Int. J. Oral Surg. 1984: 13: 355-359 (Key words: c1lorlstollla; osteoma, lingual; surgery, oral)
Lingual osseous choristoma Report of 2 cases MASAKI SHIMONO, TAKANORI TSUJI, YUICHI IGUCHI, TAKEO YAMAMURA, MIYUKI OGASAWARA, TOMIHIKO HONDA AND TETSUO NAGAI Departments of Pathology and Oral Surgery, Tokyo Dental College, Masago, Chiba-City, Chiba; and Department of Oral Surgery, Keio University, School of Medicine, Tokyo, Japan ABSTRACT - 2 cases of lingual osseous choristoma are reported. A brief review of the literature on this lesion is presented, and possible causes for this unusual case are diSClissed.
(Recievedfor publication 8 June, accepted 29 August 1983)
The lingual osseous choristoma is a rare lesion of unknown etiology1,21.23. In 1913, the first case of an intraoral soft tissue osteoma was reported by Monserrat who termed this lesion osteoma de fa fangue 2S • This tumor occurred on the dorsal surface of the posterior portion of the tongue. Since Monserrat's original report until now, 62 additional cases of bony, cartilagenous and mixed bony-cartilage growths of the tongue have been reported in the literature, in which 20 cases of chondroma, 36 cases of osteoma and 6 cases of osteochondroma have been demonstrated 1-4 7 . KROLLS et al. 21 have compiled the findings reported in the world literature con· cerning bony and cartilage growths in the intraoral soft tissues and proposed that the term osseous choristoma is more appropriate than osteoma considering the origin, histological features and location of these lesions.
The present brief report deals with 2 additional cases of lingual osseous chari· stoma.
Case reports Case 1 A 47-year-old woman was admitted to the Section of Oral Surgery, Tochigi National Hospital for diagnosis and treatment of a painless nodular swelling located at the dorsal part of the tongue. The patient was otherwise healthy and without significant past medical history. Family history was unremarkable. The patient left the painless swelling alone as the region became gradually swollen, although she had been aware of it for about 1 year. The patient had a moderately good physique and was well-nourished. The examination of the head and neck showed no abnormalities. Routine clinical laboratory data and chest radiographs were within normal limits. A welldelineated painless nodule, measuring 1.5 em by 1.0 em in diameter, was noticed on the central dorsal surface of the base of the tongue. The overlying surface was smoother than the remain-
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Fig. 1. Radiographic appearance of Case 1. Note
Fig. 3. Intraoral view of Case 2. A pediculated
the osseous shadow revealing laminated appearance.
and lobulated swelling was noticed at the central dorsal surface.
iog dorsal part. The lesion was firm to palpation and its borders appeared well-defined. There was no history of tongue biting or other similar habits that could have contributed to the growth of the lesion. Clinical diagnosis was benign tumor ofthc tongue. Resection of the tumor was carried out under general endotracheal anesthesis. The tumor was found to be well encapsulated and easily separated from the surrounding lingual tissues. The resected tumor measuring 16 mm by 15 m.m by 10 mrn was very hard. Radiographical examination showed an osseous shadow revealing lamellar structure (Fig. 1). Histopathologically, the tumor mass contained stratified squamous epithelium that covered a well-delineated submucosal focus of the cartilage and the bone. Almost all areas of the tumor were occupied by the osseous tissues. Certain areas of the cartilagenous tissue had undergone osseous transformation. Tn a pale
cartilagenous ground substance. chondrocytes of various sizes were recognizable. The histological diagnosis was osteochondroma of the tongue (Fig. 2). Case 2 A 37-year-old woman was admiLLed to the Department of Oral Surgery, Tokyo Dental College, with a painless nodular swelling at the dorsal part of the tongue. She was healthy and without significant past medical history. Family history was nothing worthy of special mention. The patient had a moderately good physique and was well-nourished. Routine clinical laboratory data and chest radiographs were within normal limits. Although the patient had become aware of the swelling about 8 years before, she left alone it
Fig. 4. Case 2. The tumor reveals a well-
Fig. 2. Case 1. Cartilagenous tissue has undergone
osseous transformation.
circumscribed lamellated mass of dense bone. Inset shows a high magnification of Fig. 4 revealing mature lamellar bone with Harversian systems.
LINGUAL OSSEOUS CHORISTOMA
because the swelling had been painless. A pediculated and lobulated swelling, measuring 1.5 cm by 1.5 cm by 0.7 cm in diameter, was noticed at the central dorsal surface corresponding to the foramen cecum of the tongue (Fig. 3). The lesion was firm and covered with smooth healthy oral mucosa. There was no traumatic history, such as tongue biting or other similar habits that could have contributed to the growth of the lesion. The clinical impression was lingual thyroid. Under local anesthesia with lidocaine, resection of the tumor was performed. The resected tumor was a lobulated, whitish firm mass. Microscopic examination of the lesion following decalcification of the specimen revealed a well-circumscribed, lamellated mass of dense bone surrounded by fibrous connective tissue, and covered with stratified squamous epithelium. The histopathological diagnosis was osteoma of the tongue (Fig. 4).
Discussion Surveying the literature for infonnation on lingual osseous choristoma in the world, 62 previously reported examples were found 1-4 7 . In these studies, 37 patients (66%) were female, the youngest patient was 8 years, the eldest 73, and the average was 29.3+14.2 (n=57) in cases listing age and sex. Histopathological examination revealed that 36 cases (57%) were osteoma, 20 cases (32%) chondroma and 6 cases (11 %) osteochondroma. As far as size of the growths, relatively small tumors measuring about 1 em in diameter were noticed in almost all cases. This indicates that difficulty in swallowing may be induced by the location of the tumor. KROLLS et al. 21 have remarked that the tenn osteoma should be reserved for an overgrowth of bone which is closely associated with a part of the skeletal structure and the term cutaneous ossification should be used for a dermal or subcutaneous bony growth. Further, they have remarked that the tenn hamartoma for the intraoral lesions is not recommended, as the term
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should be limited to tumescences of superfluous tissue. From these ideas, they have proposed that the term osseous choristoma is more appropriate than osteoma, although the term osteoma or chondroma of the tongue has been used since the initial report of MONSERRAT 25 . Our view is in accord with that of KROLLS et al. 21 because of the origin, location and histological features of these lesions. The pathogenesis of the choristoma in the intraoral soft tissue is unknown, though hypotheses of the origin have been proposed. The close proximity of these lesions to the foramen cecum leads to the theory of a developmental malformation. That is, in the complex embryological development, multipotential cells may become enclaved and give rise to unusual proliferative lesions in later years. There are other developmental abnormalities found in this area, such as lingual thyroid, cysts which may be remnants of the thyroglossal duct, and sometimes ossifying soft tissue lesions7.14.35.45. From observations of our 2 cases, we are in substantial agreement with a malformation concept, developmental though it is surmised that heterotopic tissue is present in the tongue in which case the lesion would be considered heterotopic bone. Another possible idea of pathogenesis is that the lesion is a post-traumatic center of ossification after a hematoma has been organized as is seen in post-traumatic myositis ossificans of other muscles of the bodyl.3? However, it is tempting to deny this concept because it was not possible to find any irritant agent and no traumatic history was known in our 2 cases. In Case 1, the tumor contained a welldelineated submucosal focus of the bone and cartilage. This suggests that the cartilage may have been transformed completely to bone and may have been designated as osteoma, if the lesion had COlltinued to developl1.
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Address: Masaki Shimono Department of Pathology Tokyo Dental College 1·2·2, Masago Chiba-City 260 Japan