The Art of Block Grafting

Atypical Gingival Manifestations that Mimic Endo et al Mucocutaneous Diseases in a Patient with Contact Stomatitis Caused by Toothpaste Hiroyasu Endo, DDS, PhD1 2 Fi ta Sisilia, DDS, PhD3 3 4 4 6 5 3 Abstract Background: The clinical features of contact diagnostic. The direct immunofluorescence find- stomatitis are generally nonspecific. The follow- ings indicated a linear pattern of antifibrinogen ing case report describes the gingival manifesta- in the basement membrane zone; a finding con- tions in a patient with contact stomatitis caused sistent with but not diagnostic for oral lichen pla- by toothpaste that closely mimicked the clini- nus. When the patient discontinued use of a new cal appearance of a mucocutaneous disease. toothpaste on her own volition, the lesions sub- sided, causing us to suspect a contact stomati- Methods: A 74 year-old female presented with tis. Rechallenge with the implicated toothpaste a one-month history of burning, erythematous resulted in the recurrence of the gingival symp- gingiva. Oral examination revealed localized ery- toms. A diagnosis of contact stomatitis caused thematous and edematous lesions on the maxil- by toothpaste was made based on these findings. lary and mandibular labial gingivae. The differential diagnosis included erosive lichen planus, mucous Conclusions: The gingival lesions described membrane pemphigoid, and pemphigus vulgaris. in this report were similar to those sometimes found in several oral mucocutaneous disorders. Results: A gingival biopsy was obtained from a Therefore contact hypersensitivity to oral hygiene lesional and perilesional site. The histopathologi- products should be considered when evaluat- cal findings were nonspecific and therefore not ing patients for oral mucocutaneous diseases. KEY WORDS: Gingival Diseases, Gingivitis, Toothpaste, Hypersensitivity 1. Department of Periodontology, Nihon University, School of Dentistry at Matsudo, JAPAN 2. Department of Periodontics, Baylor College of Dentistry, Texas A&M Health Science Center, Dallas, TX, USA 3. Department of Oral Pathology, Nihon University, School of Dentistry at Matsudo, JAPAN 4. Department of Oral Diagnosis, Nihon University, School of Dentistry at Matsudo, JAPAN 5. Department of Oral and Maxillofacial Implantology, Nihon University, School of Dentistry at Matsudo, JAPAN 6. Department of Preventive Management, Nihon University, School of Dentistry at Matsudo, JAPAN The Journal of Implant & Advanced Clinical Dentistry 101

Endo et al Figure 1a: Initial presentation right maxilla. The initial Figure 1b: Initial presentation left maxilla. The initial examination revealed localized erythematous and examination revealed localized erythematous and edematous lesions on the labial gingiva. edematous lesions on the labial gingiva. INTRODUCTION mucocutaneous diseases in a patient with contact stomatitis caused by toothpaste. Contact stomatitis is an inflammatory response of the oral mucosa caused by external sub- CASE REPORT stances. The substances act either as irritant or allergic contactants. The clinical features A 74 year-old female presented with a one-month of contact stomatitis are generally nonspe- history of burning, erythematous gingiva. Oral cific, and therefore, their symptoms are consid- examination revealed localized erythematous ered to be of unknown cause in many cases. and edematous lesions confined to the ante- rior maxillary and the mandibular labial gingivae Contact stomatitis caused by toothpaste (figures 1a, 1b). Neither the anterior palatal/ occurs at various sites, but the gingiva is the lingual gingivae nor the facial or lingual molar most common.1 The reported clinical features area were affected. The lesions occurred mainly include gingivitis involving the full width of on the attached gingiva, although the erythema the attached gingivae,2 erythema,1,3,4 hyper- extended into the alveolar mucosa. The lesions plasia,3 edema,5 desquamation,3 and ulcer- were not seen on the free and papillary gingiva, ation.4 According to the classification of and did not appear to be related to plaque accu- periodontal diseases and conditions, allergic mulation. Nikolsky’s sign, which involves the reactions due to oral hygiene products are application of a shear force on normal-appearing included in the non-plaque-induced gingival gingiva producing epithelial desquamation, was lesion category.6 In general, however, there positive. These observations were consistent are few reports concerning gingival manifesta- with mild desquamative gingivitis. A review of tions with contact stomatitis in the dental lit- the patient’s medical history was unremarkable. erature. The following case report describes She did not smoke or take any medications. The atypical gingival manifestations that mimic 102 Vol. 2, No. 2 March 2010

Endo et al Figure 2a: Histopathologic specimen from the gingiva Figure 2b: Histopathologic specimen from the gingiva (H&E stain). Histopathological ndings revealed di use (H&E stain). Perivascular in ltration in the deep layer was subepithelial in ltration composed of lymphocytes. observed (arrow). differential diagnosis included erosive lichen pla- Figure 3: Direct immuno uorescence (DIF) with deposits nus (LP), mucous membrane pemphigoid (MMP) of brinogen. The DIF ndings indicated a linear pattern of and pemphigus vulgaris (PV). A gingival biopsy anti brinogen in the basement membrane zone. was made that included lesional and perile- sional tissue. The specimen was submitted for two weeks after the toothpaste was discontin- routine histopathology and direct immunofluo- ued, contact stomatitis was suspected (figures rescence (DIF). The histopathological exami- 4a, 4b). After informed consent, the patient nation revealed hyperparakeratosis and diffuse was asked to use the implicated toothpaste subepithelial infiltration composed of lympho- again twice daily for several days. This rechal- cytes (figure 2a). Perivascular infiltration was lenge with the toothpaste resulted in the recur- observed deep in the submucosa (figure 2b). rence of gingival signs and symptoms (figures DIF testing was performed using conjugates 5a, 5b). A diagnosis of toothpaste induced con- for IgG, IgA, IgM, C3, and fibrinogen. The DIF tact stomatitis was made based on these find- findings indicated a linear deposition of fibrino- gen in the basement membrane zone (figure 3). One week after the biopsy, the erythema- tous lesions were diminished. Two weeks later, most of the erythema had disappeared. When asked about her daily oral hygiene practices, the patient reported that she had switched to a new kind of toothpaste about one month ago but quit using it after the biopsy was done. Since the gingival lesions disappeared within The Journal of Implant & Advanced Clinical Dentistry 103

Endo et al Figure 4a: Right maxilla after discontinuing toothpaste. Figure 4b: Left maxilla after discontinuing toothpaste. Note disappearance of the gingival lesions. Note disappearance of the gingival lesions. Figure 5a: Right maxilla after 7 days of rechallenge with Figure 5b: Left maxilla after 7 days of rechallenge with the implicated toothpaste. Note recurrence of gingival the implicated toothpaste. Note recurrence of gingival erythema. erythema. ings. The patient’s symptoms had not recurred erosive, vesiculobullous and/or desquamative seven months after the toothpaste was dis- lesions of the gingiva is a clinical manifestation continued (figures 6a, 6b). She returned to caused by several diseases, mainly those related the toothpaste that she had used for years. to mucocutaneous diseases.7 Toothpaste con- tact stomatitis has also been reported as a DISCUSSION cause of desquamative gingivitis.1 Histopatho- logical & DIF examinations may be required The gingival manifestations described in this to facilitate the diagnosis.7 Non-specific histo- report are similar to those of mucocutaneous pathologic findings with submucosal perivas- diseases such as PV, MMP and LP. Desquama- cular inflammatory cell infiltration should raise tive gingivitis characterized by erythematous, 104 Vol. 2, No. 2 March 2010

Endo et al Figure 6a: Right maxilla seven months after discontinuing Figure 6b: Left maxilla seven months after discontinuing toothpaste. No recurrence of initially presented gingival toothpaste. No recurrence of initially presented gingival symptoms. symptoms. suspicion of a contact hypersensitivity etiology. uefaction. The DIF findings are only suggestive, PV is an autoimmune blistering disease char- rather than diagnostic, of LP. Characteristic DIF findings in LP include a linear deposition of fibrin acterized by acantholysis in the epithelium.8 Most or fibrinogen in the basement membrane zone. patients with PV are middle-aged or elderly. The disease is equally common in men and women. Few reports have been made on the histo- Histopathologically, PV is characterized by intraep- pathological findings of contact stomatitis.1,11-13 ithelial bulla formation. In the DIF examination of These reports exhibit some similarities, though PV patients, the deposition of IgG and/or C3 is most tissue reactions are nonspecific. Four types found in the intercellular spaces of the epithelium. of tissue reactions are reported: 1) psoriasi- form tissue reaction;12 2) lichenoid tissue reac- MMP is an autoimmune, subepithelial blister- tion;13 3) perivascular infiltration in the deep ing disease that affects the mucous membranes.9 layer;3 and 4) non-caseating epitheloid granu- Most patients with MMP are in their 50s, and the lomas and multinucleated giant cells in the oral majority of them are women. Histopathologically, mucosa.11 In the case reported in this article, MMP is characterized by subepithelial bulla for- the histopathological findings were nonspecific mation. In DIF testing of MMP, the linear depo- and therefore not diagnostic. The histopatho- sition of C3, IgG or other immune globulin is logical findings revealed hyperparakeratosis and observed along the basement membrane zone. diffuse subepithelial inflammatory cell infiltration consisting mainly of lymphocytes. Perivascular LP is a chronic inflammatory disease caused infiltration in the deep layer was also observed, by unknown etiology.10 Most patients with LP are alerting one to the possibility of a contact hyper- middle-aged or older, and women are affected sensitivity reaction. The DIF findings consisted about twice as often as men. Histopathologi- of a linear deposition of fibrinogen along the cally, LP is characterized by band-like lymphocyte infiltration below the epithelium and basal cell liq- The Journal of Implant & Advanced Clinical Dentistry 105

Endo et al basement membrane zone, which is suggestive Disclosure of, but not diagnostic for, a lichenoid reaction. The authors report no conflicts of interest with anything mentioned in this article. References Although it is possible for any of the ingredi- 1. Endo H, Rees TD. Clinical features of cinnamon-induced contact stomatitis. ents found in toothpaste to become allergens, the most common allergens are flavors and pre- Compend Contin Educ Dent 2006;27(7): 403-10. servatives.14, 15 Patch testing can be performed 2. Lamey PJ, Lewis MA, Rees TD, Fowler C, Binnie WH, Forsyth A. Sensitivity to identify the specific allergens in toothpaste.1 Menthol, sodium benzoate and paraben were reaction to the cinnamonaldehyde component of toothpaste. Br Dent J ingredients in the toothpaste that this patient was 1990;168(3):115-8. using, and all of these have been reported to be 3. Macleod RI, Ellis JE. Plasma cell gingivitis related to the use of herbal allergens.14-17 In this case, however, the allergen toothpaste. Br Dent J 1989;20;166(10):375-6. could not be definitively identified because the 4. Thyne G, Young DW, Ferguson MM. Contact stomatitis caused by toothpaste. patient did not agree to undergo patch testing. N Z Dent J 1989;85(382):124-6. 5. Monti M, Berti E, Carminati G, Cusini M. Occupational and cosmetic dermatitis CONCLUSIONS from propolis. Contact Dermatitis 1983;9(2):163. 6. Holmstrup P. Non-plaque-induced gingival lesions. Ann Periodontol We reported on a case of contact stomatitis 1999;4:20-31. caused by toothpaste. This case showed local- 7. Lo Russo L, Fedele S, Guiglia R, Ciavarella D, Lo Muzio L, Gallo P, Di Liberto ized erythematous and edematous lesions in C, Campisi G. Diagnostic pathways and clinical significance of desquamative the attached gingiva and extending into the gingivitis. J Periodontol 2008;79(1):4-24. alveolar mucosa. These lesions were similar to 8. Endo H, Rees TD, Matsue M, Kuyama K, Nakadai M, Yamamoto H. Early those found in mucocutaneous disorders. We detection and successful management of oral pemphigus vulgaris: a case suggest that the adverse reactions induced report. J Periodontol 2005;76(1):154-60. by gingival contact hypersensitivity should be 9. Endo, H., Rees, T.D., Kuyama, K., Kono, Y. and Yamamoto, H.: Clinical and considered in diagnosis of individuals with diagnostic features of mucous membrane pemphigoid. Compend Contin Educ apparent gingival mucocutaneous disease Dent 2006;27(9):512-517. 10. Endo H, Rees TD, Kuyama K, Matsue M, Yamamoto H. Successful treatment Correspondence: Dr. Hiroyasu Endo using occlusive steroid therapy in patients with erosive lichen planus: A report Dept. of Periodontology on 2 cases. Quintessence Int 2008;39(4):e162-72. Nihon University School of Dentistry at 11. Endo H, Rees TD. Cinnamon products as a possible etiologic factor in Matsudo orofacial granulomatosis. Med Oral Patol Oral Cir Bucal 2007;12(6):E440-4. 2-870-1 Sakaecho Nishi, 12. Allen CM, Blozis GG. Oral mucosal reactions to cinnamon-flavored chewing Matsudo, Chiba, Japan, 271-8587 gum. J Am Dent Assoc 1988;116:664-667. TEL: 81-47-368-6111 13. Miller RL, Gould AR, Bernstein ML. Cinnamon-induced stomatitis venenata. FAX: 81-47-364-6295 Clinical and characteristic histopathologic features. Oral Surg Oral Med Oral e-mail address: [email protected] Pathol 1992;73:708-716. 14. Sainio EL, Kanerva L. Contact allergens in toothpastes and a review of their hypersensitivity. Contact Dermatitis 1995;33(2):100-5. 15. Rees TD. Orofacial granulomatosis and related conditions. Periodontol 2000 1999;21:145-57. 16. Wilkinson SM, Beck MH. Allergic contact dermatitis from menthol in peppermint. Contact Dermatitis 1994;30(1):42-3. 17. Munoz FJ, Bellido J, Moyano JC, Alvarez M, Fonseca JL. Perioral contact urticaria from sodium benzoate in a toothpaste. Contact Dermatitis 1996;35(1):51. 106 Vol. 2, No. 2 March 2010

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