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Mandibular osteomyelitis caused by Actinomyces israelii
Mandibular osteomyelitis caused by
Actinomyces israelii Susannah Walker, M.D., THE
EDWARD
MALLINCKRODT
OF MEDICINE, CHILDREN’S
J. N. Middelkamp,
AND
THE
DEPARTMENT
DIVISIONS
M.D.,
and Allen Sclaroff,
OF PEDIATRICS,
OF AMBULATORY
D.D.S.,
WASHINGTON
PEDIATRICS
AND
ORAL
St. Louis, MO.
UNIVERSITY
SCHOOL
SURGERY,
ST. LOUIS
HOSPITAL
Mandibular osteomyelitis due to Actinomyces israelii is considered rare in the pediatric population. The initial complaint of the 7-year-old girl described here was increased mobility of several permanent teeth. A specific diagnosis was aggressively pursued with biopsy and anaerobic cultures to prevent the loss of these permanent teeth.
A
ctinomyces israelii is a gram-positive, nonacidfast, filamentous branching facultative anaerobe which is part of the normal flora of the oral cavity.’ This organism has been isolated from multiple sites, including tooth pockets, carious teeth, gingival crevices, and tonsillar crypts. All of these sites may serve as a portal of entry for soft-tissue infection when trauma and/or disease occur in conjunction with these anaerobes. Actinomyces infection occurred in the cervical region in approximately one half of the patients reported in several series,2-4 with soft tissue being more commonly involved than bone. Our patient, a 7-year-old girl, developed osteomyelitis of the mandible due to Actinomyces israelii. CASE REPORT
A 7-year-old girl was seen following a sudden onset of fever, mild erythema, and swelling over the angle of the mandible. She complained of loose left mandibular lateral incisor and canine teeth. A minimally tender, indurated area extended from the left lateral incisor to the left second deciduous molar. Oral penicillin V, 250 mg. four times daily, was given for a periapical abscess,but subsequentlycarious teeth were not in evidence. There was no history of traumaor dental manipulation; however, the patient was on therapeutic doses of aspirin (70 mg./kg./day) for treatment of juvenile rheumatoid arthritis which was in remission. One month later (December, 1978) the left mandibular permanentcentral incisor and the first and second deciduous molars were exfoliated. There was no evidence of root system resorption. Panoramic and occlusal roentgenograms of the mandible ‘demonstrated periosteal new bone growth along the buccal aspect of the mandible anterior to the first and second left mandibular deciduous molars and canine, together with 0030-4220/81/030243+02$00.20/0
0
1981 The C. V. Mosby Co
radiolucency of the bone anterior to the left mandibular lateral incisor. Initially, curettagewas performed on Feb. 1, 1979, on the left mandibular alveolar ridge. The provisional clinical diagnosis was “hyperplastic gingivitis.” Material from a repeat biopsy of the anterior mandibular gingiva and bone on Feb. 19, 1979, was submitted for aerobic and anaerobic cultures and histopathologic examination. Anaerobic cultures of the biopsy specimen demonstratedActinomyces isruelii, which was confirmed by the Center for Disease Control Actinomyces Laboratory in Atlanta, Georgia. A histopathologic diagnosis of focal fibrosis of bone was made. Treatment with oral penicillin V, 250 mg. four times a day for 3 months was initiated. Following 1 month of this antibiotic therapy, sclerosis of the mandibular lesion was evident on panoramic views and was interpretedas healing osteomyelitis. The patient has been asymptomaticfor a period of 11monthsfollowing completion of the penicillin therapy. Panoramic views taken 3 months after discontinuation of treatment were interpreted as indicative of an increasein new bone formation and sclerosisof the alveolar ridge. DISCUSSION
Osteomyelitis due to actinomyces has been reported infrequently in adults.5 Bone involvement occurred in 1 to 15 percent of those series reviewed by Lewis and associates. l6 Actinomyces infection in pediatric patients is also rare, even when appropriate cultures and laboratory methods are usedsi A review of inpatient medical records for the past 20 years at St. Louis Children’s Hospital revealed only one other case of actinomycotic infection, and that involved the thorax. Hematogenous spread of actinomyces with intraos243
244
Walker, Middelkamp, and Sclaroff
Oral Surg.
March,1981 seous granuloma formation and minimal subperiosteal bone reaction has been reported.8 However, the involvement of the mandible in this patient was probably by direct extension from a soft-tissue focus, although our patient demonstrated none of the usual predisposing factors associated with breaks in the mucosal barrier previously mentioned. A high index of suspicion for infection with this organism is necessary if one is to make the correct diagnosis. A biopsy should be performed on any persistent periapical lesion associated with osteomyelitis, even though a chronic draining sinus or cervicofacial abscess does not exist. Proper collection, transport, and culture of specimens under anaerobic conditions are essential for the growth of Actinomyces israelii. Actinomyces infection penetrates tissue planes with ease and characteristically produces a subacute tissue reaction with connective tissue fibrosis.3 Early diagnosis with vigorous antibiotic therapy may prevent sinus tract formation. Mobility of permanent teeth may be a presenting complaint in children. Wesley and colleagues,g in their review of the literature, reported that four of nine patients under 20 years of age with periapical actinomycosis had mobility of permanent teeth as a symptom. A substantial amount of bony destruction may occur prior to the onset of the classic symptoms of fever, trismus, and brawny swelling about the ramus of the mandible. Early loss of permanent teeth, bone de-
struction, and draining sinus tracts may be averted by aggressive diagnosis and appropriate treatment. REFERENCES 1. Slack, J. M.: Genus 1 Actinomyces. In Buchanan, R. E.. and Gibbons, N. E. (co-editors): Bergey’s Manual of Determinative Bacteriology, ed. 8, Baltimore, 1974, Williams & Wilkins Company, p. 660. 2. Weese, W. C., and Smith, 1. M.: A Study of 57 Cases of Actinomycosis Over a 36-Year Period, Arch. Intern. Med. 135: 1562-1568, 1975. 3. Cope, Z.: Actinomycosis, London, 1938, Oxford University Press, p. 57. 4. McQuarrie, D. B., and Hall, W. H.: Actinomycosis of the Lung and Chest Wall, Surgery 64: 905911, 1968. 5. Yakata, H., Nakajima, T., Yamada, H., and Tokiwa, N.: Actinomycotic Osteomyelitis of the Mandible: Report of Case, J. Oral Surg. 36: 720-724, 1978. 6. Lewis, R. P., Sutter, V. L., and Finegold, S. M.: Bone Infections Involving Anaerobic Bacteria, Medicine 57: 279-305, 1978. 7. Dunkle, L. M., Bmtherton, T. J., and Feigin, R. D.: Anaerobic Infections in Children: A Prospective Study, J. Pediatr. 57: 31 l320, 1976. Nathan, M. H., Radman, W. P., and Barton, H. L.: Osseous Actinomycosis of the Head and Neck. In Bronner, M., and Bronner, M. (editors): Actinomycosis, ed. 2, Bristol, 1971, John Wright & Sons, Ltd., p. 277. Wesley, R. K., Osborn, T. P., and Dylewski, J. J.: Periapical Actinomycosis: Clinical Considerations, J. Endod. 3: 352-355, 1977. Reprint requests to: Dr. Susannah Walker St. Louis Children’s Hospital P.O. Box 14871 St. Louis, MO. 63178
Actinomyces israelii Susannah Walker, M.D., THE
EDWARD
MALLINCKRODT
OF MEDICINE, CHILDREN’S
J. N. Middelkamp,
AND
THE
DEPARTMENT
DIVISIONS
M.D.,
and Allen Sclaroff,
OF PEDIATRICS,
OF AMBULATORY
D.D.S.,
WASHINGTON
PEDIATRICS
AND
ORAL
St. Louis, MO.
UNIVERSITY
SCHOOL
SURGERY,
ST. LOUIS
HOSPITAL
Mandibular osteomyelitis due to Actinomyces israelii is considered rare in the pediatric population. The initial complaint of the 7-year-old girl described here was increased mobility of several permanent teeth. A specific diagnosis was aggressively pursued with biopsy and anaerobic cultures to prevent the loss of these permanent teeth.
A
ctinomyces israelii is a gram-positive, nonacidfast, filamentous branching facultative anaerobe which is part of the normal flora of the oral cavity.’ This organism has been isolated from multiple sites, including tooth pockets, carious teeth, gingival crevices, and tonsillar crypts. All of these sites may serve as a portal of entry for soft-tissue infection when trauma and/or disease occur in conjunction with these anaerobes. Actinomyces infection occurred in the cervical region in approximately one half of the patients reported in several series,2-4 with soft tissue being more commonly involved than bone. Our patient, a 7-year-old girl, developed osteomyelitis of the mandible due to Actinomyces israelii. CASE REPORT
A 7-year-old girl was seen following a sudden onset of fever, mild erythema, and swelling over the angle of the mandible. She complained of loose left mandibular lateral incisor and canine teeth. A minimally tender, indurated area extended from the left lateral incisor to the left second deciduous molar. Oral penicillin V, 250 mg. four times daily, was given for a periapical abscess,but subsequentlycarious teeth were not in evidence. There was no history of traumaor dental manipulation; however, the patient was on therapeutic doses of aspirin (70 mg./kg./day) for treatment of juvenile rheumatoid arthritis which was in remission. One month later (December, 1978) the left mandibular permanentcentral incisor and the first and second deciduous molars were exfoliated. There was no evidence of root system resorption. Panoramic and occlusal roentgenograms of the mandible ‘demonstrated periosteal new bone growth along the buccal aspect of the mandible anterior to the first and second left mandibular deciduous molars and canine, together with 0030-4220/81/030243+02$00.20/0
0
1981 The C. V. Mosby Co
radiolucency of the bone anterior to the left mandibular lateral incisor. Initially, curettagewas performed on Feb. 1, 1979, on the left mandibular alveolar ridge. The provisional clinical diagnosis was “hyperplastic gingivitis.” Material from a repeat biopsy of the anterior mandibular gingiva and bone on Feb. 19, 1979, was submitted for aerobic and anaerobic cultures and histopathologic examination. Anaerobic cultures of the biopsy specimen demonstratedActinomyces isruelii, which was confirmed by the Center for Disease Control Actinomyces Laboratory in Atlanta, Georgia. A histopathologic diagnosis of focal fibrosis of bone was made. Treatment with oral penicillin V, 250 mg. four times a day for 3 months was initiated. Following 1 month of this antibiotic therapy, sclerosis of the mandibular lesion was evident on panoramic views and was interpretedas healing osteomyelitis. The patient has been asymptomaticfor a period of 11monthsfollowing completion of the penicillin therapy. Panoramic views taken 3 months after discontinuation of treatment were interpreted as indicative of an increasein new bone formation and sclerosisof the alveolar ridge. DISCUSSION
Osteomyelitis due to actinomyces has been reported infrequently in adults.5 Bone involvement occurred in 1 to 15 percent of those series reviewed by Lewis and associates. l6 Actinomyces infection in pediatric patients is also rare, even when appropriate cultures and laboratory methods are usedsi A review of inpatient medical records for the past 20 years at St. Louis Children’s Hospital revealed only one other case of actinomycotic infection, and that involved the thorax. Hematogenous spread of actinomyces with intraos243
244
Walker, Middelkamp, and Sclaroff
Oral Surg.
March,1981 seous granuloma formation and minimal subperiosteal bone reaction has been reported.8 However, the involvement of the mandible in this patient was probably by direct extension from a soft-tissue focus, although our patient demonstrated none of the usual predisposing factors associated with breaks in the mucosal barrier previously mentioned. A high index of suspicion for infection with this organism is necessary if one is to make the correct diagnosis. A biopsy should be performed on any persistent periapical lesion associated with osteomyelitis, even though a chronic draining sinus or cervicofacial abscess does not exist. Proper collection, transport, and culture of specimens under anaerobic conditions are essential for the growth of Actinomyces israelii. Actinomyces infection penetrates tissue planes with ease and characteristically produces a subacute tissue reaction with connective tissue fibrosis.3 Early diagnosis with vigorous antibiotic therapy may prevent sinus tract formation. Mobility of permanent teeth may be a presenting complaint in children. Wesley and colleagues,g in their review of the literature, reported that four of nine patients under 20 years of age with periapical actinomycosis had mobility of permanent teeth as a symptom. A substantial amount of bony destruction may occur prior to the onset of the classic symptoms of fever, trismus, and brawny swelling about the ramus of the mandible. Early loss of permanent teeth, bone de-
struction, and draining sinus tracts may be averted by aggressive diagnosis and appropriate treatment. REFERENCES 1. Slack, J. M.: Genus 1 Actinomyces. In Buchanan, R. E.. and Gibbons, N. E. (co-editors): Bergey’s Manual of Determinative Bacteriology, ed. 8, Baltimore, 1974, Williams & Wilkins Company, p. 660. 2. Weese, W. C., and Smith, 1. M.: A Study of 57 Cases of Actinomycosis Over a 36-Year Period, Arch. Intern. Med. 135: 1562-1568, 1975. 3. Cope, Z.: Actinomycosis, London, 1938, Oxford University Press, p. 57. 4. McQuarrie, D. B., and Hall, W. H.: Actinomycosis of the Lung and Chest Wall, Surgery 64: 905911, 1968. 5. Yakata, H., Nakajima, T., Yamada, H., and Tokiwa, N.: Actinomycotic Osteomyelitis of the Mandible: Report of Case, J. Oral Surg. 36: 720-724, 1978. 6. Lewis, R. P., Sutter, V. L., and Finegold, S. M.: Bone Infections Involving Anaerobic Bacteria, Medicine 57: 279-305, 1978. 7. Dunkle, L. M., Bmtherton, T. J., and Feigin, R. D.: Anaerobic Infections in Children: A Prospective Study, J. Pediatr. 57: 31 l320, 1976. Nathan, M. H., Radman, W. P., and Barton, H. L.: Osseous Actinomycosis of the Head and Neck. In Bronner, M., and Bronner, M. (editors): Actinomycosis, ed. 2, Bristol, 1971, John Wright & Sons, Ltd., p. 277. Wesley, R. K., Osborn, T. P., and Dylewski, J. J.: Periapical Actinomycosis: Clinical Considerations, J. Endod. 3: 352-355, 1977. Reprint requests to: Dr. Susannah Walker St. Louis Children’s Hospital P.O. Box 14871 St. Louis, MO. 63178