Orofacial granulomatosis and erythema multiforme in an adolescent with Crohn's disease

Georgesen, Corey; Huang, Jin; Avarbock, Andrew; Harp, Joanna; Magro, Cynthia M.

doi:10.1111/pde.13548

Cited by 3 publications

(1 citation statement)

References 15 publications

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“…Lip edema is the most common finding (over 90%), although it is rarely the sole clinical feature. Oral examination may reveal cobblestoning, ulceration, tags, a fissured tongue, and chronic erythematous gingivitis [5,6], with frequently alternating acute inflammation and remission periods. A global prevalence of 0.8% has been suggested [7].…”

Section: Introductionmentioning

confidence: 99%

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

et al. 2023

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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 anti-Saccharomyces cerevicae 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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Section: Introductionmentioning

confidence: 99%

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

et al. 2023

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Superimposed infections in inflammatory bowel diseases

Kulkarni

Shen

2020

Atlas of Endoscopy Imaging in Inflammatory Bowel Disease

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Pediatric cutaneous Crohn disease: A case series of 89 patients and review

McKay,

Liu,

Shaw

et al. 2024

Pediatric Dermatology

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BackgroundCutaneous (or “Metastatic”) Crohn disease (CCD) is a rare and underrecognized disease characterized by cutaneous granulomatous inflammation. We describe patient demographics, clinical characteristics, histology, and treatment of 89 pediatric cases of CCD, including 78 previously reported and 11 new cases seen at four academic institutions. We emphasize the efficacy of biologic mono‐ and dual therapy.MethodsPubMed identified cases using keywords including “metastatic Crohn disease” and “cutaneous Crohn disease”. Patients were identified by retrospective review of the electronic health record including histopathologic diagnosis consistent with CCD. Chart review collected demographic, clinical, and histologic data.ResultsMost pediatric patients with CCD are male 55% (49/89), present with edema (73/89, 82%) and erythema (47/89, 53%) of the genitals (33/49, 67%), and have intestinal Crohn disease (69/89, 78%). Oral corticosteroids (53/75, 71%) and metronidazole (29/75, 39%) are the most frequently prescribed medications. Of the 17 patients treated with tumor necrosis factor (TNF)‐blockade, 94% (16/17) had partial or total clearance. Ustekinumab resulted in clearance of cutaneous disease in two patients (2/3, 67%) and partial clearance in one patient (1/3, 33%). Two cases achieved total clearance with the use of dual biologic therapy defined as the use of two biologic therapies with differing mechanisms of action or the use of a biologic therapy and small molecule inhibitor.ConclusionsTNF blockade is an effective treatment for pediatric CCD, and interleukin‐12/23 inhibitors may be similarly effective. Consideration of dual biologic therapy may be useful in pediatric patients requiring discordant therapies for their intestinal and cutaneous CD.

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Orofacial granulomatosis and erythema multiforme in an adolescent with Crohn's disease

Cited by 3 publications

References 15 publications

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

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

Superimposed infections in inflammatory bowel diseases

Pediatric cutaneous Crohn disease: A case series of 89 patients and review

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