Intraoral osseous and cartilaginous choristomas: Report of three cases and review of the literature Mary Jane Tohill, D.D.S.,* James G. Green, D.D.S.,** and Donald M. Cohen, D.M.D., M.S.D.,*** Omaha and Lincoln, Neb. UNIVERSITY
OF NEBRASKA
MEDICAI.
CENTER
COLLEGE
OF DENTISTRY
Intraoral osseous and cartilaginous choristomas are rare lesions, with only 72 cases previously reported the literature. Three additional cases are presented, and the literature is reviewed. One case represents the first reported osseous choristoma on the retromolar pad. (ORAL SURG. ORAL MED. ORAL PATHOL. 1987;63:506-IO)
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sseousand cartilaginous lesions of the intraoral soft tissues are uncommon. Krolls and coworkers’ reviewed the literature in 1971 and reported 24 cases of intraoral soft tissue osseouslesions. They favored the term choristoma for these lesions since they represented tumorlike growths of histologically normal tissue in an abnormal location. Since Monserrat’s2 original case report in 1913, 72 cases of osseous,cartilaginous, or osseocartilaginous choristomas have been reported in the world literature.‘-55 Three additional cases are presented in this article with two choristomas occurring in unusual locations: the retromolar pad and the anterior lateral portion of the tongue.
located slightly to the left of the midline and anterior to the circumvallate papillae. Palpation of the lesion could not be performed becauseof the patient’s hyperactive gag reflex. The differential diagnosis included fibroma, lingual thyroid nodule, and salivary gland neoplasm. The lesion was excised with the patient under local anesthetic and submitted for histologic examination. Gross examination of the submitted specimen revealed a calcified mass within the soft tissue. Histologic evaluation of the decalcified tissue showed a spherical massof dense, viable bone surrounded by connective tissue and skeletal muscle, covered with stratified squamous epithelium. The bony mass contained a well-developed haversian canai system and had numerous osteocytes within the lacunae (Fig. 2). The diagnosis was osseouschoristoma.
CASE REPORTS Case 1
Case 2
A 31-year-old black woman was seen at the University of Nebraska Medical Center Adult General Dentistry Clinic for initial dental evaluation. During examination of soft tissues of the head and neck, an asymptomatic 0.7 X 0.8 X 1.Ocm nodule of unknown duration was noted on the dorsum of the tongue (Fig. 1). The lesion was
*Resident, Adult General Dentistry, University of Nebraska Medical Center College of Dentistry, Omaha, Neb. **Assistant Professor, Department of Pathology, Diagnosis, and Radiology, and Director, Adult General Dentistry, University of Nebraska Medical Center College of Dentistry, Omaha, Neb. ***Associate Professor, Department of Pathology, Diagnosis, and Radiology, University of Nebraska Medical Center College of Dentistry, Lincoln, Neb.
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A 26-year-old white woman with a 1 cm firm, raised nodule on the right anterior and lateral surface of the tongue was seen in a private dental office. The lesion was excised and submitted for histologic examination with a provisional diagnosis of irritation fibroma. Histologic examination of the 0.9 X 0.9 x 0.5 cm nodule revealed a well-encapsulated mass of mature cartilage surrounded by dense fibrous connective tissue and covered with parakeratinized stratified squamous epithelium. The cartilage showed single nucleated chondrocytes within the lacunae (Fig. 3). The diagnosis was cartilaginous choristoma. Case 3
A 68-year-old edentulous white man with two separate lesions on the left retromolar pad adjacent to his denture was seen in a private dental office. Clinically, the lesions
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Fig.
1. Case 1. Clinical appearance of asymptomatic
Fig. 2. Case 1. Photomicrograph of lesion showing mature, viable bone with well-developed Haversian canal system.
nodule on tongue.
Fig. 3. Case 2. Photomicrograph of lesion demonstrating mature, viable cartilage surrounded by dense connective tissue.
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Fig. 4. Case 3. Photomicrograph of retromolar pad lesion showing mature, viable bone and associated fatty
marrow.
were describedas exophytic, pedunculatedmasseswith verrucal surfaces.The lesions measured1.5 X 1.2 X 0.5 cm and 0.7 X 0.5 X 0.3 cm in size.The patient had been awareof the lesionsfor 2 years.The lesionswereexcised and submittedfor histologicreview,eachwith provisional diagnosisof a “large” papilloma. Histologic examination of the larger tissue specimen
revealeda pedunculatedlesioncomposedof a central mass of calcified material with fatty marrow surroundedby connectivetissue (Fig. 4). The lesion was coveredwith stratified squamousepithelium.The diagnosiswasosseous choristoma. DISCUSSION
The intraoral osseous,cartilaginous, or osseocartilaginous choristoma is an uncommon lesion of debatable pathogenesis. Most authors have suggestedthat the intraoral choristoma is a developmental lesion because it is frequently located near the foramen caecum. Monserrat* postulated that the “osteoma” of the tongue developed from ossification of remnants of the branchial arches. Engel and Cherrick14 supported this view by detailing the ossifying poten-
Oral Surg. April. 1987
tial of many of the branchial arch derivatives. Cataldo and coworkers”’ and Jahnke and Daly2” suggested that the intraoral choristoma developed from calcifying remnants of the thyroid gland becauseof the gland’s development near the foramen caecum. Roy and coworkers4’supported the developmental theory for choristomas in the posterior portion of the tongue but thought that lesions in the anterior part of the tongue developed secondary to trauma. He indicated that either pluripotential cells or ectopic mesenchymal cells were probably present at these sites and, when stimulated by trauma, produced bone or cartilage. Additional theories have been advanced to explain the development of the intraoral choristoma. These include epignathous formation19and ossification of irritational fibromas” or lymphatic channels.9 The age range for reported cases of intraoral choristoma is between 8 and 73 years; however, the majority have been diagnosed in women between the ages of 20 and 40. Most intraoral choristomas develop in the area of the foramen caecum, but lesions have been reported in the anterior7,“. “, 39and ventrolateral”, “, 54 surface of the tongue, in the buccal mucosa,‘,12.2Rand in the lingual alveolus of the mandible.44Clinically, the choristoma develops as a firm nodular or pedunculated lesion between 0.5 and 2.0 cm in size. Most patients are unaware of the lesion, but symptoms of pain, dysphagia, gagging, choking, and nausea have been reported. Since the intraoral choristoma is rare, most such lesions are misdiagnosed as other soft tissue tumors. The differential diagnosis for the intraoral choristoma depends on the location of the lesion. When the lesion is located near the foramen caecum, lingual
thyroid nodule must be considered and evaluation for a normal and functional thyroid gland may be required before treatment. Hyperplastic lingual tonsil and salivary gland neoplasm should also be included in the differential diagnosis. When the lesion is located on the anterior and lateral aspect of the tongue, fibroma, granular cell tumor, and neural tumor should be considered. Lesions on the ventral surface of the tongue may resemble salivary gland neoplasms, mucous retention phenomena, lipomas, and neural tumors. When the lesion is located on the alveolar ridge, peripheral giant cell granuloma or fibrous hyperplasia should be included in the differential diagnosis. When the lesion is pedunculated and has a verrucal surface, it may clinically resemble a papilloma. Histologically, the intraoral choristoma consists of a well-circumscribed mass of viable lamellar bone with a well-developed haversian canal system, a
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well-developed mass of mature viable cartilage, or a mixture of bone and cartilage surrounded by dense fibrous connective tissue. Occasionally, hematopoietic or fatty marrow has been reported in the osseous lesions. Osteoblastic or osteoclastic activity is rarely seen. The intraoral choristoma is treated by means of surgical excision and, in all cases with follow-up, recurrence has not been reported. SUMMARY
The intraoral choristoma is an uncommon benign lesion of unknown origin. A review of the literature and three additional cases of intraoral choristomas are reported. Although most develop near the foramen caecum, lesions from other intraoral locations have been described. The first case demonstrates the classic lingual osseouschoristoma. The second and third cases represent unusual locations for such lesions. The occurrence of the osseouschoristoma on the retromolar pad represents the first reported lesion in that location. REFERENCES 1. Krolls SO, Jacoway JR, Alexander WN. Osseouschoristomas (osteomas) of intraoral soft tissues. ORAL SURGORAL MED ORAL PATHOL 1971;32:588-95. 2. Monserrat M. Osteome de la langue. Bull Sot Anat 1913; 88:282-3. 3. Alfred0 E, Espinoza E, Guillermo D. Lingual osteoma. J Oral Med 1982;37:27-9. 4. Begel H, Wilson H, Statigos G, Zambito RF. Osteoma of the tongue: report of a case. J Oral Surg 1968;26:662-4. 5. Berry J. Fibro-chondroma of the tongue. Trans Path SOC London 1890;41:81-2. 6. Breckenridge RL, Lukens RM. Osteoma of the tongue. Arch Otolaryngol 1950;52:19-24. 7. Bruce KW, McDonald JR. Chondroma of the tongue. ORAL SURGORAL MED ORAL PATHOL 1953;6:1281-3. 8. Bussuttil A. An osteoma of the tongue. J Laryngol Otol 1977;91:259-61. 9. Camus M, Ertzbischoff P. Chondroma de la pointe de la langue chez une jeune femme. Bull Mem Sot Anat Paris 1905;80:301-2. 10. Cataido E, Shklar G, Meyer I. Osteoma of the tongue. Arch Otolaryngol 1967;85:202-6. 11. Church LE. Osteoma of the tongue: report of a case. ORAL SURG ORAL MED ORAL PATHOL 1964;17:768-80.
12. Davis GB. Intraoral osseouschoristoma: report of a case. J Oral Surg 1980;38:144. 13. Del Rio CE. Chondroma of the tongue: review of the literature and a case report. J Oral Med 1978;33:54-6. 14. Engel P, Cherrick HM. Extraosseous osteomasof the tongue. J Oral Med 1976;31:99-103. 15. Gabriele R, Kaufman PS. Osteochondroma of the tongue: report of case. J Oral Surg 1978;36:476. 16. Gentscheff C. Uber Skeletreste in der menschlichen Zunge. Virchows Arch Path01 1934;293:129-34. 17. Goldberg AF, Skuble DF, Latronica RJ. Osteoma of the tongue: report of a case. J Oral Surg 1970;28:457. 18. Gutmann J, Cifuentes C, Balzarini MA, Sobarzo V, Vicuna R. Chondroma of the tongue. ORAL SURG ORAL MED ORAL PATHOL 1974;37:75-7.
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39. Rosen MD. Chondroma of the tongue: report of a case.J Oral Surg 1961;19:157-9. 40. Routier M. Enchondrome de la langue. Bull Mem SOCChir Paris 1905;31:369-70. 41. Roy JJ, Klein HZ, Tipton DL. Osteochondroma of the tongue. Arch Path01 1970;89:565-8. 42. Samant HC, Gupta OP. Chondroma of the tongue. ORAL SURG ORAL MED ORAL PATHOL 1971:32:450-2. 43. Sato Y, Ozawa S, Arai N, Toguchi L, Fukuda T, Ueda Y, Yoshimoto T. Osseous choristoma of the tongue: report of case. Jpn J Oral Surg 1981;27:93-5. 44. Sheridan SM. Osseouschoristoma: a report of two cases.Br J Oral Maxillofac Surg 1984;22:99-102. 45. Shimono M, Tsuji T, Iguchi Y, Yamamura T, Ogasawara M, Honda T, Nagai T. Lingual osseouschoristoma: report of 2 cases. Int J Oral Surg 1984;13:355-9. 46. Singh SM, Doyle JL. Osteoma of the tongue. Two case reports. NY State Dent J 1972;38:599-600. 47. Sugita H, Yamamoto E, Sunakawa H, Matsubara T, Furuta I, Kohama G. Lingual osseouschoristoma: report of a case. Jpn J Oral Surg 1979;25:1417-21. 48. Toyoda K, Nonoyama J, Hayashi K, Nakane H. A case of choristoma in the tongue. Zibi Rinsyo 1964;57:307-10. 49. Viglioglia PA, Stirparo MA. Chondroma of the tongue. ORAL SURG ORAL MED ORAL PATHOL 1970;29:820-6. 50. Wasserstein MH, SunderRa‘j M, Rajinder J, Yamane G, Chaundhry AP. Lingual osseous choristoma. J Oral Med 1983;38:87-9.
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51. Wesley RK, Zielinski RJ. Osteocartilaginous choristoma of the tongue: Clinical and histopathologic considerations. J Oral Surg 1978;36:59-61. 52. Witchell IS. Osteoma of the tongue. Arch Otolaryngol 1949;50:453-6. 53. Yoel J, Pundyk C. Chondroma of the tongue. ORAL SURG ORAL MED ORAL PATHOL 1965;20:578-82.
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