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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 5  |  Issue : 1  |  Page : 102-105

Primary oral tuberculosis in a patient with lepromatous leprosy: Diagnostic dilemma


1 Department of Microbiology, Jawaharlal Institute of Postgraduate Medicine and Research, Puducherry; Department of Microbiology, Velammal Medical College Hospital and Research Institute, Madurai, Tamil Nadu, India
2 Department of Microbiology, Jawaharlal Institute of Postgraduate Medicine and Research, Puducherry, India

Date of Web Publication8-Feb-2017

Correspondence Address:
Vithiya Ganesan
Number 4/437, Babu Nagar Main Road, Sourashtra Teachers Colony, Anuppanadi, Madurai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.1016/j.ijmyco.2015.10.006

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  Abstract 


Pulmonary tuberculosis (TB) is the most common form of TB. Primary infection can also affect the pharynx, cervical lymph node, intestine, or oral mucosa. Historically, the observed incidence of concomitant infection with leprosy and TB is high. However, reports of concomitant infection in modern literature remain scarce. Most cases reported in the literature had borderline/lepromatous leprosy and pulmonary tuberculosis. Extrapulmonary tuberculosis is reported in only 3.2% of leprosy cases. To the best of our knowledge, this is the first case report of primary oral tuberculosis of the tongue in a patient with lepromatous leprosy with Type 2 lepra reaction. The patient was referred to Directly Observed Treatment, Short-Course clinic and started on Category I treatment. She received oral prednisolone for lepra reaction, which was subsequently tapered and stopped, however, she continued to receive other antileprotic drugs (thalidomide and clofazimine). The patient's general condition improved and she is on regular follow up.

Keywords: Co-infection, Leprosy, Oral tuberculosis


How to cite this article:
Ganesan V, Mandal J. Primary oral tuberculosis in a patient with lepromatous leprosy: Diagnostic dilemma. Int J Mycobacteriol 2016;5:102-5

How to cite this URL:
Ganesan V, Mandal J. Primary oral tuberculosis in a patient with lepromatous leprosy: Diagnostic dilemma. Int J Mycobacteriol [serial online] 2016 [cited 2021 Oct 28];5:102-5. Available from: https://www.ijmyco.org/text.asp?2016/5/1/102/199735




  Case Report Top


A 45-year-old female came to the outpatient department with complaints of low-grade fever for 2 weeks, erythema and edema of the whole body, and painless ulcers in the tip of the tongue. She had a history of leprosy, which was treated and cured 15 years ago. In the dermatology outpatient department, she was evaluated for leprosy. Slit-skin smear was taken from different sites such as the nose, eyebrows, earlobes, and the results indicated 4+ grading for all the aforementioned samples. Based on this result, the patient was diagnosed as a case of lepromatous leprosy with Type 2 reaction and we decided to treat her with multidrug therapy.

To examine the pathology of the ulcer of the tongue, sample tissue from the tip of the tongue was collected and sent for various examinations such as KOH mount and fungal culture, Giemsa stain for histoplasmosis, bacterial culture and acid-fast staining, and histopathological examination. Surprisingly, Ziehl–Neelsen staining showed acid-fast bacilli, which were beaded and not uniformly stained, a finding characteristic of tubercle bacilli. Histopathological examination of the sample showed a granulomatous reaction with central caseous necrosis and epithelioid and giant cells suggestive of tuberculosis([Figure 1]).
Figure 1: Histopathological examination showing central caseous necrosis and epithelioid and giant cells suggestive of tuberculosis.

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Nested polymerase chain reaction was performed after extracting the DNA from the sample tissue. The extracted DNA was amplified using two sets of primers coding for IS6110 (insertion sequence), which is specific for Mycobacterium tuberculosis. After amplification, gel electrophoresis was carried out, which showed a band corresponding to IS6110 (219 bp; [Figure 2]). Based on this result, oral tuberculosis of the tongue was diagnosed. To identify the primary focus of infection, we carried out further examinations. Chest X-ray showed clear lung fields without features of tuberculosis. Computed tomography of the thorax and abdomen did not reveal any primary focus. Venereal Disease Research Laboratory and human immunodeficiency virus (HIV) tests were not reactive. A provisional diagnosis of primary oral tuberculosis of the tongue in lepromatous leprosy was made and we initiated treatment for both leprosy and tuberculosis. The patient was advised to stop taking dapsone and instead received oral prednisolone, thalidomide, and clofazimine. The patient was referred to the Directly Observed Treatment, Short-Course clinic and started on Category I treatment. The oral prednisolone dose was subsequently tapered and stopped, however, she continued to receive the other antileprotic drugs. The patient's general condition improved and she was on regular follow up. We obtained Institutional Ethical Committee approval and informed consent from the patient for reporting this case.
Figure 2: Gel electrophoresis of nested PCR amplicons showing bands corresponding to IS6110 (219 bp). Note: IS = insertion sequence; M = molecular ladder; PC = positive control; PCR = polymerase chain reaction; T = test.

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  Discussion Top


Historically, the observed incidence of concomitant infection with leprosy and tuberculosis (TB) is high. However, reports of concomitant infection in modern literature remain scarce. Estimation of the annual new case detection rate (ANCDR) in India, where both TB (ANCDR 181/100,000 in 2011) [1] and leprosy (ANCDR 10–35/100,000 in 2011) [2] remain endemic, suggests that only 0.019 cases of concomitant infection/100,000 population would be detected.

It is hypothesized that reduced cell-mediated immunity plays a role in reactivation of latent TB or superinfection with TB in multibacillary patients. Trindade et al. [3] recently investigated the cell-mediated responses of two patients who were diagnosed with borderline leprosy and TB [3]. However, they were unable to find any aberrant response of the interferon-gamma/interleukin-12/23 axis on immunological evaluation. Therefore, further studies are needed to support this hypothesis.

Although there are many well-known risk factors for TB, including HIV infection, diabetes mellitus, transplant patients on immunosuppression, and birth and travel in the developing world, the use of steroids and development of TB are controversial. One weakness in the argument for steroids increasing the risk of developing TB is that a large number of leprosy patients, especially those with multibacillary leprosy, go on to develop lepra reactions, which require steroid treatment. This means that there is a high rate of steroid prescriptions in leprosy cases. With an estimated ANCDR for co-infection of 0.019/100,000 patients (in India), the incidence of patients started on steroids is approximately 66 times greater than the estimated incidence of co-infection with both diseases [4]. Therefore, longitudinal work with active screening of patients before commencing treatment is necessary to identify any temporal relationship between steroids and TB development.

Most co-infection cases reported had borderline/lepromatous leprosy and pulmonary tuberculosis. Extrapulmonary tuberculosis was reported in only 3.2% of leprosy cases. This includes cutaneous, lymph node, and central nervous system tuberculosis [4]. To date, only one case of co-infection of leprosy and oral tuberculosis (larynx) [5] was reported in the literature.

Oral manifestation of tuberculosis is a rare occurrence, observed only in 0.05–5% of the TB population [6]. Saliva, saprophytes, and the very high integrity of oral epithelium with striated muscles resist the bacterial invasion. Usually, oral tuberculosis is secondary to pulmonary involvement and presents as a single indurated painful ulcer especially in middle aged and elderly individuals. Primary oral tuberculosis presenting as a single painless ulcer in younger individuals is uncommon. The most common site is the lateral margins of the tongue, which rest against rough, sharp, and broken teeth. Any area of chronic irritation or inflammation may favor localization of Mycobacterium associated with the disease [7].

Infectious etiologies to be evaluated include primary syphilis and deep fungal diseases such as histoplasmosis. Noninfectious etiology includes traumatic ulcer and squamous cell carcinoma. When oral lesions of tuberculosis are the sole manifestations of the disease, the clinician may face a diagnostic challenge. Another concern is whether there is an increased risk of rifampicin resistance in undiagnosed tuberculosis co-infection due to multidrug therapy. Although this may be an unlikely consequence of monthly rifampicin treatment in India, it may be a greater concern in regions where leprosy patients take daily rifampicin, such as in the United States.


  Conclusion Top


The relationship between the two mycobacterial diseases continues to be enigmatic despite decades of research. Nowadays, oral manifestations of TB are reappearing alongside many forgotten extrapulmonary infections. Recognition of oral tuberculosis is important to prevent emergence of rifampicin-resistant tuberculosis during treatment of leprosy. These patients require interdisciplinary management and social support.

Conflicts of interest

None.



 
  References Top

1.
World Health Organization, Global Tuberculosis Report 2012, World Health Organization, Geneva, Switzerland. Available from: <http://www.who.int/tb/publications/global_report/en/>,(last accessed: 9.11.2015).  Back to cited text no. 1
    
2.
National Leprosy Eradication Programme, NLEP – Progress report for the year 2011–2012, Central Leprosy Division, New Delhi, India.  Back to cited text no. 2
    
3.
M.A. Trindade, D. Miyamoto, G. Benard, et al, Leprosy and tuberculosis coinfection: clinical and immunological report of two cases and review of the literature, Am. J. Trop. Med. Hyg. 88 (2013) 236–240.  Back to cited text no. 3
    
4.
M.R. Timothy, A. Vaseem, H. Jasmin, et al, Leprosy and tuberculosis concomitant infection: a poorly understood, ageold relationship, Lepr. Rev. 85 (2014) 288–295.  Back to cited text no. 4
    
5.
P.M. Flanagan, J.C. McIlwain, Tuberculosis of the larynx in a lepromatous patient, J. Laryngol. Otol. 107 (1993) 845–847.  Back to cited text no. 5
    
6.
F.A. Ito, C.R. de Andrade, P.A. Vargas, et al, Primary tuberculosis of the oral cavity, Oral Dis. 11 (2005) 50–53.  Back to cited text no. 6
    
7.
S. Kapoor, S. Gandhi, N. Gandhi, et al, Oral manifestations of tuberculosis, CHRISMED J. Health Res. 1 (2014) 11–14.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2]


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