Abstract
This paper reports on a case of Orofacial Granulomatosis (OFG) in which the presence of amalgam fillings appears to have played a part in the aetiology. Once these restorations were removed and replaced with an alternative composite restorative material, all symptoms and signs of OFG resolved completely. This case highlights the necessity to include dental metals in the patch test battery when performing delayed patch testing on patients with OFG.
Keywords
Introduction
Orofacial granulomatosis (OFG) typically presents with diffuse lip swelling. This is often a clinical diagnosis but, upon biopsy, it is characterised histologically by non-caseating epithelioid cell granulomas and oedema of the tissues. 1 The time of onset varies from infancy to old age but, it is being seen increasingly in young adults in whom it is often found to be a result of hypersensitivity to food additives such as cinnamaldehyde and benzoates.2,3 These additives are found in carbonated soft drinks, which can be consumed in high quantities by some individuals. The importance of immediate open patch testing to detect such hypersensitivity has been reported previously.4,5 However, as exemplified in this case, patch testing should also be performed to exclude delayed hypersensitivity.
Case presentation
An 18-year old man presented to the Oral Medicine service at Dundee Dental Hospital in January 2009 with a seven-month history of discomfort affecting the gingivae in addition to an intermittent swelling of the upper lip. Crohn’s disease was excluded by referral to a gastroenterologist. The possibility of Sarcoidosis was excluded by a normal level of serum angiotensin converting enzyme (SACE). Lip biopsy confirmed a chronic granulomatous inflammatory reaction and a diagnosis of OFG was made. Delayed patch testing was performed to the British Society for Cutaneous Allergy (BSCA) standard series and the departmental dental and oral series. Patch tests were applied with Finn Chambers® on Scanpore® tape, occluded for two days and readings performed at 48 and 96 h according to the methodology of the International Contact Dermatitis Research Group (see Figures 1 and 2). Open patch testing to food additives for possible intermediate non-immunological hypersensitivity were also performed and readings recorded at 30 mins. There was a positive (+) reaction to 0.5% mercury in petrolatum at 48 and 96 h. All other readings were negative.
Preparation of Finn chambers. Patch test application, Finn chambers in place.
The patient had 13 (mercury containing) amalgam surfaces in his mouth. A six-month period of avoidance by replacing his amalgam restorations with composite resulted in a complete resolution of the lip swelling with only some remaining lip dryness and angular cheilitis. Further review after six months revealed the patient had no further symptoms and no stigmata at all of OFG.
Conclusion
Clinically relevant positive patch tests to mercury and amalgam are not uncommon in patients with oral lichenoid reactions; replacement of amalgam fillings may lead to resolution of symptoms and signs.
6
However, although the association in patients with OFG has been reported rarely to date,7,8 this case adds to a number of cases of OFG which have resolved upon removal of amalgam fillings after it was found that the patient had a positive allergic response to mercury and/or dental amalgam. In our patient, testing only to food additives and the BSCA standard series would have missed a clinically relevant allergen. We suggest that in patients with OFG, dental metals should be added to the patch test battery.
Footnotes
Declaration of conflicting interests
None declared.
Funding
This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.