Jordi | Journal of Oral DiagnosisOnline SubmissionReview an ArticleSOBEP - Sociedade Brasileira de Estomatologia e Patologia Oral
Volume 2 - 2017


Case Report

DOI: 10.5935/2525-5711.20170007

Reemerging syphilis: diagnosis from oral lesions

Ingrid da Silva Santos1; Daniela Brito Bastos1; Vitor Bonetti Valente1; Solange Pires D'Ávila2; Kellen Cristine Tjioe1; Éder Ricardo Biasoli1; Glauco Issamu Miyahara1; Daniel Galera Bernabé1

1. Universidade Estadual Paulista, School of Dentistry, Araçatuba, Brazil
2. Private Clinic, Araçatuba, São Paulo, Brazil

Corresponding authors: Oral Oncology Center, Department of Pathology and Clinical Propedeutics
Universidade Estadual Paulista (UNESP), School of Dentistry
Araçatuba, Brazil 1193 José Bonifácio St, Araçatuba, SP 15050-015
E-mail: danielbernabe@foa.unesp.br

Article received on January 20, 2017
Article accepted on May 11, 2017

 

ABSTRACT

BACKGROUND: Syphilis cases have increasing worldwide recently, indicating a resurgence of the disease. Its clinical features vary widely and often challenge the establishment of the diagnosis.
CASE REPORT: A 23-year-old woman presented with multiple erosive and patchy oral lesions, dysphagia, and pain with onset of two months. Her medical history revealed that she also presented skin macules which were misdiagnosed as pityriasis by a dermatologist. Nontreponemic test was reagent. An incisional biopsy of an oral lesion was performed and the histopathological aspects were suggestive of syphilis. Treponemic test was positive, confirming secondary syphilis. The treatment consisted of penicillin for 3 weeks. After the 6-months follow-up, clinical and serological exams did not show any sign of the disease.
CONCLUSIONS: Diagnosing syphilis by oral lesions can be difficult once it can mimic other affections. Additionally, the reemergence of this sexually transmitted disease in the last years demands its inclusion in the differential diagnosis of either erosive or ulcerate lesions.

Keywords: Syphilis; Treponema pallidum; Diagnosis, Oral; Sexually Transmitted Diseases.

 

INTRODUCTION

Syphilis is a systemic chronic infection caused by the anaerobic spirochete Treponema pallidum and can involve different organs and tissues depending on the stage of the disease1,2. It can be sexually transmitted (acquired syphilis), vertically spread via the placenta (congenital syphilis) or, uncommonly, transmitted by contaminated objects used in blood transfusions2-4. This disease can be classified into four stages according to its activity and infectivity: primary, secondary, latent, and tertiary. The presence of oral lesions may be a feature of all phases of syphilis and can often be the first manifestation of the disease1,5.

The primary stage of infection has an average incubation period of 3 weeks. It is characterized by a painless ulcer called hard chancre that occurs at the site of inoculation, heals spontaneously, and is classically located in the genital region. Although not common, hard chancre can also be extragenital and affect mainly anal or oral sites5,6. After 2 to 12 weeks after the primary infection, the treponemes spread through blood and lymphatic vessels6,7 giving rise to the secondary syphilis phase, characterized by widely variable systemic and oral manifestations. The classic cutaneous manifestation of this phase is the maculopapular rash that often involves the palms and soles6. Oral lesions occur more frequently at this stage of the disease, and their clinical aspects can be quite heterogeneous4-6. Tertiary syphilis, which occurs after a highly variable latent period, presents severe complications that may involve different systems. Its oral manifestation is characterized by gumma formation, which often affects the hard palate8. Although the diagnosis of syphilis may be challenging due to its variable clinical findings, it is effectively treated with antibiotic therapy.

Here, we report a case of syphilis diagnosed by our team from an oral manifestation in a young patient emphasizing the importance of diagnosing this reemerging sexually transmitted disease (STD).


CASE REPORT

A 23-year-old female sought our Stomatology outpatient clinic complaining about multiple lesions in the oral mucosa that appeared approximately two months before the consultation. At the history of present illness, she reported the previous onset of painful lesions in the palate, which were treated as pharyngitis with oral azithromycin and sodium diclofenac by an otolaryngologist. The clinical conditions had only partially improved. The patient also revealed that 1 month before the consultation she had hair, eyebrows, and eyelashes loss. A dermatologist treated the case as hypovitaminosis. After this period, the oral lesions progressed to other sites causing pain, intense dysphagia, and feeding impairment. By that time, the patient was referred to our service.

On extra-oral examination, lymphadenopathies were not observed and the patient denied genital and cutaneous lesions. Intraoral examination revealed multiple and painful erosions in the labial mucosa (Figure 1a), hard and soft palate (Figure 1b), and in the right lateral border of the oral tongue (Figure 1c) in addition to red plates in the oropharynx. The clinical hypotheses included immuno-mediated diseases such as systemic lupus and discoid lupus erythematosus. Hematologic and serological tests were requested. In the following week, most of the lesions had regressed. However, erythrocyte sedimentation rate (ESR) and C-reactive protein showed high levels, and rheumatoid factor and venereal disease research laboratory (VDRL) test (titer 1:32) were reactive.


Figure 1. A. Clinical features of the "mucous patches" form on the labial mucosa and commissure. B. Erosive lesions and mucous patches covered by gray pseudomembrane on the hard and soft palate. C. Serpiginous lesions affecting the right edge of the tongue, described as snail track ulcers.



Thus, the patient's sexual behavior was questioned, but she denied having any previous sexual activity. Furthermore, her medical history was further reviewed and, in this opportunity, she reported to have had maculopapular skin eruptions in abdominal region one month ago, which were diagnosed and treated as pityriasis rosea by a dermatologist. The patient also reminded that her first oral lesion appeared in the lower lip (two months ago) and provided photographs of the cutaneous (Figure 2a) and lip (Figure 2b) lesions.


Figure 2. A. Maculopapular skin lesions in the abdomen. (Photo courtesy of patient). B. Clinical appearance of the first lesion of Oral Syphilis. Ulcer fibrin-covered, surrounded by erythematous halo in the lower lip. (Photo courtesy of patient).



The photograph from lip showed a solitary, painless, fibrin-covered ulcer in the left lower lip measuring approximately 0.5cm in diameter and surrounded by an erythematous halo, which healed spontaneously after 1 week according to patient (Figure 2b). At this time, the treponemal test Fluorescent Treponemal Antibody-absorption (FTA-Abs) was requested and an incisional biopsy of one of the palatal lesions was performed.

Microscopic analysis showed irregular acanthosis of the oral epithelium and lymphocytes and neutrophil exocytosis (Figure 3a,b). The histopathological diagnosis was suggestive of syphilis. The treponemic FTA-Abs was reactive and the diagnosis of secondary syphilis was confirmed. Once more, her sexual history was reviewed, but the patient still denied any previous sexual contact or high-risk behavior to STD. The patient was referred to an infectologist who prescribed weekly doses of intramuscular penicillin G benzathine, 2.4 million units, for 3 weeks. Gynecological evaluation to investigate genital lesions was negative. The 2-weeks follow-up showed total remission of the oral lesions and absence of symptomatology. After 6 months, clinical and serological exams did not show any sign of the disease (Figure 4).


Figure 3. A. Photomicrograph showing (H & E x 10) chronical mucositis activity, characterized by an irregular acanthosis. B. Photomicrograph showing (H & E x 40) proliferated congested capilars, surrounded by inflammatory infiltrate composed of lymphocytes and plasma cells and exocytosis of lymphocytes.


Figure 4. No clinical evidence of disease after 6 months' follow-up.



DISCUSSION

In this report, we presented a 23-year-old female patient diagnosed with syphilis by our team after the onset of oral lesions and previous consultation with other professionals. In recent years, syphilis cases have been increasing in different parts of the world mainly in USA, Canada, China, Russia, parts of Europe, Australia, and Brazil2,3,5,9.

Particularly in Brazil, there was a significant increase between June 2010 and 2016, almost 230,000 new cases of the disease were reported. The states of São Paulo, Minas Gerais, Rio de Janeiro, Paraná, Rio Grande do Sul have presenting the highest increasing rates of syphilis in the year 201510. In the last years, there were no other syphilis cases diagnosed at our service what may indicate a tendency of reappearance of this disease.

The at-risk population includes young, pregnant women, and men who have sex with men2,5,9. Some of the factors that could explain the increase of syphilis cases are the high-risk sexual behavior, such as multiple concurrent sexual partnerships, and the reduction of the use of barrier protection (e.g. condoms) in the anogenital sex besides the increasing practice of unprotected oral sex2,5,11. However, when our patient was recalled about her sex life, she still denied any history of sexual contact, high-risk behavior, including contact with contaminated objects or any other factor.

Primary syphilis, clinically characterized by chancre, is usually the consequence of orogenital or oroanal contact with a Treponema pallidum infected lesion. Extragenital chancres are rare and, when present, the lip is most commonly involved site, as result of oral sex practice and often goes unnoticed by the patient2,12. It was not possible to clinically observe the chancre in our patient once she came to our Institution in a more advanced stage of the disease. Interestingly, her clinical history and self-photographed first oral lesion were compatible with primary chancre of syphilis.

Although oral lesions and skin manifestations may occur at any stage of the disease, they are mainly associated with secondary syphilis13,14, which occurs in approximately 30% of untreated patients. In this phase, several systemic and mucocutaneous manifestations of the disease, such as fever, malaise, weight loss, pharyngitis, and lymphadenopathy5,6,14,15 occur. Some of these events were reported by our patient, additionally to hair, eyebrows, and eyelashes loss, which have been reported in about 5-6% of the patients with syphilis5.

In addition, approximately 75% of patients with secondary syphilis develop skin manifestations16. Its most common presentation is called syphilitic roseola which is characterized by symmetrical, asymptomatic, and pink or red macules that last about two weeks and usually affect the trunk and extremities5,14. The skin lesions from our patient were diagnosed as pityriasis rosea by a dermatologist, but we assume that these lesions were suggestive of syphilitic roseola, which is the first clinical sign of secondary syphilis and accompanying oral lesions2,17.

Different from primary syphilis, that is presented as a single painless chancre, the oral lesions of secondary syphilis are usually symptomatic and multiple11. The most common oral lesions associated with secondary syphilis are ulcers or erosions and "mucous patches" with two subtypes: slightly elevated-type plaques with ulcerated surface occasionally; or multiple mucous patches that may coalesce to give rise to serpiginous lesions, described as snail track ulcers4-6,12-14. As seen in our patient, the usually affected sites are soft palate, tongue, lips, buccal mucosa, and vestibular mucosa5,14.

In the reported case, it was possible to observe the diversity of clinical characteristics of oral lesions, a fact that challenges the clinical diagnosis of syphilis. In fact, syphilis is known as the ''great imitator''5 due to this clinical variability. In this report, the initial clinical diagnostic hypotheses were immune-mediated diseases based on clinical characteristics of the lesions with periods of remission and exacerbation and also in her medical history. The differential diagnosis of oral lesions of secondary syphilis includes lupus erythematosus, erythema multiform, stomatitis, pemphigus, idiopathic leukoplakia, lichen planus, candidiasis, and other STD2,5,6,14,18.

The confirmation of syphilis diagnosis is based on clinical signs and symptoms, sexual history, and specific serologic tests6. Histopathological characteristics of secondary syphilis can be very variable and are not sufficient for the diagnosis. The presence of plasma cell perivascular infiltrate associated with exocytosis of lymphocytes and neutrophils, in a psoriasis form appearance or not6,14 are suggestive features of syphilis. In our particular case, only serologic tests concluded the diagnosis4,6.

There are 2 types of serologic tests for syphilis diagnosis. Nontreponemic tests (e.g. VDRL), and treponemic tests (e.g. FTA-Abs). The treponemic tests are used for confirmatory purposes if the nontreponemic tests are positive6,18. In this case, VDRL in association with other sorological tests was requested because of the suspicion of immune-mediated disease. However, only VDRL was positive, indicating a possible veneral disease. Thus, FTA-Abs was requested and it was reactive, confirming the diagnosis of syphilis.

Syphilis treatment depends on the disease stage. As the untreated, disease may progress to severe stages, long-term complications may develop11 and require careful management. Penicillin is the first choice of treatment due to its efficacy at all stages of syphilis. Therapy with penicillin G benzathine, for 3 weeks, administered in our patient, resulted in the remission of symptoms and oral lesions after the first dose.

This report shows different manifestations of syphilis, a STD that is frightfully reemerging, and reinforces the urgent need to include this disease in the differential diagnosis of oral mucosa as well as systemic lesions. The variable clinical presentation of the disease, the vertiginous and recent increase in the number of cases reported added to the incomplete information provided by the patient to us and maybe to the other health professionals consulted by her delayed the diagnosis. For this reason, the awareness of the resurgence of syphilis may be propagated in order to avoid the dissemination of this STD and prevent a public health issue.


ACKNOWLEDGMENTS

Ingrid da Silva Santos (Decision 001/2015) is a recipient of a Master's fellowship from Fundação de Amparo à Pesquisa do Estado do Amazonas (FAPEAM)

The authors thank Dr. Stelios Fikaris, M.D, for the support in clinical approaching of the patient.


REFERENCES

1. Singh PV, Patil R. Atypical oral manifestations in secondary syphilis. Indian J Dent Res. 2013;24:142-4.

2. Strieder LR, León JE, Carvalho YR, Kaminagakura E. Oral syphilis: report of three cases and characterization of the inflammatory cells. Ann Diagn Pathol. 2015;19:76-80.

3. Minicucci EM, Vieira RA, Oliveira DT, Marques SA. Oral manifestations of secondary syphilis in the elderly - a timely reminder for dentists. Aust Dent J. 2013;58:368-70.

4. Siqueira CS, Saturno JL, de Sousa SC, da Silveira FR. Diagnostic approaches in unsuspected oral lesions of syphilis. Int J Oral Maxillofac Surg. 2014;43:1436-40.

5. Ficarra G, Carlos R. Syphilis: the renaissance of an old disease with oral implications. Head Neck Pathol. 2009;3:195-206.

6. Kelner N, Rabelo GD, da Cruz Perez DE, Assunção JN Jr, Witzel AL, Migliari DA, et al. Analysis of nonspecific oral mucosal and dermal lesions suggestive of syphilis: a report of 6 cases. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;117:1-7.

7. Kollipara R, Guidry JA, Tyring SK. Erythematous Papules and Vesicles on the Palms, Soles, and Oropharynx. JAMA Dermatol. 2015;151(7):783-4.

8. Compilato D, Amato S, Campisi G. Resurgence of syphilis: a diagnosis based on unusual oral mucosa lesions. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009;108(3):e45-9.

9. Araujo JP, Jaguar GC, Alves FA. Syphilis related to atypical oral lesions affecting an elderly man. a case report. Gerodontology. 2015;32:73-5.

10. Brazil. Ministry of Health. Secretaria de Vigilância em Saúde. Boletim Epidemiológico. 2016;47(35):28 p. [cited 2017 Nov 7]. Available from: http://www.aids.gov.br/sites/default/files/anexos/publicacao/2016/59209/dst_aids_boletim_de_sifil_1_pdf_32008.pdf

11. Hertel M, Matter D, Schmidt-Westhausen AM, Bornstein MM. Oral syphilis: a series of 5 cases. J Oral Maxillofac Surg. 2014;72:338-45.

12. Leão JC, Gueiros LA, Porter SR. Oral manifestations of syphilis. Clinics (Sao Paulo). 2006;61:161-6.

13. de Paulo LF, Servato JP, Oliveira MT, Durighetto AF Jr, Zanetta-Barbosa D. Oral Manifestations of Secondary Syphilis. Int J Infect Dis. 2015;35:40-2.

14. Pires FR, da Silva PJ, Natal RF, Alves FA, Pinto CA, Rumayor A, et al. Clinicopathologic features, microvessel density, and immunohistochemical expression of ICAM-1 and VEGF in 15 cases of secondary syphilis with oral manifestations. Oral Surg Oral Med Oral Pathol Oral Radiol. 2015;121:274-81.

15. Cohen SE, Klausner JD, Engelman J, Philip S. Syphilis in the modern era: an update for physicians. Infect Dis Clin North Am 2013;27:705-22.

16. Lautenschlager S. Diagnosis of syphilis: clinical and laboratory problems. J Dtsch Dermatol Ges. 2006;4:1058-75.

17. Carbone PN, Capra GG, Nelson BL. Oral Secondary Syphilis. Head Neck Pathol. 2016;10:206-8.

18. Seibt CE, Munerato MC. Secondary syphilis in the oral cavity and the role of the dental surgeon in STD prevention, diagnosis and treatment: a case series study. Braz J Infect Dis. 2016;20:393-8.

 

© Copyright 2017 - Jordi - All rights reserved GN1