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CASE OF THE MONTH

Pemphigus vulgaris is rare in childhood,Dr. Jeffrey Callen said.Boy tested positive for pemphigus antibodies on direct immunofluorescence and the indirect immunofluorescence was greater than 1:640. His chest x-ray was normal.

BANFF, ALTA. — Pemphigus vulgaris is rare in childhood, Dr. Jeffrey Callen said in a poster session at the annual meeting of the Society for Pediatric Dermatology. The disease usually occurs between 40 and 60 years of age.

Dermatologically, oral lesions are often the initial manifestation. They are followed by erosions and crusts with or without flaccid blisters on the skin. The diagnosis is often made in the oral stage; treatment may result in prevention of the cutaneous blistering.

After biopsy revealed pemphigus vulgaris, this patient was sent to the division of dermatology at the University of Louisville Health Sciences Center for management.

At the initial consultation, the patient’s lesions were limited to the oral cavity Dr. Callen and his associates noted mild, non-inflammatory acne.

The boy tested positive for pemphigus antibodies on direct immunofluorescence and the indirect immunofluorescence was greater than 1:640. His chest x-ray was normal.

Biopsy revealed markedtombstoningof the basal keratinocytes. Acantholytic cells were observed in the suprabasilar cleft.

Initial therapy of prednisone 60 mg/day led to partial improvement, but Cushingoid features, including an acne flare, occurred early in the course. The boy had developed some skin lesions, but the primary problem remained his intraoral disease, said Dr. Callen, who is chief of the university’s division of dermatology.

The boy seemingly failed dapsone, methotrexate, and azathioprine therapies, but his compliance to drug administration was questionable. Mycophenolate mofetil also did not seem to help.

During his first semester in college, the patient attempted suicide while on low doses of prednisone. He was then given a 6-month course of monthly high-dose intravenous immune globulin in combination with daily prednisone (15-30 mg) and azathioprine (300 mg). He was able to taper the prednisone slightly during the intravenous immune globulin therapy He is now taking azathioprine 300 mg/day and prednisone 15 mg/day and is receiving intramuscular gold 50 mg/week.

This patient illustrates some of the difficulties in dealing with serious disease in adolescents,” Dr. Callen observed. “His suicide attempt is mirrored by his lack of taking medications that might improve his condition. In addition, he has difficulties keeping appointments, which is another symptom of his abnormal psyche.


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