Bilateral chancre of the tongue: An unusual presentation of primary syphilis

Introduction

Syphilis is a sexually transmitted infectious disease caused by the spirochete bacterium Treponema pallidum. It can also be acquired by non-sexual contact viz blood transfusion, occupational or non-occupational exposure, or in-utero exposure of fetus from infected mother. The natural course of sexually acquired syphilis includes primary, secondary, latent (early and late), and tertiary syphilis. ¹ The characteristics lesion of primary syphilis is the chancre. It can develop over genital or extragenital sites, depending on the site of contact with the infectious agent. ² We here report an unusual case of primary syphilis presented with a painless indurated oral ulcer over the lateral borders of the tongue.

Case

A 47-year-old married female presented to the outpatient department with the complaint of non-healing painless lesions on the lateral borders of the tongue for one month. She complained of minimal burning sensation and discomfort. On examination, two ulcers were present on the lateral border of the tongue, one on each side. The ulcers were well-circumscribed, round to oval shaped, with raised indurated borders. The margins were sloping with a clear floor (Figures 1(a)–(c)). There was no oozing or bleeding from the ulcer or regional lymphadenopathy. Systemic examination was unremarkable. Before her presentation to our center, she had been treated for jagged teeth, considered a traumatic ulcer, though the response was unsuccessful. The patient denied a history of unprotected oro-genital contact. There were no similar lesions elsewhere, including genital and perianal areas. Based on clinical presentation, the possibilities of primary syphilitic ulcer, traumatic ulceration, aphthous ulcer, and malignancy were considered. Tzanck smear prepared from the base of the ulcer did not show any abnormal cells. Histopathological examination revealed parakeratosis, acanthosis, and dense mixed inflammatory infiltrate in the dermis comprised of lymphocytes, plasma cells, and neutrophils (Figure 2). Screening for human immunodeficiency virus (HIV), hepatitis B surface antigen, and hepatitis C antibody was negative. Her rapid plasma reagin (RPR) titer came out strongly positive (1:16). In addition, Treponema pallidum hemagglutination assay (TPHA) was also positive. Unfortunately, due to a lack of resources, immunostaining or PCR was not done. Based on clinical presentation and laboratory findings, the diagnosis of primary syphilitic chancre was proffered. She was then treated with a single dose of injection Benzathine penicillin 2.4 million units intramuscularly. The ulcers responded well within a week of administration of penicillin and resolved completely within three weeks of treatment (Figures 3(a) and (b)). Serological tests of the husband also came out to be positive. Although the patient and her partner both denied any history of oro-genital sexual contact, however, this does not invalidate the clinical findings and the positive serological tests.OPEN IN VIEWER

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Figure 2.

Photo micrograph showing dense mixed inflammatory infiltrate in dermis comprised of lymphocytes, plasma cells and few neutrophils (Hematoxylin and eosin stain; 100×).

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Figure 3.

(a, b) Resolution of ulcers after treatment. (a) Left lateral surface of tongue. (b) Right lateral surface of tongue.

Discussion

Chancre typically develops at the site of inoculation on contact with an infectious sex partner. It usually occurs over genital areas. However, in approximately 15–20% of cases, it can occur over extragenital sites such as the rectum, perirectal area, fingers, mouth, and nipples, reflecting sexual practices. The oral cavity is the most frequent extragenital site, and in 1–2%, it has been reported as the first and sole manifestation of primary syphilis. Mainly oral chancre develops over lips but can occur over the tongue and palate.

In contrast to secondary syphilis, oral lesions in primary syphilis are usually single, painless, and ulcerative.^3,5 Painless regional lymphadenopathy occurs in up to 80% of patients and is usually associated with genital lesions. This may explain the absence of lymphadenopathy in our patient. After an extensive literature search, only two cases of primary oral syphilis were reported with multiple oral lesions.^6,7 Veraldi et al. reported a case of multiple oral syphilitic chancres with aphthoid-like presentation. ⁷

Although sexual contact is the most common mode of transmission through which it is acquired, it can also be acquired following the use of unsterilized surgical instruments, common drinking vessels, straws, and breastfeeding in children.^8,9 Extragenital chancres may have an atypical presentation, and diagnosis is often missed due to a lack of consideration of sexually transmitted infection in the absence of genital lesions.^10,11 Primary syphilis of the mouth must be differentiated from the gummatous lesion, traumatic ulceration, oral tubercular ulcer, herpes simplex, aphthous ulcers, and Bechet diseases. Serologic testing and microscopic and histopathological examination are needed to confirm the diagnosis. Microscopy in oral lesions is controversial due to the presence of saprophytic organisms.^1,12 Histology findings in various studies have also been found to be non-specific, viz inflammatory infiltrate mainly comprising plasma cells, lymphocytes, and histiocytes. Hence, clinical and laboratory findings must be considered together to reach a diagnosis.

In our patient, we had a suspicion of chancre because of indurated ulcer on the tongue, which was confirmed with serology and histopathology findings. It is also noteworthy that the ulcers in our case were present bilaterally on the lateral border of the tongue, which is an uncommon finding for chancre on the tongue. The possibility of inoculation of organisms at the site of trauma due to jagged teeth can not be ruled out in this patient, i.e., the pseudo-Koerner phenomenon. To our knowledge, bilateral symmetrical presentation of oral chancre over lateral borders of the tongue has never been reported.

Conclusion

The present case highlights the importance of considering syphilis in the differential diagnosis of any ulcerative lesion in the oral cavity.