|Year : 2021 | Volume
| Issue : 2 | Page : 58-61
A case of extrapulmonary tuberculosis presenting as multiple pleural nodules and esophageal ulcer
Jesin Kumar Chakkamadathil1, Ravindran Chetambath1, Sanjeev Shivashankaran1, C Girija2, Christopher Mathew3
1 Department of Pulmonary Medicine, DM Wayanad Institute of Medical Sciences, Wayanad, Kerala, India
2 Department of Pathology, DM Wayanad Institute of Medical Sciences, Wayanad, Kerala, India
3 Department of Internal Medicine, DM Wayanad Institute of Medical Sciences, Wayanad, Kerala, India
|Date of Submission||27-Mar-2021|
|Date of Decision||21-Apr-2021|
|Date of Acceptance||04-May-2021|
|Date of Web Publication||21-Jun-2021|
Dr. Jesin Kumar Chakkamadathil
Department of Pulmonary Medicine, Karuna Medical College, Vilayodi, Chittur, Palakkad - 678 103, Kerala
Source of Support: None, Conflict of Interest: None
Tuberculosis (TB) can affect various organs besides the lungs. Among the extrapulmonary sites, pleura and gastrointestinal tract are the second- and sixth-most common sites, respectively. Pleural involvement usually occurs in the form of pleural effusion, and the sites most commonly involved in gastrointestinal TB are the peritoneum and ileocecal area. Here we present an interesting case of a 23-year-old female who presented to the gastroenterologist with symptoms of dysphagia, regurgitation of food, and weight loss. Esophagogastro duodenoscopy showed an esophageal ulcer which was confirmed to be due to TB. Incidentally, this patient also had multiple pleural nodules due to TB. This case is unique since there is a simultaneous occurrence of two different sites of occurrence of extrapulmonary TB.
Keywords: Esophageal ulcer, extrapulmonary, pleural nodules, tuberculosis
|How to cite this article:|
Chakkamadathil JK, Chetambath R, Shivashankaran S, Girija C, Mathew C. A case of extrapulmonary tuberculosis presenting as multiple pleural nodules and esophageal ulcer. J Adv Lung Health 2021;1:58-61
|How to cite this URL:|
Chakkamadathil JK, Chetambath R, Shivashankaran S, Girija C, Mathew C. A case of extrapulmonary tuberculosis presenting as multiple pleural nodules and esophageal ulcer. J Adv Lung Health [serial online] 2021 [cited 2021 Jul 28];1:58-61. Available from: http://www.jalh.com/text.asp?2021/1/2/58/318914
| Introduction|| |
Tuberculosis (TB) outranks even human immunodeficiency virus as a leading cause of death from a single infectious agent. Despite being a preventable and curable disease, it claims close to 4000 lives each day. A common hindrance faced in the diagnosis of TB is its varied presentation due to the various organs it affects besides the lungs. Twenty-five percent of adults with TB present with the extrapulmonary form. Following lymph nodes, pleura is the second most common site of involvement in extrapulmonary TB.
Pleural involvement most commonly occurs in the form of pleural effusion. It is often accompanied by parenchymal involvement. Other forms of pleural involvement include empyema, pleural thickening, and calcifications. The pathogenesis currently hypothesized is the rupture of sub pleural caseous foci and delayed hypersensitivity reaction to mycobacterial antigens. Hence, the tubercle bacilli is rarely found in the pleural fluid or biopsy specimens of pleural tissue. Esophageal TB is also a rare occurrence, accounting for only about 2.8% of all cases of gastrointestinal TB. Direct spread from mediastinal lymph nodes is the pathogenesis currently speculated. However, it can also occur due to spread through the lungs or blood stream. We are now reporting a rare case of pleural TB present as multiple pleural nodules with an esophageal ulcer without pleural effusion or any evidence of parenchymal or lymphatic disease.
| Case Report|| |
A 23-year-old female college student with no significant comorbidities presented with symptoms of dysphagia, regurgitation of food, and significant weight loss for 3 months. She had no respiratory symptoms including chest pain. Her physical examination revealed no significant findings except for a low body mass index. Blood investigations were normal, and erythrocyte sedimentation rate was 18 mm. She presented to the gastroenterologist with the above complaints. Esophagogastroduodenoscopy was done which revealed an ulcerated lesion from the midesophagus from which a biopsy was taken [Figure 1]. Since the suspicion of TB was high, a pulmonology consultation was sought. Chest X-ray [Figure 2] done showed a nodule in the right lower zone which was evaluated further with a contrast-enhanced computed tomography (CT) scan of the thorax. CT revealed multiple pleural-based nodules largest measuring 2.7 cm × 1.4 cm with peripheral enhancement [Figure 3] and [Figure 4]. No pleural effusion or mediastinal lymph nodes were noted.
|Figure 3: Lung window of the computed tomography thorax showing pleural nodules|
Click here to view
|Figure 4: Mediastinal window of the computed tomography thorax showing pleural nodules. No significant lymph nodes were noted|
Click here to view
An ultrasound-guided fine-needle aspiration was done from one of the nodules. Aspirate smears showed round-to-spindle cells and epithelioid cells arranged in sheets and clusters. Well defined granulomas with multinucleated giant cells of Langhan's type were also seen [Figure 5]. Aspiration smears from another pleural nodule, which had necrotic changes, yielded mainly eosinophilic, granular, and necrotic material in the smears. A Modified Ziehl Neelson (Acid-fast Bacilli [AFB]) staining was performed which revealed singly scattered as well as clumps of beaded, rod-shaped bacilli which were positive for AFB stain [Figure 6]. Cytology showed granulomatous lesion with multinucleate Langhans's giant cells. Fluorescence staining for AFB showed singly scattered rod-shaped bacilli. Cartridge-based nucleic acid amplification test (CBNAAT) was also done on the aspirate, which also detected Mycobacterium TB.
|Figure 5: (a) Cytology Smears-Low power view with multinucleated giant cells and epithelioid cells, (b and c) Multinucleate cells of Langhan's type, (d) Well-defined granuloma with slipper-shaped nuclei|
Click here to view
Endoscopic biopsy specimen from ulcerated lesion in the midesophagus showed squamous epithelium lined tissue with ulcerations. Subepithelium was infiltrated by dense mixed inflammatory cells predominantly lymphocytes along with few neutrophils and histiocytes. Many congested vessels were also noted in the subepithelium. No granulomas or giant cells were seen in the sections [Figure 7]a and [Figure 7]b. An AFB staining was performed in these slides too and it revealed beaded, AFB-positive bacilli [Figure 7]c and [Figure 7]d.
|Figure 7: (a) Esophageal mucosa lined by squamous epithelium and subepithelial inflammatory infiltrates. (b) Ulcerated mucosa and neutrophilic infiltrates. (c and d) Acid-fast Bacilli positive bacilli in esophageal tissue|
Click here to view
She was started on antituberculous medication as per the National TB Elimination Program regimen, following which her symptoms improved. An esophago-gastro-duodenoscopy was repeated after 5 months of treatment with anti-tuberculous medication which showed resolution of the ulcer [Figure 8]. A CT thorax was also repeated after completion of the continuation phase of treatment which showed reduction in the size of the pleural nodules.
|Figure 8: Endoscopic view of the esophagus after 5 months of ATT showing resolution of the ulcer (arrowhead)|
Click here to view
| Discussion|| |
In clinical practice, pleural masses are encountered when we are dealing with primary malignancies such as mesothelioma, pleural lymphomas or due to hematogenous spread from malignancies in the lung, breast, or gastrointestinal tract. Mesothelioma is the most common primary malignancy but is less common than metastasis to the pleura. Rarely, even sarcoidosis can present as pleural masses.
A diagnosis of malignancy was entertained initially considering the presence of an esophageal ulcer at the initial stage. However, the age group of the patient, lack of exposure to asbestos, and absence of atypical cells in the esophageal biopsy specimen made this possibility unlikely. The availability of diagnostic modalities such as CBNAAT enabled us to make a diagnosis early without resorting to invasive modalities like thoracoscopic pleural biopsy.
Pleural TB most commonly presents as pleural effusion. A similar case of tuberculous pleural masses with hemorrhagic pleural effusion was reported by Patel and Choudhury. A case of esophageal ulcer, later diagnosed as TB, was also reported by Khanna et al. However, there was associated mediastinal lymph nodes, unlike our case. Hence, to the best of our knowledge, a case of extrapulmonary TB presenting as pleural nodules and an esophageal ulcer without effusion, mediastinal lymph nodes or a parenchymal lesion has not been reported previously. The presence of two such rare occurrences makes this an extremely rare case. Furthermore, early diagnosis in such cases is imperative due to its complications such as esophageal perforation, fistula formation, stricture formation, aspiration pneumonia, and hematemesis. These may require surgical correction and may prove to be fatal. In our case, early diagnosis and initiation of treatment enabled us to successfully treat this patient and prevent these complications.
| Conclusion|| |
In the era of the COVID-19 pandemic, TB still remains a relevant disease claiming many lives daily. As discussed in this case, it can present as unusual lesions at unusual sites. A thorough knowledge regarding the varied presentations of this illness is often needed to suspect and eventually confirm this illness. When suspected, an attempt should be made to obtain a microbiological confirmation, through AFB staining or CBNAAT, even in the absence of typical histological features.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Organisation WH. Global Tuberculosis Report 2020. Available from: https://apps.who.int/iris/bitstream/handle/10665/336069/9789240013131-eng.pdf. [Last accessed on 2021 Mar 21].
Vorster MJ, Allwood BW, Diacon AH, Koegelenberg CF. Tuberculous pleural effusions: Advances and controversies. J Thorac Dis 2015;7:981-91.
Stead WW, Eichenholz A, Stauss HK. Operative and pathologic findings in twenty-four patients with syndrome of idiopathic pleurisy with effusion, presumably tuberculous. Am Rev Tuberc 1955;71:473-502.
Berger HW, Mejia E. Tuberculous pleurisy. Chest 1973;63:88-92.
Patel N, Amarapurkar D, Agal S, Baijal R, Kulshrestha P, Pramanik S, et al.
Gastrointestinal luminal tuberculosis: Establishing the diagnosis. J Gastroenterol Hepatol 2004;19:1240-6.
Fang HY, Lin TS, Cheng CY, Talbot AR. Esophageal tuberculosis: A rare presentation with massive hematemesis. Ann Thorac Surg 1999;68:2344-6.
Dynes MC, White EM, Fry WA, Ghahremani GG. Imaging manifestations of pleural tumors. Radiographics 1992;12:1191-201.
Loughney E, Higgins BG. Pleural sarcoidosis: A rare presentation. Thora×1997;52:200-1.
Patel A, Choudhury S. Pleural tuberculosis presented as multiple pleural masses: An atypical presentation. Lung India 2013;30:54-6.
] [Full text]
Khanna V, Kumar A, Alexander N, Surendran P. A case report on esophageal tuberculosis-A rare entity. Int J Surg Case Rep 2017;35:41-3.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]