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Pathologic fracture of the mandible associated with simultaneous occurrence of an odontogenic keratocyst and traumatic bone cyst
J Oral Maxillofnc 45:69-71,
Surg
1987
Pathologic Fracture of the Mandible Associated with Simultaneous Occurrence of an Odontogenic Keratocyst and Traumatic Bone Cyst JOSEPH L. MATISE, DDS,* LOUIS M. BETO, DMD,t JOHN E. FANTASIA, ALLEN F. FIELDING, DMD§
The oclontogenic keratocyst (OKC) and traumatic bone cyst (TBC) are well-recognized entities that have been extensively reviewed. 1-9 However, a search of the literature failed to reveal simultaneous occurrence of these lesions. A case involving a patient with a pathologic fracture of the mandible secondary to an adjacent TBC and OKC is presented.
lesion (Fig. 1). There was an unfavorable parasymphyseal fracture extending between the roots of the canine and first premolar, and on the right side there was a minimally displaced subcondylar fracture. A closed reduction was performed two days following admission under local anesthesia and intravenous sedation. Aspiration and biopsy of the area anterior to the impaction also were carried out at that time. No aspirant was obtained and a small amount of tissue was curetted and submitted for histologic examination. Microscopic examination of the fragments revealed slender strips of granulation tissue which exhibited an inflammatory component consisting of plasma cells, lymphocytes, and some neutrophils. The tissue was lined on one side by a relatively acellular fibrinous material (Fig. 2). A diagnosis of traumatic bone cyst was made. The patient did not give a history of previous trauma to the jaw. Two days post-biopsy, the patient was taken to the operating room where, under general anesthesia. a routine extraoral approach to the left mandible was done. The fracture site was visualized, with evidence of significant thinning of the cortices. A buccal cortical window was created in the area of the parasymphysis and carried posteriorly to the ramus and coronoid notch. On exposure of the anterior aspect of the cavity. it was noted that medullary space from the parasymphysis to the area of the impaction was devoid of soft tissue except for a blood clot from the previous biopsy. However, posteriorly there was a demonstrable cystic lining attached to the impaction. Due to the absence of medullary bone and the presence of thin cortices, especially in the area of the fracture, packing the defect with cancellous bone was considered to give the best chance of fracture healing. The cystic structure and impaction were enucleated and the entire bony defect was filled with cancellous bone from the left iliac crest. An intraosseous wire was placed for fracture stabilization (Fig. 3). Histologic examination of the material removed from the posterior compartment revealed a cystic lesion lined by squamous epithelium and exhibiting a distinct basal cell layer. The epithelial lining was four to six cell layers thick and had a parakeratotic surface (Fig. 4). Epithelial proliferation into the subjacent connective tissue was absent; however, there were multiple smaller satellite cysts in the connective tissue wall. No inflammatory compo-
Report of a Case A 30-year-old white male was admitted to Temple University Hospital following an altercation in which he sustained multiple blows to the face. He complained of pain on the left side of his jaw and inability to approximate his teeth. The past medical history was noncontributory. Physical examination revealed a well-developed, wellnourished. white male in a moderate amount of distress. The heart and lung examinations were unremarkable. Oral examination revealed an obvious occlusal discrepancy between the lower left canine and first premolar, with gross mobility of the mandible noted upon manipulation. All teeth in the affected quadrant were vital. Routine admission tests were within normal limits. Radiographic examination revealed a large radiolucency of the left mandible extending from the parasymphysis to the left condyle and including the coronoid process. The unerupted
third molar was contained
DDS,$ AND
within the
* Former Resident, Department of Oral and Maxillofacial Surgery, Temple University Hospital/School of Dentistry, Philadelphia, Pennsylvania. Now in private practice in Medford, New Jersey. t Former Resident, Department of Oral and Maxillofacial Surgery, Temple University Hospital/School of Dentistry. Now in private practice in Danville, Kentucky. $ Assistant Professor, Department of Pathology, Temple University School of Dentistry,.Philadelphia. Pennsylvania. 9 Professor and Chairman, DeDartment of Oral and Maxillofacial Surgery. Temple Universit; Hospital/School of Dentistry, Philadelphia, Pennsylvania. Address correspondence and reprint requests to Dr. Matise: Jackson Commons, Ste. C3-A. 30 Jackson Road, Medford, NJ 08055. 0278-2391i87 $0.00 + .25
69
SIMULTANEOUS
OCCUKRENCE
OF OKC AND TBC
FIGURE I (fop left). Panoramic radiograph showing the radiolucency extending from the left parasymphysis to the ramus onoid process. The unfavorable fracture of the parasymphysis and a right subcondylar fracture can also be seen. FIGURE 2 (lop riglzt). Photomicrograph of tissue curetted from the anterior compartment an acellular fibrinous material. (Hematoxylin and eosin. Original magnification. x 50.) FIGURE 3 (borron~ 1@). with cancellous bone.
Postoperative
radiograph
after enucleation
FIGURE 4 (botrorn uiahr). Photomicrograph of odontogenic (Hematoxylin and eosin. Original magnification. x 63.)
nent was noted. A diagnosis was made.
of odontogenic
keratocyst
keratocyst
Discussion
Traumatic bone cyst, simple bone cyst, hemorrhagic cyst, extravasation cyst and unicameral bone cyst are terms that have been used in the literature to describe the same pathologic entity.lO The cause of the TBC is unknown although several theories have been proposed.” A widely-held theory suggesting trauma as the etiologic factor finds support from cases with such a history; however, this is not always easy to establish.“,’ The TBC is thought to originate from intramedullary hemorrhage following injury to the bone. Rather than undergoing reorganization and osteogenesis, the clot liquefies. The lesion increases in size by steady expansion produced by restriction of venous drainage. 5.12The occurrence of TBC in pa-
of the OKC. showing
consisting
reduction
a well-defined
of granulation
of the fracture
tissue
and packing
and cor-
surfaced
by
of the defect
basal layer and a parakeratotic
surface.
tients with florid osseous dysplasia has led some investigators to postulate the role of altered venous hemodynamics as a causative basis.13 Other theories include: 1) origin from degenerating bone tumors; 2) faulty calcium metabolism; 3) necrosis due to ischemic fatty marrow: 4) low-grade chronic infection; and 5) osteoclasis.1° Traumatic bone cyst usually occurs in young, male patients, and is often located between the canine and angle of the mandible.5*‘4 Most show little or no subjective findings and are diagnosed only during radiographic examination on or after pathologic fracture. Radiographically, there is frequently a scalloping of the lesion between the roots of the involved teeth. The teeth usually respond positively to vitality testing. During surgical exploration, the cavity appears empty or may contain a serosanguinous fluid.5 Histologic examination usually reveals a thin connective tissue lining but no epithelium is present. I4
71
MATISE ET AL.
Treatment modalities range from biopsy and curettage to close observation.4,6,11 With surgical intervention, bleeding is re-established and healing of the defect. is expected in several months. If the cavity is very large, bone chips can be placed to help fill the defect. lo Conservative treatment involves “watchful waiting” and can be justified by the fact that very few TBCs are noted in older individuals. l5 The OKC usually presents with a peak incidence between the second and third decades, and there is a slight predilection for males. The lesion is located predominantly in the’ mandibular third molar-ascending ramus area.‘v3J6 Studies have reported that 8.5-19% of dentigerous cysts are OKCs. Furthermore, the literature reports an incidence of keratinization among primordial cysts varying from 4.4 to 75%.**17 Some investigators consider all OKCs to be primordial in origin and view the tooth-cyst association as a result of collision of an erupting tooth with the expanding cyst. These findings suggest that OKCs that present radiographically as dentigerous cysts are actually misrepresentations. l8 This view has been disputed to varying degrees. I6919 The OKC is potentially a destructive lesion.20 Frequently, it may expand extensively in an anteroposterior direction before cortical expansion of bone is noted.1,3 This was the type of lesion presented by our patient. Of clinical importance is the significant rate of recurrence associated with the OKC. The reported incidence of recurrence ranges from 12 to 62%. Therefore, follow-up of the patient is extremely important.1,3,15.16 Our patient was presumed lost to follow-up but returned approximately one year postoperatively. His chief complaint was pain and swelling in the area of the left mandible. A panoramic radiograph revealed good osseous fill within the region of the bone graft, with no evidence of other gross pathology. Clinically, the fracture site was stable. There was a moderate cellulitis overlying the left mandible. On further investigation, the cause was determined to be pulpal and related to tooth number 20. Purulence eventually developed, and the abscess was incised and drained. The intraosseous wire was removed at that time. Endodontic therapy was accomplished on the involved tooth. A radiograph taken six months post-endodontic treatment showed no recurrence of the lesions (Fig. 5). Summary A case is presented which involved a pathologic fracture of the mandible associated with a large radiolucent lesion extending from the parasymphysis to the coronoid notch. The osseous defect was found to involve two separate lesions, identified
FIGURE 5. Radiograph of left mandible six months after endodontic treatment, one year and six months after initial treatment. Good osseous fill is seen, with no recurrence of the cysts.
histologically as a traumatic bone cyst in the anterior portion and an odontogenic keratocyst in the posterior aspect. The clinical course and treatment are discussed. References I. Brannon 2. 3.
4. 5. 6. 7. 8. 9.
10. II. 12. 13. 14. 15. 16. 17.
18 19. 20.
R: The odontogenic keratocyst-a clinicopathologic study of 312 cases, part 1. Oral Surg 42:54. 1976 Brannon R: The odontogenic keratocyst-a clinicopathologic study of 312 cases, part II. Oral Surg 43:233. 1977 Browne R: The odontogenic keratocyst, clinical aspects. Br Dent J 128:225, 1970 Goodstein D. Himmelfarb R: Paresthesia and the traumatic bone cyst. Oral Surg 42:442, 1976 Grass0 A, Demkee D. Finnegan J: Traumatic cyst of the mandible: report of a case. J Oral Surg 27:341. 1969 Howe GL: Haemorrhagic cysts of the mandible--I. Br J Oral Surg 355. 1965 Howe GL: Haemorrhagic cysts of the mandible--K Br J Oral Surg 3:77, 1965 Hughes C: Hemorrhagic bone cyst and pathologic fracture of the mandible. J Oral Surg 27:345. 1969 Zachariades N, Papanicolaou S, Triantafyllou D: Odontogenie keratocysts: review of the literature and report of sixteen cases. J Oral Maxillofac Surg 43:177, 1985 Shafer W, Hine M, Levy B: A Textbook of Oral Pathology. 4th ed. Philadelphia, WB Saunders, 1974, pp 494-496 Narang R: Large traumatic bone cyst of the mandible. .I Oral Surg 38:617. 1980 Whinery J: Progressive bone cavities of the mandible. Oral Surg 8:903. 1955 Melrose R, Abrams A, Mills B: Florid osseous dysplasia. Oral Surg 41:6382, 1976 Waldron C: Solitary (hemorrhagic) cyst of the mandible. Oral Surg 7%. 1954 Szerlip L: Traumatic bone cysts. Oral Surg 21:201. 1966 Rud J, Pindborg J: Odontogenic keratocysts: a follow-up study of 21 cases. Oral Surg 27:323. 1969 Payne T: An analysis of the clinical and histopathologic parameters of the odontoaenic keratocvst. Oral Surg 33:538. 1972 Soskolne WA, Shear M: Observations of the pathogenesis of primordial cvsts. Br Dent J 123. 1967 Robinson H: Primordial cyst versus keratocyst. Oral Surg 40:362, 1975 Ahlfors E. Larsson A. Sjogren S: The odontogenic keratocysts: a benign cystic tumor? Oral Maxillofac Surg 42: IO, 1984
Surg
1987
Pathologic Fracture of the Mandible Associated with Simultaneous Occurrence of an Odontogenic Keratocyst and Traumatic Bone Cyst JOSEPH L. MATISE, DDS,* LOUIS M. BETO, DMD,t JOHN E. FANTASIA, ALLEN F. FIELDING, DMD§
The oclontogenic keratocyst (OKC) and traumatic bone cyst (TBC) are well-recognized entities that have been extensively reviewed. 1-9 However, a search of the literature failed to reveal simultaneous occurrence of these lesions. A case involving a patient with a pathologic fracture of the mandible secondary to an adjacent TBC and OKC is presented.
lesion (Fig. 1). There was an unfavorable parasymphyseal fracture extending between the roots of the canine and first premolar, and on the right side there was a minimally displaced subcondylar fracture. A closed reduction was performed two days following admission under local anesthesia and intravenous sedation. Aspiration and biopsy of the area anterior to the impaction also were carried out at that time. No aspirant was obtained and a small amount of tissue was curetted and submitted for histologic examination. Microscopic examination of the fragments revealed slender strips of granulation tissue which exhibited an inflammatory component consisting of plasma cells, lymphocytes, and some neutrophils. The tissue was lined on one side by a relatively acellular fibrinous material (Fig. 2). A diagnosis of traumatic bone cyst was made. The patient did not give a history of previous trauma to the jaw. Two days post-biopsy, the patient was taken to the operating room where, under general anesthesia. a routine extraoral approach to the left mandible was done. The fracture site was visualized, with evidence of significant thinning of the cortices. A buccal cortical window was created in the area of the parasymphysis and carried posteriorly to the ramus and coronoid notch. On exposure of the anterior aspect of the cavity. it was noted that medullary space from the parasymphysis to the area of the impaction was devoid of soft tissue except for a blood clot from the previous biopsy. However, posteriorly there was a demonstrable cystic lining attached to the impaction. Due to the absence of medullary bone and the presence of thin cortices, especially in the area of the fracture, packing the defect with cancellous bone was considered to give the best chance of fracture healing. The cystic structure and impaction were enucleated and the entire bony defect was filled with cancellous bone from the left iliac crest. An intraosseous wire was placed for fracture stabilization (Fig. 3). Histologic examination of the material removed from the posterior compartment revealed a cystic lesion lined by squamous epithelium and exhibiting a distinct basal cell layer. The epithelial lining was four to six cell layers thick and had a parakeratotic surface (Fig. 4). Epithelial proliferation into the subjacent connective tissue was absent; however, there were multiple smaller satellite cysts in the connective tissue wall. No inflammatory compo-
Report of a Case A 30-year-old white male was admitted to Temple University Hospital following an altercation in which he sustained multiple blows to the face. He complained of pain on the left side of his jaw and inability to approximate his teeth. The past medical history was noncontributory. Physical examination revealed a well-developed, wellnourished. white male in a moderate amount of distress. The heart and lung examinations were unremarkable. Oral examination revealed an obvious occlusal discrepancy between the lower left canine and first premolar, with gross mobility of the mandible noted upon manipulation. All teeth in the affected quadrant were vital. Routine admission tests were within normal limits. Radiographic examination revealed a large radiolucency of the left mandible extending from the parasymphysis to the left condyle and including the coronoid process. The unerupted
third molar was contained
DDS,$ AND
within the
* Former Resident, Department of Oral and Maxillofacial Surgery, Temple University Hospital/School of Dentistry, Philadelphia, Pennsylvania. Now in private practice in Medford, New Jersey. t Former Resident, Department of Oral and Maxillofacial Surgery, Temple University Hospital/School of Dentistry. Now in private practice in Danville, Kentucky. $ Assistant Professor, Department of Pathology, Temple University School of Dentistry,.Philadelphia. Pennsylvania. 9 Professor and Chairman, DeDartment of Oral and Maxillofacial Surgery. Temple Universit; Hospital/School of Dentistry, Philadelphia, Pennsylvania. Address correspondence and reprint requests to Dr. Matise: Jackson Commons, Ste. C3-A. 30 Jackson Road, Medford, NJ 08055. 0278-2391i87 $0.00 + .25
69
SIMULTANEOUS
OCCUKRENCE
OF OKC AND TBC
FIGURE I (fop left). Panoramic radiograph showing the radiolucency extending from the left parasymphysis to the ramus onoid process. The unfavorable fracture of the parasymphysis and a right subcondylar fracture can also be seen. FIGURE 2 (lop riglzt). Photomicrograph of tissue curetted from the anterior compartment an acellular fibrinous material. (Hematoxylin and eosin. Original magnification. x 50.) FIGURE 3 (borron~ 1@). with cancellous bone.
Postoperative
radiograph
after enucleation
FIGURE 4 (botrorn uiahr). Photomicrograph of odontogenic (Hematoxylin and eosin. Original magnification. x 63.)
nent was noted. A diagnosis was made.
of odontogenic
keratocyst
keratocyst
Discussion
Traumatic bone cyst, simple bone cyst, hemorrhagic cyst, extravasation cyst and unicameral bone cyst are terms that have been used in the literature to describe the same pathologic entity.lO The cause of the TBC is unknown although several theories have been proposed.” A widely-held theory suggesting trauma as the etiologic factor finds support from cases with such a history; however, this is not always easy to establish.“,’ The TBC is thought to originate from intramedullary hemorrhage following injury to the bone. Rather than undergoing reorganization and osteogenesis, the clot liquefies. The lesion increases in size by steady expansion produced by restriction of venous drainage. 5.12The occurrence of TBC in pa-
of the OKC. showing
consisting
reduction
a well-defined
of granulation
of the fracture
tissue
and packing
and cor-
surfaced
by
of the defect
basal layer and a parakeratotic
surface.
tients with florid osseous dysplasia has led some investigators to postulate the role of altered venous hemodynamics as a causative basis.13 Other theories include: 1) origin from degenerating bone tumors; 2) faulty calcium metabolism; 3) necrosis due to ischemic fatty marrow: 4) low-grade chronic infection; and 5) osteoclasis.1° Traumatic bone cyst usually occurs in young, male patients, and is often located between the canine and angle of the mandible.5*‘4 Most show little or no subjective findings and are diagnosed only during radiographic examination on or after pathologic fracture. Radiographically, there is frequently a scalloping of the lesion between the roots of the involved teeth. The teeth usually respond positively to vitality testing. During surgical exploration, the cavity appears empty or may contain a serosanguinous fluid.5 Histologic examination usually reveals a thin connective tissue lining but no epithelium is present. I4
71
MATISE ET AL.
Treatment modalities range from biopsy and curettage to close observation.4,6,11 With surgical intervention, bleeding is re-established and healing of the defect. is expected in several months. If the cavity is very large, bone chips can be placed to help fill the defect. lo Conservative treatment involves “watchful waiting” and can be justified by the fact that very few TBCs are noted in older individuals. l5 The OKC usually presents with a peak incidence between the second and third decades, and there is a slight predilection for males. The lesion is located predominantly in the’ mandibular third molar-ascending ramus area.‘v3J6 Studies have reported that 8.5-19% of dentigerous cysts are OKCs. Furthermore, the literature reports an incidence of keratinization among primordial cysts varying from 4.4 to 75%.**17 Some investigators consider all OKCs to be primordial in origin and view the tooth-cyst association as a result of collision of an erupting tooth with the expanding cyst. These findings suggest that OKCs that present radiographically as dentigerous cysts are actually misrepresentations. l8 This view has been disputed to varying degrees. I6919 The OKC is potentially a destructive lesion.20 Frequently, it may expand extensively in an anteroposterior direction before cortical expansion of bone is noted.1,3 This was the type of lesion presented by our patient. Of clinical importance is the significant rate of recurrence associated with the OKC. The reported incidence of recurrence ranges from 12 to 62%. Therefore, follow-up of the patient is extremely important.1,3,15.16 Our patient was presumed lost to follow-up but returned approximately one year postoperatively. His chief complaint was pain and swelling in the area of the left mandible. A panoramic radiograph revealed good osseous fill within the region of the bone graft, with no evidence of other gross pathology. Clinically, the fracture site was stable. There was a moderate cellulitis overlying the left mandible. On further investigation, the cause was determined to be pulpal and related to tooth number 20. Purulence eventually developed, and the abscess was incised and drained. The intraosseous wire was removed at that time. Endodontic therapy was accomplished on the involved tooth. A radiograph taken six months post-endodontic treatment showed no recurrence of the lesions (Fig. 5). Summary A case is presented which involved a pathologic fracture of the mandible associated with a large radiolucent lesion extending from the parasymphysis to the coronoid notch. The osseous defect was found to involve two separate lesions, identified
FIGURE 5. Radiograph of left mandible six months after endodontic treatment, one year and six months after initial treatment. Good osseous fill is seen, with no recurrence of the cysts.
histologically as a traumatic bone cyst in the anterior portion and an odontogenic keratocyst in the posterior aspect. The clinical course and treatment are discussed. References I. Brannon 2. 3.
4. 5. 6. 7. 8. 9.
10. II. 12. 13. 14. 15. 16. 17.
18 19. 20.
R: The odontogenic keratocyst-a clinicopathologic study of 312 cases, part 1. Oral Surg 42:54. 1976 Brannon R: The odontogenic keratocyst-a clinicopathologic study of 312 cases, part II. Oral Surg 43:233. 1977 Browne R: The odontogenic keratocyst, clinical aspects. Br Dent J 128:225, 1970 Goodstein D. Himmelfarb R: Paresthesia and the traumatic bone cyst. Oral Surg 42:442, 1976 Grass0 A, Demkee D. Finnegan J: Traumatic cyst of the mandible: report of a case. J Oral Surg 27:341. 1969 Howe GL: Haemorrhagic cysts of the mandible--I. Br J Oral Surg 355. 1965 Howe GL: Haemorrhagic cysts of the mandible--K Br J Oral Surg 3:77, 1965 Hughes C: Hemorrhagic bone cyst and pathologic fracture of the mandible. J Oral Surg 27:345. 1969 Zachariades N, Papanicolaou S, Triantafyllou D: Odontogenie keratocysts: review of the literature and report of sixteen cases. J Oral Maxillofac Surg 43:177, 1985 Shafer W, Hine M, Levy B: A Textbook of Oral Pathology. 4th ed. Philadelphia, WB Saunders, 1974, pp 494-496 Narang R: Large traumatic bone cyst of the mandible. .I Oral Surg 38:617. 1980 Whinery J: Progressive bone cavities of the mandible. Oral Surg 8:903. 1955 Melrose R, Abrams A, Mills B: Florid osseous dysplasia. Oral Surg 41:6382, 1976 Waldron C: Solitary (hemorrhagic) cyst of the mandible. Oral Surg 7%. 1954 Szerlip L: Traumatic bone cysts. Oral Surg 21:201. 1966 Rud J, Pindborg J: Odontogenic keratocysts: a follow-up study of 21 cases. Oral Surg 27:323. 1969 Payne T: An analysis of the clinical and histopathologic parameters of the odontoaenic keratocvst. Oral Surg 33:538. 1972 Soskolne WA, Shear M: Observations of the pathogenesis of primordial cvsts. Br Dent J 123. 1967 Robinson H: Primordial cyst versus keratocyst. Oral Surg 40:362, 1975 Ahlfors E. Larsson A. Sjogren S: The odontogenic keratocysts: a benign cystic tumor? Oral Maxillofac Surg 42: IO, 1984