Issue |
Med Buccale Chir Buccale
Volume 22, Number 2, avril 2016
|
|
---|---|---|
Page(s) | 113 - 117 | |
Section | Cas clinique et revue de la littérature / Up-to date review and case report | |
DOI | https://doi.org/10.1051/mbcb/2015055 | |
Published online | 10 February 2016 |
Syndrome APECED et lichen plan atrophique : un cas
1
UFR d’odontologie, université Victor Segalen Bordeaux, Pôle
d’odontologie et santé buccale, hôpital Pellegrin, CHU de Bordeaux,
France
2
Service de dermatologie, hôpital Saint-André,
CHU de Bordeaux,
France
3
Service d’anatomie pathologique, hôpital Pellegrin,
CHU de Bordeaux,
France
* Correspondance :
emmanuelmr@hotmail.fr
Reçu :
15
Novembre
2015
Accepté :
30
Novembre
2015
Introduction : Le syndrome APECED (Autoimmune Polyendocrinopathy Candidiasis Ectodermal Dystrophy) est une maladie autosomique récessive caractérisée par l’association d’une atteinte endocrinienne auto-immune, d’une candidose cutanéo-muqueuse et d’une atteinte des tissus ectodermiques. Observation : Le cas d’un patient de 19 ans atteint d’un syndrome APECED est rapporté. Les signes retrouvés étaient une maladie d’Addison, une kératopathie et une alopécie. Le diagnostic de candidose cutanéo-muqueuse a été plusieurs fois porté mais jamais formellement prouvé chez ce patient. Un lichen plan atrophique a été diagnostiqué cliniquement et histologiquement sur la muqueuse buccale. Il a été traité par clobétasol topique. Discussion : L’apparition d’un lichen plan est inhabituelle dans ce syndrome. La question du caractère fortuit ou non du lichen et de la confusion diagnostique avec une candidose est posée. Conclusion : Les praticiens spécialistes de la cavité orale devraient connaître cette dystrophie muqueuse car elle peut être associée à des manifestations orales plus communes comme le lichen plan ou la candidose orale.
Abstract
Introduction: Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED) is an autosomal recessive disease defined by two of the following conditions: hypoparathyroidism, adrenocortical failure and chronic mucocutaneous candidiasis. Other autoimmune disorders may be associated with it. Chronic candidiasis is usually the first and most common aspect of the syndrome. Case report: We report the case of a 19-year-old man whose clinical manifestations of APECED comprised adrenocortical failure, keratopathy and alopecia. Chronic candidiasis was previously clinically suspected, but never demonstrated with mucocutaneous scraping and cultures. APECED status was confirmed after identification of a mutation in the AIRE gene. In the oral mucosa, atrophic and erosive lesions were observed, which were compatible with oral lichen planus. This was histologically confirmed. Discussion: The finding of oral lichen planus in an APECED patient is unusual, and it poses the question of the occurrence of misdiagnosis of candidiasis in some cases of APECED, which could be due to the lack of histological examinations of these lesions. Conclusion: Dentists should be aware of this dystrophy, as oral manifestations are not specific but can be related to more common oral diseases such as oral lichen planus or candidiasis.
Mots clés : APECED / lichen plan atrophique / candidose
Key words: APECED / oral lichen planus / candidiasis
© Les auteurs, 2016
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited
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