Circumcision as treatment for Zoon’s balanitis in an HIV-positive teenage patient

Introduction

Plasma cell balanitis, first described by Zoon in 1952, is defined as an inflammation of the glans, which often also involves the foreskin. This inflammatory process may be caused by infectious, mechanical or irritative factors. ¹ Clinically, the Zoon’s balanitis is characterized by the presence of well-circumscribed erythematous lesions of orange-red with glazed appearance and occasional epidermal atrophy. ² It is infrequent and occurs in uncircumcised middle-aged males. ³

The differential diagnosis includes contact dermatitis, candidiasis, Queyrat’s erythroplasia (squamous cell carcinoma in situ or Bowen's disease of the glans), fixed drug eruptions, herpes simplex virus, lichen planus, pemphigus vulgaris, psoriasis vulgaris, circinate balanitis and secondary syphilis. ⁴ Association with malignancies has been rarely reported. ²

Different therapeutic approaches are used in Zoon’s balanitis, including local conservative treatment (topical or injectable corticosteroids, CO₂ laser, cyclosporin and other creams) and surgery (circumcision). While topical therapies are not always effective, circumcision has been proven effective for Zoon’s balanitis and for several other skin conditions affecting the glans and the foreskin. ⁵

We describe the evolution of an atypical case of Zoon’s balanitis in an 11-year-old boy with human immunodeficiency virus (HIV) successfully treated with circumcision.

Case report

In this report, we described the case of an 11-year-old boy with acquired immunodeficiency syndrome (AIDS B2) acquired by vertical transmission. The child had been diagnosed with AIDS (category B, CD4 17%) aged one year seven months when he presented with recurrent pneumonia. He was immediately started on zidovudine monotherapy and his CD4 increased to 20%. After four years, he was switched to dual therapy with AZT + DDI. At age nine, he was treated for clinically and epidemiologically confirmed tuberculosis (RIP regimen for 24 weeks) and switched to AZT + 3TC + efavirenz, achieving clinical response with VL <50 copies/ml and CD4 of 700 (32%). From 9 to 11 years of age, he presented lesions on the glans, characterized by painful erythematous patches also affecting the urinary meatus. Genital herpes was initially suspected, but first attempt at treatment with topical acyclovir for three months was unsuccessful.

Despite the use of acyclovir, erythematous macules covered with hyaline discharge persisted on the glans. Some lesions were whitish and appeared also on the foreskin. The sample collected for rapid analysis showed fungal contamination, and oral treatment with fluconazole was initiated (Figure 1). After two months of treatment with fluconazole, the condition persisted. The glans lesions were then biopsied for histopathology, and a new sample of the discharge from the balanopreputial sulcus and glans was collected for bacterial and fungal cultures. OPEN IN VIEWER

The histopathology report indicated marked integument surface inflammation with predominance of plasma cells. The hematoxylin-eosin stain showed integument fragments covered with ulcerated squamous stratified epithelium, associated with a marked, well-circumscribed, band-like, lymphohistiocytic chronic inflammatory infiltrate rich in plasma cells, on the surface stroma (Figure 2). The special stain (GMS) for fungal structures showed a negative result. Fungal culture and examination of the fresh fragment were also negative. OPEN IN VIEWER

Based on the biopsy report, circumcision was recommended. Care was taken to ensure removal of excess prepuce during the surgical procedure, to allow for total exposure of the glans and the balanopreputial sulcus.

At one-week follow-up, the glans lesions were healed, scabs covered the ulcerations, and there was a significant improvement in pain sensation. One month postoperatively, there was a complete healing of all lesions and the patient had no symptoms of pain or itch.

Discussion

Zoon’s balanitis is a disease condition of unknown etiology affecting the glans and prepuce. Chronic infection by Mycobacterium smegmatis, mechanical trauma, and lack of hygiene are the suspected causes. ⁴ The lesions tend to be chronic and are often present and practically unchanged for one to two years before diagnosis. ⁶ According to the literature, the mean age of patients presenting with balanitis ¹ is highly variable but it usually occurs in patients over 30 years old. ³ During a review of the literature, we found one report of Zoon’s balanitis associated with the HIV infection ⁷ and one in a 12-year-old patient. ⁸ Therefore, our patient with Zoon’s balanitis and HIV infection represents an unusual case.

Specific tests for mycobacteria were not performed. The fungal cultures were always negative. The biopsy provided a definitive diagnosis. In this case, the lesions had also spread to the foreskin. Even though the lesions were suggestive of Zoon’s balanitis, the rarity of the disease in this age group and its association with HIV infection made it difficult for us to raise this suspicion before the biopsy.

Clinical and histopathological aspects are well known and documented. In general, balanitis is characterized by well-circumscribed, glazed, erythematous, orange-red lesions presented in the form of patches or ulcers, located mainly in the glans, and also in the glans and foreskin, or foreskin only. ⁶ Pain, irritation, and discomfort are frequent symptoms. Our patient initially developed lesions on the glans and urinary meatus, characterized by painful erythematous patches. The treatment for herpes simplex virus (acyclovir) and two courses of oral and topical antifungal agents were all unsuccessful.

The histopathological examination of our patient’s biopsy specimen showed features suggestive of Zoon’s balanitis, including a band-like plasma cell infiltrate in the upper dermis with dilated capillaries and red blood cell leakage, associated with haemosiderin deposition. There was a dense dermal infiltrate without atypical cells. The predominance of plasma cells in the subepidermal infiltrate is typical of this type of balanitis.^3,6,9

The biopsy was thus a key element in the diagnosis and therapeutic approach.

In such cases, either medical or surgical therapy is recommended. Different modalities of conservative treatment may be used, including topical corticosteroids^10,11 and other topical creams, such as imiquimod 5% for example, ¹² with variable results. The use of the CO₂ laser has shown promise as a definitive therapy^13,14 and its advantage is the lower risk of HIV contamination. ⁴

The treatment proposed for our patient was circumcision. Circumcision is a safe procedure that can be performed on an outpatient basis, under local anaesthesia. Moreover, it allows removal of excess foreskin and facilitates local hygiene. Conservative treatment should be reserved for patients with high surgical risk or refusing circumcision.