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CASE REPORT
Melkersson-Rosenthal syndrome with coeliac and allergic diseases
  1. Joana Albuquerque Martins1,2,
  2. António Azenha1,2,
  3. Rui Almeida3 and
  4. João Páscoa Pinheiro1,2
  1. 1 Physical and Rehabilitation Medicine Department, Centro Hospitalar e Universitario de Coimbra EPE, Coimbra, Portugal
  2. 2 Faculty of Medicine, University of Coimbra, Coimbra, Portugal
  3. 3 Department of Pathology, Centro Hospitalar e Universitario de Coimbra EPE, Coimbra, Portugal
  1. Correspondence to Dr Joana Albuquerque Martins, joanamrmamartins{at}hotmail.com

Abstract

A 45-year-old man presented with a 10-year history of relapsing oedema of the lips. Moreover, he exhibited recurrent facial nerve palsy since the age of 10 years, coeliac disease since the age of 12 years, atopic eczema, allergic rhinitis and asthma. Physical examination revealed lip swelling and lingua plicata. Thus, he presented the classic triad of Melkersson-Rosenthal syndrome which includes recurrent orofacial oedema, facial nerve palsy and fissured tongue. A lip biopsy confirmed our clinical diagnosis.

This case is particularly rare, as the classic triad is seen only in a minority of the cases. Moreover, allergic and coeliac diseases were observed concomitantly. This paper illustrates a potential pathophysiological interconnection between these pathologies in which interferon gamma could play a key role. To our knowledge, this is the first case report in which Melkersson-Rosenthal syndrome has been observed concurrently with coeliac disease.

  • coeliac disease
  • mouth
  • cranial nerves
  • allergy
  • asthma
  • pathology
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Footnotes

  • JAM and AA contributed equally.

  • Contributors JAM and AA performed the clinical study, diagnosis of the patient and drafted the manuscript. RA performed anatomopathological analysis and was involved in clinical data acquisition. JPP revised the manuscript. All authors read and approved the final manuscript.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Patient consent for publication Obtained.

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