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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MDPI AG
2023-03-01
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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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