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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 7  |  Issue : 1  |  Page : 60-62

Lingual tuberculosis: A rare presentation of disseminated form of tuberculosis


1 Department of ENT, Yashoda Hospital, Hyderabad, Telangana, India
2 Department of Laboratory Medicine, Yashoda Hospital, Hyderabad, Telangana, India
3 Department of Radiology, Yashoda Hospital, Hyderabad, Telangana, India

Date of Submission08-Feb-2019
Date of Decision24-Oct-2019
Date of Acceptance16-Dec-2019
Date of Web Publication05-Feb-2020

Correspondence Address:
Dr. Majed Abdul Basit Momin
Department of Laboratory Medicine, Yashoda Hospitals, Malakpet, Nalgonda Cross Road, Hyderabad - 500 036, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/sjos.SJOralSci_11_19

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  Abstract 


Tuberculosis (TB) is a common infectious disease, affecting almost any organ or system. Here, we report a case of an 18-year-old female presenting with a swelling in the tongue, which on fine-needle aspiration cytology (FNAC) and special stain for acid-fast bacilli was diagnosed as TB of the tongue. Further extensive imaging study, finally, confirmed the diagnosis as disseminated TB presenting as TB of the tongue. This case was reported because of its rare incidence and to be included in the differential diagnosis of tongue swellings. This case emphasized the utility of simple outpatient procedure such as FNAC ,special staining and extensive imaging, to diagnose such a rare disseminated form of tuberculosis in an immunocompetent host.

Keywords: Disseminated tuberculosis, fine-needle aspiration cytology, lingual tuberculosis


How to cite this article:
Mahendra N, Momin MA, Reddy G A, Borad DK. Lingual tuberculosis: A rare presentation of disseminated form of tuberculosis. Saudi J Oral Sci 2020;7:60-2

How to cite this URL:
Mahendra N, Momin MA, Reddy G A, Borad DK. Lingual tuberculosis: A rare presentation of disseminated form of tuberculosis. Saudi J Oral Sci [serial online] 2020 [cited 2020 Jun 6];7:60-2. Available from: http://www.saudijos.org/text.asp?2020/7/1/60/277790




  Introduction Top


Tuberculosis (TB) is an infectious bacterial disease caused by Mycobacterium tuberculosis, which commonly affects the lungs. In 2017, 10 million fell ill with TB, and 1.6 million died from the disease.[1] TB of the tongue, irrespective of primary or disseminated form, is quite rare encounter, even in areas or countries, in which TB is endemic, like India. Indeed, among ear-nose–throat (ENT) area, cervical lymphadenitis is the most common extrapulmonary TB and tongue is the least probable site. Clinical presentation of TB tongue seen in the form of an ulcer, fissure, or nodular mass mimicking neoplastic lesion.[2] These lesions require fine-needle aspiration cytological (FNAC) examinations with or without histopathological examinations of representative biopsy, to confirm final diagnosis.[3] FNAC has emerged as the first-line investigation and special stain (Ziehl–Neelsen [Z-N] stain) and Gene Xpert real-time polymerase chain reaction plays a promising role for rapid diagnosis in addition to detect rifampicin-resistant species.[4]


  Case Report Top


The case of young, female aged 18 years presented to an ENT surgeon with the complaint of swelling on the right side of the tongue for 3 months. Swelling was painless and gradually increasing in size. There was no history of fever, mouth ulcer, or trauma to tongue. Oral examination revealed a nodular swelling over the right lateral side of tongue, measuring 2 cm × 2 cm [Figure 1]. Other oropharyngeal examinations were normal with no evidence of cervical lymphadenopathy.
Figure 1: Tongue swelling with side view (inlet box)

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Her vital signs and systemic examination were normal. Laboratory tests including complete hemogram, coagulation profile, liver function test, kidney function test, and electrolytes are within normal limit except low hemoglobin (9.5 g/dl). Erythrocyte sedimentation rate was 40 mm at the end of 1st h. Viral serology for hepatitis B surface antigen and HIV was nonreactive. Mantoux test was positive, with 20 mm induration at the end of 48 h.

Chest X-ray and Ultrasound abdomen were normal. Contrast enhanced computerized tomography scan (CECT) neck show well defined peripherally enhancing hypodense lesion in tongue on right side measuring 19 x 22 mm) not crossing mid line [Figure 2]a and [Figure 2]b. Few enlarged mediastinal and right hilar lymph nodes), right paratracheal lymph node measuring 19 x 10 mm and right hilar lymph node measuring 17 x 11 mm [Figure 2]c and [Figure 2]d.
Figure 2: (a-d) Contrast-enhanced computerized tomography neck and chest. (a and b) Hypodense lesion in tongue (red arrow); (c and d) paratracheal and hilar lymph node (green arrow)

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FNA from the tongue swelling was performed, aspirated 1.0 ml of purulent material, which on cytology showed suppurative inflammation [Figure3] and smears were positive for acid fast bacilli in Ziehl- Neelsen (Z-N) stain [Figure 3] inlet box].
Figure 3: Fine-needle aspiration cytology smear showing suppurative inflammation with positive acid-fast bacilli (inlet, blue arrow)

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Gene Xpert testing performed on the aspirated material was positive for M. tuberculosis with no resistance to rifampicin drug.

Based on clinical, cytological, microbiological, and radiological findings, the patient was finally diagnosed as lingual TB with disseminated forms and had been treated with antituberculous treatment (rifampicin 600 mg/day, isoniazid 600 mg/day, pyrazinamide 1500 mg/day, and ethambutol 1200 mg/day) and follow-up after 1 month showed good initial response in the form reduced tongue nodular swelling [Figure 4] After 1 month, repeat CECT neck showed no lymphadenopathy completely after 1 month post-CT. Once the patient completed the initial phase of treatment, she was maintained on isoniazid and rifampicin for a total of 12 months.
Figure 4: One month after treatment with markedly reduced swelling

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  Discussion Top


Despite major global burden of TB, still TB tongue is rare 0.1%.[5] It is believed to be due to continuous protection of oral mucosa by saliva, submucosal antibodies, and variable normal flora.[6] First case of TB tongue was described by Morgagni in 1761.[7] TB tongue occurs due to contaminated sputum, hematogenous route, or neighboring TB focus in the oral cavity.[3] The common clinical presentation of lingual TB includes ulcerated lesion, fissure, nodular swelling either due to tuberculoma or cold abscess, which further ruptured to form an ulcer.[2] Clinically and radiologically, it is difficult to differentiate such lesions from benign or malignant neoplasms, lymphomas, and metastatic deposits. The common sites of lingual TB are the tip, lateral borders, the dorsum, or the base of the tongue.[8]

Diagnostic workup includes FNAC or representative biopsy for histopathological examinations. Image-guided FNAC has emerged as the first line of investigation in an assessment of detected lesions.[9] This is a safe, less traumatic, rapid, and easy method compared to larger core or open biopsy. This procedure is cost-effective as well as easier to repeat, if necessary. Special stain (Z-N) for AFB and Gene Xpert testing helps not only for diagnosing but also rules out drug resistance for rifampicin, respectively. Although the culture is gold standard for final determination, it is slow and may take up 2–8 weeks.[7] Thus, rapid identification is essential not only for early initiation of treatment but to improved patient outcome.

The treatment of disseminated tuberculosis follows the same lines as for pulmonary TB. Conventional antitubercular therapy for at least 6 months including initial 2 months of isoniazid, rifampicin, ethambutol, and pyrazinamide followed by 4 months. Isoniazid, rifampicin is recommended in all patients with disseminated tuberculosis.[10] The patient has now completely recovered and doing well.


  Conclusion Top


Disseminated TB presenting as lingual TB is less commonly encountered and should be considered in differential diagnosis of tongue swellings. Extensive imaging studies are needed to rule out possible secondary or disseminated forms. FNAC, special staining, and Gene Xpert testing on aspirate samples have promising roles in early diagnosis and rifampicin resistance status in a host to improve clinical outcome.

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

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
WHO. Global Tuberculosis Report: 2017. Geneva, Switzerland: World Health Organization; 2018.  Back to cited text no. 1
    
2.
Gupta A, Shinde KJ, Bhardwaj I. Primary lingual tuberculosis: A case report. J Laryngol Otol 1998;112:86-7.  Back to cited text no. 2
    
3.
Nalini B, Vinayak S. Tuberculosis in ear, nose, and throat practice: Its presentation and diagnosis. Am J Otolaryngol 2006;27:39-45.  Back to cited text no. 3
    
4.
Centers for Disease Control and Prevention (CDC). Updated guidelines for the use of nucleic acid amplification tests in the diagnosis of tuberculosis. MMWR Morb Mortal Wkly Rep 2009;58:7-10.  Back to cited text no. 4
    
5.
Prada JL, Kindelan JM, Villanueva JL, Jurado R, Sánchez-Guijo P, Torre-Cisneros J. Tuberculosis of the tongue in two immunocompetent patients. Clin Infect Dis 1994;19:200-2.  Back to cited text no. 5
    
6.
Sezer B, Zeytinoglu M, Tuncay U, Unal T. Oral mucosal ulceration: A manifestation of previously undiagnosed pulmonary tuberculosis. J Am Dent Assoc 2004;135:336-40.  Back to cited text no. 6
    
7.
Titche LL. Tuberculosis of the tongue. Am Rev Tuberc 1945;52:377.  Back to cited text no. 7
    
8.
Soni NK, Chatterji P, Nahata SK. Tuberculosis of the tongue. Indian J Tub 1981;28:22-5.  Back to cited text no. 8
    
9.
Cramer H, Lampe H, Downing P. Intraoral and transoral fine needle aspiration. A review of 25 cases. Acta Cytol 1995;39:683-8.  Back to cited text no. 9
    
10.
Centers for Disease Control and Prevention. Treatment of Tuberculosis, American Thoracic Society, CDC, and Infectious Diseases Society of America. MMWR Morb Mortal Wkly Rep 2003;52:1-15.  Back to cited text no. 10
    


    Figures

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



 

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