Abstract
Dermatological conditions are more common and can present atypically, in human immunodeficiency virus-infected individuals. This case report describes a 22-year-old human immunodeficiency virus-positive Caucasian female who presented with a vulval lesion eight weeks after starting antiretroviral treatment. Clinical examination revealed a 2 cm well-demarcated plaque on the outer aspect of the left labium minus. The lesion was tender, no contact bleeding or ulceration present. She was presumptively treated for chancroid and herpes simplex with 500 mg ceftriaxone IM stat, 1 g azithromycin PO stat, and valacyclovir 500 mg BD for five days. The lesion persisted despite treatment, and during follow-up, a punch biopsy was carried out. She was diagnosed with pseudoepitheliomatous hyperplasia of the epidermis. In addition to highlighting this condition that has been previously reported in human immunodeficiency virus/herpes simplex virus co-infection, this case demonstrates that unusual skin presentations must be considered in human immunodeficiency virus-infected individuals and illustrates the importance of biopsy for any non-healing lesions.
A 22-year-old Caucasian female presented with a vulval lesion of three weeks duration, increasing in size and becoming very tender. Sexual history was restricted to a single regular male partner of two years, with whom she had oral intercourse, vaginal intercourse, and anal intercourse without condoms. She was diagnosed with HIV infection eight weeks prior to the onset of the lesion, her CD4 cell count at diagnosis was 540 with an HIV-1 RNA viral load 5400 copies/ml. Antiretroviral treatment (ART) was commenced four weeks following diagnosis. One month after starting ART, her CD4 cell count had risen to 630 and viral load declined to 40 copies per ml. There was no other medical history and no tobacco, alcohol, or recreational drug use. Moreover, there was no history of previous dermatological conditions or travel to remote areas in Australia or other countries.
On examination, there was a single well-demarcated, raised, tender lesion on the left outer aspect of the labium minus. There was no ulceration, no contact bleeding, no induration, or lymphadenopathy. Her systemic examination was unremarkable. The following investigations were carried out: standard microscopy and culture, polymerase chain reaction testing for Haemophilus ducreyi, Klebsiella granulomatis, Treponema pallidum (T. pallidum), Herpes simplex (HSV), Herpes zoster, Chlamydia trachomatis, Neisseria gonorrhoeae, and serology for T. pallidum. The patient was presumptively treated for chancroid and genital herpes on initial presentation with ceftriaxone 500 mg IM stat, azithromycin 1 g PO stat, and valacyclovir 500 mg BD for five days. Her partner was treated with azithromycin 1 g PO stat, and they were both advised not to have sexual intercourse for seven days.
Junction between a squamous proliferation of pseudoepitheliomatous hyperplasia involving the epidermis and extending into the dermis (left) and normal skin (right). H + E × 100. Deep edge of the lesion shows tentacles and separated nests of a cytologically bland squamous proliferation extending into the dermis. H + E × 100.
During a follow-up visit seven days later, the lesion was still present and remained painful and showed no morphological change. All investigations for sexually transmitted infections (listed above) were negative. A 3 mm vulval punch biopsy was performed and sent for histopathology.
The histopathology report revealed pseudoepitheliomatous hyperplasia (PEH) of the epidermis arising from adjacent normal skin (Figures 1 and 2). There was marked proliferation of squamous epithelium forming tentacles and nests growing down into the dermis and associated with oedema of the stroma and a few lymphocytes. There was no atypia desmoplastic stroma, eosinophils, or koilocytosis seen. No organisms were seen on Periodic acid-Schiff. No viral inclusions were seen on routine histology. The herpes and Cytomegalovirus immunoperoxidase stains were negative. The p16 showed a non-specific staining pattern. The histopathology findings did not suggest a specific cause of her PEH. The lesion resolved spontaneously post punch biopsy, and the patient was lost to further follow-up.
PEH is a benign condition that causes hyperplasia of epidermis and resembles squamous cell carcinoma. The lesion is usually a well-demarcated plaque or nodule with or without scaling or crusting. PEH lesions are usually skin coloured or tan pink and they can increase in size or spontaneously regress. 1 Many different conditions are associated with PEH including infections, neoplasia, dermatoses, and other miscellaneous processes. 1
We have presented a case of an HIV-positive female with a single hypertrophic genital lesion due to PEH which did not respond to initial treatment, but subsequently spontaneously resolved six weeks later. Previous case reports have described PEH in association with herpes simplex virus (HSV) and HIV co-infection (HSV).2–9 Lesions are usually exophytic and have been reported to be painful. 3 Herpes simplex virus type 2 is known to affect up to 90% of HIV-infected individuals and diagnosing genital lesions in these patients can be very challenging in view of their atypical presentation, particularly in the context of immune reconstitution inflammatory syndrome.2,3
Despite HSV PCR and immunostaining not detecting HSV, it remains likely this was the cause of her PEH reaction given the painful nature of the lesion and high prevalence of HSV infection. HSV can be difficult to detect in biopsy samples due to the effect not occurring uniformly across the lesion, 4 the presence of marked granulomatous tissue 2 and an exaggerated response in the presence of minimal organisms due to IRIS. 2
Footnotes
Declaration of conflicting interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.