References

Wiesenfeld D, Ferguson MM, Mitchell DN, MacDonald DG, Scully C, Cochran K, Russell RI. Orofacial granulomatosis – a clinical and pathological analysis. Q J Med. 1985; 54:101-113
Campbell H, Escudier M, Patel P, Nunes C, Elliot TR, Barnard K Distinguishing orofacial granulomatosis from Crohn's disease: two separate disease entities?. Inflamm Bowel Dis. 2011; 17:2109-2115
Gibson J, Wray D. Human leucocyte antigen typing in orofacial granulomatosis. Br J Dermatol. 2000; 143:1119-1121
James J, Patton DW, Lewis CJ, Kirkwood EM, Ferguson MM. Oro-facial granulomatosis and clinical atopy. J Oral Med. 1986; 41:29-30
Campbell H, Escudier M, Patel P, Challacombe SJ, Sanderson JD, Lomer MC Review article: cinnamon- and benzoate-free diet as a primary treatment for orofacial granulomatosis. Aliment Pharmacol Ther. 2011; 34:687-701
Guttman-Yassky E, Weltfriend S, Bergman R. Resolution of orofacial granulomatosis with amalgam removal. J Eur Acad Dermatol Venereol. 2003; 17:344-347
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Orofacial Granulomatosis in an Orthodontic Patient: a Case Report

From Volume 12, Issue 1, January 2019 | Pages 25-28

Authors

Aman Ulhaq

BDS, MFDS, MSc, MOrth, FDOrth

Consultant in Orthodontics, Edinburgh Dental Institute, Edinburgh, UK

Articles by Aman Ulhaq

Katy Tidbury

BDS, MFDS RCS(Ed)

Specialty Registrar in Orthodontics, King's College Dental Institute, Bessemer Road, Camberwell, London SE5 9RW, UK

Articles by Katy Tidbury

Abstract

A case is presented of a 14-year-old male patient being treated with a fixed orthodontic appliance who presented with a persistent upper lip swelling. The patient was experiencing discomfort from the appliance and the appliance was debonded. A diagnosis of orofacial granulomatosis was established and the patient was managed carefully with a cinnamon- and benzoate-free diet leading to resolution of the swelling. Orofacial granulomatosis is a rare condition and its presentation, diagnosis, and management are discussed.

CPD/Clinical Relevance: Orthodontists should be aware of the presentation of orofacial granulomatosis and the possible challenges associated with managing these patients.

Article

Orofacial granulomatosis (OFG) is an uncommon chronic granulomatous inflammatory condition affecting the lips, face and oral mucosa in the absence of any recognized systemic condition.1 Swelling of the oral tissues is the most common presentation; erythema, cobblestoning, mucosal tags, ulceration, fissures and nodules may also be evident.2 The presence of long-standing disease may lead to the development of mucosal scarring. Histologically, OFG is characterized by non-caseating granulomas with multinucleated giant cells of macrophage origin, and oedema of the tissues.

The aetiology of OFG remains unclear with several theories having been postulated. Weak evidence exists for a genetic aetiology, as certain Human Leucocyte Antigen (HLA) alleles are more common in OFG patients than in the general population.3 An association with food intolerances and a history of atopy are also linked with this condition. A previous study has illustrated that up to 60% of patients with OFG were atopic, showing IgE reactions to common allergens.4 Food substances may be causative, or lead to the precipitation of the disease process. Common sensitivities include: benzoic acid, cinnamaldehyde, cinnamon, benzoates and chocolate.5 These can be found in foods which form part of a regular diet and may also be ‘hidden’ when used as food additives and labelled as ‘E-numbers’.

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