Background: Tuberculous (TB) oesophagitis is a rare manifestation of dysphagia occurring in 0.3% of all gastro- intestinal tract TB infections as well as 0.15% of all cases of dysphagia and often is misdiagnosed. This report presents a rare manifestation of TB as a cause of oesophageal stricture. Case presentation: We describe a rare presentation of a patient with grade IV dysphagia due to an oesophageal stricture. Oesophagoscopy revealed a pinhole stricture with evidence of high grade dysplasia on histology. Subsequently an Ivor-Lewis oesophagectomy was performed and histology revealed evidence of active oesophageal tuberculosis. The patient had an uneventful recovery and completed anti-TB medication. Conclusions: Oesophageal TB is a rare but curable cause of dysphagia. It may mimic cancer of the oesophagus and it is usually missed as a possible cause of oesophageal strictures. There needs to be an increased index of suspicion among patients with dysphagia in TB endemic regions. Keywords: Tuberculous oesophagitis, Oesophageal stricture, TB stricture Background the 1 year period and also had no history of treatment Tuberculous (TB) oesophagitis is a rare manifestation of for TB. There was no family history of oesophageal dis- dysphagia occurring in 0.3% of all gastro-intestinal tract eases such as cancer. TB infections as well as 0.15% of all cases of dysphagia and often is misdiagnosed . This report presents a Clinical findings rare manifestation of TB as a cause of oesophageal stric- On physical examination, she was in fair general condi- ture. Due to the rarity of this disease, often there may be tion with moderate wasting, she was not anaemic and delays in diagnosis which may as a result be associated neither was she jaundiced. with complications. Occasionally diagnostic challenges Overall, the systemic exam was unremarkable with no may also be encountered however there should be an in- clinically significant findings in all systems. creased index of suspicion in endemic populations. The clinical presentation, diagnosis and treatment are Diagnostic assesment discussed. Initial blood work up included complete blood count, liver function, renal function and serum electrolytes Case presentation which were all normal. She was sero-negative for Hu- Patient information man Immunodeficiency Virus (HIV). We report on a 36-year old house-wife who presented to Imaging investigations that were done included a bar- our hospital with a one-and-half year history of progres- ium swallow which revealed a 5 cm long mid-to-distal sive dysphagia, first to solids and later to liquids. At the third stricture of the oesophagus. Following that, an time of presentation she had Grade IV dysphagia. This upper gastrointestinal endoscopy was done which re- was associated with progressive weight loss and retro- vealed a tight stricture at 30 cm with a pin-hole opening, sternal pain. She had no history of a chronic cough in minimal mucosal inflammation above the stricture with no ulceration or obvious mass. The scope could not be * Correspondence: email@example.com 1 advanced beyond the narrowing (Fig. 1). The initial hist- Department of Surgery, Makerere University, Kampala, Uganda Full list of author information is available at the end of the article ology taken off at the time of endoscopy showed a high © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Mbiine et al. Journal of Cardiothoracic Surgery (2018) 13:58 Page 2 of 5 Fig. 1 Upper GI endoscopy showing a pin-hole stricture with minimal mucosal inflammation at the proximal opening of the stricture. Legend: Fig. 1 a and b show a pin hole stricture of the oesophagus at 30 cm from the upper incisor teeth. Figure 1 c shows a patent oesophagus proximal to the stricture grade dysplasia (TIS). However, due to a delay in under- week following discharge, she had complete healing of taking an oesophageal resection, a subsequent second the surgical incisions without any wound infection. She endoscopy was done with repeat histology revealing a was continuously reviewed to ensure she didnot develop chronic inflammatory process. A staging chest and ab- any short term and long term post surgical complica- dominal CT scan done showed an enhancing and mark- tions. Surgical OPD reviews were conducted first weekly edly thickened oesophageal wall in the area of the for a month and then monthly for 6 months, TB treat- stricture, but no abnormally enlarged intra-thoracic and ment reviews were also done at the time of review of the intra-abdominal lymph nodes nor distant metastases surgical aspects. She took the anti-TB’s for 6 months were seen. and was reviewed monthly for treatment adherence, de- At that point basing on the findings of the initial histo- velopment of side effects. She adhered to her treatment logical findings of a high grade dysplasia, with no evi- and didnot develop any TB treatment complication. She dence of local or distant invasion, the patient was had a complete recovery after the 6 months and has pro- treated worked up and prepared for oesophagectomy. gressively resumed her daily activities. The follow up duration didnot exceed the 6 months following TB treat- Therapeutic intervention ment and as a result the article was authored before fol- Initially, a temporary feeding gastrostomy was placed for low up for any long term complications could be nutritional rehabilitation and full optimisation for sur- assessed. gery. Due to the suspicion of an oesophageal carcinoma and the grade IV dysphagia, the patient was worked up Discussions and conclusions for oesophagectomy. We performed an Ivor-Lewis oeso- Oesophageal stricture is a very rare manifestation of phagectomy (upper midline laparotomy plus right thora- extra-pulmonary tuberculosis globally. Despite the high cotomy with two-stage systematic lymph node prevalence of TB in sub-Saharan Africa, this is the first dissection) with pyloplasty and a feeding jejunostomy. time oesophageal TB stricture is being documented in The resected oesophagus along with the dissected lymph our population. The overall prevalence of TB nodes were sent for histology. The patient was extubated oesophagus is less than 0.3% of all forms of gastrointes- on table and transferred to the ICU where she spent 2 tinal TB . This has been attributed to the rapid clear- days. The results of the histology revealed active TB dis- ance of oesophageal contents during swallowing. It has ease both at the stricture site and in the station 7 (sub-- also shown to be present in up to 0.5% of the patients carinal) group of lymphnodes (Fig. 2). A barium swallow that present with dysphagia . TB oesophagus may done on day 7 post surgery showed normal propulsion occur either as primary TB disease without evidence of of barium, no stenosis and no anastomotic leak (Fig. 3). pulmonary TB which has been doubted to exist. The The patient was started on anti-TB medications and other cases occur as either direct extension of medias- was discharged on day 10 post surgery without any tinal TB often presenting with complications such as complications. trachea-oesophageal fistula. In our patient, a female, 36 years old, post oesophagectomy histology revealed ac- Follow up and outcomes tive TB disease in the presence of an oesophageal stric- The patient was subsequently reviewed on numerous ture. The patient did not have evidence of active visits in the outpatient clinic. By the end of the first pulmonary TB. This thus could point to the possibility Mbiine et al. Journal of Cardiothoracic Surgery (2018) 13:58 Page 3 of 5 c d Fig. 2 Histology of the surgical specimens. Legend: In Fig. 2 a and Fig. 2 b, histology of subcarinal lymph nodes shows well formed epitheliod granulomas and Langerhans giant cells (arrows) and caseous necrosis; Fig. 2 c stricture- shows normal stratified squamous epithelium but with areas of well formed epitheliod granulomas and Langerhans giant cells (arrows) and caseous necrosis; Fig. 2 d gastric/celiac lymph nodes- show marked reactive follicular hyperplasia of primary TB oesophagitis without underlying lung dis- which occurred in the distal third as well . This is im- ease. The presence and the nature of active mediastinal portant as it questions the purported possibility of direct nodal disease in this patient implies either spread from TB extension from the tracheobronchial tree or hillar the oesophagus to the mediastinal nodes but not vice lymph nodes. versa. In 90% of the patients, dysphagia is the commonest The aetiology has been demonstrated to be mycobac- presentation , while other patients present with ody- terium tuberculosis however the mode of infection can- nophagia, retrosternal pain and in some cases bleeding. not be clearly elucidated. Primary inoculation following Some cases will present with complications of tracheo- ingestion of infected material is the mode of infection bronchial fistula and perforation . Constitutional for primary disease while direct extension from infected symptoms are often present but may not be ascribed to mediastinal structures has been attributed to the second- TB oeophagitis. Weight loss for example may be attrib- ary type of infection . Typically infection takes on uted to the malnutrition due to inadequate intake. similar pattern usually resulting in chronic granuloma- Otherwise presence of weight loss, drenching night tous inflammation with giant cells and granuloma for- sweats following evening fevers have all been reported to mation with areas of caseous necrosis. This usually is be present . However, in retrospective review of her associated with ulceration of the mucosa which maybe history, over a period of 2 years she had had evening fe- symptomatic and often picked at endoscopy. In other vers, occasional night sweats, and household contact cases, healing occurs with fibrosis with resultant devel- with a pulmonary TB patient who had, at the time of opment of the oesophageal stricture. Erosion into the her surgery, completed an 8-month course of TB trachea-bronchial tree with development of treatment. trachea-esophageal fistula or perforation into the medi- The presence of active pulmonary or other astinum may also occur . The most affected segment extra-pulmonary TB disease need to be sought and his- of the oesophagus is the middle third  owing to its tory of chronic cough needs to be explored. Our patient proximity to the tracheobronchial tree and mediastinal did not have any chronic cough but had reported prior nodes. However our case presented with a mid-distal history of evening fevers and night sweats associated third stricture and also concurs with a similar case with weight loss. The diagnosis of oesophageal TB is Mbiine et al. Journal of Cardiothoracic Surgery (2018) 13:58 Page 4 of 5 usually made on oesophagoscopy and biopsy, where hist- ology shows epithelioid granulomas with Langhans cells, central necrosis and acid-fast bacilli [4, 7, 8]. In our case, prior histology revealed high grade dysplasia and subse- quently inflammatory tissue without conclusion of TB. The inability to advance the scope into the lumen of the stricture prevented us from taking biopsies from the cul- prit mucosa. In other cases, the histological confirmation is made following surgery with resection of the oesophageal stricture [9, 10]. Once TB diagnosis is made, chest x-ray, abdominal ultra sound scan are done to rule out presence of TB in other areas. The chest x-ray may be normal in the majority of the patients  while upper gastrointestinal-contrast studies often show stricture that may mimic a malignant stricture. Strictures maybe due to luminal, intramural or extrinsic compression de- pending on the form of TB involvement . Contrast leakage into the mediastinum or into the tracheobron- chial tree may also be demonstrated in the presence of fistulation. Thoracic contrast enhanced CT scan may also de- lineate presence of the oesophageal lesion or presence of extrinsic mediastinal nodes compressing the oesophagus. The oesophageal thickness is also assessed. In our patient, chest CT scan showed a markedly thickened and enhancing oesophageal wall at the stricture level, but no obvious extrinsic masses or lymphadenopathy. However, at operation, patho- logical mediastinal lymphnodes were found especially at station 7 (sub-carinal). Typically once the diagnosis is made, the patient is started on anti-TB medication Fig. 3 Day 7 post-operative barium swallow showing no and response has been described as sufficient [2, 4]. anastomotic leak and no stenosis. Legend Fig. 3 a and b are However the presence of complications warrants sur- anteroposterior and lateral views showing an intact anastomotic site as indicated by the black arrows gical intervention [5, 10] and this maybe in the form of treatment of the stricture following principles of stricture management. In our case, an Ivor-Lewis oesophagectomy was performed because of total necessary. Overall the outcome is excellent once proper oesophageal occlusion with suspicion of malignancy. . diagnosis is made. The patient had an un-eventful recovery in the im- mediate postoperative period and was discharged on Abbreviations CT: Computed tomography; HIV: Human immunodeficiency virus; MDR the 10th post-operative day. She was enrolled on TB: Multi drug resistant tuberculosis; TB: Tuberculosis; TIS: Tumour in situ anti-TB medication and was followed up post TB treatment with a successful recovery. Acknowledgments We acknowledge the nurses and other care providers of the Cardiothoracic Department of Mulago National Referral Hospital. These participated in Conclusion caring for the above patient. Oesophageal TB is a rare but curable cause of dysphagia. It may mimic cancer of the oesophagus and it is usually Funding There wasn’t any funding for writing this report. missed as a possible cause of oesophageal strictures. There needs to be an increased index of suspicion among patients with dysphagia in TB endemic regions. Availability of data and materials Data sharing is not applicable to this article as no datasets were generated In uncomplicated cases, anti-tubercular treatment is ad- or analysed during the case report. equate for cure and resolution of dysphagia while once Data sharing is not applicable to this article as no datasets were generated complications occur, surgical intervention may be or analysed during the current study. Mbiine et al. Journal of Cardiothoracic Surgery (2018) 13:58 Page 5 of 5 Authors’ contributions RM was a major contributor in writing the manuscript. MW was involved in critically revising the manuscript for important intellectual content and was the lead surgeon in the management of the patient. RK participated in the literature review and revising the manuscript. All authors were directly responsible in the management of the patient and they all read and approved the final manuscript. Ethics approval and consent to participate Not applicable as this was a case written following the completion of follow up of the patient and the original contact with the patient wasn’t research based. Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the consent form is available for review by the Editor-in-Chief of this journal. Competing interests The authors declare that they have no competing interests. Publisher’sNote Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. Author details 1 2 Department of Surgery, Makerere University, Kampala, Uganda. Uganda Heart Institute, Kampala, Uganda. Received: 14 January 2018 Accepted: 31 May 2018 References 1. Marshall JB. Tuberculosis of the gastrointestinal tract and peritoneum. Am J Gastroenterol. 1993;88(7):989–99. 2. Jain SK, Jain S, Jain M, Yaduvanshi A. Esophageal tuberculosis: is it so rare? Report of 12 cases and review of the literature. Am J Gastroenterol. 2002; 97(2):287–91. 3. Welzel TM, Kawan T, Bohle W, Richter GM, Bosse A, Zoller WG. An unusual cause of dysphagia: esophageal tuberculosis. J Gastrointestin Liver Dis. 2010; 19(3):321–4. 4. Grubbs BC, Baldwin DR, Trenkner SW, McCabe RP Jr, Maddaus MA. Distal esophageal perforation caused by tuberculosis. J Thorac Cardiovasc Surg. 2001;121(5):1003–4. 5. Ni B, Lu X, Gong Q, Zhang W, Li X, Xu H, Zhang S, Shao Y. 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Journal of Cardiothoracic Surgery
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Published: Jun 5, 2018