Allergic contact stomatitis is a rare disorder,

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Allergic Contact Stomatitis: A Case Report and Review of Literature P Lokesh, T Rooban, Joshua Elizabeth, K Umadevi, K Ranganathan Abstract Allergic contact stomatitis is a well-recognized entity, which may be easily overlooked by the clinician since its signs and symptoms are similar to various other oral lesions. Accurate diagnosis warrants adequate treatment that will help in providing prompt relief and will also prevent further recurrences. We present a case report of a 27-year-old South Indian male student, who presented with multiple erythematous erosions involving much of the nonkeratinized oral mucosa. History revealed that there was a previous episode of a similar lesion, associated with intake of food with flavoring agents. Based on the history and clinical features, we arrived at a diagnosis of allergic contact stomatitis and successfully treated the lesions with topical and systemic antihistamines. Keywords: Allergic contact stomatitis, oral mucosa, antihistamines Allergic contact stomatitis is a rare disorder, which most clinicians are not familiar with. A wide variety of substances are known to elicit adverse oral mucosal reactions. Flavoring agents, preservatives and dental materials are the most common causes of allergic/hypersensitivity reactions related to oral mucosa. Flavoring agents and preservatives have been used widely in commercially available personal hygiene products and foods, thereby increasing the risk hypersensitivity reactions. Previous exposure with an allergen is essential for diagnosis of allergic contact stomatitis. Sensitization usually occurs through contact of allergen with the oral mucosa. Rarely, sensitization may also occur by contact of allergen with skin. Memory T cells are activated soon after the initial exposure. On re-exposure to the same allergen, a type IV hypersensitivity reaction occurs. This reaction may be delayed by at least 48 hours and the clinical presentation may vary depending on the severity of the reaction. Case report A 26-year-old dental postgraduate student presented with a complaint of pain and diffuse intraoral erythematous lesions for the past three days. The patient first experienced roughness and discomfort in the left Dept. of Oral and Maxillofacial Pathology, Ragas Dental College and Hospital, Chennai buccal mucosa four days ago. The following day he developed erythematous lesions and pain in the left buccal mucosa, followed by lesions on the anterior part of ventral tongue, soft palate, right buccal mucosa and lower labial mucosa. Difficulty in brushing, speech and burning sensation while eating were experienced, for which 2% benzocaine gel was applied 3-4 times every day before food intake. Intraoral examination revealed carious 26, glass ionomer cement (GIC) Class I restoration in 46, buccally inclined 18 and 28. Diffuse erythema involving the whole of soft palate, without extension on to the hard palate was seen (Fig. 1). Ventral surface of anterior tongue was bright red in color with few small whitish plaques, suggestive of necrosis (Fig. 2). Lower labial mucosa exhibited irregular zones of erythema. Large oval to irregular bright red patches surrounded by whitish edematous zones were seen on buccal mucosa, extending some distance into the vestibule on both right (Fig. 3) and left side (Fig. 4). The keratinized mucosa of the hard palate, gingiva and dorsum of the tongue was not involved. Further questioning did not reveal history of any change or use of oral hygiene products, recent dental treatment or drug intake. Eventually, patient did recall an episode of having food at a restaurant 2-3 days before developing the lesions. Patient also gave a history of episodes of recurrent minor aphthous ulcers. One such episode occurred about two years ago and was characterized by multiple painful oral lesions, 458 Indian Journal of Clinical Practice, Vol. 22, No. 9, February 2012

Figure 1. Diffuse erythema of soft palate. Figure 3. Erythematous lesions on right buccal mucosa. Figure 2. Erosive lesions on ventral surface of tongue. which developed soon after intake of specific food, which the patient has been avoiding since then. Based on the history and clinical features, a provisional diagnosis of allergic stomatitis was made. Management Patient was advised to avoid foods with preservatives and flavoring agents. Cetirizine hydrochloride 10 mg tablet hs, 5 ml of diphenhydramine hydrochloride syrup mixed with equal amount of an antacid liquid in a swish and swallow method 3-4 times daily were prescribed to alleviate the symptoms. During the follow-up visit, four days later, most of the initial lesions had healed without any scarring Figure 4. Erythematous lesions on left buccal mucosa. (Figs. 5-8). A mild increase in the pigmentation was seen on the buccal mucosa. Two new lesions, which were not present during the initial examination, were seen on both the left and right sides of buccal mucosa, adjacent to the upper canine and premolars (Figs. 9 and 10). These lesions were erythematous areas, 2 1 cm in size with whitish necrotic plaques. Patient was advised to continue the same medications for three more days, following which all the oral lesions healed completely. Discussion Contact stomatitis is an inflammation of the oral mucosa caused by external substances. It can be caused by a Indian Journal of Clinical Practice, Vol. 22, No. 9, February 2012 459

Figure 5. Soft palate - 4 days after therapy. Figure 7. Right buccal mucosa - 4 days after therapy. Figure 6. Ventral surface of tongue - 4 days after therapy. variety of substances, which can either act as irritant or allergic agents. These substances include dental materials, preservatives and flavoring agents in foods or oral hygiene products. Oral mucosa is less commonly prone to contact allergic reactions, when compared to skin, though the latter is exposed to a wide variety of antigenic stimuli. This can be attributed to the various biologic and physiologic differences between the two. Saliva acts as a solvent that solubilizes, dilutes and also starts digesting potential allergens and helps to wash them there by limiting the duration and number of molecules that contact oral mucosa. Limited keratinization makes hapten binding more difficult and the limited number of antigen presenting cells Figure 8. Left buccal mucosa - 4 days after therapy. in the oral mucosa decreases the chance of antigen recognition. Irritants and allergens that do contact the oral mucosa are removed more quickly because of higher vascularity and faster epithelial renewal rates than in keratinized skin. Balsam of peru, cinnamon, cinnamic aldehyde, menthol, peppermint and eugenol are some of the common oral flavoring allergens. These reactions can be either acute or chronic. Clinical presentations vary based on the nature of reaction, type of allergen site and duration of contact. Patients with acute lesions may present with burning or redness. Vesicles are rarely seen and if present rupture in a short while after formation. Some patients 460 Indian Journal of Clinical Practice, Vol. 22, No. 9, February 2012

Table 1. Differential Diagnoses Pemphigus Lupus erythematosus Pemphigoid Syphilis Lichen planus Friction-induced Drug reactions Contact stomatitis Erythema multiforme Erythematous candidiasis lesions during clinical examination. Erosions can also be caused by trauma arising from friction between the teeth or irregular dental restorations. Burns from hot foods, radiation and caustic chemicals also cause similar erosions. Figure 9. Lesions on right buccal mucosa, during review visit. Figure 10. Lesions on left labial vestibule, during review visit. may experience edema, itching or stinging sensation. Contact allergy lesions occur directly at the site of exposure to the causative agent. Acute lesions develop soon after antigenic exposure; diagnosis of these may be straightforward since a cause-and-effect relationship can be easily established. Chronic lesions typically present as areas of erythema, edema, desquamation and occasionally ulceration. In addition, allergic contact stomatitis can also present as erosions with rough surface and irregular borders, often surrounded by a red halo. These lesions may be indistinguishable from aphthous ulcers and other Hence, it is essential to elicit a thorough history and exclude other pathosis presenting with similar lesions clinically (Table 1). Patch testing of oral mucosa is difficult and may yield false-negative results. Some common conditions which can present as erosive lesions in the oral mucosa are listed in Table 1. Identification and elimination of the allergen that initiated the reaction is essential to treat the condition, as well as to prevent recurrences. If an association is not established, cutaneous patch testing may be useful. Lesions respond well once the antigenic stimulus is eliminated. Antihistamines, topical anesthetics and topical corticosteroids are the commonly used pharmacological agents. Use of antihistamine suspensions in a swish and swallow method provide the advantage of both local and systemic action. Some of these agents may not be tolerable when there is a mucosal breach. Hence, a well-tolerated, flavored antacid was included in the prescription. Conclusion Allergic contact stomatitis is a well-recognized entity, the incidence of which could be far more than that reported. Clinical presentation and histopathologic features are not always specific. Hence, a high-degree of suspicion and careful history taking to establish a cause-and-effect relationship is essential. Biopsy findings may be confirmatory but not always essential. Health practitioners should consider contact allergic stomatitis in the differential diagnosis of nonspecific oral lesions so as to provide proper treatment and avoid recurrences. Indian Journal of Clinical Practice, Vol. 22, No. 9, February 2012 461

Consent Written informed consent was obtained from the patient for publication of this case report and accompanying images. Competing Interests The authors declare that they have no competing interests. Acknowledgments We thank our Principal, Dr S Ramachandran, for encouraging the publication of this case report and Dr Yakob Martin, for the images. suggested reading 1. 2. 3. LeSueur BW, Yiannias JA. Contact stomatitis. Dermatol Clin 2003;21(1):105-14, vii. Tosti A, Piraccini BM, Peluso AM. Contact and irritant stomatitis. Semin Cutan Med Surg 1997;16(4):314-9. Ophaswongse S, Maibach HI. Allergic contact cheilitis. Contact Dermatitis 1995;33(6):365-70. 4. 5. 6. 7. 8. 9. 10. Nevelle B, Damm D, Allen C, Bouquot J. Oral and Maxillofacial Pathology. 3rd edition. Ostman PO, Anneroth G, Skoglund A. Amalgamassociated oral lichenoid reactions. Clinical and histologic changes after removal of amalgam fillings. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1996;81(4): 459-65. Kowitz G, Jacobson J, Meng Z, Lucatorto F. The effects of tartar-control toothpaste on the oral soft tissues. Oral Surg Oral Med Oral Pathol 1990;70(4):529-36. Tremblay S, Avon SL. Contact allergy to cinnamon: case report. J Can Dent Assoc 2008;74(5):445-61. De Rossi SS, Greenberg MS. Intraoral contact allergy: a literature review and case reports. J Am Dent Assoc 1998;129(10):1435-41. Raap U, Stiesch M, Reh H, Kapp A, Werfel T. Investigation of contact allergy to dental metals in 206 patients. Contact Dermatitis 2009;60(6):339-43. Torgerson RR, Davis MD, Bruce AJ, Farmer SA, Rogers RS 3rd. Contact allergy in oral disease. J Am Acad Dermatol 2007;57(2):315-21. 462 Indian Journal of Clinical Practice, Vol. 22, No. 9, February 2012