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Article

Unique development: Pyoderma gangrenosum appear during ulcerative colitis

In a recent case study, a patient receiving infliximab for the treatment of ulcerative colitis paradoxically developed widespread pyoderma gangrenosum lesions, a disease which normally responds well to this biologic therapy.

Medellin, Colombia - Biologics are one of the current therapies that prove to be effective in the treatment and management of ulcerative colitis and have also been successfully used in the treatment of pyoderma gangrenosum.

In a recent case study, a patient receiving infliximab for the treatment of ulcerative colitis developed widespread cutaneous lesions resembling pyoderma gangrenosum, which normally responds very well to biologic therapy. In this case report however, the exception appears to make the rule.

"This is an interesting case in the sense that patients who have pyoderma gangrenosum normally respond well to treatment with biologics, such as infliximab; however, our patient paradoxically developed widespread cutaneous pyoderma gangrenosum lesions," says Natalia Jaimes M.D., a dermatology resident of the Universidad Pontificia Bolivariana, department of dermatology, Hospital Pablo Tabón Uribe, Medellin, Colombia.

The 47-year-old Hispanic male patient with a history of ulcerative colitis was resistant to medical and surgery treatments and, therefore, the surgery staff started the patient on a course of infliximab. The intestinal symptoms such as diarrhea and cramping markedly improved with the infliximab therapy.

However, six months following the first dose, the patient developed erythemato-violaceous ulcers on the trunk, abdomen, genitalia, gluteus, extremities, left preauricular region and peristomally. Histopathology exam revealed an ulcerated epidermis with a dense cellular infiltrate composed of giant cells, plasma cells, lymphocytes, neutrophils and abscesses. Though diagnostically inconclusive, the histology raised the possibility of pyoderma gangrenosum.

The patient was taken off infliximab therapy and subsequently received methylprednisolone for five days and topical tacrolimus 0.1 percent bid. The ulcerative lesions slowly improved except for the peristomal lesion, which proved very difficult to treat, likely due to the constant trauma involved with the area. The patient is currently doing well and the plan is to start the patient on another biologic therapy soon.

Anti-TNF drugs such as Infliximab are designed to stimulate the immune system and interfere with specific cytokines involved in the inflammatory response seen in inflammatory bowel disease.

The precise etiology of pyoderma gangrenosum remains unknown; however, more than half of the cases are associated with systemic disease, especially inflammatory bowel disease.

According to Dr. Jaimes, the clinical presentation of pyoderma gangrenosum is variable and the histologic features are not pathognomonic. Infliximab, etanercept and adalimumab are nonapproved biologic drugs that have been used in the treatment of sterile inflammatory diseases with cutaneous manifestations such as pyoderma gangrenosum, and have all shown to be effective in various case reports and randomized studies.

"The majority of the patients with pyoderma gangrenosum normally improve with anti-TNF therapy. It is difficult to explain the paradox seen in this case but one theory is that the hereditary predisposition of the inflammatory bowel disease is so strongly expressed in our patient that the infliximab could simply not take hold," Dr. Jaimes says.

This case underscores the fact that paradoxical reactions do occur in dermatologic therapy. According to Dr. Jaimes, physicians should treat each patient individually, follow-up each patient carefully and be vigilant for such cases and expect the unexpected. DT

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