.
			11% delayed
			complications
			(oesophageal stricture
			and recurrence)

		Complications of	Three out of seven
		conservative	asymptomatic patients
		management	followed up developed
			symptoms; four patients
			lost to follow-up

Suen et al., Ann	42 patients from one	Presenting complaint	50% symptomatic (pain,	Complete excision
Thorac Surg,	centre, over 30 years		cough, fever, dysphagia,	recommended in most
1993, USA, [11]			purulent sputum,	cases to relieve
			haemoptysis, dyspnoea).	symptoms, prevent
Retrospective			26% complicated	complications and
cohort study (2b)			(dysphagia, haemorrhage,	confirm diagnosis
			infection and one patient
			with adenocarcinoma in
			cyst)

		Preoperative	59% no preoperative
		diagnosis	diagnosis (more recent
			cases, better success
			rate)

		Postoperative	5% postoperative
		complications	complications (wound
			infection and C. Diff
			colitis).
			No recurrence.
			No deaths

		Complications of	Two patients treated
		conservative	conservatively (one
		management	follow-up only and one
			drainage) with no
			complications

St-Georges et al.,	86 patients from one	Presenting complaint	72% patients	35/86 (41%) of patients
Ann Thorac Surg,	centre, over 20 years		symptomatic by time of	operated on had major
1991, Canada, [12]			surgery (57%	operative difficulties: all
			progressive symptoms	these were patients with
Retrospective			and 15% acute).	symptoms
cohort study (2b)			53% patients with >1	preoperatively
			symptom (chest pain,
			cough, dyspnoea, fever,	Resection recommended
			sputum, anorexia/weight	as majority of patients
			loss, dysphagia,	eventually develop
			haemoptysis).	symptoms or
			38% with complications	complications
			(fistula with airway,
			ulceration cyst wall,
			haemorrhage, infection,
			bronchial atresia)
		Preoperative	57% patients presumed
		diagnosis	diagnosis at surgery after
			CT and angiography.
			Positive diagnosis never
			made preoperatively

		Operative findings	41% had complicated
			cysts (fistula, ulcer,
			haemorrhage, infection,
			atresia) at operation

		Postoperative	11% intraoperative
		complications	complications (vagal
			trunk division, segmental
			bronchus laceration,
			oesophageal mucosal
			laceration).
			9% postoperative
			complications
			(atelectasis, pleural
			effusion, wound
			infection, transient
			Horner's syndrome,
			respiratory failure
			requiring tracheostomy
			and haemothorax) with
			major postoperative
			complications in
			symptomatic patients

		Complications of	37 patients were
		conservative	followed up
		management	conservatively, and
			13 (35%) were still
			asymptomatic at time of
			operation

Cartmill and	20 patients from one	Presenting complaint	75% symptomatic (chest	Advocate surgery for
Hughes, Aust N Z J	centre, over 10 years.		pain, cough,	symptom relief,
Surg, 1989,	Included some		haemoptysis, dysphagia).	exclusion of malignancy
Australia, [13]	paediatric patients (not		5% serious complications	and prevention of
	known how many)			catastrophic
Retrospective		Preoperative	45% diagnosed with CT,	complications
cohort study (2b)	Mean postoperative	diagnosis	barium swallow and
	follow-up 67 months		aortography

		Postoperative	5% (one patient) multiple
		complications	PE.
			No recurrence.
			No death

		Complications of	5% (one patient) declined
		conservative	surgery initially but
		management	returned for elective
			treatment after six years.
			Reason not given

Coselli et al., Ann	Eight patients from one	Presenting complaint	75% symptomatic	Recommend excision to
Thorac Surg, 1987,	centre, over 11 years		(dysphagia, epigastric	establish diagnosis,
USA, [14]			pain, respiratory distress,	alleviate symptoms and
			dyspnoea, chest pain)	prevent complications
Case series (4)
		Preoperative	With use of CT; operated
		diagnosis	on to confirm diagnosis

		Operative findings	Previously infected cysts
			more difficult and
			hazardous to excise


Ge et al., Chin Med	22 patients from one	Presenting complaint	91% symptomatic (chest	Recommend that
Sci J, 1995, China	centre, over 20 years.		pain, dyspnoea, cough,	asymptomatic
[15]	Included paediatric		fever, infection,	bronchogenic cysts
	patients, but not		dysphagia, haemoptysis)	should be excised
Retrospective	specified how many			because of the high risk
cohort study (2b)		Preoperative	36.4% diagnosed with	of complications,
	Mean postoperative	diagnosis	CT	although this conclusion
	follow-up seven years			was not tested in the
		Postoperative	No recurrence.	study
		complications	No complications.
			No mortality

Takeda et al.,	105 patients (with any	Presenting complaint	40% symptomatic (chest	Heterogenous population.
Chest, 2003, USA	mediastinal cyst) from		pain, dyspnoea, cough,	Data extracted for
[16]	one centre including		fever, sputum,	bronchogenic cysts only.
	45 adults and six		dysphagia, haemoptysis)	Three patients had
Retropective cohort	paediatric patients with			complicated surgery due
study (2b)	bronchogenic cysts,	Preoperative	Could not extract data	to peri-cystic adhesions.
	over 50-year period	diagnosis	but improved diagnostic	Preventative resection
			capabilities of MRI	preferred because of
			acknowledged	unpredictable clinical
				behaviour
		Postoperative	Could not extract data
		complications	but described as
			‘acceptable’ by authors

		Complications of	Two patients refused
		conservative	treatment: outcome not
		management	described

Gursoy et al., Saudi	28 adult patients with	Presenting complaint	71% symptomatic	Six additional patients
Med J, 2009,	preoperative diagnosis		(dyspnoea, chest pain,	excluded as preoperative
Turkey, [17]	of bronchogenic cyst		cough, fever)	diagnosis of
	from one centre over			bronchogenic cyst not
Retrospective	seven years	Preoperative	82.4% correctly	confirmed histologically
cohort study (2b)		diagnosis	diagnosed with CT
	36-month mean			Surgical resection
	postoperative	Postoperative	11% early complication	recommended in
	follow-up	complications	rate (wound infection,	asymptomatic patients
			prolonged air leak,	because of the possibility
			pneumoperitoneum).	of malignant
			7% late complication	transformation and
			(dyspnea,	anatomic complications
			pneumothorax).	of delayed surgery
			No mortality

		Complications of	No patients
		conservative	conservatively managed
		management

Liu et al., Chin Med	50 adult patients with	Presenting complaint	66% symptomatic	Recommend surgical
Sci J, 2009, China,	histopathologically		(cough, chest pain,	resection to confirm
[18]	proven bronchogenic		hemoptysis, dyspnoea,	diagnosis, avoid
	cyst from one centre of		fever, dysphagia,	development of
Retrospective	24 years		paralysis, hoarseness).	symptoms or malignant
cohort study (2b)			26% serious	change. Conclusions
	6.5-year mean		complications	drawn upon findings of
	follow-up			other studies
		Preoperative	40% diagnosed
		diagnosis	preoperatively.
			14% misdiagnosed after
			all investigations

		Postoperative	4% early complication
		complications	(persistent air leak,
			hoarseness).
			No late complications.
			No mortality


		Complications of	No patients
		conservative	conservatively managed
		management

Kosar et al., Heart	29 patients from one	Presenting complaint	86% symptomatic	The authors suggest
Lung Circ, 2009	centre, including 13		(cough, sputum, pain,	there is a ‘general
Turkey, [19]	paediatric patients,		breathlessness,	consensus’ that all
	over 15 years treated		haemoptysis, fever)	bronchogenic cysts
Retrospective	with either resection			should be operated on to
cohort study (2b)	or de-epithelialisation	Preoperative	89.7% diagnosed	avoid development of
		diagnosis	preoperatively using	symptoms or
			predominantly CT	complications

		Postoperative	17% early complication
		complications	rate (pneumonia, wound
			infections, prolonged air
			leak); higher in
			complicated cysts.
			No late complications.
			No mortality.
			Recurrence in
			de-epithelialised group

		Complications of	No patients
		conservative	conservatively managed
		management

Limaïauiem et al.,	33 patients from one	Presenting complaint	94% symptomatic (chest	Management of all
Lung, 2008,	centre over six years		pain, cough,	bronchogenic cysts
Tunisia, [20]			haemoptysis, dyspnoea,	based on complete
	Follow-up between		fever, dysphagia)	surgical excision.
Retrospective	one and 51 months			Conclude that definitive
cohort study (2b)		Preoperative	Correct diagnosis in	diagnosis is by histology
		diagnosis	33.3%	only and that complete
				surgical excision is
		Postoperative	14% Early complications	mandatory, although
		complications	(pneumothorax,	conclusions based on
			haemorrhage, pleural	findings of other studies
			effusion, seizure).
			No late complications.
			No mortality

		Complications of	No patients
		conservative	conservatively managed
		management

Granato et al.,	30 adult patients treated	Presenting complaint	30% symptomatic	Two symptomatic cysts
Asian Cardiovasc	for bronchogenic cysts		(cough, sputum, pain,	complicated
Thorac Ann, 2009,	from one centre, over		fever, weakness)	intraoperatively by severe
Italy, [21]	32 years			adhesions
		Preoperative	Correct diagnosis in
Retrospective		diagnosis	100% (CT or CT and	One case of large-cell
cohort study (2b)			MRI)	anaplastic carcinoma in
				wall of cyst
		Postoperative	10% intraoperative
		complications	complications.	Excision of
			10% postoperative	asymptomatic cysts
			complications	advocated to avoid
				complex surgery and
		Complications of	No patients	complications, and also
		conservative	conservatively managed	to reduce malignant
		management		potential

De Giacomo et al.,	30 adult patients from	Presenting complaint	37% symptomatic	Authors feel that patients
Eur J Cardiothorac Surg	one centre over		(cough, pain, dysphagia)	cannot be completely
2009, Italy, [22]	12 years			assured about
		Postoperative	None	conservative
Retrospective	Follow-up	complications		management but
cohort study (2b)	3–120 months			acknowledge the

		Complications of	Asymptomatic patients	management is
		conservative	requested surgery	controversial
		management	because of enlarging
			cysts, risk of
			complication or fear of
			malignancy

Costa Júnior Ada	60 patients with	Presenting complaint	92% symptomatic	Heterogenous group of
et al., J Bras	pulmonary		(cough, dyspnoea, pain,	patients and disorders.
Pneumol, 2008,	malformations		infection)	Difficult to extract
Brazil, [23]	(including 27 with			bronchogenic cyst data
	bronchogenic cyst)	Preoperative	‘Frequent’ misdiagnosis
Retrospective	from one centre	diagnosis		Prognosis noted to be
cohort study (2b)	over 35 years,			unpredictable. One
	including 40 paediatric	Postoperative	23% (pneumonia,	patient found to have
	patients	complications	atelectasis, empyema,	adenocarcinoma in wall
			sepsis).	of cyst
			3.3% mortality

		Complications of	Diagnosis/treatment
		conservative	delayed in three patients
		management	up to 36 months (mean
			15): outcome in these
			patients not specified

Weber et al.,	12 patients from a	Presenting complaint	42% symptomatic	Agree that management
Ann Thorac Surg,	single centre		(cough, pain, pneumonia)	of asymptomatic cysts is
2004, Switzerland,	undergoing			controversial but that
[24]	video-assisted	Preoperative	100% correct diagnosis	there appears to be no
	thoracoscopic surgery	diagnosis	with CT with or without	need for urgent surgery
Retrospective	for bronchogenic		MRI. MRI noted to be	in these cases provided
cohort study (2b)	cysts, over seven years		superior	that a simple cyst has
				been clearly diagnosed
	40.5-month mean	Postoperative	None
	follow-up	complications

		Complications of	Six patients observed for
		conservative	between two and
		management	22 years without
			complications.Three
			developed mild
			symptoms.
			Three patients eventually
			requested surgery
			because of fear of
			malignancy/complications
			or enlarging cyst

CT, computed tomography; EUS, endoscopic ultrasound; USS, ultrasound scan; FNA, fine needle aspiration.

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Table 1.

Summary of the 20 papers derived from the search strategy

Author, date and country	Patient group	Outcomes	Key results	Comments
Study type
(level of evidence)
Laberge et al.,	Review of literature	Presenting complaint	Dysphagia, dyspnoea,	Highly heterogenous
Semin Pediatr Surg,	regarding asymptomatic		infection, haemoptysis	group including all adult
2005, Canada, [2]	congenital lung		and haemothorax (no	and paediatric congenital
	malformations,		figures of incidence)	lung malformations
Review (2a)	including bronchogenic
	cysts, in a paediatric	Postoperative	Adult postoperative	Two cases of
	population	complications	complications not	malignancy in
			discussed	bronchogenic cysts
	Includes papers on adult			noted in adult patients
	congenital lung	Complications of	One adult patient with
	malformations	conservative	abnormal chest X-ray	Conclusions based on
		management	eventually presented	management of
			10 years later with	paediatric malformations
			dyspnoea and was found	but recommend early
			to have	excision of bronchogenic
			bronchioloalveolar	cysts due to progression
			carcinoma associated	to symptoms, malignant
			with bronchogenic cyst.	potential and
			Another patient had ‘long	complications of
			standing’ history of cyst	symptomatic cysts
			infections and later found
			to have associated
			mesenchymal malignancy

Sarper et al., Tex	22 patients from one	Presenting complaint	45% presented with	Recommended surgical
Heart Inst J, 2003,	centre, over 15 years		severe haemoptysis,	resection of all suspected
Turkey, [3]			pneumothorax and	bronchogenic cysts in
	5.2-year median		pleuritis, oesophageal	operable candidates due
Retrospective	postoperative follow-up		compression, infected	to difficulties in
cohort study (2b)			cyst, or postobstructive	establishing definitive
			pneumonia.	diagnoses and frequency
			82% presented with	of complications
			symptoms of cough,
			pain, dyspnoea,
			dysphagia or infection

		Postoperative	5% (one patient)
		complications	persistent air leak
			No recurrence
			No late sequelae

		Complications of	No patients
		conservative	conservatively managed
		management

Kanemitsu et al.,	17 patients (16 adults	Presenting complaint	29% symptomatic (9%	It is noted that the
Surg Today, 1999,	and one paediatric) from		of mediastinal cysts, 67%	advanced age of some
Japan, [4]	one centre, over		of intrapulmonary cysts)	patients at presentation
	30 years		with cough, sputum,	suggests that these cysts
Retrospective			fever, pain or weight loss	can remain
cohort study (2b)	30-month median			asymptomatic forever.
	postoperative follow-up	Preoperative	69% diagnosed with CT	The authors state that the
		diagnosis	100% diagnosed with	most appropriate
			MRI	treatment for
				asymptomatic cysts is
		Operative findings	41% adhesions, 6%	controversial but due to
			complicated	diagnostic limitations and
				the possibility of
		Postoperative	No postoperative	symptoms arising or
		complications	complications.	malignant transformation
			No recurrence	that surgery is advocated

		Complications of	None in one patient
		incomplete resection	where residual tissue
			ablated with
			electro-cautery

		Complications of	No patients
		conservative	conservatively managed
		management
Cioffi et al., Chest,	27 adults of whom	Presenting complaint	50% chest pain, 13%	Conclude that all patients
1998, Italy, [5]	16 with bronchogenic		cough, 6% epigastric	should have surgical
	cyst from one centre,		pain.	resection for definitive
Retrospective	over 20 years		44% asymptomatic	diagnosis and to
cohort study (2b)			(incidental finding)	minimise complications
	Four-year median			that might arise from
	postoperative follow-up	Preoperative	100% preoperatively	symptomatic cysts
		diagnosis	diagnosed with CT, EUS

		Postoperative	No postoperative
		complications	complications

		Complications of	No patients
		conservative	conservatively managed
		management

Aktogu et al., Eur	31 patients (30 adults	Presenting complaint	19% superior vena cava	Thirteen patients
Respir J, 1996,	and one paediatric) from		syndrome, tracheal	symptomatic at the time
Turkey, [6]	one centre, over		compression,	of surgery had complex
	19 years		pneumothorax, pleurisy	peri-cystic adhesions or
Retrospective			or pneumonia.	fistulisation.
cohort study (2b)	Follow-up 2–10 years		81% cough, infection,	Surgical treatment of
	postoperatively		pain, dyspnoea, anorexia/	asymptomatic cysts is
			weight loss, haemoptysis	recommended to avoid
				potentially
		Preoperative	19% asymptomatic	life-threatening
		diagnosis	(incidental finding).	complications and for
			71% undiagnosed with	definitive diagnosis
			CT

		Postoperative	No recurrence
		complications

		Complications of	Two of six initially
		conservative	asymptomatic patients
		management	either had cyst
			enlargement or became
			symptomatic

Ribet et al., Ann	41 patients from one	Presenting complaint	80% symptomatic	Uncertain what
Thorac Surg, 1996,	centre, including 20		(cough, pain, purulent	proportion of
France, [7]	paediatric cases, over		sputum, haemoptysis and	bronchogenic cysts
	25 years		dyspnoea) in adult life	remain asymptomatic
Retrospective				and long-term prognosis
cohort study (2b)	2.6-year mean	Preoperative	45% undiagnosed	unpredictable.
	postoperative follow-up	diagnosis		Preventative surgery
				recommended
		Postoperative	5% (one patient)
		complications	bronchial fistula.
			No recurrence in 88%
			(remainder lost to
			follow-up).
			No deaths

		Complications of	15% (three patients)
		conservative	followed for 11 months,
		management	five years and unknown
			length of time since
			diagnosis developed no
			symptoms but were
			operated on.
			15% (three patients)
			initially with cough,
			recurrent bronchitis and
			no symptoms
			(respectively) refused
			surgery but lost to
			follow-up


Cuypers et al., Eur	20 adult patients from	Presenting complaint	30% symptomatic	The authors found the
J Cardiothorac	one centre, over		(pneumonia, abscess,	risk of malignancy and
Surg, 1996,	18 years		dysphagia and cough)	cyst-related
Belgium, [8]			70% asymptomatic	complications to be
	No long-term follow-up			justification for operative
Retrospective		Preoperative	25% undiagnosed after	treatment in all cases of
cohort study (2b)		diagnosis	CT, bronchoscopy,	bronchogenic cysts
			barium swallow or echo
			(all cases had CT with
			dense fluid)

		Postoperative	No postoperative
		complications	morbidity; one case
			histologically associated
			with squamous cell
			carcinoma

		Complications of	No patients
		conservative	conservatively managed
		management

Ribet et al., J	69 patients from one	Presenting complaint	63.7% symptomatic	Due to the frequency of
Thorac Cardiovasc	centre, including 24		pain, respiratory tract	late complications with
Surg, 1995, France,	paediatric cases, over		infection, cough,	asymptomatic cysts and
[9]	25 years		dyspnoea, dysphagia,	the unpredictable
			heartburn)	prognosis, preventative
Retrospective	4.2-year mean			surgery was
cohort study (2b)	postoperative follow-up	Preoperative	22% initially	recommended
		diagnosis	misdiagnosed (11% not
			as bronchogenic cyst;
			11% as bronchogenic
			cyst when actually cystic
			neurogenic tumour,
			benign lymphoma and
			haemolymphangioma)

		Postoperative	13.4% postoperative
		complications	morbidity (infection,
			chylothorax and phrenic
			paresis).
			12% symptoms of pain
			or dyspnoea
			postoperative (7%
			symptomatic
			preoperative, 5%
			asymptomatic)

		Complications of	11% refused operation,
		conservative	only 4% followed up
		management	(two patients): one
			remained asymptomatic,
			one died of generalised
			malignancy of unknown
			origin.
			7% had incomplete
			operations but cyst
			remained stable or had no
			recurrence

Patel et al., Chest,	18 adult patients from	Presenting complaint	44% symptomatic	There was no statistical
1994, USA, [10]	one centre, over		(cough, pain) of which	difference in the
	19-year period		11% serious (dyspnea,	frequency of
Retrospective			infection).	intraoperative difficulties
cohort study (2b)	Follow-up 12		56% asymptomatic	or postoperative
	months to 11 years			complications between
	(mean not given)	Preoperative	37.5% undiagnosed with	asymptomatic and
		diagnosis	CT, angio, USS, FNA,	symptomatic patients.
			barium swallow,	The authors concluded,
			bronchoscopy or	however, that surgery is
			mediastinoscopy	advocated in
		Operative difficulties	No difference in	asymptomatic cysts due
			frequency of operative	to the potential for
			difficulties between	complications, incorrect
			symptomatic and	diagnosis or progression
			asymptomatic patients	to symptoms
			(P=0.0656)

		Postoperative	No difference (P=0.596)
		complications	in complication rate
			between preoperatively
			asymptomatic (14%) or
			symptomatic (27%)
			patients. Complications
			included phrenic nerve
			paresis.
			11% delayed
			complications
			(oesophageal stricture
			and recurrence)

		Complications of	Three out of seven
		conservative	asymptomatic patients
		management	followed up developed
			symptoms; four patients
			lost to follow-up

Suen et al., Ann	42 patients from one	Presenting complaint	50% symptomatic (pain,	Complete excision
Thorac Surg,	centre, over 30 years		cough, fever, dysphagia,	recommended in most
1993, USA, [11]			purulent sputum,	cases to relieve
			haemoptysis, dyspnoea).	symptoms, prevent
Retrospective			26% complicated	complications and
cohort study (2b)			(dysphagia, haemorrhage,	confirm diagnosis
			infection and one patient
			with adenocarcinoma in
			cyst)

		Preoperative	59% no preoperative
		diagnosis	diagnosis (more recent
			cases, better success
			rate)

		Postoperative	5% postoperative
		complications	complications (wound
			infection and C. Diff
			colitis).
			No recurrence.
			No deaths

		Complications of	Two patients treated
		conservative	conservatively (one
		management	follow-up only and one
			drainage) with no
			complications

St-Georges et al.,	86 patients from one	Presenting complaint	72% patients	35/86 (41%) of patients
Ann Thorac Surg,	centre, over 20 years		symptomatic by time of	operated on had major
1991, Canada, [12]			surgery (57%	operative difficulties: all
			progressive symptoms	these were patients with
Retrospective			and 15% acute).	symptoms
cohort study (2b)			53% patients with >1	preoperatively
			symptom (chest pain,
			cough, dyspnoea, fever,	Resection recommended
			sputum, anorexia/weight	as majority of patients
			loss, dysphagia,	eventually develop
			haemoptysis).	symptoms or
			38% with complications	complications
			(fistula with airway,
			ulceration cyst wall,
			haemorrhage, infection,
			bronchial atresia)
		Preoperative	57% patients presumed
		diagnosis	diagnosis at surgery after
			CT and angiography.
			Positive diagnosis never
			made preoperatively

		Operative findings	41% had complicated
			cysts (fistula, ulcer,
			haemorrhage, infection,
			atresia) at operation

		Postoperative	11% intraoperative
		complications	complications (vagal
			trunk division, segmental
			bronchus laceration,
			oesophageal mucosal
			laceration).
			9% postoperative
			complications
			(atelectasis, pleural
			effusion, wound
			infection, transient
			Horner's syndrome,
			respiratory failure
			requiring tracheostomy
			and haemothorax) with
			major postoperative
			complications in
			symptomatic patients

		Complications of	37 patients were
		conservative	followed up
		management	conservatively, and
			13 (35%) were still
			asymptomatic at time of
			operation

Cartmill and	20 patients from one	Presenting complaint	75% symptomatic (chest	Advocate surgery for
Hughes, Aust N Z J	centre, over 10 years.		pain, cough,	symptom relief,
Surg, 1989,	Included some		haemoptysis, dysphagia).	exclusion of malignancy
Australia, [13]	paediatric patients (not		5% serious complications	and prevention of
	known how many)			catastrophic
Retrospective		Preoperative	45% diagnosed with CT,	complications
cohort study (2b)	Mean postoperative	diagnosis	barium swallow and
	follow-up 67 months		aortography

		Postoperative	5% (one patient) multiple
		complications	PE.
			No recurrence.
			No death

		Complications of	5% (one patient) declined
		conservative	surgery initially but
		management	returned for elective
			treatment after six years.
			Reason not given

Coselli et al., Ann	Eight patients from one	Presenting complaint	75% symptomatic	Recommend excision to
Thorac Surg, 1987,	centre, over 11 years		(dysphagia, epigastric	establish diagnosis,
USA, [14]			pain, respiratory distress,	alleviate symptoms and
			dyspnoea, chest pain)	prevent complications
Case series (4)
		Preoperative	With use of CT; operated
		diagnosis	on to confirm diagnosis

		Operative findings	Previously infected cysts
			more difficult and
			hazardous to excise


Ge et al., Chin Med	22 patients from one	Presenting complaint	91% symptomatic (chest	Recommend that
Sci J, 1995, China	centre, over 20 years.		pain, dyspnoea, cough,	asymptomatic
[15]	Included paediatric		fever, infection,	bronchogenic cysts
	patients, but not		dysphagia, haemoptysis)	should be excised
Retrospective	specified how many			because of the high risk
cohort study (2b)		Preoperative	36.4% diagnosed with	of complications,
	Mean postoperative	diagnosis	CT	although this conclusion
	follow-up seven years			was not tested in the
		Postoperative	No recurrence.	study
		complications	No complications.
			No mortality

Takeda et al.,	105 patients (with any	Presenting complaint	40% symptomatic (chest	Heterogenous population.
Chest, 2003, USA	mediastinal cyst) from		pain, dyspnoea, cough,	Data extracted for
[16]	one centre including		fever, sputum,	bronchogenic cysts only.
	45 adults and six		dysphagia, haemoptysis)	Three patients had
Retropective cohort	paediatric patients with			complicated surgery due
study (2b)	bronchogenic cysts,	Preoperative	Could not extract data	to peri-cystic adhesions.
	over 50-year period	diagnosis	but improved diagnostic	Preventative resection
			capabilities of MRI	preferred because of
			acknowledged	unpredictable clinical
				behaviour
		Postoperative	Could not extract data
		complications	but described as
			‘acceptable’ by authors

		Complications of	Two patients refused
		conservative	treatment: outcome not
		management	described

Gursoy et al., Saudi	28 adult patients with	Presenting complaint	71% symptomatic	Six additional patients
Med J, 2009,	preoperative diagnosis		(dyspnoea, chest pain,	excluded as preoperative
Turkey, [17]	of bronchogenic cyst		cough, fever)	diagnosis of
	from one centre over			bronchogenic cyst not
Retrospective	seven years	Preoperative	82.4% correctly	confirmed histologically
cohort study (2b)		diagnosis	diagnosed with CT
	36-month mean			Surgical resection
	postoperative	Postoperative	11% early complication	recommended in
	follow-up	complications	rate (wound infection,	asymptomatic patients
			prolonged air leak,	because of the possibility
			pneumoperitoneum).	of malignant
			7% late complication	transformation and
			(dyspnea,	anatomic complications
			pneumothorax).	of delayed surgery
			No mortality

		Complications of	No patients
		conservative	conservatively managed
		management

Liu et al., Chin Med	50 adult patients with	Presenting complaint	66% symptomatic	Recommend surgical
Sci J, 2009, China,	histopathologically		(cough, chest pain,	resection to confirm
[18]	proven bronchogenic		hemoptysis, dyspnoea,	diagnosis, avoid
	cyst from one centre of		fever, dysphagia,	development of
Retrospective	24 years		paralysis, hoarseness).	symptoms or malignant
cohort study (2b)			26% serious	change. Conclusions
	6.5-year mean		complications	drawn upon findings of
	follow-up			other studies
		Preoperative	40% diagnosed
		diagnosis	preoperatively.
			14% misdiagnosed after
			all investigations

		Postoperative	4% early complication
		complications	(persistent air leak,
			hoarseness).
			No late complications.
			No mortality


		Complications of	No patients
		conservative	conservatively managed
		management

Kosar et al., Heart	29 patients from one	Presenting complaint	86% symptomatic	The authors suggest
Lung Circ, 2009	centre, including 13		(cough, sputum, pain,	there is a ‘general
Turkey, [19]	paediatric patients,		breathlessness,	consensus’ that all
	over 15 years treated		haemoptysis, fever)	bronchogenic cysts
Retrospective	with either resection			should be operated on to
cohort study (2b)	or de-epithelialisation	Preoperative	89.7% diagnosed	avoid development of
		diagnosis	preoperatively using	symptoms or
			predominantly CT	complications

		Postoperative	17% early complication
		complications	rate (pneumonia, wound
			infections, prolonged air
			leak); higher in
			complicated cysts.
			No late complications.
			No mortality.
			Recurrence in
			de-epithelialised group

		Complications of	No patients
		conservative	conservatively managed
		management

Limaïauiem et al.,	33 patients from one	Presenting complaint	94% symptomatic (chest	Management of all
Lung, 2008,	centre over six years		pain, cough,	bronchogenic cysts
Tunisia, [20]			haemoptysis, dyspnoea,	based on complete
	Follow-up between		fever, dysphagia)	surgical excision.
Retrospective	one and 51 months			Conclude that definitive
cohort study (2b)		Preoperative	Correct diagnosis in	diagnosis is by histology
		diagnosis	33.3%	only and that complete
				surgical excision is
		Postoperative	14% Early complications	mandatory, although
		complications	(pneumothorax,	conclusions based on
			haemorrhage, pleural	findings of other studies
			effusion, seizure).
			No late complications.
			No mortality

		Complications of	No patients
		conservative	conservatively managed
		management

Granato et al.,	30 adult patients treated	Presenting complaint	30% symptomatic	Two symptomatic cysts
Asian Cardiovasc	for bronchogenic cysts		(cough, sputum, pain,	complicated
Thorac Ann, 2009,	from one centre, over		fever, weakness)	intraoperatively by severe
Italy, [21]	32 years			adhesions
		Preoperative	Correct diagnosis in
Retrospective		diagnosis	100% (CT or CT and	One case of large-cell
cohort study (2b)			MRI)	anaplastic carcinoma in
				wall of cyst
		Postoperative	10% intraoperative
		complications	complications.	Excision of
			10% postoperative	asymptomatic cysts
			complications	advocated to avoid
				complex surgery and
		Complications of	No patients	complications, and also
		conservative	conservatively managed	to reduce malignant
		management		potential

De Giacomo et al.,	30 adult patients from	Presenting complaint	37% symptomatic	Authors feel that patients
Eur J Cardiothorac Surg	one centre over		(cough, pain, dysphagia)	cannot be completely
2009, Italy, [22]	12 years			assured about
		Postoperative	None	conservative
Retrospective	Follow-up	complications		management but
cohort study (2b)	3–120 months			acknowledge the

		Complications of	Asymptomatic patients	management is
		conservative	requested surgery	controversial
		management	because of enlarging
			cysts, risk of
			complication or fear of
			malignancy

Costa Júnior Ada	60 patients with	Presenting complaint	92% symptomatic	Heterogenous group of
et al., J Bras	pulmonary		(cough, dyspnoea, pain,	patients and disorders.
Pneumol, 2008,	malformations		infection)	Difficult to extract
Brazil, [23]	(including 27 with			bronchogenic cyst data
	bronchogenic cyst)	Preoperative	‘Frequent’ misdiagnosis
Retrospective	from one centre	diagnosis		Prognosis noted to be
cohort study (2b)	over 35 years,			unpredictable. One
	including 40 paediatric	Postoperative	23% (pneumonia,	patient found to have
	patients	complications	atelectasis, empyema,	adenocarcinoma in wall
			sepsis).	of cyst
			3.3% mortality

		Complications of	Diagnosis/treatment
		conservative	delayed in three patients
		management	up to 36 months (mean
			15): outcome in these
			patients not specified

Weber et al.,	12 patients from a	Presenting complaint	42% symptomatic	Agree that management
Ann Thorac Surg,	single centre		(cough, pain, pneumonia)	of asymptomatic cysts is
2004, Switzerland,	undergoing			controversial but that
[24]	video-assisted	Preoperative	100% correct diagnosis	there appears to be no
	thoracoscopic surgery	diagnosis	with CT with or without	need for urgent surgery
Retrospective	for bronchogenic		MRI. MRI noted to be	in these cases provided
cohort study (2b)	cysts, over seven years		superior	that a simple cyst has
				been clearly diagnosed
	40.5-month mean	Postoperative	None
	follow-up	complications

		Complications of	Six patients observed for
		conservative	between two and
		management	22 years without
			complications.Three
			developed mild
			symptoms.
			Three patients eventually
			requested surgery
			because of fear of
			malignancy/complications
			or enlarging cyst

Author, date and country	Patient group	Outcomes	Key results	Comments
Study type
(level of evidence)
Laberge et al.,	Review of literature	Presenting complaint	Dysphagia, dyspnoea,	Highly heterogenous
Semin Pediatr Surg,	regarding asymptomatic		infection, haemoptysis	group including all adult
2005, Canada, [2]	congenital lung		and haemothorax (no	and paediatric congenital
	malformations,		figures of incidence)	lung malformations
Review (2a)	including bronchogenic
	cysts, in a paediatric	Postoperative	Adult postoperative	Two cases of
	population	complications	complications not	malignancy in
			discussed	bronchogenic cysts
	Includes papers on adult			noted in adult patients
	congenital lung	Complications of	One adult patient with
	malformations	conservative	abnormal chest X-ray	Conclusions based on
		management	eventually presented	management of
			10 years later with	paediatric malformations
			dyspnoea and was found	but recommend early
			to have	excision of bronchogenic
			bronchioloalveolar	cysts due to progression
			carcinoma associated	to symptoms, malignant
			with bronchogenic cyst.	potential and
			Another patient had ‘long	complications of
			standing’ history of cyst	symptomatic cysts
			infections and later found
			to have associated
			mesenchymal malignancy

Sarper et al., Tex	22 patients from one	Presenting complaint	45% presented with	Recommended surgical
Heart Inst J, 2003,	centre, over 15 years		severe haemoptysis,	resection of all suspected
Turkey, [3]			pneumothorax and	bronchogenic cysts in
	5.2-year median		pleuritis, oesophageal	operable candidates due
Retrospective	postoperative follow-up		compression, infected	to difficulties in
cohort study (2b)			cyst, or postobstructive	establishing definitive
			pneumonia.	diagnoses and frequency
			82% presented with	of complications
			symptoms of cough,
			pain, dyspnoea,
			dysphagia or infection

		Postoperative	5% (one patient)
		complications	persistent air leak
			No recurrence
			No late sequelae

		Complications of	No patients
		conservative	conservatively managed
		management

Kanemitsu et al.,	17 patients (16 adults	Presenting complaint	29% symptomatic (9%	It is noted that the
Surg Today, 1999,	and one paediatric) from		of mediastinal cysts, 67%	advanced age of some
Japan, [4]	one centre, over		of intrapulmonary cysts)	patients at presentation
	30 years		with cough, sputum,	suggests that these cysts
Retrospective			fever, pain or weight loss	can remain
cohort study (2b)	30-month median			asymptomatic forever.
	postoperative follow-up	Preoperative	69% diagnosed with CT	The authors state that the
		diagnosis	100% diagnosed with	most appropriate
			MRI	treatment for
				asymptomatic cysts is
		Operative findings	41% adhesions, 6%	controversial but due to
			complicated	diagnostic limitations and
				the possibility of
		Postoperative	No postoperative	symptoms arising or
		complications	complications.	malignant transformation
			No recurrence	that surgery is advocated

		Complications of	None in one patient
		incomplete resection	where residual tissue
			ablated with
			electro-cautery

		Complications of	No patients
		conservative	conservatively managed
		management
Cioffi et al., Chest,	27 adults of whom	Presenting complaint	50% chest pain, 13%	Conclude that all patients
1998, Italy, [5]	16 with bronchogenic		cough, 6% epigastric	should have surgical
	cyst from one centre,		pain.	resection for definitive
Retrospective	over 20 years		44% asymptomatic	diagnosis and to
cohort study (2b)			(incidental finding)	minimise complications
	Four-year median			that might arise from
	postoperative follow-up	Preoperative	100% preoperatively	symptomatic cysts
		diagnosis	diagnosed with CT, EUS

		Postoperative	No postoperative
		complications	complications

		Complications of	No patients
		conservative	conservatively managed
		management

Aktogu et al., Eur	31 patients (30 adults	Presenting complaint	19% superior vena cava	Thirteen patients
Respir J, 1996,	and one paediatric) from		syndrome, tracheal	symptomatic at the time
Turkey, [6]	one centre, over		compression,	of surgery had complex
	19 years		pneumothorax, pleurisy	peri-cystic adhesions or
Retrospective			or pneumonia.	fistulisation.
cohort study (2b)	Follow-up 2–10 years		81% cough, infection,	Surgical treatment of
	postoperatively		pain, dyspnoea, anorexia/	asymptomatic cysts is
			weight loss, haemoptysis	recommended to avoid
				potentially
		Preoperative	19% asymptomatic	life-threatening
		diagnosis	(incidental finding).	complications and for
			71% undiagnosed with	definitive diagnosis
			CT

		Postoperative	No recurrence
		complications

		Complications of	Two of six initially
		conservative	asymptomatic patients
		management	either had cyst
			enlargement or became
			symptomatic

Ribet et al., Ann	41 patients from one	Presenting complaint	80% symptomatic	Uncertain what
Thorac Surg, 1996,	centre, including 20		(cough, pain, purulent	proportion of
France, [7]	paediatric cases, over		sputum, haemoptysis and	bronchogenic cysts
	25 years		dyspnoea) in adult life	remain asymptomatic
Retrospective				and long-term prognosis
cohort study (2b)	2.6-year mean	Preoperative	45% undiagnosed	unpredictable.
	postoperative follow-up	diagnosis		Preventative surgery
				recommended
		Postoperative	5% (one patient)
		complications	bronchial fistula.
			No recurrence in 88%
			(remainder lost to
			follow-up).
			No deaths

		Complications of	15% (three patients)
		conservative	followed for 11 months,
		management	five years and unknown
			length of time since
			diagnosis developed no
			symptoms but were
			operated on.
			15% (three patients)
			initially with cough,
			recurrent bronchitis and
			no symptoms
			(respectively) refused
			surgery but lost to
			follow-up


Cuypers et al., Eur	20 adult patients from	Presenting complaint	30% symptomatic	The authors found the
J Cardiothorac	one centre, over		(pneumonia, abscess,	risk of malignancy and
Surg, 1996,	18 years		dysphagia and cough)	cyst-related
Belgium, [8]			70% asymptomatic	complications to be
	No long-term follow-up			justification for operative
Retrospective		Preoperative	25% undiagnosed after	treatment in all cases of
cohort study (2b)		diagnosis	CT, bronchoscopy,	bronchogenic cysts
			barium swallow or echo
			(all cases had CT with
			dense fluid)

		Postoperative	No postoperative
		complications	morbidity; one case
			histologically associated
			with squamous cell
			carcinoma

		Complications of	No patients
		conservative	conservatively managed
		management

Ribet et al., J	69 patients from one	Presenting complaint	63.7% symptomatic	Due to the frequency of
Thorac Cardiovasc	centre, including 24		pain, respiratory tract	late complications with
Surg, 1995, France,	paediatric cases, over		infection, cough,	asymptomatic cysts and
[9]	25 years		dyspnoea, dysphagia,	the unpredictable
			heartburn)	prognosis, preventative
Retrospective	4.2-year mean			surgery was
cohort study (2b)	postoperative follow-up	Preoperative	22% initially	recommended
		diagnosis	misdiagnosed (11% not
			as bronchogenic cyst;
			11% as bronchogenic
			cyst when actually cystic
			neurogenic tumour,
			benign lymphoma and
			haemolymphangioma)

		Postoperative	13.4% postoperative
		complications	morbidity (infection,
			chylothorax and phrenic
			paresis).
			12% symptoms of pain
			or dyspnoea
			postoperative (7%
			symptomatic
			preoperative, 5%
			asymptomatic)

		Complications of	11% refused operation,
		conservative	only 4% followed up
		management	(two patients): one
			remained asymptomatic,
			one died of generalised
			malignancy of unknown
			origin.
			7% had incomplete
			operations but cyst
			remained stable or had no
			recurrence

Patel et al., Chest,	18 adult patients from	Presenting complaint	44% symptomatic	There was no statistical
1994, USA, [10]	one centre, over		(cough, pain) of which	difference in the
	19-year period		11% serious (dyspnea,	frequency of
Retrospective			infection).	intraoperative difficulties
cohort study (2b)	Follow-up 12		56% asymptomatic	or postoperative
	months to 11 years			complications between
	(mean not given)	Preoperative	37.5% undiagnosed with	asymptomatic and
		diagnosis	CT, angio, USS, FNA,	symptomatic patients.
			barium swallow,	The authors concluded,
			bronchoscopy or	however, that surgery is
			mediastinoscopy	advocated in
		Operative difficulties	No difference in	asymptomatic cysts due
			frequency of operative	to the potential for
			difficulties between	complications, incorrect
			symptomatic and	diagnosis or progression
			asymptomatic patients	to symptoms
			(P=0.0656)

		Postoperative	No difference (P=0.596)
		complications	in complication rate
			between preoperatively
			asymptomatic (14%) or
			symptomatic (27%)
			patients. Complications
			included phrenic nerve
			paresis.
			11% delayed
			complications
			(oesophageal stricture
			and recurrence)

		Complications of	Three out of seven
		conservative	asymptomatic patients
		management	followed up developed
			symptoms; four patients
			lost to follow-up

Suen et al., Ann	42 patients from one	Presenting complaint	50% symptomatic (pain,	Complete excision
Thorac Surg,	centre, over 30 years		cough, fever, dysphagia,	recommended in most
1993, USA, [11]			purulent sputum,	cases to relieve
			haemoptysis, dyspnoea).	symptoms, prevent
Retrospective			26% complicated	complications and
cohort study (2b)			(dysphagia, haemorrhage,	confirm diagnosis
			infection and one patient
			with adenocarcinoma in
			cyst)

		Preoperative	59% no preoperative
		diagnosis	diagnosis (more recent
			cases, better success
			rate)

		Postoperative	5% postoperative
		complications	complications (wound
			infection and C. Diff
			colitis).
			No recurrence.
			No deaths

		Complications of	Two patients treated
		conservative	conservatively (one
		management	follow-up only and one
			drainage) with no
			complications

St-Georges et al.,	86 patients from one	Presenting complaint	72% patients	35/86 (41%) of patients
Ann Thorac Surg,	centre, over 20 years		symptomatic by time of	operated on had major
1991, Canada, [12]			surgery (57%	operative difficulties: all
			progressive symptoms	these were patients with
Retrospective			and 15% acute).	symptoms
cohort study (2b)			53% patients with >1	preoperatively
			symptom (chest pain,
			cough, dyspnoea, fever,	Resection recommended
			sputum, anorexia/weight	as majority of patients
			loss, dysphagia,	eventually develop
			haemoptysis).	symptoms or
			38% with complications	complications
			(fistula with airway,
			ulceration cyst wall,
			haemorrhage, infection,
			bronchial atresia)
		Preoperative	57% patients presumed
		diagnosis	diagnosis at surgery after
			CT and angiography.
			Positive diagnosis never
			made preoperatively

		Operative findings	41% had complicated
			cysts (fistula, ulcer,
			haemorrhage, infection,
			atresia) at operation

		Postoperative	11% intraoperative
		complications	complications (vagal
			trunk division, segmental
			bronchus laceration,
			oesophageal mucosal
			laceration).
			9% postoperative
			complications
			(atelectasis, pleural
			effusion, wound
			infection, transient
			Horner's syndrome,
			respiratory failure
			requiring tracheostomy
			and haemothorax) with
			major postoperative
			complications in
			symptomatic patients

		Complications of	37 patients were
		conservative	followed up
		management	conservatively, and
			13 (35%) were still
			asymptomatic at time of
			operation

Cartmill and	20 patients from one	Presenting complaint	75% symptomatic (chest	Advocate surgery for
Hughes, Aust N Z J	centre, over 10 years.		pain, cough,	symptom relief,
Surg, 1989,	Included some		haemoptysis, dysphagia).	exclusion of malignancy
Australia, [13]	paediatric patients (not		5% serious complications	and prevention of
	known how many)			catastrophic
Retrospective		Preoperative	45% diagnosed with CT,	complications
cohort study (2b)	Mean postoperative	diagnosis	barium swallow and
	follow-up 67 months		aortography

		Postoperative	5% (one patient) multiple
		complications	PE.
			No recurrence.
			No death

		Complications of	5% (one patient) declined
		conservative	surgery initially but
		management	returned for elective
			treatment after six years.
			Reason not given

Coselli et al., Ann	Eight patients from one	Presenting complaint	75% symptomatic	Recommend excision to
Thorac Surg, 1987,	centre, over 11 years		(dysphagia, epigastric	establish diagnosis,
USA, [14]			pain, respiratory distress,	alleviate symptoms and
			dyspnoea, chest pain)	prevent complications
Case series (4)
		Preoperative	With use of CT; operated
		diagnosis	on to confirm diagnosis

		Operative findings	Previously infected cysts
			more difficult and
			hazardous to excise


Ge et al., Chin Med	22 patients from one	Presenting complaint	91% symptomatic (chest	Recommend that
Sci J, 1995, China	centre, over 20 years.		pain, dyspnoea, cough,	asymptomatic
[15]	Included paediatric		fever, infection,	bronchogenic cysts
	patients, but not		dysphagia, haemoptysis)	should be excised
Retrospective	specified how many			because of the high risk
cohort study (2b)		Preoperative	36.4% diagnosed with	of complications,
	Mean postoperative	diagnosis	CT	although this conclusion
	follow-up seven years			was not tested in the
		Postoperative	No recurrence.	study
		complications	No complications.
			No mortality

Takeda et al.,	105 patients (with any	Presenting complaint	40% symptomatic (chest	Heterogenous population.
Chest, 2003, USA	mediastinal cyst) from		pain, dyspnoea, cough,	Data extracted for
[16]	one centre including		fever, sputum,	bronchogenic cysts only.
	45 adults and six		dysphagia, haemoptysis)	Three patients had
Retropective cohort	paediatric patients with			complicated surgery due
study (2b)	bronchogenic cysts,	Preoperative	Could not extract data	to peri-cystic adhesions.
	over 50-year period	diagnosis	but improved diagnostic	Preventative resection
			capabilities of MRI	preferred because of
			acknowledged	unpredictable clinical
				behaviour
		Postoperative	Could not extract data
		complications	but described as
			‘acceptable’ by authors

		Complications of	Two patients refused
		conservative	treatment: outcome not
		management	described

Gursoy et al., Saudi	28 adult patients with	Presenting complaint	71% symptomatic	Six additional patients
Med J, 2009,	preoperative diagnosis		(dyspnoea, chest pain,	excluded as preoperative
Turkey, [17]	of bronchogenic cyst		cough, fever)	diagnosis of
	from one centre over			bronchogenic cyst not
Retrospective	seven years	Preoperative	82.4% correctly	confirmed histologically
cohort study (2b)		diagnosis	diagnosed with CT
	36-month mean			Surgical resection
	postoperative	Postoperative	11% early complication	recommended in
	follow-up	complications	rate (wound infection,	asymptomatic patients
			prolonged air leak,	because of the possibility
			pneumoperitoneum).	of malignant
			7% late complication	transformation and
			(dyspnea,	anatomic complications
			pneumothorax).	of delayed surgery
			No mortality

		Complications of	No patients
		conservative	conservatively managed
		management

Liu et al., Chin Med	50 adult patients with	Presenting complaint	66% symptomatic	Recommend surgical
Sci J, 2009, China,	histopathologically		(cough, chest pain,	resection to confirm
[18]	proven bronchogenic		hemoptysis, dyspnoea,	diagnosis, avoid
	cyst from one centre of		fever, dysphagia,	development of
Retrospective	24 years		paralysis, hoarseness).	symptoms or malignant
cohort study (2b)			26% serious	change. Conclusions
	6.5-year mean		complications	drawn upon findings of
	follow-up			other studies
		Preoperative	40% diagnosed
		diagnosis	preoperatively.
			14% misdiagnosed after
			all investigations

		Postoperative	4% early complication
		complications	(persistent air leak,
			hoarseness).
			No late complications.
			No mortality


		Complications of	No patients
		conservative	conservatively managed
		management

Kosar et al., Heart	29 patients from one	Presenting complaint	86% symptomatic	The authors suggest
Lung Circ, 2009	centre, including 13		(cough, sputum, pain,	there is a ‘general
Turkey, [19]	paediatric patients,		breathlessness,	consensus’ that all
	over 15 years treated		haemoptysis, fever)	bronchogenic cysts
Retrospective	with either resection			should be operated on to
cohort study (2b)	or de-epithelialisation	Preoperative	89.7% diagnosed	avoid development of
		diagnosis	preoperatively using	symptoms or
			predominantly CT	complications

		Postoperative	17% early complication
		complications	rate (pneumonia, wound
			infections, prolonged air
			leak); higher in
			complicated cysts.
			No late complications.
			No mortality.
			Recurrence in
			de-epithelialised group

		Complications of	No patients
		conservative	conservatively managed
		management

Limaïauiem et al.,	33 patients from one	Presenting complaint	94% symptomatic (chest	Management of all
Lung, 2008,	centre over six years		pain, cough,	bronchogenic cysts
Tunisia, [20]			haemoptysis, dyspnoea,	based on complete
	Follow-up between		fever, dysphagia)	surgical excision.
Retrospective	one and 51 months			Conclude that definitive
cohort study (2b)		Preoperative	Correct diagnosis in	diagnosis is by histology
		diagnosis	33.3%	only and that complete
				surgical excision is
		Postoperative	14% Early complications	mandatory, although
		complications	(pneumothorax,	conclusions based on
			haemorrhage, pleural	findings of other studies
			effusion, seizure).
			No late complications.
			No mortality

		Complications of	No patients
		conservative	conservatively managed
		management

Granato et al.,	30 adult patients treated	Presenting complaint	30% symptomatic	Two symptomatic cysts
Asian Cardiovasc	for bronchogenic cysts		(cough, sputum, pain,	complicated
Thorac Ann, 2009,	from one centre, over		fever, weakness)	intraoperatively by severe
Italy, [21]	32 years			adhesions
		Preoperative	Correct diagnosis in
Retrospective		diagnosis	100% (CT or CT and	One case of large-cell
cohort study (2b)			MRI)	anaplastic carcinoma in
				wall of cyst
		Postoperative	10% intraoperative
		complications	complications.	Excision of
			10% postoperative	asymptomatic cysts
			complications	advocated to avoid
				complex surgery and
		Complications of	No patients	complications, and also
		conservative	conservatively managed	to reduce malignant
		management		potential

De Giacomo et al.,	30 adult patients from	Presenting complaint	37% symptomatic	Authors feel that patients
Eur J Cardiothorac Surg	one centre over		(cough, pain, dysphagia)	cannot be completely
2009, Italy, [22]	12 years			assured about
		Postoperative	None	conservative
Retrospective	Follow-up	complications		management but
cohort study (2b)	3–120 months			acknowledge the

		Complications of	Asymptomatic patients	management is
		conservative	requested surgery	controversial
		management	because of enlarging
			cysts, risk of
			complication or fear of
			malignancy

Costa Júnior Ada	60 patients with	Presenting complaint	92% symptomatic	Heterogenous group of
et al., J Bras	pulmonary		(cough, dyspnoea, pain,	patients and disorders.
Pneumol, 2008,	malformations		infection)	Difficult to extract
Brazil, [23]	(including 27 with			bronchogenic cyst data
	bronchogenic cyst)	Preoperative	‘Frequent’ misdiagnosis
Retrospective	from one centre	diagnosis		Prognosis noted to be
cohort study (2b)	over 35 years,			unpredictable. One
	including 40 paediatric	Postoperative	23% (pneumonia,	patient found to have
	patients	complications	atelectasis, empyema,	adenocarcinoma in wall
			sepsis).	of cyst
			3.3% mortality

		Complications of	Diagnosis/treatment
		conservative	delayed in three patients
		management	up to 36 months (mean
			15): outcome in these
			patients not specified

Weber et al.,	12 patients from a	Presenting complaint	42% symptomatic	Agree that management
Ann Thorac Surg,	single centre		(cough, pain, pneumonia)	of asymptomatic cysts is
2004, Switzerland,	undergoing			controversial but that
[24]	video-assisted	Preoperative	100% correct diagnosis	there appears to be no
	thoracoscopic surgery	diagnosis	with CT with or without	need for urgent surgery
Retrospective	for bronchogenic		MRI. MRI noted to be	in these cases provided
cohort study (2b)	cysts, over seven years		superior	that a simple cyst has
				been clearly diagnosed
	40.5-month mean	Postoperative	None
	follow-up	complications

		Complications of	Six patients observed for
		conservative	between two and
		management	22 years without
			complications.Three
			developed mild
			symptoms.
			Three patients eventually
			requested surgery
			because of fear of
			malignancy/complications
			or enlarging cyst

CT, computed tomography; EUS, endoscopic ultrasound; USS, ultrasound scan; FNA, fine needle aspiration.

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6. Results

The prevalence rates for bronchogenic cysts are not known, with this rare condition thought to remain largely undiagnosed in an asymptomatic population. From the papers reviewed, the incidence was, on average, approximately 30 cases per institution (population sizes unknown) over a 20-year period (approx. 11–55). Some cysts were found incidentally (6–79%) but the remainder presented with a variety of symptoms including chest pain, dyspnoea, haemoptysis and recurrent chest infections. Rarely, the presentation was of a serious complication of the cysts, such as sepsis or compressive symptoms.

In total, the papers identified for this systematic review included 683 adult patients with bronchogenic cysts of whom 74 were either treated conservatively, or had diagnosis and/or treatment delayed up to 22 years. Of those treated conservatively, 31 (45%) subsequently developed symptoms and proceeded to surgical treatment. A number of studies determined that complicated bronchogenic cysts (for example ruptured or infected at the time of surgery) were associated with intraoperative difficulties. Only one study [10] compared the frequency of postoperative complications between patients who were symptomatic or asymptomatic preoperatively (27% and 14%, respectively), and found that these were not significantly different (P=0.596). There was no evidence in any of the reviewed papers that prolonged observation increased the incidence of complications.

Historically, it has been appropriate to operate on these asymptomatic bronchogenic cysts for both diagnostic and therapeutic purposes and the majority of the studies cited did proceed to operate on patients, both with and without symptoms. Ponn [25] commented on this dogmatic approach to tradition, making reference to a number of the studies described here [9, 12, 16]. With inclusion of only surgical cases in several of the smaller studies, the data presented is likely to be skewed.

Bronchogenic or benign cyst was diagnosed preoperatively in 50–100% of cases. Several studies made reference to the improvement in imaging over the last two decades, in particular to the benefits of magnetic resonance. Patients investigated in the latter part of studies were often noted to have more accurate preoperative diagnosis than those investigated 20 years earlier. Kanemitsu et al. [4] scanned all patients with MRI and reported 100% success rate in preoperative diagnosis, while Granato et al. [21] and Weber et al. [24] also commented on the superior diagnostic ability of MRI. In studies in which computerised tomography was used with or without other diagnostic procedures including ultrasound and mediastinoscopy, the diagnosis was made preoperatively with varying degrees of success.

Postoperative complications in the studies ranged from 0 to 27%. In patients who did suffer complications of surgery, the majority were generic operative complications with two cases attributed directly to cysts. In five patients (0.7% of all those studied), bronchogenic cysts were associated with malignancies: one squamous cell, one adenocarcinoma, two bronchoalveolar carcinomas and one large-cell anaplastic carcinoma. It was not stated whether these were due to malignant transformation or incidental finding, but the malignancies were usually found in the cyst wall.

Surgical excision of bronchogenic cysts has historically been performed for three main reasons:

to confirm diagnosis;
to prevent development of symptoms and/or complications and to pre-empt the possibility of surgery on complex inflammatory lesions;
to avoid any potential for malignant transformation.

Advances in non-invasive diagnostic techniques, in particular the role of MRI, may have rendered the first reason obsolete.

Nonetheless, the literature available demonstrates that the traditional approach of surgical treatment of all bronchogenic cysts gives acceptable results with low rates of recurrence, morbidity and postoperative mortality. Approximately half of patients who are asymptomatic at presentation will eventually go on to develop symptoms and/or complications, with a body of evidence which suggests that complications make surgery more challenging. No studies, however, have demonstrated that this is associated with a worse postoperative outcome than that of elective procedures.

Because of these concerns, only a very small proportion of the patients studied were followed up with conservative management instead of surgical excision (74/683).

7. Clinical bottom line

While there is good evidence to support the excision of symptomatic cysts, the evidence for conservative management of asymptomatic bronchogenic cysts is very limited, both because the majority of bronchogenic cysts present with bothersome symptoms and because conventional treatment has tended towards surgical management. Concerns that these benign entities may develop complications are not unfounded as 45% of asymptomatic patients studied here went on to develop symptoms. However, the success, morbidity and mortality of surgery are no different if operated on whilst asymptomatic or once complications have arisen. The other major concern – of missed malignancy – should be weighed in balance of its 0.7% risk, compared with the approximately 20% morbidity of surgery. Many surgeons have and continue to recommend surgery to asymptomatic adults with bronchogenic cysts and the literature would suggest this to be an acceptable approach. However, conservative management is a potentially viable alternative if close follow-up is possible.

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Article Contents

Should asymptomatic bronchogenic cysts in adults be treated conservatively or with surgery?

Abstract

1. Introduction

2. Three-part question

3. Clinical scenario

4. Search strategy

5. Search outcome

6. Results

7. Clinical bottom line

References

Citations

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Article Contents

Should asymptomatic bronchogenic cysts in adults be treated conservatively or with surgery?

Abstract

1. Introduction

2. Three-part question

3. Clinical scenario

4. Search strategy

5. Search outcome

6. Results

7. Clinical bottom line

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