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Pyodermatitis–Pyostomatitis Vegetans: Two Cases and a Review of the Literature

  • Case Report
  • Published:
Journal of Cutaneous Medicine and Surgery: Incorporating Medical and Surgical Dermatology

Abstract

Background: Pyodermatitis–pyostomatitis vegetans (PD–PSV) is a rare vegetating, pustular, eosinophilic, mucocutaneous dermatosis characterized by mucocutaneous lesions of the genital, axillary, and oral regions, as well as on the scalp. Objective: We report two patients who were diagnosed with PD–PSV. The published cases of this rare disorder are reviewed. Results: The first patient presented with vegetating pustular plaques of the scalp, vulva, and mouth. The second patient initially showed pustules on fingers and then on toes. Vulva and mouth were later involved. There was no gastrointestinal involvement in either case. In both cases, histology revealed eosinophilic spongiosis with eosinophilic microabscesses and pseudoepitheliomatous hyperplasia. Direct immunofluorescence was negative. Conclusion: Pyodermatitis–pyostomatitis vegetans is a rare disorder of unknown cause. It bears similarities to pemphigus vegetans but histology, direct immunofluorescence, and clinical history show the differences. Response to treatment is generally rapid. In several reported cases, an inflammatory gastrointestinal disorder is found in association with the mucocutaneous signs.

Antécédents: La pyodermite-pyostomatite végétante (PD-PSV) est une dermatose cutanéo-muqueuse éosinophilique rare, caractérisée par des lésions cutanéo-muqueuses des régions génitale, axillaires et buccale, ainsi que du scalp. Objectifs: Nous rapportons le cas de deux patientes atteintes de PD-PSV. Les cas publiés de cette affection rare sont revus. Rsultats: La première patiente avait des plaques pustuleuses végétantes du scalp, de la vulve et de la bouche. La seconde patiente présentait initialement des pustules sur les doigts ensuite sur les orteils. L’affection a impliqué plus tard la vulve et la bouche. Aucune des patientes n’a présenté une atteinte gastro-intestinale. Dans les deux cas, l’histologie a révélé une spongiose éosinophilique, avec miocroabcès éosinophilique et hyperplastie pseudoépithéliomateuse. L’immunofluorescence directe s’est révélée négative. Conclusion: La pyodermite-pyostomatite végétante (PD-PSV) est un trouble rare d’origine inconnue. Les similarités avec le pemphigus végétant sont différenciées par l’histologie, l’immunofluorescence directe et l’historique clinique. La réponse au traitement est généralement rapide. Dans plusieurs cas rapportés, un trouble gastro-intestinal inflammatoire est associé aux signes cutanéo-muqueux.

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Authors and Affiliations

  1. Department of Dermatology, Centre Hospitalier Universitaire de Québec, pavillon Hotel-Dieu de Québec, Québec, Québec, Canada

    Simon Nigen, Yves Poulin & Éric Gagné

  2. Centre Dermatologique du Québec Métropolitain, Québec, Québec, Canada

    Linda Rochette

  3. Department of Dermatology, Centre Hospitalier Régional de Rimouski, Québec, Québec, Canada

    Marie–Hélène Lévesque

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  1. Simon Nigen
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  2. Yves Poulin
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  3. Linda Rochette
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  5. Éric Gagné
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Correspondence to Yves Poulin.

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Nigen, S., Poulin, Y., Rochette, L. et al. Pyodermatitis–Pyostomatitis Vegetans: Two Cases and a Review of the Literature . JCMS 7, 250–255 (2003). https://doi.org/10.1007/s10227-002-0112-7

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  • DOI: https://doi.org/10.1007/s10227-002-0112-7

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