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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 2  |  Page : 65-67

Uncommon etiology of upper GI bleed


Department of Medical Gastroenterology, Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu, India

Date of Submission12-Dec-2020
Date of Decision01-Dec-2021
Date of Acceptance15-Dec-2021
Date of Web Publication23-Mar-2022

Correspondence Address:
Hema Vijayalakshmi Varadarajulu
No. 18, III Main Road, Nanganallur, Chennai - 600 061, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ghep.ghep_34_20

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  Abstract 


Abnormal fistulous communications between the esophagus and respiratory tract are life-threatening conditions which need to be diagnosed at the earliest and managed appropriately. Bronchoesophageal fistula (BEF) is a rare endoscopic finding. It is commonly due to esophageal malignancy in adults, benign etiologies being very rarely encountered during endoscopy. In this case report, we discuss a BEF arising from a mid-esophageal diverticulum, which presented with recurrent hematemesis.

Keywords: Benign bronchoesophageal fistula, congenital benign bronchoesophageal fistula, etiopathogenesis, mid-esophageal diverticulum, upper gastrointestinal bleed-recurrent hematemesis


How to cite this article:
Varadarajulu HV. Uncommon etiology of upper GI bleed. Gastroenterol Hepatol Endosc Pract 2022;2:65-7

How to cite this URL:
Varadarajulu HV. Uncommon etiology of upper GI bleed. Gastroenterol Hepatol Endosc Pract [serial online] 2022 [cited 2022 May 24];2:65-7. Available from: http://www.ghepjournal.com/text.asp?2022/2/2/65/340389




  Introduction Top


The incidence of upper gastrointestinal (UGI) bleed of unknown etiology is declining and uncommon etiologies are increasingly recognized following further investigations.[1] Here, we present one such rare etiology of recurrent hematemesis.


  Case Report Top


A 39-year-old female presented with recurrent episodes of small quantity (approximately 10–20 ml/episode) hematemesis, one episode every 2–3 months. The hematemesis was not associated with melena or loss of consciousness. There was no history of dyspeptic symptoms, prior nonsteroidal anti-inflammatory drug intake, epigastric or chest pain, or symptoms suggestive of chronic liver disease. She was treated as an outpatient by her family physician with injections and did not require hospitalization. Index UGI endoscopy, done elsewhere after 6 months of onset of symptoms, reported a one cm superficial ulcer in anterior wall of the duodenal bulb with no stigmata of bleeding and a mid-esophageal diverticulum (ED). She was treated for the duodenal ulcer (DU) but the bleed persisted. Further questioning revealed a productive cough with minimal secretions preceding the bleed, occasional aspiration on ingestion of both liquids and solids, and no hemoptysis.

She had mild pallor, hemoglobin of 10.2 gm/dL and chest X-ray showed pneumonitis in the left lung. UGI endoscopy in our center showed a healed DU and the ED at 24 cm from the incisor teeth was wide-necked with a clean base and on closer inspection showed a fistulous opening about 3–4 mm in size [Figure 1].
Figure 1: Endoscopy showing a wide-necked esophageal diverticulum with a clean base and a fistulous opening

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A nonenhanced computed tomography (CT) scan was done initially which did not identify the fistula. An esophagogram with barium contrast was done subsequently which confirmed an esophagobronchial fistula and pneumonitis of the left lower lobe which was treated with antibiotics [Figure 2] and [Figure 3]. Bronchoscopy localized the fistulous opening in the left main bronchus just beyond the tracheal bifurcation with minimal mucopurulent secretions, negative for malignancy and tuberculosis, and an otherwise unremarkable bronchial airway [Figure 4]. Spurious hematemesis due to the suppurative process of the lung and entering the esophagus through bronchoesophageal fistula (BEF) in the ED was diagnosed in this patient. She was referred to a higher center for further management.
Figure 2: Barium swallow oblique view showing mildly irregular anterior wall and minimal posterior barium leakage

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Figure 3: (a and b) Coronal and axial section of nonenhanced computed tomograpraphy thorax (mediastinal window) shows barium outlining the left main bronchus lumen and adjacent lung parenchyma

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Figure 4: Bronchoscopy showing the fistulous opening in the left main bronchus, mucopurulent secretions, and a normal mucosa

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  Discussion Top


Esophagobronchial or BEF, terms describing a rare esophagorespiratory fistula (ERF), is most frequently seen in adults as a complication of malignancy-esophageal and pulmonary. BEF has also been reported in a diverse range of benign etiologies including ED of mid and lower esophagus.[2]

Mid-ED (MED) or parabronchial diverticulum constitute 15% of ED and anatomically defined as occurring within 5 cm of the carina. Often asymptomatic and detected incidentally, larger diverticulum may present with dysphagia, chest pain or regurgitation, or rarely with complications like hematemesis or fistula. Unlike Zenker's (pharyngoesophageal) and lower esophageal (epiphrenic) diverticulum which are mostly acquired pulsion type, MED comprises diverticula of different etiologies: acquired (traction and pulsion types) being more common than congenital type. Other than food stasis in diverticulum as a common factor, the pathogenesis of fistula in MED varies with each etiology.

In traction or true diverticulum (consisting of entire esophageal wall), extrinsic pull on the esophagus by chronic inflammatory diseases like tuberculosis involving the lung, mediastinum and lymph nodes, further necrosis of nodes and rupture into the esophagus leads to a fistula.[3] In pulsion type ED, high pressures generated in esophageal motility disorders cause mucosal eventration in weak spots through the muscle to create a pseudodiverticulum (consisting of mucosa and submucosa only).[4]

Recognition of congenital BEF presenting for the first time in adults led Braimbridge and Keith in 1965 to classify fistula in the base of a wide-necked diverticulum as type I congenital benign BEF (BBEF).[5] They are 25%–50% less common than tracheoesophageal fistula, compatible with normal life in the absence of esophageal atresia, and arise due to incomplete separation of primordial tracheal-esophageal components. Later onset of symptoms is attributed to oblique direction of the tract, lower position, and occlusion of the esophageal opening by mucosal fold, membrane, or muscle spasm during swallowing.[6] Congenital bronchopulmonary foregut malformations are also associated with ED with fistula to sequestered pulmonary segments and associated anomalies.

BEF is reported in all ages due to the varying etiopathogenesis and 76% of congenital BBEF are diagnosed after 17 years of age.[6] Both sexes are equally affected.

Patients with ERF present as recurrent pneumonia, bronchiectasis, lung abscesses or hemoptysis due to recurrent bronchopulmonary aspiration. The only symptom may be bouts of cough or choking during eating and drinking, known as Ono's sign which is noted in 65% of patients but subtle and recognized retrospectively in others. Hematemesis though reported is an exceedingly rare presentation and can occur with or without respiratory symptoms.[6] Massive hematemesis requiring resuscitation and urgent endotherapy or surgical management has been reported due to ulcers, erosions, or arteriovenous (AV) malformation in the base of the diverticulum. Erosion of adjacent bronchial arteries and suppuration of the lung are other contributing factors of UGI bleeding.[7],[8],[9] Clinically, patients may appear normal or have features of chronic lung diseases.

Esophagogram with diluted contrast is still the preferred method to diagnose BEF. UGI endoscopy and bronchoscopy may miss small openings and methylene blue instillation in esophagus during bronchoscopy may increase detection rate. CT scan chest with contrast confirms and rules out other lesions. Rarely, fistula is detected perioperatively. The absence of surrounding inflammation, adherent lymph nodes, and presence of an intact mucosa, submucosa on histopathological examination of the tract differentiates congenital from an acquired BEF.

In our patient, hematemesis was the only complaint without symptoms pertaining to the respiratory tract or esophagus. Ono's sign was elicited after identification of the fistula. BEF was diagnosed by all four modalities and is probably Braimbridge type 1 congenital BBEF or pulsion diverticulum complicated by fistula (though esophageal motility study was not done). The absence of visible bleeding during both UGI endoscopy and bronchoscopy might be due to the time interval between the patient presenting with the bleed and undergoing a UGI endoscopy and the course of antibiotics received during the hospitalization. In the absence of ulcers, erosions or AV malformations in the base of ED, the recurrent minor hematemesis in our patient is most likely a spurious hematemesis due to either suppuration of the lung or bleeding from erosion of adjacent arteries entering the esophagus through the fistula. CT angiography may have helped in identifying the origin of the bleed but was not done in this patient.

The management includes initial endotherapy for arresting the UGI bleed. Resolution of tuberculous fistula by antituberculosis therapy has been reported in few patients.[3] Surgery-diverticulectomy, fistulectomy, and closure of defects is the treatment of choice.[10] Less invasive endotherapy is gaining in popularity and has evolved from obliteration of the tract by injection of sclerosants, fibrin glue, and stents to argon plasma coagulation and over-the-scope-clips.[11] Factors affecting endotherapy are being studied.


  Conclusion Top


This case report attempts to highlight the various causes and evaluation required in a case of hematemesis in a MED with and without a fistula, in which endotherapy or in some cases radiological intervention might be successful rather than the more morbid but definitively curative surgical treatment.

Acknowledgements

  1. Gopinath Ganesan MD, DNB (docgopi87@gmail.com), Assistant Professor, Department of Radiology, Saveetha Medical College and Hospital, Radiologist for interpretation of CT chest and Barium Swallow investigations and providing the appropriate pictures
  2. Sreedevi Kotha, MD (drsreedevikotha@gmail.com), Assistant Professor, Department of Chest medicine, Saveetha Medical College and Hospital, Pulmonologist for bronchoscopic evaluation of the patient and providing pictures of the same.


Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Hreinsson JP, Kalaitzakis E, Gudmundsson S, Björnsson ES. Upper gastrointestinal bleeding: Incidence, etiology and outcomes in a population-based setting. Scand J Gastroenterol 2013;48:439-47.  Back to cited text no. 1
    
2.
Hendry P, Crepeau A, Beatty D. Benign bronchoesophageal fistulas. J Thorac Cardiovasc Surg 1985;90:789-91.  Back to cited text no. 2
    
3.
Narayanan S, Shiji PV, Majeed KA, Udayabhaskaran V. Tuberculosis presenting as bronchoesophageal fistula. IDCases 2017;8:19-21.  Back to cited text no. 3
    
4.
Kaye MD. Oesophageal motor dysfunction in patients with diverticula of the mid-thoracic oesophagus. Thorax 1974;29:666-72.  Back to cited text no. 4
    
5.
Braimbridge MV, Keith HI. Oesophago-bronchial fistula in the adult. Thorax 1965;20:226-33.  Back to cited text no. 5
    
6.
Kim JH, Park KH, Sung SW, Rho JR. Congenital bronchoesophageal fistulas in adult patients. Ann Thorac Surg 1995;60:151-5.  Back to cited text no. 6
    
7.
Ballehaninna UK, Shaw JP, Brichkov I. Traction esophageal diverticulum: A rare cause of gastro-intestinal bleeding. Springerplus 2012;1:50.  Back to cited text no. 7
    
8.
Kishi K, Kusunoki R, Fujishiro H, Suemitsu S, Kataoka M, Fujiwara A, et al. Mid-esophageal diverticular bleeding in a patient with kyphosis. Intern Med 2019;58:3239-42.  Back to cited text no. 8
    
9.
Alharbi SR. Tuberculous esophagomediastinal fistula with concomitant mediastinal bronchial artery aneurysm-acute upper gastrointestinal bleeding: A case report. World J Gastroenterol 2019;25:2144-8.  Back to cited text no. 9
    
10.
Sun Y, Hao S, Yang Y, Guo X, Ye B, Zhang X, et al. Surgical management of acquired tracheo/bronchoesophageal fistula associated with esophageal diverticulum. J Thorac Dis 2017;9:3684-92.  Back to cited text no. 10
    
11.
Jha AK, Suchismita A, Gupta N. Endoscopic treatment: Choosing the right method for bronchoesophageal fistula closure? Arch Gastroenterol Hepatol 2018;1:16-8.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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