- Email: [email protected]
Multiple odontomas of the mandible: A case report
338
MULTIPLE ODONTOMAS OF THE MANDIBLE
34. Lilly GE, Steiner M, Irby WB, et al: Oral health evaluation, analysis of radiographic findings. J Am Dent Assoc 71:635, 1965 35. Johnson CC: Analysis of panoramic survey. J Am Dent Assoc 81:151, 1970 36. Karmiol M, Walsh RF: Incidence of static bone defect of mandible. Oral Surg 26:225, 1968 37. Harvey W, Noble HW: Defects on the lingual surface of the mandible near the angle. Br J Oral Surg 6:75, 1968 38. Schroeder A, Sutter F, Buser D, et al: Oral Implantology, Basics, ITI Hollow Cylinder System (ed 2). Stuttggart, Germany, Thieme, 1996 39. Benque EP, Gineste M, D&fort L: A propos des bioapatites dan la chiirgie de comblement. J de Parodontologie 5:8, 1986 40. Lundgren D, Sennerby L, Falk H, et al: The use of a new bioresorbable barrier for G.B.R. in connection with implant installation. Clin Oral Implant Res 5:177, 1994 41, Abramson AS: Ectopic submaxillary gland in the mandible. J Am DentAssoc73:1114, 1966
42. Strom C, Fjellstrom CA: An unusual case of lingual mandibular depression. Oral Surg 64:159, 1987 43. Salman L, Chaudhry AP: Malposed sublingual gland in the anterior mandible: A variant of Stafne’s idiopathic bone cavity. Compendium 12:42, 1991 44. Choukas NC, Toto PD: Etiology of static bone defects of the mandible. J Oral Surg l&16, 1960 45. Choukas NC: Developmental submandibular gland defect of the mandible. J Oral Surg 31:209, 1973 46. D’Eramo M, Poidmore JS: Developmental submandibular gland defect of the mandible. Oral Surg 39:14, 1975 47. Tolman DE, Stafne EC: Developmental bone defects of the mandible. Oral Surg 24:488, 1967 48. Hansson LG: Development of lingual mandibular bone cavity in an 1l-year-old boy. Oral Surg 49:376, 1980 49. Buser D, Bragger U, Lang NP, et al: Regeneration and enlargement of jaw-bone using G.T.R. Clin Oral Implant 1:22, 1990
J Oral Maxiilofac Surg .57:338-341, 1999
Multiple
Odontomas of the Mandible: A Case Report Osamu Iwamoto, DDS, PhD, * Hiroshi Harada, DDS, f Jingo Kusukawa, DDS, PbD,f and Tadamitsu Kameyama, DDS, PbD.f
Odontomas are relatively common lesions among odontogenic tumors, but multiple odontomas rarely occur in humans, unlike in animals, and bilateral occurence is even more unusual.’ The following is a description of a case of bilateral multiple odontomas appearing in the mandible, along with a review of the literature and a discussion of their pathogenesis.
Report
of Case
A 15-year-old Japanese girl was referred to the Department of Oral and Maxillofacial Surgery, Kurnme University School of Medicine, in March 1993 with bilateral radiopaque lesions of the mandible. The patient had begun orthodontic treatment when she was 9 years old, but the radiopaque masses were not evident on routine radiographic examination at that time. Six years later, when she terminated orthodontic treatment, radiopaque masses were observed
on both sides of the mandibular body in a panoramic radiograph. She had no history of pain, swelling, or discharge in the areas, and the family history did not indicate any similar or related abnormalities. The medical history was negative. Intraoral examination showed slight bony expansion in both lower molar areas. All teeth in the lower jaw reacted normally to pulp vitality testing. Radiographic examination showed multiple radiopaque masses, which consisted of solid or ring-like hard tissues surrounded by a thin, wellcircumscribed radiolucent area, in the premolar-molar regions of both sides of the mandible. The roots of the teeth adjacent to the lesions were being resorbed by the masses (Fig 1). The lesions also appeared as solid or ring-shaped, high-density, structures on computed tomography (Fig 2). In August 1994, after a clinical diagnosis of multiple odontoma had been made, surgical removal was performed under general anesthesia. Postoperatively, there was no evidence of recurrence or complications during a 2-year follow-up (Fig 3).
PATHOLOGY Gross Findings Received from the Kurume University School of Medicine, Japan *Staff,Department of Oral and Maxillofacial Surgery. tStaff, The Second Department of Pathology. *Lecturer, Department of Oral and Maxillofacial Surgery. gProfessor,Department of Oral and Maxillofacial Surgery. Address correspondence and reprint requests to Dr Iwamoto: Department of Oral and Maxillofacial Surgery, Kurume University School of Medicine, 67 Asahi-machi, Kurume 830.0011Japan. Q 1999 American Association of Oral and Maxillofaciol Surgeons 027%2391/99/5703-0018$3 00/O
The masses consisted of globular and tooth-like hard tissue encapsulated by thick soft tissue. On the cut surface, the masses appeared as shell-like or crown-like structures surrounding empty cystic cavities (Fig 4) or as a solid structure that included fibrous tissue with focal calcilication, one of the latter showing a unique appearance consisting of irregular branch-like projections (Fig 5).
Microscopic Findings Microscopic examination showed two patterns. One (pattern A) was a shell-like structure consisting of dentine and enamel, which was lined by reticular odontogenic
lWAMOT0
FIGURE bilateral
339
ET AL
1. Panoramic radiograph at age 16 years showing odontomas composed of solid or shell-like hard tissues.
the
FIGURE branch-like
5. Cut surface prelections.
of a unique
odomtoma
with
irregular
epithelium on the inner side and covered by a thin cementum layer on the outer side (Fig 6). The other (pattern B, Fig 7) was a similar shell-like structure without a lining of odontogenic epithelium, in which trabecular-arranged osseocemetum proliferated in contact with the outer surface. There was a background of loose, immature fibrous tissue
FIGURE 2. Computed
tomography
FIGURE 3. Two-year
showing
follow-up
the bilateral
panoramic
odontomas.
radiograph
FIGURE 4. Cut surface of a shell-Irke structure
FIGURE 6. A, Close-up of the shell-like structure [pattern A) consisting of dentin with definable dentine tubules (H&E, original magnification x40). 6, Lining of odontogenic epithelium on the inner side that resembles a unicystic ameloblastoma. Gaps between the epithelium and dentin (iorge arrow] contained enamel before demineralization. A small capillary (smd arrow) is seen in the epithelium (H&E, original magnification X 160).
340
MULTIPLE ODONTOMAS OF THE MANDIBLE
FIGURE irregular
7. Whole mount of the pattern B structure showing branch-like projections (H&E, original magnification x5).
the
(Fig 8) that included tiny islets of odontogenic epithelium and “woven bone”-like material (Fig 9). The thick surrounding membrane consisted of loose fibrous tissue covered by stratified squamous epithelium and contained cords of odontogenic epithelium, which was rather dense in some areas (Fig 10).
Discussion
FIGURE
9. Tiny scattered islets of odontogenic epithelium and “woven bone”-like material in the fibrous tissue [H&E, original magnification X 80).
Baderl has described a case closely resembling this one. His classification prompted us to classify our case as follows: 1) extension of the tumor-multiple odontomas (odontomatosis); 2) relation to regular teethindependent tumor without tooth abnormality; 3) relation to bone-intraosseous tumor; and 4) histologic differentiation-mixture of both hard and soft
Even though odontomas are one of the most common odontogenic tumors, to our knowledge multiple lesions are very rare, and only 12 cases have been described in the English literature. However, no similar case to the one reported could be found in the literature. Odontomas are classified into complex and compound types, but this case could not be clearly classified because the tumors were composites of shell or crown and tooth-like structures consisting mainly of dentin; solid masses of dentin, enamel, and cementum; as well as osteoid material, and immature connective tissue, including odontogenic epithelium.
FIGURE 8. Close-up view of the same structure as Figure 7 In contact with the outer surface, osseo-cemetum is seen proliferating in a trabecular arrangements in a background of loose, immature fibrous tissue containing capillaries (H&E, original magnification x40).
FIGURE 10.
A, Surrounding connective tissue resembling a dental sac is covered by stratified epithelium resembling odontoblasts (H&E, original magnification x 160). 6, Dense trabecular arrangement of columnar odontogenic epithelium (H&E, original magnification X 160).
SAWAKI ET AL
341
odontomas. In the latter author’s case, which had been radiographically followed-up for 5 years, the odontomasis developed directly from the basal cells of the oral mucosa, and a part of the soft odontoma developed into a hard odontoma. In our case, soft radiography showed fine radiopaque material scattered in the cystic cavities within the masses. This finding suggests that if the lesions were not removed, some would have calcified into mature, solid, hard, masses. The cause of odontoma is still unknown, although local trauma or infection have been implicated.z Furthermore, multiple odontomas have been suggested to be related to systemic syndromes such as cleidocranial dysostosis or Gardner’s syndrome.3 However, no systemic symptoms were evident in this case. Odontomas have been categorized as neoplastic lesions derived from odontogenic cells; however, some investigators4,5 regard them as hamartomas or malformations rather than neoplasms. Therefore, enucleation is curative, and recurrence is not a prob-
lem. In this case, the tumors may have arisen from bilateral supernumerary tooth germs, because all permanent teeth except the lower first premolars, which had been extracted for convenience, had erupted normally. They seemed to have arisen as a regional abnormality in the mandible, neither localized to a single tooth germ nor, like Bader’s case,l to be associated with general multiple malformations. Therefore, they should be placed into an intermediate status between the two.
References Bader G: Odontomatosis
(multiple
odontomas).
Oral Surg 23:
770,1967
Marek J, Ostrowski MB: Odontoma formation in the jaw following irradiation of an adolescent. Br J Radio1 47:897, 1974 Lamberg MA, Syrjanen SM, Ripatti LT, et al: Multiple odontomasreview of the literature and report of a new case. Proc Finn Dent Soc80:169,1984
Kramer IRH, Pindborg JJ, Shear M: Histological typing of odontogenie tumors. Berurin, Springer-Verlag, 1992, p 21 Joseph AR, James S: Oral Pathology: Clinical-Pathologic Correlations. Philadelphia, PA, Saunders, 1993, p 390
J Oral Max~llofac Surg 57:341-344, 1999
Disseminated Intravascular Coagulation Presenting as Hemorrhage After Tooth Extraction Yoshihiro Sawaki, DDS, PhD, * Mitsuyuki Yamada, DDS, f Yukio Kasuya, DDS, PbD,f and Minoru Ueda, DDS, PbDJ The postoperative complications after tooth extraction can be grouped into three major categories: those related to bleeding, those related to infection and
*Formerly Head, Department
of Oral Surgery, Fukuroi Municipal
Hospital, Fukuroi, Shizuoka, Japan; Currently
Assistant Professor,
Department of Oral Surgery, Nagoya University School of Medicine, Nagoya, Japan. tStaff, Department
of Oral Surgery, Fukuroi Municipal
Hospital,
Fukuroi, Shizuoka, Japan. *Head, Department of Oral Surgery, Fukuroi Municipal Hospital,
delayed wound healing, and those related to other factors such as drug allergy, nerve paralysis, etc. Most cases of postoperative bleeding can be prevented by taking a thorough history from the patient, by using as atraumatic procedure as possible, and by properly controlled local treatment.l If bleeding or oozing persists after dental extraction, it can be stopped by additional local treatment. However, there are a few cases in which underlying diseases cause severe hemorrhage after dental extraction.*-’ We report a case in which bleeding after routine tooth extraction indicated the presence of disseminated intravascular coagulation (DIG) caused by advanced prostatic carcinoma.
Fukuroi, Shizuoka, Japan SProfessor and Chief,
Department
of Oral Surgery, Nagoya
University School of Medicine, Nagoya, Japan. Address correspondence
and reprint
Report
Department of Oral Surgery, Nagoya University School of Medicine, 65 Tsuruma-cho,
Showa-ku,
Nagoya
466-8550,
Japan; e-mail:
[email protected] o 1999Americon
Assoclotion of Oral and Maxillofacial
027%2391/99/5703~019$300/0
of Case
requests to Dr Sawaki:
A 62-year-old man visited a dental clinic in September 11, 1992. After some questions, oral examination, and a radiographic examination, his condition was diagnosed assevere, progressive
Surgeons
periodontitis,
and it was
recommended
that
some of teeth be extracted because of their mobility. Initially,
the first premolar
in the right maxilla
was extracted
MULTIPLE ODONTOMAS OF THE MANDIBLE
34. Lilly GE, Steiner M, Irby WB, et al: Oral health evaluation, analysis of radiographic findings. J Am Dent Assoc 71:635, 1965 35. Johnson CC: Analysis of panoramic survey. J Am Dent Assoc 81:151, 1970 36. Karmiol M, Walsh RF: Incidence of static bone defect of mandible. Oral Surg 26:225, 1968 37. Harvey W, Noble HW: Defects on the lingual surface of the mandible near the angle. Br J Oral Surg 6:75, 1968 38. Schroeder A, Sutter F, Buser D, et al: Oral Implantology, Basics, ITI Hollow Cylinder System (ed 2). Stuttggart, Germany, Thieme, 1996 39. Benque EP, Gineste M, D&fort L: A propos des bioapatites dan la chiirgie de comblement. J de Parodontologie 5:8, 1986 40. Lundgren D, Sennerby L, Falk H, et al: The use of a new bioresorbable barrier for G.B.R. in connection with implant installation. Clin Oral Implant Res 5:177, 1994 41, Abramson AS: Ectopic submaxillary gland in the mandible. J Am DentAssoc73:1114, 1966
42. Strom C, Fjellstrom CA: An unusual case of lingual mandibular depression. Oral Surg 64:159, 1987 43. Salman L, Chaudhry AP: Malposed sublingual gland in the anterior mandible: A variant of Stafne’s idiopathic bone cavity. Compendium 12:42, 1991 44. Choukas NC, Toto PD: Etiology of static bone defects of the mandible. J Oral Surg l&16, 1960 45. Choukas NC: Developmental submandibular gland defect of the mandible. J Oral Surg 31:209, 1973 46. D’Eramo M, Poidmore JS: Developmental submandibular gland defect of the mandible. Oral Surg 39:14, 1975 47. Tolman DE, Stafne EC: Developmental bone defects of the mandible. Oral Surg 24:488, 1967 48. Hansson LG: Development of lingual mandibular bone cavity in an 1l-year-old boy. Oral Surg 49:376, 1980 49. Buser D, Bragger U, Lang NP, et al: Regeneration and enlargement of jaw-bone using G.T.R. Clin Oral Implant 1:22, 1990
J Oral Maxiilofac Surg .57:338-341, 1999
Multiple
Odontomas of the Mandible: A Case Report Osamu Iwamoto, DDS, PhD, * Hiroshi Harada, DDS, f Jingo Kusukawa, DDS, PbD,f and Tadamitsu Kameyama, DDS, PbD.f
Odontomas are relatively common lesions among odontogenic tumors, but multiple odontomas rarely occur in humans, unlike in animals, and bilateral occurence is even more unusual.’ The following is a description of a case of bilateral multiple odontomas appearing in the mandible, along with a review of the literature and a discussion of their pathogenesis.
Report
of Case
A 15-year-old Japanese girl was referred to the Department of Oral and Maxillofacial Surgery, Kurnme University School of Medicine, in March 1993 with bilateral radiopaque lesions of the mandible. The patient had begun orthodontic treatment when she was 9 years old, but the radiopaque masses were not evident on routine radiographic examination at that time. Six years later, when she terminated orthodontic treatment, radiopaque masses were observed
on both sides of the mandibular body in a panoramic radiograph. She had no history of pain, swelling, or discharge in the areas, and the family history did not indicate any similar or related abnormalities. The medical history was negative. Intraoral examination showed slight bony expansion in both lower molar areas. All teeth in the lower jaw reacted normally to pulp vitality testing. Radiographic examination showed multiple radiopaque masses, which consisted of solid or ring-like hard tissues surrounded by a thin, wellcircumscribed radiolucent area, in the premolar-molar regions of both sides of the mandible. The roots of the teeth adjacent to the lesions were being resorbed by the masses (Fig 1). The lesions also appeared as solid or ring-shaped, high-density, structures on computed tomography (Fig 2). In August 1994, after a clinical diagnosis of multiple odontoma had been made, surgical removal was performed under general anesthesia. Postoperatively, there was no evidence of recurrence or complications during a 2-year follow-up (Fig 3).
PATHOLOGY Gross Findings Received from the Kurume University School of Medicine, Japan *Staff,Department of Oral and Maxillofacial Surgery. tStaff, The Second Department of Pathology. *Lecturer, Department of Oral and Maxillofacial Surgery. gProfessor,Department of Oral and Maxillofacial Surgery. Address correspondence and reprint requests to Dr Iwamoto: Department of Oral and Maxillofacial Surgery, Kurume University School of Medicine, 67 Asahi-machi, Kurume 830.0011Japan. Q 1999 American Association of Oral and Maxillofaciol Surgeons 027%2391/99/5703-0018$3 00/O
The masses consisted of globular and tooth-like hard tissue encapsulated by thick soft tissue. On the cut surface, the masses appeared as shell-like or crown-like structures surrounding empty cystic cavities (Fig 4) or as a solid structure that included fibrous tissue with focal calcilication, one of the latter showing a unique appearance consisting of irregular branch-like projections (Fig 5).
Microscopic Findings Microscopic examination showed two patterns. One (pattern A) was a shell-like structure consisting of dentine and enamel, which was lined by reticular odontogenic
lWAMOT0
FIGURE bilateral
339
ET AL
1. Panoramic radiograph at age 16 years showing odontomas composed of solid or shell-like hard tissues.
the
FIGURE branch-like
5. Cut surface prelections.
of a unique
odomtoma
with
irregular
epithelium on the inner side and covered by a thin cementum layer on the outer side (Fig 6). The other (pattern B, Fig 7) was a similar shell-like structure without a lining of odontogenic epithelium, in which trabecular-arranged osseocemetum proliferated in contact with the outer surface. There was a background of loose, immature fibrous tissue
FIGURE 2. Computed
tomography
FIGURE 3. Two-year
showing
follow-up
the bilateral
panoramic
odontomas.
radiograph
FIGURE 4. Cut surface of a shell-Irke structure
FIGURE 6. A, Close-up of the shell-like structure [pattern A) consisting of dentin with definable dentine tubules (H&E, original magnification x40). 6, Lining of odontogenic epithelium on the inner side that resembles a unicystic ameloblastoma. Gaps between the epithelium and dentin (iorge arrow] contained enamel before demineralization. A small capillary (smd arrow) is seen in the epithelium (H&E, original magnification X 160).
340
MULTIPLE ODONTOMAS OF THE MANDIBLE
FIGURE irregular
7. Whole mount of the pattern B structure showing branch-like projections (H&E, original magnification x5).
the
(Fig 8) that included tiny islets of odontogenic epithelium and “woven bone”-like material (Fig 9). The thick surrounding membrane consisted of loose fibrous tissue covered by stratified squamous epithelium and contained cords of odontogenic epithelium, which was rather dense in some areas (Fig 10).
Discussion
FIGURE
9. Tiny scattered islets of odontogenic epithelium and “woven bone”-like material in the fibrous tissue [H&E, original magnification X 80).
Baderl has described a case closely resembling this one. His classification prompted us to classify our case as follows: 1) extension of the tumor-multiple odontomas (odontomatosis); 2) relation to regular teethindependent tumor without tooth abnormality; 3) relation to bone-intraosseous tumor; and 4) histologic differentiation-mixture of both hard and soft
Even though odontomas are one of the most common odontogenic tumors, to our knowledge multiple lesions are very rare, and only 12 cases have been described in the English literature. However, no similar case to the one reported could be found in the literature. Odontomas are classified into complex and compound types, but this case could not be clearly classified because the tumors were composites of shell or crown and tooth-like structures consisting mainly of dentin; solid masses of dentin, enamel, and cementum; as well as osteoid material, and immature connective tissue, including odontogenic epithelium.
FIGURE 8. Close-up view of the same structure as Figure 7 In contact with the outer surface, osseo-cemetum is seen proliferating in a trabecular arrangements in a background of loose, immature fibrous tissue containing capillaries (H&E, original magnification x40).
FIGURE 10.
A, Surrounding connective tissue resembling a dental sac is covered by stratified epithelium resembling odontoblasts (H&E, original magnification x 160). 6, Dense trabecular arrangement of columnar odontogenic epithelium (H&E, original magnification X 160).
SAWAKI ET AL
341
odontomas. In the latter author’s case, which had been radiographically followed-up for 5 years, the odontomasis developed directly from the basal cells of the oral mucosa, and a part of the soft odontoma developed into a hard odontoma. In our case, soft radiography showed fine radiopaque material scattered in the cystic cavities within the masses. This finding suggests that if the lesions were not removed, some would have calcified into mature, solid, hard, masses. The cause of odontoma is still unknown, although local trauma or infection have been implicated.z Furthermore, multiple odontomas have been suggested to be related to systemic syndromes such as cleidocranial dysostosis or Gardner’s syndrome.3 However, no systemic symptoms were evident in this case. Odontomas have been categorized as neoplastic lesions derived from odontogenic cells; however, some investigators4,5 regard them as hamartomas or malformations rather than neoplasms. Therefore, enucleation is curative, and recurrence is not a prob-
lem. In this case, the tumors may have arisen from bilateral supernumerary tooth germs, because all permanent teeth except the lower first premolars, which had been extracted for convenience, had erupted normally. They seemed to have arisen as a regional abnormality in the mandible, neither localized to a single tooth germ nor, like Bader’s case,l to be associated with general multiple malformations. Therefore, they should be placed into an intermediate status between the two.
References Bader G: Odontomatosis
(multiple
odontomas).
Oral Surg 23:
770,1967
Marek J, Ostrowski MB: Odontoma formation in the jaw following irradiation of an adolescent. Br J Radio1 47:897, 1974 Lamberg MA, Syrjanen SM, Ripatti LT, et al: Multiple odontomasreview of the literature and report of a new case. Proc Finn Dent Soc80:169,1984
Kramer IRH, Pindborg JJ, Shear M: Histological typing of odontogenie tumors. Berurin, Springer-Verlag, 1992, p 21 Joseph AR, James S: Oral Pathology: Clinical-Pathologic Correlations. Philadelphia, PA, Saunders, 1993, p 390
J Oral Max~llofac Surg 57:341-344, 1999
Disseminated Intravascular Coagulation Presenting as Hemorrhage After Tooth Extraction Yoshihiro Sawaki, DDS, PhD, * Mitsuyuki Yamada, DDS, f Yukio Kasuya, DDS, PbD,f and Minoru Ueda, DDS, PbDJ The postoperative complications after tooth extraction can be grouped into three major categories: those related to bleeding, those related to infection and
*Formerly Head, Department
of Oral Surgery, Fukuroi Municipal
Hospital, Fukuroi, Shizuoka, Japan; Currently
Assistant Professor,
Department of Oral Surgery, Nagoya University School of Medicine, Nagoya, Japan. tStaff, Department
of Oral Surgery, Fukuroi Municipal
Hospital,
Fukuroi, Shizuoka, Japan. *Head, Department of Oral Surgery, Fukuroi Municipal Hospital,
delayed wound healing, and those related to other factors such as drug allergy, nerve paralysis, etc. Most cases of postoperative bleeding can be prevented by taking a thorough history from the patient, by using as atraumatic procedure as possible, and by properly controlled local treatment.l If bleeding or oozing persists after dental extraction, it can be stopped by additional local treatment. However, there are a few cases in which underlying diseases cause severe hemorrhage after dental extraction.*-’ We report a case in which bleeding after routine tooth extraction indicated the presence of disseminated intravascular coagulation (DIG) caused by advanced prostatic carcinoma.
Fukuroi, Shizuoka, Japan SProfessor and Chief,
Department
of Oral Surgery, Nagoya
University School of Medicine, Nagoya, Japan. Address correspondence
and reprint
Report
Department of Oral Surgery, Nagoya University School of Medicine, 65 Tsuruma-cho,
Showa-ku,
Nagoya
466-8550,
Japan; e-mail:
[email protected] o 1999Americon
Assoclotion of Oral and Maxillofacial
027%2391/99/5703~019$300/0
of Case
requests to Dr Sawaki:
A 62-year-old man visited a dental clinic in September 11, 1992. After some questions, oral examination, and a radiographic examination, his condition was diagnosed assevere, progressive
Surgeons
periodontitis,
and it was
recommended
that
some of teeth be extracted because of their mobility. Initially,
the first premolar
in the right maxilla
was extracted