Primary gingival tuberculosis in pregnancy: A rare combination.

Tuberculosis (TB), a common chronic-specific granulomatous disease, has become rare in the developed countries. However, it is still a common cause of morbidity and mortality in India. Although it commonly involves the lungs, its presentation in the oral cavity is quite uncommon. The very rare incidence of primary gingival TB, particularly in the absence of active pulmonary involvement often leads to misdiagnosis. Clinical features of oral lesions may include ulceration, nodules, granulomas, and fissures. TB in pregnant women is a major cause of obstetric complications and increased risk of maternal and child mortality. This report highlights a rare case of primary TB of gingiva, presenting as severe gingival overgrowth in a patient with 4th month of pregnancy. This case strongly suggests the importance of suspecting TB as the differential diagnosis of gingival overgrowth and therefore, playing a critical role in the early detection and treatment. Interdisciplinary collaboration for diagnosis and management resulted in the successful outcome and prevented the obstetric complications in this case.

INTRODUCTION

Tuberculosis (TB) is caused by Gram-positive, obligate, acid-fast, pathogenic bacteria, and Mycobacterium tuberculosis. The most common presentation is the pulmonary TB, while extrapulmonary TB occurs in only 10%–15% of all cases.[1] TB involving head and neck forms approximately 10% of all extrapulmonary manifestations of disease.[2] Oral TB usually involves tongue and gingiva, followed by the floor of mouth, soft palate, tonsillar pillars, and buccal mucosa.[34] In most of the cases, oral tuberculous lesion presents as painless ulcer of long duration and is associated with regional lymphadenopathy.[5]

India has one of the highest prevalence of TB in the world. It is a leading cause of maternal-child mortality.[6] Although it is a common disease in the Indian population, its gingival involvement is extremely rare.[7] Especially when there is no pulmonary involvement, diagnosis of such cases becomes extremely difficult.

The goal of the present report is to describe a rare occurrence of primary gingival TB, presenting as gingival overgrowth without any pulmonary involvement, in a patient with 4th month of pregnancy. To the best of authors' knowledge and search of various databases, this is the first article reporting primary gingival TB in pregnancy. This paper also highlights the role of interdisciplinary collaboration in the management of primary gingival TB in pregnancy.

CASE REPORT

A 25-year-old female presented with massive, painless swelling of gingiva, gradually growing over the past 4 months. She had a history of bleeding gums for the past 6 months that increased over time. She reported being in the 4th month of pregnancy. There were mild fever and cough with no expectoration for 15 days. There was no history of any previous dental treatment except for ultrasonic scaling 2 years ago. Her body mass index was 18, suggestive of being underweight. Extraoral examination revealed mild swelling of upper and lower lips; however, cervical lymph nodes were not palpable.

Intraoral examination revealed diffuse enlargement of gingiva of both arches affecting both labial and lingual aspects [Figure 1 and 2]. Surprisingly, the right side of arch was more severely affected than the left side [Figure 3a and b]. The gingiva was reddish and lobular in appearance covering more than half of the clinical crown length of maxillary and mandibular right posterior and mandibular anterior teeth [Figure 3]. On palpation, the growth was tender, soft, friable, and had a tendency for spontaneous bleeding. She was not able to maintain adequate oral hygiene due to massive enlargement and bleeding on slight provocation. Angle's class III malocclusion was present. No other relevant finding was observed.

Figure 1

Diffuse enlargement involving both the arches with lobulated appearance of the mandibular labial gingiva

Figure 2

Mandibular occlusal view showing gingival enlargement affecting both labial and lingual aspects

Figure 3

(a) Right lateral view showing diffuse enlargement of gingiva covering more than half of the clinical crown length of maxillary and mandibular posterior and mandibular anterior teeth and extending up to the second molar. (b) Left lateral view showing the absence of gingival enlargement in both the arches

On the basis of history and clinical examination, differential diagnosis of the condition included pregnancy-induced gingival enlargement, chronic inflammatory gingival enlargement, leukemia-associated enlargement, and granulomatous infection. Blood cell counts were in the normal range, except a marginal rise in total leukocyte count and raised erythrocyte sedimentation rate (ESR-34 mm/h). Peripheral blood smear examination ruled out leukemia-associated gingival enlargement and suggested the possibility of one of the common causes of high ESR, i.e., TB. Serologic test for human immunodeficiency virus was also advised which came out to be nonreactive. As the patient was pregnant an incisional biopsy was planned before treating the condition.

A biopsy sample was taken from the labial gingiva around the right mandibular canine. On histopathologic examination, focally acanthotic stratified squamous epithelium with dense lymphoplasmacytic infiltrate was observed. A large number of Langhans type of giant cells were present in the clusters of epithelioid cells. There was no evidence of caseating necrosis suggestive of a “hard tubercle” [Figure 4]. Acid-fast staining of tissue was positive for Mycobacteria. The Mantoux test was weakly positive, suggesting tubercular infection. Posteroanterior view of shielded chest radiograph revealed no abnormalities [Figure 5]. Considering these findings, the diagnosis of primary tuberculous gingival enlargement was made.

Figure 4

Histopathologic photograph of biopsy specimen showing Langhans giant cells present in the clusters of epithelioid cells

Figure 5

Postero-anterior view of chest radiograph revealed no relevant findings, suggesting the absence of pulmonary TB

Coordinating with the gynecologist and TB and pulmonary disease specialist, anti-tubercular therapy (ATT) was initiated following the current guidelines of the Center for Disease Control and Prevention (CDC) for antitubercular drug regimen in pregnant women.[8] Treatment included isoniazid, rifampin, and ethambutol daily for 2 months. This was followed by Isoniazid and Rifampin, daily for 7 months. Streptomycin and Pyrazinamide are not recommended as these have not been confirmed to be safe for the fetus.[89]

During this period, supragingival scaling was done with hand scalers, and the patient was instructed about appropriate home care oral hygiene measures. She was advised not to undergo any elective dental procedures until the completion of ATT. She was also informed about the chances of transmission of disease through aerosol and salivary contamination.

After starting the ATT, there was continuous improvement in general health and a significant regression of the gingival overgrowth in both the arches. Patient delivered a systemically healthy infant who was negative for tuberculous disease and its symptoms. One-month postpartum, she still had mild gingival enlargement and discomfort while brushing. Gingivectomy and gingivoplasty were performed to contour the residual enlargement. Healing after surgery was uneventful. No signs of recurrence were observed during 1 year of follow-up [Figure 6].

Figure 6

Intraoral photograph at 1-year follow-up showing no recurrence of the lesion

DISCUSSION

The occurrence of tuberculous lesions in the oral cavity is rare.[1] It may be attributed to the resistance of intact squamous epithelium, pH, and cleansing action of saliva. Antibodies present in saliva and gingival crevicular fluid also play an important role in this defense mechanism.[10] Any event leading to loss of this natural barrier, such as trauma, exposed pulp of a carious tooth, inflammatory conditions such as gingivitis or periodontitis, ulcers, or surgical procedures may provide a route of entry for the Mycobacterium bacilli. This case was of clinical interest due to the severity and extent of tissue proliferation in both the arches, that too in a pregnant female. In addition, the resemblance of the lesions to pregnancy-induced gingival enlargement might have led to misdiagnosis.

Oral TB usually presents as chronic irregular ulceration. Primary tuberculous lesion in gingiva often present as enlargement with fiery red, irregular, pebbled, or granular appearance.[7111213] The presence of a soft and friable mass as seen in this case constitutes a less common clinical presentation. Similar clinical features have been reported previously in cases of postpartum gingival enlargement.[14] It may be attributed to hormonal changes in pregnancy which increase the proliferation and vascular permeability of capillaries leading to increase in gingival edema.[14] Similar to previously reported cases of primary gingival TB, there was no relevant finding on clinical or radiographic examination of chest which might have suggested the pulmonary involvement.[7111213]

In general, the chances of finding the acid-fast bacilli in biopsy specimens are low (7.8%).[15] However, the granulomatous reaction observed on histopathologic examination and clear identification of Mycobacterium bacilli in acid-fast staining led to the presumptive diagnosis of TB in our case.

The diagnosis of extrapulmonary TB is challenging because of variable clinical presentation. Further, the typical constitutional features are not apparent in most of the cases. Moreover, clinical presentation of TB in pregnancy may be confusing due to similarities of clinical findings between the two, such as tachycardia, anemia, and elevated ESR. Pregnancy further masks the constitutional symptoms such as fever and night sweats.[16] There are some dissimilar features (such as weight gain during pregnancy and weight loss due to TB, hypertension in pregnancy, and hypotension in TB) which also make it difficult to establish the diagnosis.[17]

Although primary oral TB is rare, pulmonary TB is not uncommon in pregnancy. Pregnancy has been considered as a factor associated with the development and flaring up of TB, especially by the stress, poor nutrition, inadequate immunity, or other coexistent diseases. Pregnancy suppresses the T-helper 1 proinflammatory response, leading to masking of the symptoms while increasing susceptibility to new infections and reactivation of TB.[1819]

Pregnant women with pulmonary or extrapulmonary TB have high risk of complications including stillbirths, premature rupture of membranes, and preterm labor.[1620] The fetus may have high risk of getting congenital TB, intrauterine growth retardation, low birth weight, and increased risk of mortality.[2021] Babies born to mothers who have completed ATT before delivery or have received ATT for at least 2 weeks duration before delivery are less likely to have congenital TB compared to babies of untreated mothers.[20] Moreover, if ATT is started early in pregnancy, the outcome can be expected to be same as that in non-pregnant patients. Whereas late diagnosis and treatment has been found to result in a 4-fold increase in obstetric morbidity.[22]

A large number of TB cases involving oropharyngeal region might get misdiagnosed since these lesions are associated with nonspecific clinical presentation. Delayed diagnosis often occurs in cases where the disease is established before any systemic signs and symptoms become apparent. Identification of TB is important for the clinicians who work in proximity to the oral cavity of such patients as routine operative procedures can be a potential source for the spread of infection through the generation of infectious aerosols. Dental clinicians should be encouraged to always follow the universal aseptic protocol to avoid the risk of contracting this disease.

CONCLUSION

Although primary gingival tuberculous is a rare entity, clinicians need to consider this possibility and should include it in the differential diagnosis of gingival overgrowth. Considering the high prevalence of TB in the Indian subcontinent, a thorough evaluation and interdisciplinary collaboration to rule out the involvement of other primary sites should always be ensured. Since the occurrence of TB in pregnancy carries additional risk to the fetus, along with obstetric complications, an interdisciplinary approach becomes mandatory for accurate diagnosis, risk assessment, and treatment planning.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.