- Email: [email protected]
Clinicopathologic conference: bluish gingival nodule
Vol. 120 No. 5 November 2015
Clinicopathologic conference: bluish gingival nodule Michelle Agostini, DDS, PhD, Aline Corrêa Abrahão, DDS, PhD, Márcia Grillo Cabral, DDS, PhD, Bruno Augusto Benevenuto de Andrade, DDS, PhD, and Mário José Romañach, DDS, PhD (Oral Surg Oral Med Oral Pathol Oral Radiol 2015;120:544-547)
CLINICAL PRESENTATION A 35-year-old woman was referred for management of a slow-growing nodule of 6 months’ duration in the maxillary gingiva. The medical history and extraoral examination of the patient were unremarkable, with no history of trauma in the region. Intraoral examination revealed a dome-shaped, well-defined bluish nodule measuring 1 cm, localized in the buccal aspect of the attached gingiva between the upper right canine and first premolar. The nodule was asymptomatic, soft in consistency, and covered by a smooth surface displaying discrete capillaries. The gingival papilla was preserved, and there was no sign of dental plaque-induced gingival inflammation. Adjacent teeth did not show evidence of calculus or carious lesions. Periapical radiography revealed discrete, well-defined, superficial bone resorption of the alveolar crest, indicating the extraosseous location of the lesion (Figure 1). DIFFERENTIAL DIAGNOSIS The differential diagnosis in the current case included peripheral bluish gingival lesions, such as peripheral giant cell lesion, gingival cyst of the adult, melanocytic nevus, angioleiomyoma, Kaposi sarcoma, and melanoma. Peripheral giant cell lesion is a reactive lesion that occurs in the gingiva or alveolar ridge of adult patients and may present a bluish to reddish color and smooth surface,1 as in the present case. However, in some cases, the lesions occur as lobulated nodules and may show superficial ulceration, and most are associated with local irritants such as dental plaque or trauma,2 features not seen in this case. Furthermore, most cases involve the marginal gingiva of the attached gingival mucosa, a clinical feature that can distinguish both lesions, which, in the present case, was absent.1,2 Gingival cyst of the adult is an uncommon odontogenic cyst that frequently appears as a small, circumscribed, bluish nodule in the buccal attached gingiva of the premolar region of adults.3 No radiographic features are expected, but some occasional cases may exhibit mild superficial regular bony erosion,3 as in the current case. Department of Oral Diagnosis and Pathology, Federal University of Rio de Janeiro School of Dentistry (UFRJ), Rio de Janeiro, Brazil. Received for publication Jan 27, 2015; returned for revision Apr 9, 2015; accepted for publication Jun 16, 2015. Ó 2015 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter http://dx.doi.org/10.1016/j.oooo.2015.06.032
544
Oral melanocytic nevus is a benign proliferation of nevus cells of the oral mucosa.4 As in the present case, oral melanocytic nevus usually appears as a single macule or nodule with color variations from brown to blue, bluish-gray, or black, measuring less than 0.6 cm and affecting patients with a mean age of 35 years.4 However, the most common sites affected by melanocytic nevus are the hard palate and buccal mucosa; the commitment of the gingiva is found in about 12% of cases.4,5 Angioleiomyomas are benign smooth muscle tumors with a distinct vascular component that might reflect a pigmented clinical appearance.6 They are considered uncommon in the oral cavity and usually affect the lips and palate of middle-aged individuals.7 The main clinical appearance is of a bluish painless nodule measuring less than 2 cm, being rarely observed in the gingiva, as in this case.6,7 Kaposi sarcoma (KS) is an angioproliferative disorder caused by human herpesvirus 8, usually related to human immunodeficiency virus infection.8,9 KS of the oral cavity is characterized by the appearance of purplish, reddish blue, or dark brown macules, plaques, or nodules mainly localized in the hard palate and gingiva.9 Uncommon examples of oral KS might affect the gingiva as a well-delimited, small, reddish nodule, but a regular smooth surface is usually not expected.9 Melanoma is usually composed by malignant melanocytes of the skin, but it may also arise from different mucosal surfaces, including the oral cavity.10 Oral melanoma commonly appears as a dark bluish or black macule or nodule affecting the palate and gingiva of adult patients, as also observed in this case.11 However, oral melanoma tends to become progressively irregular in shape, exhibiting ulcerated surface, irregular borders, variable coloration, and fast growth,10,11 features not observed in the present case. In summary, in the current case, that of a 35-year-old female patient with a slow-growing bluish nodule in the posterior maxillary gingiva, our favored clinical diagnosis was of gingival cyst of the adult.
MANAGEMENT AND DIAGNOSIS The pigmented clinical appearance of the lesion was unchanged after diascopy, which made the hypothesis of a vascular lesion less likely. The patient underwent an excisional biopsy under local anesthesia; during the surgical procedure, clear fluid and a whitish semi-solid
OOOO Volume 120, Number 5
CLINICOPATHOLOGIC CONFERENCE Agostini et al. 545
Fig. 1. A, Clinical appearance of a well-delimited bluish nodule localized in the attached gingiva between the maxillary right canine and first premolar, covered by a smooth surface, which exhibited delicate and small superficial blood vessels. B, Periapical radiography depicting superficial resorption of the alveolar bone crest produced by a peripheral lesion between the premolar and canine.
material drained out from the lesion, with the lesion decreasing in size immediately. Microscopic evaluation of the gingival connective tissue revealed proliferation of mature sebaceous glands of different sizes surrounding a central cystic formation lined by parakeratinized stratified squamous epithelium. The sebaceous acini were of varying sizes, some of them located within the cystic lining. Interestingly, a minor duct-like structure covered by double-layered cuboidal epithelium with areas of apocrine metaplasia was observed adjacent to the sebaceous structure. In addition, a structure consistent with an excretory duct was present in the epithelium of the oral mucosa (Figure 2). The final diagnosis was gingival sebaceous choristoma. There was no sign of recurrence 18 months after surgical removal.
DISCUSSION Sebaceous glands are adnexal structures of the skin that are commonly identified as clusters of small, yellowish papules (Fordyce granules) in the buccal mucosa and the vermilion border of the lip of most healthy adults.12 Although considered a normal variation, the presence of sebaceous glands has also been rarely described under different terms in oral sites, such as the retromolar region,13 tongue,14 hard and soft palates,15 and gingiva.16,17 The occurrence of sebaceous glands in human gingiva is exceedingly rare, and most of these cases exhibit a clinical appearance compatible with Fordyce granules.16-18 When it produces a nodular lesion, as in
the present case, the preferred term is sebaceous choristoma, which is defined as a tumor-like hyperplasia of mature sebaceous glands of different sizes in a location where they are not usually seen.18 There are few reports of oral sebaceous choristoma in the English language literature, and most of the lesions were located in the posterior midline region of the tongue of adult patients.19,20 To the best of our knowledge, this is the first report of a gingival sebaceous choristoma with the clinical appearance of a bluish nodule. The development of oral sebaceous choristoma is attributed to the entrapment of ectodermal structures during embryogenesis of the oral structures, but the exact mechanism of sebaceous gland proliferation in the gingiva is uncertain.18 Clinically, it is usually characterized by an asymptomatic dome-shaped swelling, covered by a smooth surface and measuring less than 2 cm.18 The color usually varies from yellowish to bluish, and an interesting clinical finding of white fluid discharge from the lesion has been described in some cases,18 as also observed in the present case. Microscopically, sebaceous choristomas are predominantly composed of numerous branching lobules of well-differentiated sebaceous glands disposed around a main excretory duct lined by parakeratinized stratified squamous epithelium and connected to the surface epithelium.18 Additional features include lymphoid tissue and duct-like structures resembling the ducts of apocrine sweat glands,18 the latter being also observed in the current case adjacent to the main excretory duct, which was directly connected to the superficial oral epithelium.
CLINICOPATHOLOGIC CONFERENCE 546 Agostini et al.
OOOO November 2015
Fig. 2. A, Proliferation of mature sebaceous glands of different sizes in the connective tissue surrounding a central cystic formation lined by parakeratinized stratified squamous epithelium (H&E, 40). B, A minor duct-like structure covered by cuboidal cells was observed adjacent to the sebaceous structure (H&E, 40). C, Microscopic features of the sectioned specimen exhibiting mature sebaceous glands of varying sizes surrounding a dilated central excretory duct (H&E, 200). D, A duct-like structure with apocrine metaplasia is present just below the superficial epithelium (H&E, 200). A high-resolution version of this slide for use with the Virtual Microscope is available as eSlide: VM00733.
The microscopic features of dominant sebaceous glands with cystic dilation of the main excretory duct and the duct-like structure with apocrine metaplasia represented a diagnostic challenge and led us to consider other sebaceous lesions with cystic components, such as dermoid cyst, steatocystoma simplex, and folliculosebaceous cystic hamartoma. Dermoid cyst is usually a subcutaneous lesion that is present from birth, occurring in the head and neck region. The epithelial lining of dermoid cysts is almost completely covered by an orthokeratinized layer and shows more than one cutaneous adnexal structure, such as eccrine and apocrine sweat glands and hair follicles.21 The present case exhibited a parakeratinized lining and the presence of only sebaceous glands after evaluation of serial cuts of the lesion. Steatocystoma simplex is a solitary cystic lesion, mainly observed in the skin of the face but rarely reported in the oral cavity, and is microscopically characterized by a thin layer of stratified squamous epithelium and usually presenting a superficial acellular hyalinized cuticle with an undulant architecture,22 features not identified in the present case. Folliculosebaceous cystic hamartoma is a rare lesion of the skin that consists of many dilated
folliculosebaceous structures intermingled with mature adipocytes,23 which were absent in the present case. Indeed, some authors have reported infestation of the pilosebaceous parasite Demodex folliculorum within oral sebaceous glands.24 We did not identify evidence of parasite infection within sebaceous cells after evaluation of different sections of the lesion. The clinical aspect of a bluish-colored nodule is occasionally seen in sebaceous-predominant lesions and has not been described in the gingiva so far.12,14,16,18 The presence of fluid within the duct-like structure with apocrine metaplasia, cystic dilation, and sebaceous glands is likely to be the main reason for this clinical appearance, which might be justified by a phenomenon analogous to the Tyndall effect, also observed in different oral lesions with considerable cystic or pigmented components.
CONCLUSIONS We presented here a rare case of the ectopic presence of hyperplastic sebaceous glands in the maxillary gingiva, containing cystic ductal dilation and adjacent duct-like structures with apocrine metaplasia. Oral pathologists
OOOO Volume 120, Number 5
and clinicians should consider sebaceous choristoma when evaluating bluish-colored nodules of the gingiva. REFERENCES 1. Lester SR, Cordell KG, Rosebush MS, Palaiologou AA, Maney P. Peripheral giant cell granulomas: a series of 279 cases. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118:475-482. 2. Katsikeris N, Kakarantza-Angelopoulou E, Angelopoulos AP. Peripheral giant cell granuloma. Clinicopathologic study of 224 new cases and review of 956 reported cases. Int J Oral Maxillofac Surg. 1988;17:94-99. 3. Buchner A, Hansen LS. The histomorphologic spectrum of the gingival cyst in the adult. Oral Surg Oral Med Oral Pathol. 1979;48:532-539. 4. Buchner A, Hansen LS. Pigmented nevi of the oral mucosa: a clinicopathologic study of 36 new cases and review of 155 cases from the literature. Part II: analysis of 191 cases. Oral Surg Oral Med Oral Pathol. 1987;63:676-682. 5. Meleti M, Mooi WJ, Casparie MK, van der Waal I. Melanocytic nevi of the oral mucosadno evidence of increased risk for oral malignant melanoma: an analysis of 119 cases. Oral Oncol. 2007;43:976-981. 6. Brooks JK, Nikitakis NG, Goodman NJ, Levy BA. Clinicopathologic characterization of oral angioleiomyomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002;94:221-227. 7. Gueiros LA, Romañach MJ, Pires-Soubhia AM, Pires FR, Paes-de-Almeida O, Vargas PA. Angioleiomyoma affecting the lips: report of 3 cases and review of the literature. Med Oral Patol Oral Cir Bucal. 2011;16:e482-e487. 8. Rockafellow A, Ko E, Philipone E. Asymptomatic gingival lesion occurring in an 83-year-old man. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118:5-8. 9. Lager I, Altini M, Coleman H, Ali H. Oral Kaposi’s sarcoma: a clinicopathologic study from South Africa. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;96:701-710. 10. de-Andrade BA, Toral-Rizo VH, León JE, et al. Primary oral melanoma: a histopathological and immunohistochemical study of 22 cases of Latin America. Med Oral Patol Oral Cir Bucal. 2012;17:e383-e388. 11. Sortino-Rachou AM, Cancela M de C, Voti L, Curado MP. Primary oral melanoma: population-based incidence. Oral Oncol. 2009;45:254-258. 12. Daley T. Pathology of intraoral sebaceous glands. J Oral Pathol Med. 1993;22:241-245.
CLINICOPATHOLOGIC CONFERENCE Agostini et al. 547 13. Koutlas IG, Yaholnitsky B. Oral sebaceous retention phenomenon. J Periodontol. 1994;65:186-188. 14. Kovero O. A sebaceous gland in the dorsal surface of the tongue. Int J Oral Maxillofac Surg. 1989;18:266. 15. Daley TD. Intraoral sebaceous hyperplasia. Diagnostic criteria. Oral Surg Oral Med Oral Pathol. 1993;75:343-347. 16. Moskow BS, Baden E. Gingival choristoma. Report of a case. Oral Surg Oral Med Oral Pathol. 1964;18:504-516. 17. Cohen RE, Aguirre A, Drinnan AJ, Neiders ME, Mastroianni AJ. Ectopic gingival sebaceous glands presenting as localized periodontitis. J Periodontol. 1990;61:58-60. 18. Chou LS, Hansen LS, Daniels TE. Choristomas of the oral cavity: a review. Oral Surg Oral Med Oral Pathol. 1991;72:584-593. 19. Leider AS, Lucas JW, Eversole LR. Sebaceous choristoma of the thyroglossal duct. Oral Surg Oral Med Oral Pathol. 1977;44: 261-266. 20. Azorin D, Enriquez de Salamanca J, de Prada I, Colmenero I, Gonzalez Mediero I. Congenital melanotic macules and sebaceous choristoma arising on the tongue of a newborn: epidermal choristoma? J Cutan Pathol. 2005;32:251-253. 21. King RC, Smith BR, Burk JL. Dermoid cyst in the floor of the mouth. Review of the literature and case reports. Oral Surg Oral Med Oral Pathol. 1994;78:567-576. 22. Olsen DB, Mostofi RS, Lagrotteria LB. Steatocystoma simplex in the oral cavity: a previously undescribed condition. Oral Surg Oral Med Oral Pathol. 1988;66:605-607. 23. Ansai S, Kimura T, Kawana S. A clinicopathologic study of folliculosebaceous cystic hamartoma. Am J Dermatopathol. 2010;32:815-820. 24. Franklin CD, Underwood JC. Demodex infestation of oral mucosal sebaceous glands. Oral Surg Oral Med Oral Pathol. 1986;61:80-82.
Reprint requests: Mário José Romañach, DDS, PhD Department Oral Diagnosis and Pathology Federal University of Rio de Janeiro School of Dentistry. Av. Carlos Chagas Filho 373. Prédio do CCS Bloco K, 2 andar Sala 56. Ilha da Cidade Universitária Rio de Janeiro-RJ Brasil [email protected]
Clinicopathologic conference: bluish gingival nodule Michelle Agostini, DDS, PhD, Aline Corrêa Abrahão, DDS, PhD, Márcia Grillo Cabral, DDS, PhD, Bruno Augusto Benevenuto de Andrade, DDS, PhD, and Mário José Romañach, DDS, PhD (Oral Surg Oral Med Oral Pathol Oral Radiol 2015;120:544-547)
CLINICAL PRESENTATION A 35-year-old woman was referred for management of a slow-growing nodule of 6 months’ duration in the maxillary gingiva. The medical history and extraoral examination of the patient were unremarkable, with no history of trauma in the region. Intraoral examination revealed a dome-shaped, well-defined bluish nodule measuring 1 cm, localized in the buccal aspect of the attached gingiva between the upper right canine and first premolar. The nodule was asymptomatic, soft in consistency, and covered by a smooth surface displaying discrete capillaries. The gingival papilla was preserved, and there was no sign of dental plaque-induced gingival inflammation. Adjacent teeth did not show evidence of calculus or carious lesions. Periapical radiography revealed discrete, well-defined, superficial bone resorption of the alveolar crest, indicating the extraosseous location of the lesion (Figure 1). DIFFERENTIAL DIAGNOSIS The differential diagnosis in the current case included peripheral bluish gingival lesions, such as peripheral giant cell lesion, gingival cyst of the adult, melanocytic nevus, angioleiomyoma, Kaposi sarcoma, and melanoma. Peripheral giant cell lesion is a reactive lesion that occurs in the gingiva or alveolar ridge of adult patients and may present a bluish to reddish color and smooth surface,1 as in the present case. However, in some cases, the lesions occur as lobulated nodules and may show superficial ulceration, and most are associated with local irritants such as dental plaque or trauma,2 features not seen in this case. Furthermore, most cases involve the marginal gingiva of the attached gingival mucosa, a clinical feature that can distinguish both lesions, which, in the present case, was absent.1,2 Gingival cyst of the adult is an uncommon odontogenic cyst that frequently appears as a small, circumscribed, bluish nodule in the buccal attached gingiva of the premolar region of adults.3 No radiographic features are expected, but some occasional cases may exhibit mild superficial regular bony erosion,3 as in the current case. Department of Oral Diagnosis and Pathology, Federal University of Rio de Janeiro School of Dentistry (UFRJ), Rio de Janeiro, Brazil. Received for publication Jan 27, 2015; returned for revision Apr 9, 2015; accepted for publication Jun 16, 2015. Ó 2015 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter http://dx.doi.org/10.1016/j.oooo.2015.06.032
544
Oral melanocytic nevus is a benign proliferation of nevus cells of the oral mucosa.4 As in the present case, oral melanocytic nevus usually appears as a single macule or nodule with color variations from brown to blue, bluish-gray, or black, measuring less than 0.6 cm and affecting patients with a mean age of 35 years.4 However, the most common sites affected by melanocytic nevus are the hard palate and buccal mucosa; the commitment of the gingiva is found in about 12% of cases.4,5 Angioleiomyomas are benign smooth muscle tumors with a distinct vascular component that might reflect a pigmented clinical appearance.6 They are considered uncommon in the oral cavity and usually affect the lips and palate of middle-aged individuals.7 The main clinical appearance is of a bluish painless nodule measuring less than 2 cm, being rarely observed in the gingiva, as in this case.6,7 Kaposi sarcoma (KS) is an angioproliferative disorder caused by human herpesvirus 8, usually related to human immunodeficiency virus infection.8,9 KS of the oral cavity is characterized by the appearance of purplish, reddish blue, or dark brown macules, plaques, or nodules mainly localized in the hard palate and gingiva.9 Uncommon examples of oral KS might affect the gingiva as a well-delimited, small, reddish nodule, but a regular smooth surface is usually not expected.9 Melanoma is usually composed by malignant melanocytes of the skin, but it may also arise from different mucosal surfaces, including the oral cavity.10 Oral melanoma commonly appears as a dark bluish or black macule or nodule affecting the palate and gingiva of adult patients, as also observed in this case.11 However, oral melanoma tends to become progressively irregular in shape, exhibiting ulcerated surface, irregular borders, variable coloration, and fast growth,10,11 features not observed in the present case. In summary, in the current case, that of a 35-year-old female patient with a slow-growing bluish nodule in the posterior maxillary gingiva, our favored clinical diagnosis was of gingival cyst of the adult.
MANAGEMENT AND DIAGNOSIS The pigmented clinical appearance of the lesion was unchanged after diascopy, which made the hypothesis of a vascular lesion less likely. The patient underwent an excisional biopsy under local anesthesia; during the surgical procedure, clear fluid and a whitish semi-solid
OOOO Volume 120, Number 5
CLINICOPATHOLOGIC CONFERENCE Agostini et al. 545
Fig. 1. A, Clinical appearance of a well-delimited bluish nodule localized in the attached gingiva between the maxillary right canine and first premolar, covered by a smooth surface, which exhibited delicate and small superficial blood vessels. B, Periapical radiography depicting superficial resorption of the alveolar bone crest produced by a peripheral lesion between the premolar and canine.
material drained out from the lesion, with the lesion decreasing in size immediately. Microscopic evaluation of the gingival connective tissue revealed proliferation of mature sebaceous glands of different sizes surrounding a central cystic formation lined by parakeratinized stratified squamous epithelium. The sebaceous acini were of varying sizes, some of them located within the cystic lining. Interestingly, a minor duct-like structure covered by double-layered cuboidal epithelium with areas of apocrine metaplasia was observed adjacent to the sebaceous structure. In addition, a structure consistent with an excretory duct was present in the epithelium of the oral mucosa (Figure 2). The final diagnosis was gingival sebaceous choristoma. There was no sign of recurrence 18 months after surgical removal.
DISCUSSION Sebaceous glands are adnexal structures of the skin that are commonly identified as clusters of small, yellowish papules (Fordyce granules) in the buccal mucosa and the vermilion border of the lip of most healthy adults.12 Although considered a normal variation, the presence of sebaceous glands has also been rarely described under different terms in oral sites, such as the retromolar region,13 tongue,14 hard and soft palates,15 and gingiva.16,17 The occurrence of sebaceous glands in human gingiva is exceedingly rare, and most of these cases exhibit a clinical appearance compatible with Fordyce granules.16-18 When it produces a nodular lesion, as in
the present case, the preferred term is sebaceous choristoma, which is defined as a tumor-like hyperplasia of mature sebaceous glands of different sizes in a location where they are not usually seen.18 There are few reports of oral sebaceous choristoma in the English language literature, and most of the lesions were located in the posterior midline region of the tongue of adult patients.19,20 To the best of our knowledge, this is the first report of a gingival sebaceous choristoma with the clinical appearance of a bluish nodule. The development of oral sebaceous choristoma is attributed to the entrapment of ectodermal structures during embryogenesis of the oral structures, but the exact mechanism of sebaceous gland proliferation in the gingiva is uncertain.18 Clinically, it is usually characterized by an asymptomatic dome-shaped swelling, covered by a smooth surface and measuring less than 2 cm.18 The color usually varies from yellowish to bluish, and an interesting clinical finding of white fluid discharge from the lesion has been described in some cases,18 as also observed in the present case. Microscopically, sebaceous choristomas are predominantly composed of numerous branching lobules of well-differentiated sebaceous glands disposed around a main excretory duct lined by parakeratinized stratified squamous epithelium and connected to the surface epithelium.18 Additional features include lymphoid tissue and duct-like structures resembling the ducts of apocrine sweat glands,18 the latter being also observed in the current case adjacent to the main excretory duct, which was directly connected to the superficial oral epithelium.
CLINICOPATHOLOGIC CONFERENCE 546 Agostini et al.
OOOO November 2015
Fig. 2. A, Proliferation of mature sebaceous glands of different sizes in the connective tissue surrounding a central cystic formation lined by parakeratinized stratified squamous epithelium (H&E, 40). B, A minor duct-like structure covered by cuboidal cells was observed adjacent to the sebaceous structure (H&E, 40). C, Microscopic features of the sectioned specimen exhibiting mature sebaceous glands of varying sizes surrounding a dilated central excretory duct (H&E, 200). D, A duct-like structure with apocrine metaplasia is present just below the superficial epithelium (H&E, 200). A high-resolution version of this slide for use with the Virtual Microscope is available as eSlide: VM00733.
The microscopic features of dominant sebaceous glands with cystic dilation of the main excretory duct and the duct-like structure with apocrine metaplasia represented a diagnostic challenge and led us to consider other sebaceous lesions with cystic components, such as dermoid cyst, steatocystoma simplex, and folliculosebaceous cystic hamartoma. Dermoid cyst is usually a subcutaneous lesion that is present from birth, occurring in the head and neck region. The epithelial lining of dermoid cysts is almost completely covered by an orthokeratinized layer and shows more than one cutaneous adnexal structure, such as eccrine and apocrine sweat glands and hair follicles.21 The present case exhibited a parakeratinized lining and the presence of only sebaceous glands after evaluation of serial cuts of the lesion. Steatocystoma simplex is a solitary cystic lesion, mainly observed in the skin of the face but rarely reported in the oral cavity, and is microscopically characterized by a thin layer of stratified squamous epithelium and usually presenting a superficial acellular hyalinized cuticle with an undulant architecture,22 features not identified in the present case. Folliculosebaceous cystic hamartoma is a rare lesion of the skin that consists of many dilated
folliculosebaceous structures intermingled with mature adipocytes,23 which were absent in the present case. Indeed, some authors have reported infestation of the pilosebaceous parasite Demodex folliculorum within oral sebaceous glands.24 We did not identify evidence of parasite infection within sebaceous cells after evaluation of different sections of the lesion. The clinical aspect of a bluish-colored nodule is occasionally seen in sebaceous-predominant lesions and has not been described in the gingiva so far.12,14,16,18 The presence of fluid within the duct-like structure with apocrine metaplasia, cystic dilation, and sebaceous glands is likely to be the main reason for this clinical appearance, which might be justified by a phenomenon analogous to the Tyndall effect, also observed in different oral lesions with considerable cystic or pigmented components.
CONCLUSIONS We presented here a rare case of the ectopic presence of hyperplastic sebaceous glands in the maxillary gingiva, containing cystic ductal dilation and adjacent duct-like structures with apocrine metaplasia. Oral pathologists
OOOO Volume 120, Number 5
and clinicians should consider sebaceous choristoma when evaluating bluish-colored nodules of the gingiva. REFERENCES 1. Lester SR, Cordell KG, Rosebush MS, Palaiologou AA, Maney P. Peripheral giant cell granulomas: a series of 279 cases. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118:475-482. 2. Katsikeris N, Kakarantza-Angelopoulou E, Angelopoulos AP. Peripheral giant cell granuloma. Clinicopathologic study of 224 new cases and review of 956 reported cases. Int J Oral Maxillofac Surg. 1988;17:94-99. 3. Buchner A, Hansen LS. The histomorphologic spectrum of the gingival cyst in the adult. Oral Surg Oral Med Oral Pathol. 1979;48:532-539. 4. Buchner A, Hansen LS. Pigmented nevi of the oral mucosa: a clinicopathologic study of 36 new cases and review of 155 cases from the literature. Part II: analysis of 191 cases. Oral Surg Oral Med Oral Pathol. 1987;63:676-682. 5. Meleti M, Mooi WJ, Casparie MK, van der Waal I. Melanocytic nevi of the oral mucosadno evidence of increased risk for oral malignant melanoma: an analysis of 119 cases. Oral Oncol. 2007;43:976-981. 6. Brooks JK, Nikitakis NG, Goodman NJ, Levy BA. Clinicopathologic characterization of oral angioleiomyomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002;94:221-227. 7. Gueiros LA, Romañach MJ, Pires-Soubhia AM, Pires FR, Paes-de-Almeida O, Vargas PA. Angioleiomyoma affecting the lips: report of 3 cases and review of the literature. Med Oral Patol Oral Cir Bucal. 2011;16:e482-e487. 8. Rockafellow A, Ko E, Philipone E. Asymptomatic gingival lesion occurring in an 83-year-old man. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118:5-8. 9. Lager I, Altini M, Coleman H, Ali H. Oral Kaposi’s sarcoma: a clinicopathologic study from South Africa. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;96:701-710. 10. de-Andrade BA, Toral-Rizo VH, León JE, et al. Primary oral melanoma: a histopathological and immunohistochemical study of 22 cases of Latin America. Med Oral Patol Oral Cir Bucal. 2012;17:e383-e388. 11. Sortino-Rachou AM, Cancela M de C, Voti L, Curado MP. Primary oral melanoma: population-based incidence. Oral Oncol. 2009;45:254-258. 12. Daley T. Pathology of intraoral sebaceous glands. J Oral Pathol Med. 1993;22:241-245.
CLINICOPATHOLOGIC CONFERENCE Agostini et al. 547 13. Koutlas IG, Yaholnitsky B. Oral sebaceous retention phenomenon. J Periodontol. 1994;65:186-188. 14. Kovero O. A sebaceous gland in the dorsal surface of the tongue. Int J Oral Maxillofac Surg. 1989;18:266. 15. Daley TD. Intraoral sebaceous hyperplasia. Diagnostic criteria. Oral Surg Oral Med Oral Pathol. 1993;75:343-347. 16. Moskow BS, Baden E. Gingival choristoma. Report of a case. Oral Surg Oral Med Oral Pathol. 1964;18:504-516. 17. Cohen RE, Aguirre A, Drinnan AJ, Neiders ME, Mastroianni AJ. Ectopic gingival sebaceous glands presenting as localized periodontitis. J Periodontol. 1990;61:58-60. 18. Chou LS, Hansen LS, Daniels TE. Choristomas of the oral cavity: a review. Oral Surg Oral Med Oral Pathol. 1991;72:584-593. 19. Leider AS, Lucas JW, Eversole LR. Sebaceous choristoma of the thyroglossal duct. Oral Surg Oral Med Oral Pathol. 1977;44: 261-266. 20. Azorin D, Enriquez de Salamanca J, de Prada I, Colmenero I, Gonzalez Mediero I. Congenital melanotic macules and sebaceous choristoma arising on the tongue of a newborn: epidermal choristoma? J Cutan Pathol. 2005;32:251-253. 21. King RC, Smith BR, Burk JL. Dermoid cyst in the floor of the mouth. Review of the literature and case reports. Oral Surg Oral Med Oral Pathol. 1994;78:567-576. 22. Olsen DB, Mostofi RS, Lagrotteria LB. Steatocystoma simplex in the oral cavity: a previously undescribed condition. Oral Surg Oral Med Oral Pathol. 1988;66:605-607. 23. Ansai S, Kimura T, Kawana S. A clinicopathologic study of folliculosebaceous cystic hamartoma. Am J Dermatopathol. 2010;32:815-820. 24. Franklin CD, Underwood JC. Demodex infestation of oral mucosal sebaceous glands. Oral Surg Oral Med Oral Pathol. 1986;61:80-82.
Reprint requests: Mário José Romañach, DDS, PhD Department Oral Diagnosis and Pathology Federal University of Rio de Janeiro School of Dentistry. Av. Carlos Chagas Filho 373. Prédio do CCS Bloco K, 2 andar Sala 56. Ilha da Cidade Universitária Rio de Janeiro-RJ Brasil [email protected]