Pyostomatitis vegetans (PSV)-pyodermatitis vegetans (PDV): A clinicopathologic study of 7 cases at a tertiary referral center

J Am Acad Dermatol. 2016 Sep;75(3):578-584. doi: 10.1016/j.jaad.2016.03.047. Epub 2016 Jun 24.

Abstract

Background: Pyostomatitis vegetans (PSV)-pyodermatitis vegetans (PDV) is a rare inflammatory mucocutaneous disease associated with inflammatory bowel disease.

Objective: We sought to evaluate the clinicopathologic findings of PSV-PDV in a series of 7 patients.

Methods: We conducted a retrospective review of all cases of PSV-PDV at the Mayo Clinic from 1995 to 2014.

Results: Seven patients with PSV-PDV were included, and all had inflammatory bowel disease. Three had Crohn's disease and 4 had ulcerative colitis. Three patients had peripheral blood eosinophilia. Two had concomitant pyoderma gangrenosum in which pyoderma gangrenosum lesions were recalcitrant to therapy. Primary sclerosing cholangitis was seen in 3 patients. Two patients had direct and 3 had indirect immunofluorescence findings. Tissue eosinophilia was seen in the majority of mucosal and cutaneous lesions.

Limitations: Limited sample size and retrospective study design are limitations.

Conclusions: PSV-PDV is associated with inflammatory bowel disease and primary sclerosing cholangitis and may precede gastrointestinal symptoms. Immunofluorescence findings in select PSV-PDV cases may indicate possible overlap with autoimmune bullous disease. Tissue eosinophilia may be helpful in distinguishing PSV-PDV from pyoderma gangrenosum. Strict control of bowel disease and close monitoring of patients with subclinical disease is warranted.

Keywords: Crohn's disease; inflammatory bowel disease; pemphigus; primary sclerosing cholangitis; pyoderma gangrenosum; pyodermatitis; pyostomatitis; ulcerative colitis; vegetans.

MeSH terms

  • Adult
  • Age Distribution
  • Biopsy, Needle
  • Comorbidity
  • Female
  • Humans
  • Immunohistochemistry
  • Incidence
  • Inflammatory Bowel Diseases / diagnosis
  • Inflammatory Bowel Diseases / epidemiology*
  • Male
  • Middle Aged
  • Prognosis
  • Pyoderma / diagnosis
  • Pyoderma / epidemiology*
  • Pyoderma / pathology*
  • Retrospective Studies
  • Risk Assessment
  • Sampling Studies
  • Severity of Illness Index
  • Sex Distribution
  • Stomatitis / diagnosis
  • Stomatitis / epidemiology*
  • Stomatitis / pathology*
  • Tertiary Care Centers
  • Young Adult