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Treatment of Painful and Recurrent Oral Mucoceles With a High-Potency Topical Corticosteroid: A Case Report
1737
LUIZ ET AL
J Oral Maxillofac Surg 66:1737-1739, 2008
Treatment of Painful and Recurrent Oral Mucoceles With a High-Potency Topical Corticosteroid: A Case Report Ana C. Luiz, DDS,* Karen R.N. Hiraki, DDS,† Celso A. Lemos, Jr, DDS, DSc,‡ Sílvio K. Hirota, DDS, MS,§ and Dante A. Migliari, DDS, DSc¶ Single mucoceles are a relatively common disorder of the oral mucosa, but the occurrence of multiple mucoceles is considered an uncommon event. There are only a few reports in the literature.1-6 Although no etiologic factor has been associated with multiple mucoceles, some authors reported on this condition in patients with oral lichen planus and in association with graft-versus-host disease.1,6,7 There is a hypothesis that an inflammatory process may play a role in the pathogenesis of recurrent mucoceles,6,7 although it was also argued that changes in minor salivary-gland function may contribute to the development of these lesions.2 We report on a case of multiple, painful, and recurrent mucoceles. Our main focus was on assessing the effectiveness of a topical high-potency corticosteroid for the treatment of this clinical condition.
Report of a Case A 33-year-old female was referred to our clinic for diagnosis of a recurrent “bullous” formation, accompanied by ulcerations, which had been present for about 1 month. The ulcerations had caused her severe pain and difficulty in chewing. Several translucent vesicles at multiple sites, dis-
Received from the School of Dentistry, University of São Paulo, São Paulo, Brazil. *Research Fellow, Department of Oral Diagnosis. †Doctoral Student, Department of Oral Pathology. ‡Assistant Professor, Department of Oral Diagnosis. §Research Fellow, Department of Oral Diagnosis. ¶Assistant Professor, Department of Oral Diagnosis. Address correspondence and reprint requests to Dr Migliari: Disciplina de Semiologia, Departamento de Estomatologia, Faculdade de Odontologia, Universidade de São Paulo, Av Prof Lineu Prestes 2227, Cidade Universitária, 05580-900 São Paulo, São Paulo, Brazil; e-mail: [email protected] © 2008 American Association of Oral and Maxillofacial Surgeons
0278-2391/08/6608-0027$34.00/0 doi:10.1016/j.joms.2008.01.050
tributed on the lower lip, buccal mucosa (bilaterally), and floor of the mouth, were observed in association with white striae and papules (Figs 1A-C). This clinical picture suggested either a combination of mucoceles with reticular oral lichen planus, or bullous lichen planus. Any other vesiculobullous diseases were clinically ruled out. Apart from her oral lesions, the patient was in good health. A biopsy was taken involving a vesicle together with a sample of the striae. Microscopic examination showed features only of a mucocele without any characteristics of lichen planus (Fig 1D). Thus, the final diagnosis was restricted to multiple mucoceles.
Management A high-potency topical corticosteroid (0.05% clobetasol propionate in Orabase; Colgate, New York, NY) was used mainly for controlling the symptomatology. The patient was instructed to apply the medication 3 times a day. After 4 weeks of treatment, she reported that the symptoms and frequency of recurrences had diminished. In addition, the white areas (striae and papules) had disappeared completely. Eventually, she was free of lesions and recurrences, and was taken off the medication. When the lesions recurred later, the possibility of an allergy-related mechanism for the mucoceles was investigated. The patient underwent skin-patch testing for standard (Brazilian) allergens and for dental materials (Trolab; Hermal, Reinbeck, Germany); the results were negative. She was also asked to avoid the use of toothpaste8 for a while and to refrain from potentially allergenic foods (eg, milk, eggs, citrus fruit, wheat, cinnamon, and seafood). Despite these measures, the mucoceles kept reappearing. Clobetasol propionate was restarted 3 times a day. As soon as improvement was observed, the medication was tapered. Three weeks later, she was again free of lesions, and was then instructed to use the medication only when she noticed initial symptoms of the lesions. For a time, she used the medication twice a week (on average) to control some flareups. At her latest appointment, after 13 months of
1738
TOPICAL CORTICOSTEROID FOR ORAL MUCOCELES
FIGURE 1. A, Cluster of superficial mucoceles on the labial mucosa. B, Single mucocele surrounded by white patches on the buccal mucosa. C, Ulcerations resulting from ruptured mucoceles, along with white striae and papules suggesting oral lichen planus, seen on the left side of the buccal mucosa. A similar pattern was seen on the right side (not shown). D, A pool of mucin surrounded by granulation tissue. Note many foamy macrophages in the lumen (hematoxylin and eosin stain; original magnification, ⫻40). Luiz et al. Topical Corticosteroid for Oral Mucoceles. J Oral Maxillofac Surg 2008.
follow-up, she reported feeling well regarding her present clinical status. The flare-ups have been quite infrequent, and she rarely needs to use the medication. A clinical examination showed an oral mucosa free of any signs of lesions (Fig 2). Her serum cortisol level was checked 3 times during follow-up, and remained within normal values.
Discussion For single mucoceles, surgical excision is the principal treatment, and recurrence is rarely reported. The
treatment of recurrent, multiple mucoceles has mostly focused on control of the recurrences. For this purpose, some authors have used laser vaporization, corticosteroid injection, or gamma-linolenic acid, with recurrences reported in 2 of these studies.4,9,10 Considering both the recurrent nature of the mucoceles and the considerable pain that would ensue from surgery and corticosteroid injections, both these procedures were ruled out for the present case. The decision was then made to use a high-potency corticosteroid (clobetasol propionate) in a mucosal adhesive base, which proved to be very helpful in control-
FIGURE 2. A (patient’s right side) and B (patient’s left side), buccal mucosae free of lesions after 13 months of follow-up. Luiz et al. Topical Corticosteroid for Oral Mucoceles. J Oral Maxillofac Surg 2008.
1739
MENDONÇA-CARIDAD ET AL
ling the pain and recurrences. The efficacy of topical corticotherapy in controlling the recurrences is likely attributable to the high-potency vasoconstriction and anti-inflammatory properties of the clobetasol propionate, reducing the inflammatory process involved in the pathogenesis of the mucoceles.6,7,9 A previous study6 dealing with 3 cases of recurrent, superficial mucoceles in patients with graftversus-host disease (GVHD) did not report satisfactory results using either systemic (prednisone) or topical (clobetasol propionate and betamethasone) corticosteroids in combination with an immunosuppressive agent (cyclosporine mouthwash). This lack of benefit was likely because the coexistence of GVHD acted as a factor in producing the reappearances of mucoceles. Although it must be emphasized that the rarity of this condition has precluded any controlled study on which to base any definitive conclusions, the satisfactory result obtained in the present case suggests that a highpotency topical corticosteroid may be considered an option for the treatment of recurrent mucoceles with multiple-site involvement, especially in the absence of any immunologically active factor such as GVHD.
References 1. Bermejo A, Aguirre JM, Lopez P, et al: Superficial mucocele. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 88:469, 1999 2. Mandel L: Multiple superficial oral mucoceles: Case report. J Oral Maxillofac Surg 59:928, 2001 3. Porter SR, Scully C, Kainth B, et al: Multiple salivary mucoceles in a young boy. Int J Paediatr Dent 8:149, 1998 4. Mc Caul JA, Lamey PJ: Multiple oral mucoceles treated with gamma-linolenic acid: Report of a case. Br J Oral Maxillofac Surg 32:392, 1994 5. Tal H, Altini M, Lemmer J: Multiple mucous retention cysts of the oral mucosa. Oral Surg Oral Med Oral Pathol 58:692, 1984 6. Campana F, Sibaud V, Chauvel A, et al: Recurrent superficial mucoceles associated with lichenoid disorders. J Oral Maxillofac Surg 64:1830, 2006 7. García-Villalta MJ, Pascual-López M, Elices M, et al: Superficial mucoceles and lichenoid graft versus host disease: Report of three cases. Acta Derm Venereol (Stockh) 82:453, 2002 8. Navazesh M: Tartar-control toothpaste as a possible contributory factor in the onset of superficial mucoceles: A case report. Spec Care Dent 15:74, 1995 9. Jinbu Y, Tsukiuoki K, Kusama M, et al: Recurrent multiple superficial mucoceles on the palate: Histopathology and laser vaporization. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 95:193, 2003 10. Wilcox JW, Hickory JE: Nonsurgical resolution of mucoceles. J Oral Surg 36:478, 1978 J Oral Maxillofac Surg 66:1739-1743, 2008
Swordfish Bill Injury Involving the Pterygomaxillary Fossae: Surgical Management and Case Report Jose Joaquín Mendonça-Caridad, MD, DMD, PhD,* Pedro Juiz Lopez, MD,† Luis Francos, MD,‡ and Mercedes Rodriguez, MD§ Maxillofacial foreign body injuries are relatively common clinical events. The nature of the causal agent depends on the particular circumstances to which the individual has been exposed. Civilian and military injuries generally differ considerably and thus should be classified in separate groups. *Consultant Surgeon, Head and Neck Surgery Unit, Polusa Hospital, Lugo, Spain; former Clinical and Research Fellow, Department of Oral and Maxillofacial Surgery, UCLA School of Dentistry, Los Angeles, CA. †Consultant Surgeon, Head and Neck Surgery Unit, Polusa Hospital, Lugo, Spain. ‡Consulant Surgeon, Vascular Surgery Unit, Polusa Hospital, Lugo, Spain.
Civilian penetrating injuries are usually related to motor vehicle accidents, labor and domestic accidents, violence of all sorts, iatrogenic impaction of dental/surgical materials and instruments, and certain types of sports. The capacity of a given material to penetrate bone depends on its density, its sharpness, §Consultant Physician, Emergency Section, Ojos Grandes Hospital, Lugo, Spain. Address correspondence and reprint requests to Dr MendonçaCaridad: Cantón Pequeño, 9-12 Entlo, La Coruña 15003, Spain; e-mail: [email protected] © 2008 American Association of Oral and Maxillofacial Surgeons
0278-2391/08/6608-0028$34.00/0 doi:10.1016/j.joms.2007.12.016
LUIZ ET AL
J Oral Maxillofac Surg 66:1737-1739, 2008
Treatment of Painful and Recurrent Oral Mucoceles With a High-Potency Topical Corticosteroid: A Case Report Ana C. Luiz, DDS,* Karen R.N. Hiraki, DDS,† Celso A. Lemos, Jr, DDS, DSc,‡ Sílvio K. Hirota, DDS, MS,§ and Dante A. Migliari, DDS, DSc¶ Single mucoceles are a relatively common disorder of the oral mucosa, but the occurrence of multiple mucoceles is considered an uncommon event. There are only a few reports in the literature.1-6 Although no etiologic factor has been associated with multiple mucoceles, some authors reported on this condition in patients with oral lichen planus and in association with graft-versus-host disease.1,6,7 There is a hypothesis that an inflammatory process may play a role in the pathogenesis of recurrent mucoceles,6,7 although it was also argued that changes in minor salivary-gland function may contribute to the development of these lesions.2 We report on a case of multiple, painful, and recurrent mucoceles. Our main focus was on assessing the effectiveness of a topical high-potency corticosteroid for the treatment of this clinical condition.
Report of a Case A 33-year-old female was referred to our clinic for diagnosis of a recurrent “bullous” formation, accompanied by ulcerations, which had been present for about 1 month. The ulcerations had caused her severe pain and difficulty in chewing. Several translucent vesicles at multiple sites, dis-
Received from the School of Dentistry, University of São Paulo, São Paulo, Brazil. *Research Fellow, Department of Oral Diagnosis. †Doctoral Student, Department of Oral Pathology. ‡Assistant Professor, Department of Oral Diagnosis. §Research Fellow, Department of Oral Diagnosis. ¶Assistant Professor, Department of Oral Diagnosis. Address correspondence and reprint requests to Dr Migliari: Disciplina de Semiologia, Departamento de Estomatologia, Faculdade de Odontologia, Universidade de São Paulo, Av Prof Lineu Prestes 2227, Cidade Universitária, 05580-900 São Paulo, São Paulo, Brazil; e-mail: [email protected] © 2008 American Association of Oral and Maxillofacial Surgeons
0278-2391/08/6608-0027$34.00/0 doi:10.1016/j.joms.2008.01.050
tributed on the lower lip, buccal mucosa (bilaterally), and floor of the mouth, were observed in association with white striae and papules (Figs 1A-C). This clinical picture suggested either a combination of mucoceles with reticular oral lichen planus, or bullous lichen planus. Any other vesiculobullous diseases were clinically ruled out. Apart from her oral lesions, the patient was in good health. A biopsy was taken involving a vesicle together with a sample of the striae. Microscopic examination showed features only of a mucocele without any characteristics of lichen planus (Fig 1D). Thus, the final diagnosis was restricted to multiple mucoceles.
Management A high-potency topical corticosteroid (0.05% clobetasol propionate in Orabase; Colgate, New York, NY) was used mainly for controlling the symptomatology. The patient was instructed to apply the medication 3 times a day. After 4 weeks of treatment, she reported that the symptoms and frequency of recurrences had diminished. In addition, the white areas (striae and papules) had disappeared completely. Eventually, she was free of lesions and recurrences, and was taken off the medication. When the lesions recurred later, the possibility of an allergy-related mechanism for the mucoceles was investigated. The patient underwent skin-patch testing for standard (Brazilian) allergens and for dental materials (Trolab; Hermal, Reinbeck, Germany); the results were negative. She was also asked to avoid the use of toothpaste8 for a while and to refrain from potentially allergenic foods (eg, milk, eggs, citrus fruit, wheat, cinnamon, and seafood). Despite these measures, the mucoceles kept reappearing. Clobetasol propionate was restarted 3 times a day. As soon as improvement was observed, the medication was tapered. Three weeks later, she was again free of lesions, and was then instructed to use the medication only when she noticed initial symptoms of the lesions. For a time, she used the medication twice a week (on average) to control some flareups. At her latest appointment, after 13 months of
1738
TOPICAL CORTICOSTEROID FOR ORAL MUCOCELES
FIGURE 1. A, Cluster of superficial mucoceles on the labial mucosa. B, Single mucocele surrounded by white patches on the buccal mucosa. C, Ulcerations resulting from ruptured mucoceles, along with white striae and papules suggesting oral lichen planus, seen on the left side of the buccal mucosa. A similar pattern was seen on the right side (not shown). D, A pool of mucin surrounded by granulation tissue. Note many foamy macrophages in the lumen (hematoxylin and eosin stain; original magnification, ⫻40). Luiz et al. Topical Corticosteroid for Oral Mucoceles. J Oral Maxillofac Surg 2008.
follow-up, she reported feeling well regarding her present clinical status. The flare-ups have been quite infrequent, and she rarely needs to use the medication. A clinical examination showed an oral mucosa free of any signs of lesions (Fig 2). Her serum cortisol level was checked 3 times during follow-up, and remained within normal values.
Discussion For single mucoceles, surgical excision is the principal treatment, and recurrence is rarely reported. The
treatment of recurrent, multiple mucoceles has mostly focused on control of the recurrences. For this purpose, some authors have used laser vaporization, corticosteroid injection, or gamma-linolenic acid, with recurrences reported in 2 of these studies.4,9,10 Considering both the recurrent nature of the mucoceles and the considerable pain that would ensue from surgery and corticosteroid injections, both these procedures were ruled out for the present case. The decision was then made to use a high-potency corticosteroid (clobetasol propionate) in a mucosal adhesive base, which proved to be very helpful in control-
FIGURE 2. A (patient’s right side) and B (patient’s left side), buccal mucosae free of lesions after 13 months of follow-up. Luiz et al. Topical Corticosteroid for Oral Mucoceles. J Oral Maxillofac Surg 2008.
1739
MENDONÇA-CARIDAD ET AL
ling the pain and recurrences. The efficacy of topical corticotherapy in controlling the recurrences is likely attributable to the high-potency vasoconstriction and anti-inflammatory properties of the clobetasol propionate, reducing the inflammatory process involved in the pathogenesis of the mucoceles.6,7,9 A previous study6 dealing with 3 cases of recurrent, superficial mucoceles in patients with graftversus-host disease (GVHD) did not report satisfactory results using either systemic (prednisone) or topical (clobetasol propionate and betamethasone) corticosteroids in combination with an immunosuppressive agent (cyclosporine mouthwash). This lack of benefit was likely because the coexistence of GVHD acted as a factor in producing the reappearances of mucoceles. Although it must be emphasized that the rarity of this condition has precluded any controlled study on which to base any definitive conclusions, the satisfactory result obtained in the present case suggests that a highpotency topical corticosteroid may be considered an option for the treatment of recurrent mucoceles with multiple-site involvement, especially in the absence of any immunologically active factor such as GVHD.
References 1. Bermejo A, Aguirre JM, Lopez P, et al: Superficial mucocele. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 88:469, 1999 2. Mandel L: Multiple superficial oral mucoceles: Case report. J Oral Maxillofac Surg 59:928, 2001 3. Porter SR, Scully C, Kainth B, et al: Multiple salivary mucoceles in a young boy. Int J Paediatr Dent 8:149, 1998 4. Mc Caul JA, Lamey PJ: Multiple oral mucoceles treated with gamma-linolenic acid: Report of a case. Br J Oral Maxillofac Surg 32:392, 1994 5. Tal H, Altini M, Lemmer J: Multiple mucous retention cysts of the oral mucosa. Oral Surg Oral Med Oral Pathol 58:692, 1984 6. Campana F, Sibaud V, Chauvel A, et al: Recurrent superficial mucoceles associated with lichenoid disorders. J Oral Maxillofac Surg 64:1830, 2006 7. García-Villalta MJ, Pascual-López M, Elices M, et al: Superficial mucoceles and lichenoid graft versus host disease: Report of three cases. Acta Derm Venereol (Stockh) 82:453, 2002 8. Navazesh M: Tartar-control toothpaste as a possible contributory factor in the onset of superficial mucoceles: A case report. Spec Care Dent 15:74, 1995 9. Jinbu Y, Tsukiuoki K, Kusama M, et al: Recurrent multiple superficial mucoceles on the palate: Histopathology and laser vaporization. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 95:193, 2003 10. Wilcox JW, Hickory JE: Nonsurgical resolution of mucoceles. J Oral Surg 36:478, 1978 J Oral Maxillofac Surg 66:1739-1743, 2008
Swordfish Bill Injury Involving the Pterygomaxillary Fossae: Surgical Management and Case Report Jose Joaquín Mendonça-Caridad, MD, DMD, PhD,* Pedro Juiz Lopez, MD,† Luis Francos, MD,‡ and Mercedes Rodriguez, MD§ Maxillofacial foreign body injuries are relatively common clinical events. The nature of the causal agent depends on the particular circumstances to which the individual has been exposed. Civilian and military injuries generally differ considerably and thus should be classified in separate groups. *Consultant Surgeon, Head and Neck Surgery Unit, Polusa Hospital, Lugo, Spain; former Clinical and Research Fellow, Department of Oral and Maxillofacial Surgery, UCLA School of Dentistry, Los Angeles, CA. †Consultant Surgeon, Head and Neck Surgery Unit, Polusa Hospital, Lugo, Spain. ‡Consulant Surgeon, Vascular Surgery Unit, Polusa Hospital, Lugo, Spain.
Civilian penetrating injuries are usually related to motor vehicle accidents, labor and domestic accidents, violence of all sorts, iatrogenic impaction of dental/surgical materials and instruments, and certain types of sports. The capacity of a given material to penetrate bone depends on its density, its sharpness, §Consultant Physician, Emergency Section, Ojos Grandes Hospital, Lugo, Spain. Address correspondence and reprint requests to Dr MendonçaCaridad: Cantón Pequeño, 9-12 Entlo, La Coruña 15003, Spain; e-mail: [email protected] © 2008 American Association of Oral and Maxillofacial Surgeons
0278-2391/08/6608-0028$34.00/0 doi:10.1016/j.joms.2007.12.016