Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case Report

Orofacial granulomatosis (OFG) represents a heterogeneous group of rare orofacial diseases. When affecting gingiva, it appears as a chronic soft tissue inflammation, sometimes combined with the enlargement and swelling of other intraoral sites, including the lips. Gingival biopsy highlights noncasea...

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Main Authors: Chiara Cecchin-Albertoni, Laetitia Pieruccioni, Thibault Canceill, Robin Benetah, Jade Chaumont, Christophe Guissard, Paul Monsarrat, Philippe Kémoun, Mathieu Marty
Format: Article
Language:English
Published: MDPI AG 2023-03-01
Series:Medicina
Subjects:
Online Access:https://www.mdpi.com/1648-9144/59/4/673
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author Chiara Cecchin-Albertoni
Laetitia Pieruccioni
Thibault Canceill
Robin Benetah
Jade Chaumont
Christophe Guissard
Paul Monsarrat
Philippe Kémoun
Mathieu Marty
author_facet Chiara Cecchin-Albertoni
Laetitia Pieruccioni
Thibault Canceill
Robin Benetah
Jade Chaumont
Christophe Guissard
Paul Monsarrat
Philippe Kémoun
Mathieu Marty
author_sort Chiara Cecchin-Albertoni
collection DOAJ
description Orofacial granulomatosis (OFG) represents a heterogeneous group of rare orofacial diseases. When affecting gingiva, it appears as a chronic soft tissue inflammation, sometimes combined with the enlargement and swelling of other intraoral sites, including the lips. Gingival biopsy highlights noncaseating granulomatous inflammation, similar to that observed in Crohn’s disease and sarcoidosis. At present, the etiology of OFG remains uncertain, although the involvement of the genetic background and environmental triggers, such as oral conditions or therapies (including orthodontic treatment), has been suggested. The present study reports the results of a detailed clinical and 2D/3D microscopy investigation of a case of gingival orofacial granulomatosis in an 8-year-old male patient after orthodontic therapy. Intraoral examination showed an erythematous hyperplasia of the whole gingiva with a granular appearance occurring a few weeks after the installation of a quad-helix. Peri-oral inspection revealed upper labial swelling and angular cheilitis. General investigations did not report ongoing extra-oral disturbances with the exception of a weakly positive <i>anti-Saccharomyces cerevicae</i> IgG auto-antibody. Two- and three-dimensional microscopic investigations confirmed the presence of gingival orofacial granulomatosis. Daily corticoid mouthwashes over a period of 3 months resulted in a slight improvement in clinical signs, despite an intermittent inflammation recurrence. This study brings new insights into the microscopic features of gingival orofacial granulomatosis, thus providing key elements to oral practitioners to ensure accurate and timely OFG diagnosis. The accurate diagnosis of OFG allows targeted management of symptoms and patient monitoring over time, along with early detection and treatment of extra-oral manifestations, such as Crohn’s disease.
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spelling doaj.art-f2c5e4ecd6e846e5a4969252e49bf2d02023-11-17T20:20:19ZengMDPI AGMedicina1010-660X1648-91442023-03-0159467310.3390/medicina59040673Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case ReportChiara Cecchin-Albertoni0Laetitia Pieruccioni1Thibault Canceill2Robin Benetah3Jade Chaumont4Christophe Guissard5Paul Monsarrat6Philippe Kémoun7Mathieu Marty8Oral Medicine Department and CHU de Toulouse, Competence Center of Oral Rare Diseases, Toulouse Institute of Oral Medicine and Science, CEDEX 9, 31062 Toulouse, FranceRESTORE Research Center, Université de Toulouse, INSERM, CNRS, EFS, ENVT, Batiment INCERE, 4bis Avenue Hubert Curien, 31100 Toulouse, FranceOral Medicine Department and CHU de Toulouse, Competence Center of Oral Rare Diseases, Toulouse Institute of Oral Medicine and Science, CEDEX 9, 31062 Toulouse, FranceOral Medicine Department and CHU de Toulouse, Competence Center of Oral Rare Diseases, Toulouse Institute of Oral Medicine and Science, CEDEX 9, 31062 Toulouse, FranceOral Medicine Department and CHU de Toulouse, Competence Center of Oral Rare Diseases, Toulouse Institute of Oral Medicine and Science, CEDEX 9, 31062 Toulouse, FranceOral Medicine Department and CHU de Toulouse, Competence Center of Oral Rare Diseases, Toulouse Institute of Oral Medicine and Science, CEDEX 9, 31062 Toulouse, FranceOral Medicine Department and CHU de Toulouse, Competence Center of Oral Rare Diseases, Toulouse Institute of Oral Medicine and Science, CEDEX 9, 31062 Toulouse, FranceOral Medicine Department and CHU de Toulouse, Competence Center of Oral Rare Diseases, Toulouse Institute of Oral Medicine and Science, CEDEX 9, 31062 Toulouse, FranceOral Medicine Department and CHU de Toulouse, Competence Center of Oral Rare Diseases, Toulouse Institute of Oral Medicine and Science, CEDEX 9, 31062 Toulouse, FranceOrofacial granulomatosis (OFG) represents a heterogeneous group of rare orofacial diseases. When affecting gingiva, it appears as a chronic soft tissue inflammation, sometimes combined with the enlargement and swelling of other intraoral sites, including the lips. Gingival biopsy highlights noncaseating granulomatous inflammation, similar to that observed in Crohn’s disease and sarcoidosis. At present, the etiology of OFG remains uncertain, although the involvement of the genetic background and environmental triggers, such as oral conditions or therapies (including orthodontic treatment), has been suggested. The present study reports the results of a detailed clinical and 2D/3D microscopy investigation of a case of gingival orofacial granulomatosis in an 8-year-old male patient after orthodontic therapy. Intraoral examination showed an erythematous hyperplasia of the whole gingiva with a granular appearance occurring a few weeks after the installation of a quad-helix. Peri-oral inspection revealed upper labial swelling and angular cheilitis. General investigations did not report ongoing extra-oral disturbances with the exception of a weakly positive <i>anti-Saccharomyces cerevicae</i> IgG auto-antibody. Two- and three-dimensional microscopic investigations confirmed the presence of gingival orofacial granulomatosis. Daily corticoid mouthwashes over a period of 3 months resulted in a slight improvement in clinical signs, despite an intermittent inflammation recurrence. This study brings new insights into the microscopic features of gingival orofacial granulomatosis, thus providing key elements to oral practitioners to ensure accurate and timely OFG diagnosis. The accurate diagnosis of OFG allows targeted management of symptoms and patient monitoring over time, along with early detection and treatment of extra-oral manifestations, such as Crohn’s disease.https://www.mdpi.com/1648-9144/59/4/673orofacial granulomatosisorthodontic devicedental treatmentcheilitislight sheet fluorescence microscopy
spellingShingle Chiara Cecchin-Albertoni
Laetitia Pieruccioni
Thibault Canceill
Robin Benetah
Jade Chaumont
Christophe Guissard
Paul Monsarrat
Philippe Kémoun
Mathieu Marty
Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case Report
Medicina
orofacial granulomatosis
orthodontic device
dental treatment
cheilitis
light sheet fluorescence microscopy
title Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case Report
title_full Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case Report
title_fullStr Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case Report
title_full_unstemmed Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case Report
title_short Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case Report
title_sort gingival orofacial granulomatosis clinical and 2d 3d microscopy features after orthodontic therapy a pediatric case report
topic orofacial granulomatosis
orthodontic device
dental treatment
cheilitis
light sheet fluorescence microscopy
url https://www.mdpi.com/1648-9144/59/4/673
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