Circinate Balanitis without Systemic Involvement: A Diagnostic Challenge

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A 35-year-old male presented with multiple asymptomatic circinate annular lesions on the glans penis for 12 years. He had no history of joint pain, urethritis, conjunctivitis or gastrointestinal and genitourinary infections, and his sexual history was unremarkable. Clinical examination revealed well-defined erythematous annular plaques with a pseudopustular border over the glans penis [Figure 1a and b]. Potassium hydroxide examination, Tzanck smear and Gram stain did not reveal any abnormality. However, with the clinical suspicion of candida balanoposthitis, the patient was treated with clotrimazole 1% cream for around 2 weeks. At follow-up visit, after 2 weeks, the patient did not notice any improvement, and the size of the erosions further increased [Figure 1c].

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Figure 1:

(a and b) Multiple well-defined erythematous circinate lesions on the glans penis. (c) Multiple well-defined erythematous erosions on the glans penis at 2 weeks. (d) Multiple regular dotted vessels over an erythematous background.

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Dermoscopic examination revealed areas of regular red dotted vessels over an erythematous background [Figure 1d].

Further investigations showed human leucocyte antigen (HLA) B27 positivity, while serology for syphilis, human immunodeficiency virus, herpes simplex and hepatitis was negative. Potassium hydroxide examination, Tzanck smear, Gram stain, fungal culture, stool culture and X-ray of pelvis did not reveal any abnormality.

The lesions responded well to topical corticosteroids and tacrolimus, with complete resolution within 3 weeks [Figure 2]; however, recurrence was noted on discontinuation of treatment. Given the chronicity, recurrence, HLA-B27 positivity and therapeutic response to topical steroids and topical immunomodulators, the possibility of circinate balanitis as an isolated manifestation of reactive arthritis (ReA) was considered.

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Figure 2:

Complete resolution of skin lesions at 3 weeks.

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Circinate balanitis is a common mucocutaneous feature of ReA but rarely occurs without systemic involvement. Its chronic and recurrent nature can pose a diagnostic challenge. HLA-B27 positivity may indicate an early stage of ReA. Early recognition is crucial for timely management and preventing progression.

Circinate balanitis presents as annular, erythematous erosive plaques with polycyclic borders affecting the glans penis and prepuce, commonly associated with ReA.^[1]

Circinate balanitis is seen in up to 40% of men with ReA.^[2] Circinate balanitis can appear as an isolated finding or in conjunction with other mucocutaneous manifestations of ReA, such as keratoderma blenorrhagicum, ulcerative vulvitis, nail dystrophy, oral ulcers, geographic tongue and conjunctivitis.^[3] Although circinate balanitis is the most common feature of ReA, it is rarely observed as an initial manifestation.^[2]

Circinate balanitis, combined with HLA-B27 positivity, may serve as an early indicator of ReA, warranting close monitoring for the potential emergence of additional disease manifestations.^[4] Carney et al. reported a similar case of circinate balanitis as the presenting complaint of sexually acquired ReA.^[5]

ReA can occur post-genitourinary (sexually acquired) or gastroenterological (enterically acquired) infections.^[2] The causative agent of the infection cannot be identified in almost half of the patients.^[2] The typical ReA triad includes urethritis, arthritis and conjunctivitis, but incomplete or atypical presentations are increasingly recognised.^[2] Less than one-third of the patients present with a classical triad, and in fact, in the majority of cases, the presentation is incomplete.^[2] In a pentad, the condition is characterised by circinate balanitis and keratoderma blenorrhagicum as characteristic lesions.

Dermoscopy reveals annular and polycyclic pustules, dotted vessels and an erythematous background, which correspond histopathologically to dermal neutrophilic infiltrates, increased papillary dermal capillaries and upper dermal capillary dilatation, respectively.^[3]

In our patient, the presence of chronic, recurrent annular lesions, HLA-B27 positivity, dermoscopic pattern and response to therapy supports the diagnosis of circinate balanitis associated with ReA.

While ReA is typically self-limiting, chronicity and recurrence can occur, particularly in HLA-B27-positive individuals.

This case suggests that isolated circinate balanitis can be a sole and chronic manifestation of an HLA-B27-associated immune-mediated process. Routine HLAB27 genetic testing of patients presenting with circinate balanitis has become important as it could help with early treatment and predict a good future prognosis.^[2] Timely recognition of the condition and early referral to a rheumatologist are crucial to prevent severe complications linked to ReA. This case report underscores the importance of recognising chronic circinate balanitis as a potential solitary presentation of HLA-B27-associated ReA. Table 1 summarises the similar case reports in the literature.^[2-5]

This case emphasises the need to consider ReA in chronic recurrent balanitis, particularly in HLA-B27-positive patients. Isolated circinate balanitis should be recognised as a possible solitary manifestation of ReA, even in the absence of systemic symptoms. Long-term management with topical immunomodulators may be necessary to prevent recurrence, and periodic evaluation for systemic involvement is advised.