Primary tuberculous osteomyelitis of the mandible

46

J. max.-fac. Surg. 11 (1983)

J. max.-fac. Surg. 11 (1983) 4 6 4 8 © 1983 Georg Thieme Verlag Stuttgart • New York

Primary Tuberculous Osteomyelitis of the Mandible L. Alagumba Nwoku, Tajudeen A. Kekere-Ekun, Danny R. Sawyer, Olusola O. Olude

Summary A case of primary tuberculous osteomyelitis in an 8year-old school girl is presented. Further investigation showed that there was no involvement of any other tissue, and no foci could he found elsewhere in the body. The unusual presentation of this lesion is also emphazised.

Key-Words Osteomyelitis - Tuberculosis - Recurrence

Department of Oral & Maxillofacial Surgery, (Head: L. Nwoku, MD, DMD, FWACS, FMCDS (Nig.)) Department of Oral Pathology & Biology, (Head: H. A. Mosadomi, DMD, MSC, FWACS, FMCDS (Nig.)) Department of Morbid Anatomy, (Head: E. O. Odunjo, MD, M. C. Path. (Eng.), F. M. C. Path. (Nig.)) University of Lagos, Nigeria

Introduction Tuberculous osteomyelitis of the jaws is rare. Many reported cases are those of secondary tuberculous involvement of the facial skeleton, the primary lesion being most frequently pulmonary (Sowray, 1967; Khosla, 1970; Trapnell and Bowerman, 1973; Sachs and Eisenbud, 1977). But still rarer is primary tuberculous involvement of the jaws and very few cases have been reported (Sowray, 1967; Bernstein, 1921; Cameron, 1939; Spilka, 1955; Taylor and Booth, 1964). A further point of interest is the unusual presentation, making early diagnosis difficult. We therefore present this case of an 8-year-old schoolgirl with primary tuberculous osteomyelitis of the mandible. A review of the literature is also presented.

Case Report First Admission: On 9 January, 1979, an 8-year-old girl attended the outpatient clinic of the Oral and Maxillofacial Department of the Lagos University Teaching Hospital with a letter of referral from a private dental practioner, requesting investigation and treatment of the patient who had completed a course of antibiotics for a swelling of the left mandible, but achieved "very little change" in the lesion. The swelling was said to have been present for about one month. She had a swollen mandible on the same side 4 years ago. No contributory medical history was given. Examination showed a frail, pale girl with a warm, tender bony-hard swelling extending from the left lower deciduous canine to the left lower first molar region. The left submandibular lymph gland was enlarged and slightly tender. The oral hygiene was good and 6EDC21/12C6 6ED 21/123DE6 were present. There were no carious teeth, but a discharging sinus was present in the buccal sulcus in the left lower second deciduous molar region. The radiological findings were bony destruction in the left premolar/molar area of the mandible, with marked expansion of the cortical plate (Figure 1). A swab taken from the discharging sinus and cultured showed Staphylococcus aureus. A clinical diagnosis of chronic osteomyelitis of the mandible was made, and the patient was placed on

Ampicillin 125 mg 6 hourly, and Paracetamol, one tablet, to be taken when necessary. But by 25 January, the mandibular swelling had extended to the angle of the mandible and the sinus was still present. The patient was therefore admitted on that day. The haemoglobin level was 10.5 gm %. On the following day, E6 were extracted under general anaesthesia, as they were mobile and sequestrectomy and curettage of the abscess cavity performed. After thorough toilet of the cavity, the muco-periosteal flap was sutured back with silk. The patient was then placed on intra-muscular injections of Ampiclox (Ampicillin/Cloxacillin combination) 250 mg tds. The post-operative course was uneventful, and she was discharged home four days later with drugs to take home, and attend the out-patients' clinic. Second admission: Although the mandibular swelling had earlier regressed significantly, on 6 March 1979 the patient had to be re-

Fig. 1 Lateral-oblique radiograph of the mandible showing bone destruction extending from the canine to the molar region on the left side. There is loss of density of bone in the first premolar region and the deciduous tooth appears to be "floating".

Primary Tuberculous Osteomyelitis of the Mandible

J. max.-fac. Surg. 11 (1983)

47

Fig. 2 Photomicrograph of the jaw lesion showing sequestered pieces of bone in the upper left hand corner and a tuberculous granuloma with Langhans giant cell (H & E; X 110).

Fig. 3 A higher magnification of the jaw lesion showing a multinucleated giant cell of the Langhans variety surrounded by epithelioid cells and lymphocytes (H & E; X 380).

admitted because of a pointing left mandibular swelling. The W. B. C. count on this admission was 10, 500 made up of poly-morphonuclear leukocytes 82%, lymphocytes 15 % and monocytes 3 %. A biochemical investigation of the blood did not show any abnormality. "Blood biochemistry investigation showed serum bicarbonate 25 m/Eq/1; chloride 102 m/Eq/l; sodium 137 m/Eq/1; potassium 4.2 m/Eq/1; calcium 9.8 mg/100 ml, and blood sugar 98 mg/100 ml. alkaline phosphatase was 7.8 KA units. Under general anaesthesia, a submandibular incision was made, and copious, thick, granular pus was drained. The pus aspirated from the abscess was again sent to the laboratory for culture and sensitivity. She was placed on a combination of Ampicillin and Cloxacillin 250 mg 6 hourly and Paracetamol elixir 5 ml 8 hourly as required. Supportive therapy of ferrous sulphate and multivitamins was also instituted. Eight days later, the wound was no longer draining, and the temperature had dropped to normal. Decortication of the left mandible was then performed. The operation and the postoperative course were uneventful. Her condition improved remarkably, and she was discharged home on 10 April 1979.

and a lot of soft tissue resembling granulation tissue, part of which was sent for histology.

Third Admission: About 4 months later, the patient reappeared in the outpatients' clinic. This time she presented with a mandibular swelling in the left anterior region crossing the midline and extending to the right lower second pre-molar. She looked frail and anaemic. At this stage, the lesion was suspected of being neoplastic. The patient was admitted for the third time. The haemoglobin level had dropped to 8.1 gm %. She was therefore transfused with two pints of packed cells. In addition, she was placed on lincomycin, ferrous sulphate and multivitamin therapy. The haemoglobin level rose to 9.5 gm %. On 17 August 1979 under general anaesthesia, throughand-through drainage was established, and the necrotic soft tissue curetted. There was again copious drainage of PUS,

Histology Report The biopsy revealed chronic granulation tissue within which could be found sequestered pieces of dead woven bone. In the area of the spicules of bone, there were numerous granulomatous foci (Figure2). Within the granulomatous foci multinucleated giant cells of the Langhans variety as well as epithelioid cells and lymphocytes were noted (Figure 3). Some of the granulomatous foci showed central areas of caseation necrosis. Even though tubercule bacilli could not be cultured, the demonstration of soft tubercules in an environment which is endemic for tuberculosis is, in the first instance, always diagnostic of tuberculosis. The morphological picture of this biopsy is diagnostic of tuberculous osteomyelitis.

Specific Examination Chest X-ray examination did not show any evidence of pulmonary tuberculosis and further screening showed the kidneys, spine and pelvis free of foci. Clinically, the Heal test was negative. Course The patient was placed on a combination of isonicotinic acid hydrazide, streptomycin and para-aminosalicylic acid, together with supportive measures including pyridoxin, multivite and ferrous sulphate. There was a good response. The mandibular swelling started to regress, and the intra- and extra-oral sinuses healed. The patient was discharged on 18 October 1979, after being admitted for about 2 months. She was given adequate antituberculous drugs to take home, and advised to attend the out-patients' clinic regularly. When the patient attended the clinic on 8 January 1980 the facial asymmetry had disappeared. She had put on weight and looked hap-

48

J. max.-fac. Surg. 11 (1983)

L.A. Nwoku et al.: Primary Tuberculous Osteomyelitis of the Mandible

pier, and there was still no evidence of pulmonary tuberculosis. A follow-up two months later showed no evidence of recurrence. She is still under observation.

Discussion The path of infection in oral tuberculosis is still debatable. In the case of tuberculous osteomyelitis of the mandible secondary to a primary focus elsewhere in the body, the mode of spread may be readily accepted (Pekarsky, 1954; Mellor and Stockdale, 1958; Khosla, 1970). In the primary lesion of the oral cavity, it is believed that the tubercle bacilli come into direct contact with the mucosa (Bernstein, 1921; Cawson, 1960) or may invade the pulp of a carious tooth (Stein, 1942; Thoma, 1960). An infected socket of an extracted tooth, and fracture sites have also been implicated as points of entry of tubercle bacilli (Thoma, 1916; Foldi, 1936; Boyes et al., 1956; Foster and Young, 1970). In this young patient, oral hygiene was good with no carious teeth and no evidence of fracture. This finding would therefore support the theory of haematogenous or lymphatic spread suggested by many other authors (Rubin, 1927; Chapotel, 1930; Darlington and Salman, 1937; Meng, 1940; Stuteville and Hulswit, 1948; Port and Euler, 1951; Thoma, 1960; Sowray, 1967). Furthermore, we did not find any other foci from which the tubercle bacilli could have spread to the mandible. In her family of four there was no history of tuberculosis and their chest radiographs were not contributory. Reports concerning sex and age incidence are not conclusive (Chapotel, 1930; Meng, 1940; Thilander and Wennstr6m, 1956; Thapnell and Bowerman, 1973). The case presented here is an 8-year-old girl. Meyer (1970) pointed out that although cervical lymphadenitis is usually present in osteomyelitis of the jaws, it is relatively less marked in tuberculous osteomyelitis. This view was supported by Foster and Young (1970). In our case, the submandibular lymph nodes were enlarged and slightly tender. The futility of treatment without recognition of the presence of tubercle bacilli has been demonstrated by the need to admit the patient three times within 8 months, and the repeated necessity to perform incision and drainage. The microbiological examination showed Staphylococcus aureus. Diagnosis was made on the basis of the histological findings. It is noteworthy that in the case presented here, the Heaf test was negative, and even after the histological diagnosis had been established. In addition, the tubercle bacilli could not be cultured. Although there was no classical tuberculous lymphadenitis, the submandibular lymph nodes were enlarged and moderately tender. The response to antituberculous treatment was very satisfactory, and also lends credence to the histological diagnosis. Conclusion Although the rarity of primary tuberculous osteomyelitis of the jaws is undisputable, the condition must be considered when conventional treatment for suppurative osteomyelitis does not seem to give satisfactory results, even when the Heal tests are negative.

Acknowledgement A special word of thanks goes to the Medical Illustrations Department of the College of Medicine, University of Lagos for producing the photographs. The authors also thank Dr. H. A. Mosadomi for his contribution to this article.

References Bernstein, B.: Primary tuberculosis of the buccal mucosa. Int. J. Orthodeut. (Abs.) 7 (192l) 103-104 Boyes, J., J. D. T. Jones, F. J. W. Miller: The recognition of primary tuberculous infection of the mouth. Arch. Dis. Child. 31 (1956) 31-36 Cameron, C.: Tuberculosis of mouth: Review with report case of chronic miliary tuberculosis complicated by the condition. Brit. J. Tuberc. 33 (1939) 157-163 Cawson, R. A.: Tuberculosis of the mouth and throat. Brit. J. Dis. Chest 54 (1960) 40-53 Chapotel: Tuberculose mandibulaire. Rev. Odont. 51 (1930) 444-454 Darlington, C.C., I. Salman: Oral tuberculous lesions. Amer. Rev. Tuberc. 35 (1937) 147-179 Foldi, A.: Sekund~ire tuberkul6se Infektion eines Granuloms. Z. Stomatol. 34 (1936) 281-283 Foster, C. F., W. C. Young: Tuberculous infection of a fractured mandible: Report of case. J. Oral Surg. 28 (1970) 686-690 Khosla, V . M . : Tuberculous osteomyelitis of the mandible. J. Oral Surg. 28 (1970) 848-853 Mellor, W. C., C. R, Stockdale: Facial sinuses secondary to pulmonary tuberculosis. Lancet 2 (1958) 62-64 Meng, C.M.: Tuberculosis of mandible. J. Bone Joint Surg. 22A (1940) 17-27 Meyer, J.: Infectious diseases of the jaws. J. Oral Surg. 28 (1970) 17-26 Pekarsky, R.L.: Tuberculous osteomyelitis of the mandible. Oral Snrg. 7 (1954) 1118-1192 Port, G., H. Euler: Lehrbuch der Zahnheilkunde. Bergmann. Munich 1951, pp. 4 1 6 - 4 1 8 , 4 5 0 - 4 5 2 Rubin, E. H.: Tuberculosis of buccal mucous membrane. Amer. Rev. Tuberc. 16 (1927) 39-52 Sachs, S. A., L. Eisenbud: Tuberculous osteomyelitis of the mandible. Oral Surg. 44 (1977) 425-429 Sowray, J. H.: Tuberculous facial sinuses. Brit. Dent. J. 123 (1967) 291-294 Spilka, C.J.: Tuberculosis of mandible: Report of case. J. Oral Surg. 13 (1955) 68-70 Stein, G.: Newer aspects of the problem of the focal infection. Amer. J. Orthodont. 28 (1942) 28-42 Stuteville, O. H., F. F. Hulswit: Tuberculous osteomyelitis of the mandible with report of a case. J. Oral Surg. 6 (1948) 255-259 Taylor, R. G., D. F. Booth: Tuberculous osteomyelitis of the mandible. Oral Surg. 18 (1964) 7-13 Thilander, H., A. Wennstr6m: Tuberculosis of the mouth and surrounding tissues. Oral Surg. 9 (1956) 858-870 Thoma, K. H.: Oral Abscesses. Ritter. Boston 1916, pp. 71 Thoma, K. H.: Oral Pathology. Mosby St. Louis (1960) pp. 733 Trapnell, D. H., J. E. Bowerman: Dental manifestations of systemic diseases. Bunerworth 1973, pp. 153-154

A. L. Nwoku, M.D., D.M.D. Department of Oral and Maxillofacial Surgery College of Medicine, University of Lagos PMB 12 003 Lagos, Nigeria