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Proliferative periostitis (Garré's osteomyelitis)
Roentgeno-oddities PROLIFERATIVE OSTEOMYELITIS)
PERIOSTITIS
(GARRk’S
G
arre’s osteomyelitis, also known as “nonsuppurative ossifying periostitis” and “periostitis ossificans,” is a disease entity that usually appears clinically as an asymptomatic, unilateral swelling of the mandible in children or young adults. This condition is usually associatedwith a carious tooth, periodontal defect, or other type of inflammatory process. GarrC’s osteomyelitis was first described by Carl GarrCl as a focal thickening of the periosteum with accompanying reactive bone formation, which results from a mild infection. Although periostitis ossificans of the tibia has been well recognized by orthopedic surgeons for many years, it was not until 1948 and 1955 that Berger* and Pe11,3respectively, described the first cases of Garri’s osteomyelitis in the mandible. A case of GarrB’s osteomyelitis with no demonstrable cause is presented here to emphasize the importance of the differential diagnosis and the ambiguity of the radiographic findings under such conditions. Case repoft In February 1983a 12-year-oldblack male patient was seen in the Billings Hospital emergency room for evaluation and treatment of a tender facial nodule on the left lateral aspect of the mandible. The patient’s mother, a pediatric nurse, described the lesions as a slowly growing bump, a few weeks old, which suddenly had become enlarged. The acute episodesubsided within 36 hours with no therapeutic intervention. There was no history of trauma to the area or infection. The patient had no history of recent travel. The physical examination revealed a firm 1.Ox 1.5 cm tender massover the lateral aspect of the left mandible. The overlying skin was unremarkable, and no fluctuance or discharge was noted. There was no lymphadenopathy, and the patient was afebrile. Chest and panoramic films were taken at this time and did not reveal any pathologic condition. The patient’s PPD was negative, and the remaining laboratory values (including a complete blood count with white count) were within normal limits. Two days later the patient was seen at the Zoller Clinic becauseof the persistenceof the previously described mass. Upon examination, a firm nodule (1.5 cm in diameter) was found on the lateral aspect of the body of the left mandible just anterior to the ascending ramus. This nodule was 258
evident only upon palpation. At this time the patient’s face was symmetrical; there was no swelling, fever, erythema, or discharge; and the area was tender only to deep pressure. Intraorally, there was no sign of gingival or periodontal infection. There was no tissue flap, operculum, or evidence of caries or associated lymphadenopathy. An occlusal film was taken at a lowered kVp and mA (65 kVp and 10 mA). An area of slight thickening or layering of the periosteum on the buccal plate opposite the lower left second molar was seen (Fig. 1). Surgical exploration of the nodule was performed with the area under local anesthesia.The periosteum was found to be thickened, and overlying soft tissue was somewhat fibrotic. The nodule had broken through the lateral cortical plate and was softer than the adjacent bone. The lesion was curetted, and multiple small fragments of tissue were submitted for histologic evaluation. The microscopic diagnosis was that of a reactive bone lesion with osteoid and osseousformation consistent with GarrC’s osteomyelitis. The patient’s postoperative course was uneventful. At 3 weeks the lesion showed a decreasein the size as well as a decreasein tenderness.A follow-up occlusal film was taken 9 weeks postoperatively and showed complete remodeling and healing of the lesion (Fig. 2). Discussion
Garr&‘s osteomyelitis most commonly occurs before the age of 25, when osteoblastic activity of the periosteum is at its peak. This form of osteomyelitis occurs predominantly over the anterior surface of the tibia and is often misdiagnosed as osteosarcoma.4In the jaws, the lateral aspect of the body of the mandible is most frequently affected. The initial presentation is usually manifested by facial asymmetry due to a focal, nonsuppurative, nontender bony enlargement.2*3,5-8In the majority of cases, a focus for an inflammatory process,such as a carious tooth, mild periodontitis, or previous dental extraction, can be found in close proximity to the bony lesion. However, there are rare instances in which no causative factors can be found.9s’o Garrt’s osteomyelitis of the jaws has fairly characteristic radiographic features. These radiographic findings depend on the duration and degree of calcification of the reactive lesion. Accordingly, the trabecular pattern in the radiographs may show varying densities.3,7The radiographs will also frequently demonstrate focal cortical expansion and
Volume 63 Number 2
1. Preoperative occlusal film taken at 60 kVp and 10 mA. Note the pericortical growth of the buccal aspect of the mandible with laminar and radial trabecular patterns. Fig.
successivelayers of new bone deposition in a manner that is sometimes referred to as an “onion skin” pattern.**‘O-I3The majority of casesof Garre’s osteomyelitis are associated with a carious tooth and periapical radiolucency, a recent extraction socket, or a periodontal defect in the vicinity of the lesion. Although the microscopic findings should provide a definitive diagnosis, the clinical presentation and the radiographic findings may be initially misleading. The differential diagnosis in this case included Garre’s osteomyelitis, Ewing’s sarcoma, osteosarcoma, cherubism, Caffey’s disease,and histiocytosis X. Ewing’s sarcoma may be confused with Garre’s osteomyelitis in that both lesions occur in patients of the same age group and both are characterized by a bony swelling in the posterior body of the mandible. Even though these two lesions may produce an “onion skin” appearance upon radiographic examination, the Ewing’s tumor is generally more osteolytic than the inflammatory process. In contrast to Garri’s osteomyelitis, rapid bony enlargement, facial neuralgias, and lip paresthesias are frequently seen in patients with Ewing’s sarcoma of the mandible 12.14
Roentgeno-oddities
259
Fig. 2. Follow-up occlusal film taken 9 weekspostoperatively at the same kVp and mA. Note the almost complete resolution of the lesion.
Although GarrC’s osteomyelitis and osteosarcoma are generally found in the same age group, the mean age of patients with osteosarcomaof the jaws is 33 years, approximately 10 years older than for patients with involvement of other sites of the body.‘* Clinically, both lesions produce hard bony swellings, and they frequently have a very short history. The bony proliferation in osteosarcomais intimately associated with a more central, ill-defined osteosclerotic and/or osteolytic lesion.I2 Unlike GarrC’s osteomyelitis, the osteoblastic forms of osteosarcomaare usually characterized by perforation and expansion of the cortical plate and by new bone formation which radiographically resembles a “sun-ray” or, more rarely, an “onion skin” pattern. Cherubism may clinically resemble GarrC’s osteomyelitis in that it presents as a hard bony swelling of the posterior body of the mandible. In contrast to the reactive bony lesion, these swellings appear in an earlier age group (2 to 6 years) and slowly progress until puberty. The lesions of cherubism are almost always bilateral and involve the maxilla as well as the mandible. This is uncommon in Garre’s osteomyelitis. Radiographically, cherubism appears as numerous cystlike, multilocular, radiolucent lesions within the mandible and/or the maxilla which tend to
260
Roentgeno-oddities
coalesce and expand, severely thinning the cortical plate. Caffey’s disease (infantile cortical hyperostosis) and GarrC’s osteomyelitis may appear quite similar radiographically, in that both lesions may produce an “onion skin” pattern. Caffey’s disease is frequently bilateral and multifocal and appears at a much earlier age than the reactive lesion, usually before the patient is 2 years old. Although active Caffey’s disease is found almost exclusively in infants, there have been reported casesin which patients manifest residual asymmetrical deformities of the mandible, most often in the area of the ramus, years after the disease has subsided.15I6 GarrC’s osteomyelitis is usually found in the posterior, tooth-bearing areas of the mandible, in contrast to Caffey’s disease, which usually develops at the angle or ramus of the mandible. Eosinophilic granuloma, a form of histiocytosis X, may resemble GarrC’s osteomyelitis in that both lesions are found in the same age group and both may appear as bony swellings in the posterior mandible. In contrast to GarrC’s osteomyelitis, eosinophilic granuloma may be associatedwith a soft tissue mass, gingivitis, or ulceration. These lesions often develop quickly and are painful. Radiographically, eosinophilic granuloma of the mandible may appear as a solitary “punched-out” lesion, but it is more commonly found to produce multilocular radiolucenties. It has been reported that eosinophilic granuloma may rarely mimic a reactive, proliferative bony lesion, producing a layering or “onion skin” radiographic appearance.4 Since the clinical and radiographic findings of the lesion are strongly suggestive but not pathognomonic of GarrC’s osteomyelitis, the histologic examination is necessary to establish a definite diagnosis. Microscopically, Garrb’s osteomyelitis consists of subperiosteal and supracortical foci of reactive bone formation with pronounced osteoblastic activity. The trabeculae may be arranged in either a parallel or a radiai pattern with respect to the cortical bone,‘7and the associated fibrous connective tissue is variably infiltrated by chronic inflammatory cells consisting primarily of lymphocytes and plasma cells. The clinical course of GarrB’s osteomyelitis is usually that of sudden onset, regeneration, spontaneous regression, and remodeling, but this cannot be considered an invariable rule.18Resolution is always enhanced by the removal of the source of inflammation and/or infection. In the case presented, the
Oral Surg. February. 1987
question arises as to whether an erupting second molar could provide the focus of inflammation that would initiate the proliferative bone response of Garr6’s osteomyelitis. It has been postulated that in casessuch as these, with no demonstrable source of inflammation, an erupting tooth may possibly be the cause of this reactive lesion.19 Patricia G. Benca, D.D.S. Reza Mosto$, D.M.D., MS. Paul C. Kuo, D.M.D., M.D. Section of Oral Pathology and Radiology Walter G. Zoller Memorial Dental Clinic University of Chicago Chicago, IL 60637 REFERENCES 1. Garrt C: Uber besondere Formen und Folgezustande der akuten infektiijsen Osteomyelitis. Beitr Z Klin Chir IO: 241-298, 1983. 2. Berger A: Perimandibular ossification of possible traumattc origin: report of a case. J Oral Surg 6: 353-356, 1948. 3. Pell GJ, et al: Garri’s osteomyelitis of the mandible: a report of a case. J Oral Surg 13: 248-252, 1955. 4. McKelvy BD, et al: Chronic disseminated histiocytosis X 01 adulthood clinically mimicking subacute osteomyelitis. J Oral Med 30: 73-76, 1975. Monteleone L, et al: Garrt’s osteomyelitis. J Oral Surg 20: 423-43 I. 1962. Gorman JM: Periostitis ossificans. ORAL SURG ORAL MED ORAL PATHOL, 10: 129-132, 1957. Perriman A Uthman A: Periostitis ossiticans. Br J Oral Surg lo: 21 l-216,
1972.
Smith SN, Farman AG: Osteomyelitis with proliferative periostitis (Garrt’s osteomyelitis): report of a case. ORAL. SURG, ORAL MED ORAL PATHOL 43: 215-318,
9. Ellis DJ, et al: Garrt’s
osteomyelitis
1977.
of the mandible. ORAL
SURG ORAL MED ORAL PATHOL 44: 183- 189, 1977.
periostitis of Garri: its 10. Eversole LR, et al: Proliferative differentiation from other nonperiostosis. J Oral Surg 37: 725-731, 1979. Il. Rowe NL, Heslop IH: Periostitis and osteomyelitis of the mandible in childhood. Br Dent J 103: 67-78, 1957. 12. Goaz P, White SC: Oral radiology: principles and interpretation, St. Louis, 1982, The C.V. Mosby Company, pp. 414426, 507-509, 532-534, 548-551, 562-567. 13. Lichty G, et al: Garrt’s osteomyelitis. ORAL SURG ORAL MED ORAL PATHOL 50: 309-313,
14. Arafut
1980.
A, et al: Ewing’s sarcoma of the jaws. ORAL SURG
ORAL MED ORAL PATHOL 55: 589-595.
1983.
15. Burbank P, Lovestedt S, Kennedy R: The dental aspects of infantile cortical hyperostosis. ORAL SURG ORAL MED ORAL PATHOL 11: 1127-1137, 1958. 16. Bowman JR, Cosby LF, Piston RE: Observations on the etiology and therapy of infantile cortical hyperostosis. J Tenn Med Assoc 48: 257-260, 1955. 17. Swartz S, Pham H: Garre’s osteomyelitis: a case report. Pediatr Dent 3: 283-286, 1981. 18. Eisenbud L, et al: Garre’s proliferative periostitis occurring simultaneously in four quadrants of the jaws. ORAL SURG, ORAL MED ORAL PATHOL 51: 172-178, 1981. 19. Loveman CE: Mandibular subperiosteal swellings occurring in children. J Am Dent Assoc. 28: 1230-1235. 1941.
PERIOSTITIS
(GARRk’S
G
arre’s osteomyelitis, also known as “nonsuppurative ossifying periostitis” and “periostitis ossificans,” is a disease entity that usually appears clinically as an asymptomatic, unilateral swelling of the mandible in children or young adults. This condition is usually associatedwith a carious tooth, periodontal defect, or other type of inflammatory process. GarrC’s osteomyelitis was first described by Carl GarrCl as a focal thickening of the periosteum with accompanying reactive bone formation, which results from a mild infection. Although periostitis ossificans of the tibia has been well recognized by orthopedic surgeons for many years, it was not until 1948 and 1955 that Berger* and Pe11,3respectively, described the first cases of Garri’s osteomyelitis in the mandible. A case of GarrB’s osteomyelitis with no demonstrable cause is presented here to emphasize the importance of the differential diagnosis and the ambiguity of the radiographic findings under such conditions. Case repoft In February 1983a 12-year-oldblack male patient was seen in the Billings Hospital emergency room for evaluation and treatment of a tender facial nodule on the left lateral aspect of the mandible. The patient’s mother, a pediatric nurse, described the lesions as a slowly growing bump, a few weeks old, which suddenly had become enlarged. The acute episodesubsided within 36 hours with no therapeutic intervention. There was no history of trauma to the area or infection. The patient had no history of recent travel. The physical examination revealed a firm 1.Ox 1.5 cm tender massover the lateral aspect of the left mandible. The overlying skin was unremarkable, and no fluctuance or discharge was noted. There was no lymphadenopathy, and the patient was afebrile. Chest and panoramic films were taken at this time and did not reveal any pathologic condition. The patient’s PPD was negative, and the remaining laboratory values (including a complete blood count with white count) were within normal limits. Two days later the patient was seen at the Zoller Clinic becauseof the persistenceof the previously described mass. Upon examination, a firm nodule (1.5 cm in diameter) was found on the lateral aspect of the body of the left mandible just anterior to the ascending ramus. This nodule was 258
evident only upon palpation. At this time the patient’s face was symmetrical; there was no swelling, fever, erythema, or discharge; and the area was tender only to deep pressure. Intraorally, there was no sign of gingival or periodontal infection. There was no tissue flap, operculum, or evidence of caries or associated lymphadenopathy. An occlusal film was taken at a lowered kVp and mA (65 kVp and 10 mA). An area of slight thickening or layering of the periosteum on the buccal plate opposite the lower left second molar was seen (Fig. 1). Surgical exploration of the nodule was performed with the area under local anesthesia.The periosteum was found to be thickened, and overlying soft tissue was somewhat fibrotic. The nodule had broken through the lateral cortical plate and was softer than the adjacent bone. The lesion was curetted, and multiple small fragments of tissue were submitted for histologic evaluation. The microscopic diagnosis was that of a reactive bone lesion with osteoid and osseousformation consistent with GarrC’s osteomyelitis. The patient’s postoperative course was uneventful. At 3 weeks the lesion showed a decreasein the size as well as a decreasein tenderness.A follow-up occlusal film was taken 9 weeks postoperatively and showed complete remodeling and healing of the lesion (Fig. 2). Discussion
Garr&‘s osteomyelitis most commonly occurs before the age of 25, when osteoblastic activity of the periosteum is at its peak. This form of osteomyelitis occurs predominantly over the anterior surface of the tibia and is often misdiagnosed as osteosarcoma.4In the jaws, the lateral aspect of the body of the mandible is most frequently affected. The initial presentation is usually manifested by facial asymmetry due to a focal, nonsuppurative, nontender bony enlargement.2*3,5-8In the majority of cases, a focus for an inflammatory process,such as a carious tooth, mild periodontitis, or previous dental extraction, can be found in close proximity to the bony lesion. However, there are rare instances in which no causative factors can be found.9s’o Garrt’s osteomyelitis of the jaws has fairly characteristic radiographic features. These radiographic findings depend on the duration and degree of calcification of the reactive lesion. Accordingly, the trabecular pattern in the radiographs may show varying densities.3,7The radiographs will also frequently demonstrate focal cortical expansion and
Volume 63 Number 2
1. Preoperative occlusal film taken at 60 kVp and 10 mA. Note the pericortical growth of the buccal aspect of the mandible with laminar and radial trabecular patterns. Fig.
successivelayers of new bone deposition in a manner that is sometimes referred to as an “onion skin” pattern.**‘O-I3The majority of casesof Garre’s osteomyelitis are associated with a carious tooth and periapical radiolucency, a recent extraction socket, or a periodontal defect in the vicinity of the lesion. Although the microscopic findings should provide a definitive diagnosis, the clinical presentation and the radiographic findings may be initially misleading. The differential diagnosis in this case included Garre’s osteomyelitis, Ewing’s sarcoma, osteosarcoma, cherubism, Caffey’s disease,and histiocytosis X. Ewing’s sarcoma may be confused with Garre’s osteomyelitis in that both lesions occur in patients of the same age group and both are characterized by a bony swelling in the posterior body of the mandible. Even though these two lesions may produce an “onion skin” appearance upon radiographic examination, the Ewing’s tumor is generally more osteolytic than the inflammatory process. In contrast to Garri’s osteomyelitis, rapid bony enlargement, facial neuralgias, and lip paresthesias are frequently seen in patients with Ewing’s sarcoma of the mandible 12.14
Roentgeno-oddities
259
Fig. 2. Follow-up occlusal film taken 9 weekspostoperatively at the same kVp and mA. Note the almost complete resolution of the lesion.
Although GarrC’s osteomyelitis and osteosarcoma are generally found in the same age group, the mean age of patients with osteosarcomaof the jaws is 33 years, approximately 10 years older than for patients with involvement of other sites of the body.‘* Clinically, both lesions produce hard bony swellings, and they frequently have a very short history. The bony proliferation in osteosarcomais intimately associated with a more central, ill-defined osteosclerotic and/or osteolytic lesion.I2 Unlike GarrC’s osteomyelitis, the osteoblastic forms of osteosarcomaare usually characterized by perforation and expansion of the cortical plate and by new bone formation which radiographically resembles a “sun-ray” or, more rarely, an “onion skin” pattern. Cherubism may clinically resemble GarrC’s osteomyelitis in that it presents as a hard bony swelling of the posterior body of the mandible. In contrast to the reactive bony lesion, these swellings appear in an earlier age group (2 to 6 years) and slowly progress until puberty. The lesions of cherubism are almost always bilateral and involve the maxilla as well as the mandible. This is uncommon in Garre’s osteomyelitis. Radiographically, cherubism appears as numerous cystlike, multilocular, radiolucent lesions within the mandible and/or the maxilla which tend to
260
Roentgeno-oddities
coalesce and expand, severely thinning the cortical plate. Caffey’s disease (infantile cortical hyperostosis) and GarrC’s osteomyelitis may appear quite similar radiographically, in that both lesions may produce an “onion skin” pattern. Caffey’s disease is frequently bilateral and multifocal and appears at a much earlier age than the reactive lesion, usually before the patient is 2 years old. Although active Caffey’s disease is found almost exclusively in infants, there have been reported casesin which patients manifest residual asymmetrical deformities of the mandible, most often in the area of the ramus, years after the disease has subsided.15I6 GarrC’s osteomyelitis is usually found in the posterior, tooth-bearing areas of the mandible, in contrast to Caffey’s disease, which usually develops at the angle or ramus of the mandible. Eosinophilic granuloma, a form of histiocytosis X, may resemble GarrC’s osteomyelitis in that both lesions are found in the same age group and both may appear as bony swellings in the posterior mandible. In contrast to GarrC’s osteomyelitis, eosinophilic granuloma may be associatedwith a soft tissue mass, gingivitis, or ulceration. These lesions often develop quickly and are painful. Radiographically, eosinophilic granuloma of the mandible may appear as a solitary “punched-out” lesion, but it is more commonly found to produce multilocular radiolucenties. It has been reported that eosinophilic granuloma may rarely mimic a reactive, proliferative bony lesion, producing a layering or “onion skin” radiographic appearance.4 Since the clinical and radiographic findings of the lesion are strongly suggestive but not pathognomonic of GarrC’s osteomyelitis, the histologic examination is necessary to establish a definite diagnosis. Microscopically, Garrb’s osteomyelitis consists of subperiosteal and supracortical foci of reactive bone formation with pronounced osteoblastic activity. The trabeculae may be arranged in either a parallel or a radiai pattern with respect to the cortical bone,‘7and the associated fibrous connective tissue is variably infiltrated by chronic inflammatory cells consisting primarily of lymphocytes and plasma cells. The clinical course of GarrB’s osteomyelitis is usually that of sudden onset, regeneration, spontaneous regression, and remodeling, but this cannot be considered an invariable rule.18Resolution is always enhanced by the removal of the source of inflammation and/or infection. In the case presented, the
Oral Surg. February. 1987
question arises as to whether an erupting second molar could provide the focus of inflammation that would initiate the proliferative bone response of Garr6’s osteomyelitis. It has been postulated that in casessuch as these, with no demonstrable source of inflammation, an erupting tooth may possibly be the cause of this reactive lesion.19 Patricia G. Benca, D.D.S. Reza Mosto$, D.M.D., MS. Paul C. Kuo, D.M.D., M.D. Section of Oral Pathology and Radiology Walter G. Zoller Memorial Dental Clinic University of Chicago Chicago, IL 60637 REFERENCES 1. Garrt C: Uber besondere Formen und Folgezustande der akuten infektiijsen Osteomyelitis. Beitr Z Klin Chir IO: 241-298, 1983. 2. Berger A: Perimandibular ossification of possible traumattc origin: report of a case. J Oral Surg 6: 353-356, 1948. 3. Pell GJ, et al: Garri’s osteomyelitis of the mandible: a report of a case. J Oral Surg 13: 248-252, 1955. 4. McKelvy BD, et al: Chronic disseminated histiocytosis X 01 adulthood clinically mimicking subacute osteomyelitis. J Oral Med 30: 73-76, 1975. Monteleone L, et al: Garrt’s osteomyelitis. J Oral Surg 20: 423-43 I. 1962. Gorman JM: Periostitis ossificans. ORAL SURG ORAL MED ORAL PATHOL, 10: 129-132, 1957. Perriman A Uthman A: Periostitis ossiticans. Br J Oral Surg lo: 21 l-216,
1972.
Smith SN, Farman AG: Osteomyelitis with proliferative periostitis (Garrt’s osteomyelitis): report of a case. ORAL. SURG, ORAL MED ORAL PATHOL 43: 215-318,
9. Ellis DJ, et al: Garrt’s
osteomyelitis
1977.
of the mandible. ORAL
SURG ORAL MED ORAL PATHOL 44: 183- 189, 1977.
periostitis of Garri: its 10. Eversole LR, et al: Proliferative differentiation from other nonperiostosis. J Oral Surg 37: 725-731, 1979. Il. Rowe NL, Heslop IH: Periostitis and osteomyelitis of the mandible in childhood. Br Dent J 103: 67-78, 1957. 12. Goaz P, White SC: Oral radiology: principles and interpretation, St. Louis, 1982, The C.V. Mosby Company, pp. 414426, 507-509, 532-534, 548-551, 562-567. 13. Lichty G, et al: Garrt’s osteomyelitis. ORAL SURG ORAL MED ORAL PATHOL 50: 309-313,
14. Arafut
1980.
A, et al: Ewing’s sarcoma of the jaws. ORAL SURG
ORAL MED ORAL PATHOL 55: 589-595.
1983.
15. Burbank P, Lovestedt S, Kennedy R: The dental aspects of infantile cortical hyperostosis. ORAL SURG ORAL MED ORAL PATHOL 11: 1127-1137, 1958. 16. Bowman JR, Cosby LF, Piston RE: Observations on the etiology and therapy of infantile cortical hyperostosis. J Tenn Med Assoc 48: 257-260, 1955. 17. Swartz S, Pham H: Garre’s osteomyelitis: a case report. Pediatr Dent 3: 283-286, 1981. 18. Eisenbud L, et al: Garre’s proliferative periostitis occurring simultaneously in four quadrants of the jaws. ORAL SURG, ORAL MED ORAL PATHOL 51: 172-178, 1981. 19. Loveman CE: Mandibular subperiosteal swellings occurring in children. J Am Dent Assoc. 28: 1230-1235. 1941.