Surgical treatment of an esophageal bronchogenic cyst with massive upper digestive tract hematoma without esophagectomy: a case report and the review of the literature

Figures & data

Figure 1 Preoperative imaging diagnoses. (A) Chest CT scan at the day before admission shows a smooth cystic tumor measuring 67 × 51 × 60 mm (white arrow). CT value was 28 HU. The esophagus was compressed by the cystic tumor. (B) An abdominal CT at the day before admission revealed a normal shape of the stomach (white arrow). (C) Transverse contrast-enhanced chest CT at 5 days after admission showed the sharply defined mass measuring 47 × 70 × 85 mm (white arrow). CT value was 31 HU. (D) Transverse contrast-enhanced abdominal CT at 5 days after admission showed a huge contusion of the stomach wall (black arrow).

Figure 2 Gastroscopy images. (A, B) After admission, gastroscopy showed a mass with a smooth surface causing a partial obstruction of the lower esophagus and stomach fundus. (C, D) The day before admission, gastroscopy showed a mass with a smooth surface causing partial obstruction of the lower esophagus and a normal stomach fundus.

Abbreviations: E, esophagus; G, gastro.

Figure 3 Intraoperative images. (A) Brown fluid was observed in the bronchogenic cyst after dissection of the cystic wall. (B) The nutrient artery (arrow) supplying the cyst was revealed distinctly. (C) The shape of the bronchogenic cyst was revealed. (D) Because of the gastric hematoma, a violet surface (arrow) was observed after incision of the diaphragm.

Figure 4 Immunohistochemical and histopathological examination of the cyst sections. (A) Positive expression of CA199, original magnification ×200. (B) Positive expression of CA125, original magnification ×200. (C, D) The cystic wall was lined with ciliated columnar epithelium. The wall also contained cartilage and bronchogenic glands (H&E stained, original magnification ×40).

Abbreviation: CA, carbohydrate antigen.

Figure 5 (A) A control esophagogram showed that the esophagus (black arrow) had neither stenosis nor leakage. (B) The shape and function of the stomach (white arrow) were normal.

Table 1 Summary of published cases with periesophageal or intramural esophageal bronchogenic cysts

Study (year)	Patient age, years	Gender	Symptoms	Location	Size	Treatment	Follow-up duration	Outcome at last follow-up
Sashiyama et al^Citation10 (2002)	34	F	Dysphagia	Submucosal cyst in middle third of esophagus	5 cm diameter	Endoscopy	1 month	Uneventful
Hallani et al^Citation11 (2004)	64	M	Chest pain	Distal esophagus	NS	Thoracotomy	2 years	Uneventful
Westerterp et al^Citation12 (2004)	67	M	Odynophagia	Intraesophageal submucosal	6.6 cm diameter	Transthoracic esophagectomy	2 weeks	Uneventful
	49	F	Dysphagia		2.9 cm diameter	Endoscopic mucosal resection	1 year	Uneventful
	49	F	Asymptomatic		3.3 cm diameter	Local enucleation	1 year	Uneventful
Melo et al^Citation13 (2005)	39	F	Rib pain	Gastric fundus	4×2.5×1 cm	Laparoscopy	NS	Uneventful
Pages et al^Citation14 (2005)	25	F	Back pain	Centro-posterior mediastinum, paraesophageal cyst, compressing esophagus w/fistulization into esophageal lumen	3 cm diameter	VATS with conversion to open posterolateral thoracotomy	3 weeks	Uneventful
Ko et al^Citation15 (2006)	19–60	6F/1M	Dysphagia and chest pain	Mid-thoracic/lower thoracic	3–4 cm diameter	Thoracotomy/VATS	1–14 years	Uneventful
Akutsu et al^Citation16 (2006)	26	M	Epigastral discomfort and mild dysphagia	Lower esophagus	0.1 cm in diameter	Thoracoscopy	NS	Uneventful
Chuang et al^Citation17 (2007)	42	M	Progressive dysphagia	Middle and lower 1/3 of esophagus	6.5×4×4 cm	L posterolateral thoracotomy	2 years	Uneventful
Grover et al^Citation18 (2007)	85	F	Dysphagia	Subcarina, communicating with esophageal lumen	7–8 cm diameter	R posterolateral thoracotomy	NS	Uneventful
Turkyilmaz et al^Citation19 (2007)	48	M	Dysphagia	Distal esophagus	3×2×1.5 cm	R thoracotomy	6 months	Uneventful
Eom et al^Citation20 (2007)	18	M	Dry cough and mild dyspnea	Middle mediastinum connected to the esophagus by a tubular esophageal duplication	3×2 cm	NS	NS	Uneventful
Kiral et al^Citation21 (2008)	44	M	Cough and hemoptysis	Paravertebral, communication between cyst and esophagus	3 cm diameter	Left thoracotomy, lower lobectomy	3 years	Uneventful
Rubin et al^Citation22 (2009)	27	F	NS	Mediastinal bronchogenic cyst perforating into esophagus	NS	Left thoracotomy	2 years	Diaphragmatic rupture
Diaz Nieto et al^Citation23 (2010)	67	M	Chronic low back pain	Lower esophagus, intra- abdominal, attached to the GE junction	6 cm diameter	Laparoscopy	NS	Uneventful
Chafik et al^Citation24 (2011)	51	M	Dysphagia and pain in the right hemithorax	Lower thorax	3.6×3.1 cm	Thoracotomy and enucleation	2 years	Uneventful
Fernandez et al^Citation25 (2011)	33	M	Intermittent epigastric and right upper quadrant pain and burning	Lesser sac near GE junction	4.3×1.7 cm	Laparoscopy	NS	Chronic chest wall pain
Barbetakis et al^Citation26 (2011)	46	M	Progressive dysphagia	Distal esophagus	NS	VATS	5 days	Uneventful
Wang et al^Citation2 (2012)	56	M	Chest pain and dysphagia	Posterolateral thoracotomy	8×7×7 cm	Lower paraesophageal segment thoracotomy	2 years	Uneventful
Ghobakhlou et al^Citation27 (2012)	23	F	Progressive dysphagia	Lower third of esophagus	3×3 cm	Thoracotomy and enucleation	NS	Uneventful
Vannucci et al^Citation4 (2013)	39	F	Dyspnea	Thoracoabdominal	25 cm diameter	R thoracotomy	3 years	Uneventful
Ballehaninna et al^Citation28 (2013)	40	F	Dysphagia	GE junction	3.5×3 cm	Laparoscopy	6 months	Uneventful
Tang et al^Citation29 (2014)	23	M	Chest discomfort and dyspnea	Distal esophagus	1.1 × 2.5 cm	Endoscopic submucosal tunnel dissection	Not mention	Uneventful
Gou et al^Citation30 (2015)	24	M	Abdominal bloating	Hepatogastric ligament	3.8×2.6 cm	Laparoscopic surgery	NS	NS
Altieri et al^Citation31 (2015)	40	F	Abdominal pain	GE junction	3 cm	Laparascopic surgery	2 weeks	Uneventful
Trehan et al^Citation32 (2015)	34	F	Heaviness in right flank	Intra-abdominal	10×6 cm	Laparoscopic surgery	2 weeks	Uneventful
Han et al^Citation33 (2016)	31	M	Dysphagia	Mediastinum	4.5×7.3 cm	R thoracotomy	3 months	Uneventful
Lin et al^Citation34 (2017)	20 months	M	Nonbilious emesis	GE junction	1.2×1.0×0.4 cm	Laparoscopic heller myotomy	7 months	Uneventful

Abbreviations: M, male; F, female; NS, not specified; VATS, video-assisted thoracoscopic surgery; GE, gastroesophageal; R, right; L, Left.

SashiyamaHMiyazakiSOkazakiYEsophageal bronchogenic cyst successfully excised by endoscopic mucosal resectionGastrointest Endosc200256114114512085056

 PubMed Web of Science ®Google Scholar

HallaniHEslickGDCoxMWyattJMLeeCHChest pain? CauseLancet2004363940745214962526

 PubMed Web of Science ®Google Scholar

WesterterpMvan den BergJGvan LanschotJJFockensPIntramural bronchogenic cysts mimicking solid tumorsEndoscopy200436121119112215578307

 PubMed Web of Science ®Google Scholar

MeloNPitmanMBRattnerDWBronchogenic cyst of the gastric fundus presenting as a gastrointestinal stromal tumorJ Laparoendosc Adv Surg Tech A200515216316515898909

 PubMed Web of Science ®Google Scholar

PagesONRubinSBaehrelBIntra-esophageal rupture of a broncho-genic cystInteract Cardiovasc Thorac Surg20054428728817670412

 PubMedGoogle Scholar

KoSFHsiehMJLinJWHuangCCLiCCCheungYCNgSHBronchogenic cyst of the esophagus: Clinical and imaging features of seven casesClin Imaging200630530931416919550

 PubMed Web of Science ®Google Scholar

AkutsuYMatsubaraHHayashiHOkazumiSAokiTKozuTOchiaiTEndoscope-assisted thoracoscopic technique for esophageal bronchogenic cyst which presented elevated CA 125Dig Surg200623420921416874000

 PubMed Web of Science ®Google Scholar

ChuangKHHuangTWChengYLEsophageal bronchogenic cyst: A rare entityZ Gastroenterol200745995896017874358

 PubMed Web of Science ®Google Scholar

GroverMGuptaAWagnerDPOrringerMBHard to swallowAm J Med2007120121023102518060919

 PubMed Web of Science ®Google Scholar

TurkyilmazAErogluASubasiMFindikGIntramural esophageal bron-chogenic cysts: A review of the literatureDis Esophagus200720646146517958719

 PubMed Web of Science ®Google Scholar

EomDWKangGHKimHWRyuDSUnusual bronchopulmonary foregut malformation associated with pericardial defect: bronchogenic cyst communicating with tubular esophageal duplicationJ Korean Med Sci200722356456717596673

 PubMed Web of Science ®Google Scholar

KiralHTezelCSKosarAKelesMClinicopathologic demonstration of complex bronchopulmonary foregut malformationAnn Thorac Surg20088562114211618498835

 PubMed Web of Science ®Google Scholar

RubinSSanduSDurandEBaehrelBDiaphragmatic rupture during labour, two years after an intra-oesophageal rupture of a bronchogenic cyst treated by an omental wrappingInteract Cardiovasc Thorac Surg20099237437619423509

 PubMedGoogle Scholar

Diaz NietoRNaranjo TorresAGómez AlvarezMIntraabdominal bronchogenic cystJ Gastrointest Surg201014475675819475460

 PubMed Web of Science ®Google Scholar

ChafikABenjellounAQassifHIntramural esophageal bron-chogenic cystsAsian Cardiovasc Thorac Ann2011191697121357324

 PubMedGoogle Scholar

FernandezJLBauzaGMcAnenyDBMinimally invasive management of lesser sac bronchogenic cystJSLS201115457157422643521

 PubMed Web of Science ®Google Scholar

BarbetakisNAsteriouCKleontasAPapadopoulouFTsilikasCVideo-assisted thoracoscopic resection of a bronchogenic esophageal cystJ Minim Access Surg20117424925222022117

 PubMed Web of Science ®Google Scholar

WangWNiYZhangLWA case report of para-esophageal bronchogenic cyst with esophageal communicationJ Cardiothorac Surg201279423013612

 PubMed Web of Science ®Google Scholar

GhobakhlouMFatemiSRDezfouliAATirgaryFZaliMRLong-term dysphagia due to bronchogenic cyst of the esophagusEndoscopy201244Suppl. 2E129E13022477181

 PubMed Web of Science ®Google Scholar

VannucciJPecorielloRTassiVCeccarelliSPumaFGiant tho-racoabdominal esophageal bronchogenic cystDis Esophagus201326334022236421

 PubMed Web of Science ®Google Scholar

BallehaninnaUKShawJPBrichkovISubdiaphragmatic bronchogenic cyst at the gastroesophageal junction presenting with dysphagia: A case reportSurg Laparosc Endosc Percutan Tech2013234e170e17223917610

 PubMed Web of Science ®Google Scholar

TangXJiangBGongWEndoscopic submucosal tunnel dissection of a bronchogenic esophageal cystEndoscopy201446Suppl. 1E626E62725502273

 PubMed Web of Science ®Google Scholar

GouYWangYFangHXuXYuWZhangKYuYBronchogenic cyst in the hepatogastric ligament masquerading as an esophageal mesenchymal tumor: A case reportInt J Clin Exp Pathol2015811153071531126823885

 PubMed Web of Science ®Google Scholar

AltieriMSZhengRPryorADHeimannAAhnSTelemDAEsophageal bronchogenic cyst and review of the literatureSurg Endosc201529103010301525669636

 PubMed Web of Science ®Google Scholar

TrehanMSinglaSSinghJGargNMahajanAA rare case of intra-abdominal bronchogenic cyst – a case reportJ Clin Diagn Res2015911PD03PD04

 Google Scholar

HanCLinRYuJA case report of esophageal bronchogenic cyst and review of the literature with an emphasis on endoscopic ultra-sonography appearanceMedicine (Baltimore)20169511e311126986156

 PubMed Web of Science ®Google Scholar

LinJSYuYRChiouEChumpitaziBPSchadyDBrandtMLIntramural esophageal bronchogenic cyst mimicking achalasia in a toddlerPediatr Surg Int201733111912327822780

 PubMed Web of Science ®Google Scholar