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Keywords:

  • Tuberculosis cutis orificialis;
  • gingival tuberculosis;
  • pulmonary tuberculosis

Abstract

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case Report
  5. Discussion
  6. Acknowledgements
  7. References

Tuberculosis cutis orificialis is a rare manifestation of cutaneous tuberculosis which occurs in the oral, perianal and adjacent mucosa. The most frequent orificial lesion location is the tongue. We report a case of a 35-year-old female with tuberculosis cutis orificialis with gingival involvement. She had a six-month history of a moderately painful gingival ulceration. Histopathological examination showed granulomatous infiltrates composed of epithelioid cells, Langhans giant cells and caseating necrosis. A purified protein derivative test was positive. Sputum culture was positive for Mycobacterium tuberculosis. Chest X-ray and high resolution computed tomography showed active pulmonary tuberculosis in both upper lung zones. The gingival specimen was positive for M. tuberculosis polymerase chain reaction. A complete resolution was achieved after six months of anti-tuberculosis therapy. Dental identification of M. tuberculosis may serve as an important aid in the first line of control of this dangerous infectious disease.


Abbreviations and acronyms:
AFB

acid-fast bacilli

CXR

chest X-ray

ESR

erythrocyte sedimentation rate

HIV

human immunodeficiency virus

HRCT

high resolution computed tomography

PAS

periodic acide Schiff

PASM

periodic acid-silver methenamine

PCR

polymerase chain reaction

PPD

purified protein derivative

TCO

tuberculosis cutis orificialis

Introduction

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case Report
  5. Discussion
  6. Acknowledgements
  7. References

Tuberculosis is a chronic specific granulomatous disease caused by Mycobacterium tuberculosis and remains a major health problem in most developing countries.1 Pulmonary tuberculosis is the most common form of the disease and extrapulmonary tuberculosis occurs in 10–15% of all cases.2 Cutaneous tuberculosis is an uncommon form (2–10%) of extrapulmonary tuberculosis,3,4 and very few cases are reported with oral involvement.5,6 Only 0.05–5.0% of tuberculosis cases may present with oral lesions.7

Oral tuberculosis can be either primary or secondary. Primary oral tuberculosis is extremely rare and more likely to occur in younger rather than older adults. It usually involves the gingiva and is associated with regional lymphadenopathy. Secondary orificial tuberculosis often involves the tongue,8,9 followed by the palate, lips, cheek, uvula, gingiva and alveolar mucosa.

Very few cases of gingival tuberculosis have been reported in the literature. Gingival tuberculosis is frequently neglected in the differential diagnosis of oral lesions and may result in delayed treatment, and therefore potentially serious consequences for patients. We report a case of tuberculosis cutis orificialis (TCO) involving gingival tuberculosis with underlying active pulmonary tuberculosis.

Case Report

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case Report
  5. Discussion
  6. Acknowledgements
  7. References

A 35-year-old female presented to the Department of Oral Medicine and Periodontology, complaining of a moderately painful gingival ulceration over six months. The ulceration had gradually increased in size, as had tooth mobility, over the last two months. No treatment had been given prior to this visit.

The patient had no history of dental trauma, allergy, cough with expectoration, surgery, fever or weight loss. Nineteen years ago she had suffered serious pneumonia for one month. Her family history revealed that her uncle had been diagnosed with pulmonary tuberculosis and died without standard treatment five years earlier.

Physical examination showed she was thin, afebrile, and otherwise in normal condition. No cervical lymphadenopathy was found. Intraoral examination revealed a large irregular, fiery red, pebbled, multiple gingival ulcerated lesion in the upper right gingiva involving the 12, 13, 14, 15. The ulcer extended to the buccal vestibule with purulent exudate on the granular surface and poorly defined tender margins. Gingival recession was found in the 12 and 13 (Fig 1). The lesion was moderately painful to touch with spontaneous bleeding. The right lateral incisor exhibited grade 2 mobility. No other abnormalities were found in the oral cavity. Laboratory investigations, including a complete blood count, blood chemistry studies, urinalysis, electrolytes and liver function testing were all normal except the elevated erythrocyte sedimentation rate (ESR) (29 mm/hr).

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Figure 1.  Tuberculous ulcer involving the upper gingiva.

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An urgent incisional biopsy of the gingival ulcer was performed under local anaesthesia and the specimens were sent for histopathological analysis. The results showed: epithelioid pseudotumour hyperplasia of the stratified squamous epithelium, along with a chronic inflammatory type of infiltrate, angiogenesis (Fig 2a); granulomas formed by epithelioid cells, Langhans giant cells (Fig 2b); caseating necrosis (Figs 2c and 2d); and actinomycetes (Fig 2e). Special stains for fungal organisms (periodic acide Schiff [PAS] and periodic acid-silver methenamine [PASM]) revealed positive findings (Figs 2f and 2g). Ziehl-Neelsen staining of paraffin-embedded tissue specimens was negative for acid-fast bacilli (AFB) (Fig 2h). An initial diagnosis of chronic granulomatous inflammation (gingival tuberculosis) accompanied by fungal (actinomycetes) infection was made.

image

Figure 2.  Histopathological examination showed: (a) epithelioid pseudotumour hyperplasia of the stratified squamous epithelium, along with a chronic inflammatory type of iniltrate, angiogenesis (H&E × 100); (b) granulomas and Langhans giant cell (H&E × 200); (c) caseating necrosis (H&E × 100); (d) caseating necrosis (H&E × 200); (e) fungal organisms (actinomycetes) (H&E × 100); (f) fungal organisms (actinomycetes) (PAS×200); (g) fungal organisms (actinomycetes) (PASM×200); (h) negative for acid-fast bacilli (Ziehl-Neelsen staining×200).

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An immunological panel to detect antibodies against Mycobacterium IgG and IgM, human immunodeficiency virus (HIV), hepatitis B and hepatitis C in the serum by ELISA were negative. However, a purified protein derivative (PPD) test was found to be positive (20 mm × 16 mm, >10 mm, evaluated after 72 hours). A chest X-ray (CXR) revealed upper patching and lobe consolidation, exudation and fibrosis propagate of the bilateral lungs (Fig 3). It was suspected the patient had active and/or obsolete pulmonary tuberculosis. Sputum culture was positive for M. tuberculosis. High resolution computed tomography (HRCT) showed an upper characteristic pattern of branching opacities with patchy distribution in the bilateral lungs and a thick-walled cavity in the left upper lobe (Fig 4).

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Figure 3.  Chest X-ray revealed bilateral upper patching and lobe consolidation, exudation and fibrosis propagate.

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image

Figure 4.  HRCT showed characteristic pattern of branching opacities with patchy distribution and lung cavity.

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A polymerase chain reaction (PCR) assay was carried out using sections of paraffin-embedded gingival tissue for M. tuberculosis. DNA was extracted by QIAamp DNA FFPE Tissue Kit (QIAGEN, USA), and PCR amplification was performed using a M. tuberculosis Fluorescent Polymerase Chain Reaction Diagnostic Kit (Da An Gene Co., Ltd. of Sun Yat-sen University, China). The PCR result of the gingival tissue was positive, which confirmed the presence of M. tuberculosis in the gingival tissue samples. Therefore, a diagnosis of secondary gingival tuberculosis accompanied by fungal infection to underlying active bilateral pulmonary tuberculosis was finally made.

The patient was given multiple anti-tuberculosis therapy with isoniazid, rifampicin, pyrazinamide and ethambutol after consultation with a pulmonologist at the hospital. The patient recovered well after six months of chemotherapy, with a complete resolution of the disease and no recurrence during follow-up.

Discussion

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case Report
  5. Discussion
  6. Acknowledgements
  7. References

Tuberculosis remains one of the leading causes of death resulting from infectious disease. The World Health Organization reported that approximately one-third of the world’s population was infected with M. tuberculosis. In 2007, approximately 9.27 million people were newly infected and 2 million people died.10 China accounts for nearly one-fourth of the global burden of tuberculosis and this needs special consideration. About 95% of tuberculosis cases remain clinically asymptomatic; the remaining 5% develop primary tuberculosis and most often localize in the lungs. Involvement of the oral cavity is relatively rare, ranging from 0.05% to 5% of all patients with tuberculosis.7 Eng et al. reported that secondary oral lesions were mostly seen in older people with an average age of 55 years,11 and more often in males than in females.12 However, our patient was a young female aged 35 years.

The clinical manifestation of TCO is non-specific; it can present as ulceration, nodules, fissures, diffuse inflammatory lesions, vesicles, tuberculomas or granulomas. Of all these lesions, the ulcerative form is the most common. TCO is characterized by painful ulcers with indurated erythematous borders and necrotic bases. Orificial tuberculosis is an extremely rare form of TCO,5,6 especially in the tongue.8,9 However, TCO can also occur in the gingiva, as in our case with non-healing gingival ulceration.

Because most patients with TCO present with concomitant pulmonary tuberculosis, a correct diagnosis might be missed if the clinician is not alert enough or if a CXR examination is not undertaken. If tuberculosis is suspected, a CXR should be performed. Chest radiography remains the first choice for initial evaluation of patients with pulmonary tuberculosis.13 In recent years, HRCT has been found to be superior to chest radiography and standard CT in the assessment of pulmonary parenchymal disease. HRCT can contribute important information when making a diagnosis and assist in early treatment planning.14 Cavitation is regarded as the most important sign of tuberculosis activity.15 Cavities are found in up to 73% of pulmonary tuberculosis patients,14 as was found in our patient. Active disease was also confirmed by sputum culture-positive for M. tuberculosis in our patient.

People with active tuberculosis can spread the infection. M. tuberculosis spreads through airborne particles, known as droplet nuclei, which can be generated while people with pulmonary or laryngeal tuberculosis sneeze, cough, speak or sing. These small particles can stay suspended in the air for hours. If a susceptible person inhales droplet nuclei containing M. tuberculosis, infection may begin if the bacilli reach the alveoli. Within 2 to 12 weeks, the body’s immunological response to M. tuberculosis often prevents further multiplication and spread.

The advent of the HIV/AIDS epidemic has accelerated the global spread of tuberculosis. HIV-infected patients are at greater risk than those with tuberculosis infection alone of progressing to active tuberculosis disease and then transmitting it to others.16 Eastern European nations have reported a rise in tuberculosis, which is related to multiple factors including the breakdown of tuberculosis control, multi-drug resistant tuberculosis strains and HIV infection.17 Our patient’s serum analysis for HIV was negative.

The mechanism of primary inoculation into the oral mucous membrane is not definitely established. Tuberculosis of the gingiva is a relatively rare entity. One reason for this may be that the intact squamous epithelium of the oral cavity resists direct penetration by bacilli.18 This resistance has been attributed to the thickness of the oral epithelium, the cleansing action of saliva, local pH and antibodies in saliva.19

Secondary orificial tuberculosis arises from another site and may reflect oral inoculation with sputum or haematogenous spread of mycobacteria.20 Bilateral upper patching, lobe consolidation and a cavitary lesion were observed in our case. Gingival tuberculosis in this case may have developed via contamination of sputum consisting of AFB or haematogenous dissemination. The patient received multiple anti-tuberculosis drugs, and gained a complete disease resolution.

Tuberculosis has been recognized for many years as an occupational risk for healthcare workers, especially dentists. The possibility that dentists may contract an infection from contact with living tubercle bacilli in the mouths of patients who have oral tuberculosis or pulmonary tuberculosis is of great concern.11

In conclusion, early diagnosis of TB can be achieved by combined examinations including histopathological and bacteriological tests, chest radiography and HRCT. TCO with various types of signs such as non-healing oral ulcers, including the gingiva, may be misdiagnosed as neoplastic or aphthous ulcers and appropriate treatment delayed. Dentists and otolaryngologists should be alert to TCO and include it in the differential diagnosis. Dental identification of M. tuberculosis may serve as an important aid in the first line of control of this dangerous and infectious disease.

Acknowledgements

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case Report
  5. Discussion
  6. Acknowledgements
  7. References

We thank the patient for participating in this project. This work is supported, in part, by a grant from Zhejiang Education Committee Projects (Y201017607) and Zhejiang Population and Family Planning Committee Projects (20080729).

References

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case Report
  5. Discussion
  6. Acknowledgements
  7. References