International Orthopaedics (SICOT) (1988) 12: 135-138

Orthopaedics
Springer-Verlag 1988

International

Multifocal osteoarticular tuberculosis

K. Kumar and M. B. L. Saxena
Krishna Institute of Medical Sciences, Karad, 415110 Maharashtra, India Mahatma Ghandi Institute of Medical Sciences, Sevagram, 442102, Wardha, Maharashtra, India

S u m m a r y . Multifocal osteoarticular tuberculosis is

Patients
Forty eight patients presented to the authors at centres in central India and south-east Iran between May 1976 and July 1985. Those with a doubtful final diagnosis or incomplete records were excluded. Their ages ranged from 4 to 42 years, with both sexes equally affected. Most were poor and uneducated. Symptoms had been present for between three months and 6 years. The patients were divided into three groups according to the distribution of the lesions (Table 1); 27% had multiple bony lesions, 29% multiple joint involvement and 44% multiple bone and joint lesions. Patients with lesions in bone tended to present with local pain, swelling and tenderness, whereas those with joint involvement were characterised by swelling, deformity, loss of movement, wasting and muscle sphsm. A few of the bony lesions were associated with discharging sinuses; some were secondarily infected. Some patients had been treated for tuberculosis; others had not. Some had been misdiagnosed as having pyogenic disease and had been treated accordingly. The diagnosis was made radiologically in most cases. Histological confirmation was obtained in those who underwent operation.

uncommonly reported despite its incidence o f 7 to 10% in the Indian population. We describe the clinical features and management o f 48 patients seen in the last nine years.
R~sum+. La tuberculose ostbo-articulaire ~ foyers

multiples n'est que rarement dbcrite, malgrb une frbquence de 7 ~t 10% dans la population hindoue. Nous prbsentons ici les caractbristiques cliniques et le traitement des 48 malades observbs au cours des derniOres neuf annbes.
Key words: Tuberculosis, Multifocal, Management, Osteoarticular

Introduction
A l t h o u g h o s t e o a r t i c u l a r t u b e r c u l o s i s is c o m m o n l y t h e r e s u l t o f b l o o d - b o r n e i n f e c t i o n , t h e f o c u s is usually solitary. This has traditionally been cons i d e r e d a f e a t u r e w h i c h d i s t i n g u i s h e s it f r o m a p y o g e n i c l e s i o n [21]. O c c a s i o n a l l y , h o w e v e r , t h e s k e l e t a l sites o f t u b e r c u l o s i s m a y a l s o b e multiple, a fact not appreciated in earlier reports [14, 15, 18]. W e p r e s e n t a s t u d y b a s e d o n 48 c a s e s o f m u l t i focal osteoarticular tuberculosis which illustrates the aetiology, pathology, clinical features, diagnosis a n d m a n a g e m e n t o f t h i s c o n d i t i o n .

Case Histories
Two illustrative cases are presented:

Case 1. A 4-year-old girl presented with a 21/2 year history of discharging sinuses over the ring and little fingers of both hands associated with swelling of the elbows and forearms. She was malnourished but showed no systemic signs of infection. Radiological examination revealed multiple cystic lesions in the bones of both upper and lower limbs with thinning of the cortex but little periosteal reaction (Fig. 1). Those in the hand were typical of "spina ventosa" while those in the left knee were lytic with little surrounding sclerosis. One lesion crossed the epiphyseal plate. The diagnosis was confirmed histologically. Case 2. A well-nourished 31 year-old man presented with multiple discharging sinuses over the left index finger and ankle, and the right little finger and elbow joint. The were no other

Offprint requests to: K. Kumar, C-7/159, Senpura, Chetganj

Ward, Varanesi - 221001 (UP) India

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K. Kumar and M. B. L. Saxena: Multifocal osteoarticular tuberculosis systemic signs of infection. Radiographs showed destructive changes in the bones of the hand typical of "spina ventosa", and in the olecranon and medial malleolus, the latter containing a feathery sequestrum (Fig. 2). The diagnosis was confirmed histologically and the patient responded well to curettage and chemotherapy.

Table 1. Distribution of multifocal osteoarticular tubercular lesions 1 Multiple bone involvement 2 - 3 bones 5 4 - 5 bones 6 6 or more bones 2 2 Multiple joint involvement 2 joints involved 3 joints involved 4 or more joints 7 4 3 21 (44%) 13 (27%)

14 (29%)

Treatment

3 Multiple bone and joint (mixed) lesions

Fig. 1. Radiographs showing multiple tuberculous cysts with periosteal reaction in the limb bones. The lesion in the left femoral metaphysis crosses the epiphyseal plate

Rest and triple drug therapy were the mainstay of treatment. Chemotherapy was continued for between 18. and 24 months on the assumption that the disease was multifocal because of poor resistance to infection and an impaired immune response. Short course chemotherapy was not therefore thought justified and was not used. Nineteen patients undergoing surgery had active disease, joint deformity a n d gross restriction of movement, 11 had operations for lesions associated with active sinuses, 6 had foci situated close to the growth plate and in 13 the disease was in subchondral bone close to a joint. Curettage was undertaken in 13 cases where a metaphyseal lesion endangered the physis or joint surface, and when a diaphyseal lesion was greater than 1 cm in diameter or contained a sequestrum. Eight small metaphyseal and diaphyseal lesions without complicating features were treated without operation. Cavities measuring more than 1.5 cm were packed with autogenous cancellous bone chips. Lesions crossing the physis were curetted through a metaphyseal window without grafting. Six cases were treated by excision with the surrounding bone (rib, acromion, proximal fibula) without loss of function. In three cases where the disease involved both hip and spine, excision arthroplasty of the affected hip was carried out with the aim of treating the hip and unloading

ers, right elbow and right

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the carious spine. Four patients with disease of the ipsilateral hip and knee were treated by arthrodesis of the knee and excision arthroplasty of the hip. In four cases there were spinal lesions at two levels; two with neurological problems were managed by myelography and anterolateral spinal decompression. Where the ipsilateral hip and sacroiliac joints were involved, the hip was treated and the sacroiliac joint ignored. The sacroiliac lesion healed in every case without operative intervention. Sixty four operative specimens from 36 patients were submitted to histological examination. In three cases, Langhans giant cells could not be identified and only chronic granulation tissue was seen; nevertheless, the patients were treated with anti-tuberculous therapy on clinical grounds and made a successful recovery.

Table 2. Analysis of two studies from India Author Period of study 1949-1957 1965-1967 No of cases 12,000 + 986 %age OAT 5.8% 1-3% %age MOAT 0% 8.8%

Sinha (1958) Tuli (1975)

OAT = Osteoarticular Tuberculosis M O A T = Multifocal Osteoarticular Tuberculosis

Results

Three patients died, one from miliary disease, one from renal involvement and one postoperatively after pleurectomy. All had chronic lesions of advanced stage. Good results were seen in young, fit, cooperative patients with disease of short duration which had been diagnosed early. Poor results were seen in older, unfit patients with complicated disease who took their medication irregularly.
Discussion

Although multifocal osteoarticular tuberculosis is common in India, its incidence of 7-10% was not appreciated until recently (Table 2), and descriptions in the literature are both brief and sparse. In about half to three-quarters of all cases of osteoarticular tuberculosis a primary focus may be found in the lungs [4, 19], and this may well clinch the diagnosis. However, failure to find a primary focus does not exclude tuberculosis. In this series, plain radiography of the chest revealed a primary focus in 19%, and abdominal disease was discovered in a further 8%, but in the remaining 73% no primary focus could be demonstrated. Bony lesions are usually solitary because sensitisation of the patient has already occurred before the onset of skeletal disease. If the host immunity is poor, the immune response may be altered [5] and lesions multiple. Since tubercle bacilli are blood-borne, individual lesions may be initiated at different times and multifocal lesions will be seen at different stages of development.

A Technecium 99 bone scan is helpful in their detection. In 1974, Fraser described four types of tuberculous lesion in bone, encysted, the commonest and chronic form, infiltrated, atrophic and hypertrophic [6]. The encysted lesions present as multiple circumscribed cysts with little surrounding sclerosis, and produce few symptoms. This condition was called "Osteitis Tuberculosa Multiple Cystica" by Jungling in 1920 and large numbers of cases were reported in the literature. Many of these patients probably suffered from sarcoidosis and it was not until the work of Van-Alstyne and Gower (1933) [22] and Law and Perham (1938) [13] that bacterial evidence of a tubercular aetiology became available. These cysts usually remain quiescent until activated by concurrent trauma [1]. Multifocal cystic tuberculosis has been reported frequently in Negroes [3, 9, 23], and sporadically in Caucasians [16, 17] and Asiatics [7, 11] but with no attempt at classification in the manner of Fraser. Though joints are involved between three and five times more frequently than bone when the lesions are single [12, 19], this difference disappears when the disease is multifocal. The ratio of lymphocytes to monocytes in the peripheral blood film should be greater than 5 : 1 if the prognosis is to be favourable [2]. A lower ratio indicates diminished host resistance. Repeated measurements of the ESR are equally helpful in determining prognosis. As high levels of antituberculous drugs are achieved in multifocal lesions [9, 20], there is no need to increase the standard dosage. Treatment was continued for two years because of the poor immune status of the patients. One patient with sickle cell disease proved to have multifocal tubercular bone disease despite the usual association of sickle cell disease with Salmonella infection. It is therefore worthwhile to look for a tubercular focus in patients from endemic areas with sickle cell disease. If several joints are involved, treatment should take into account their biomechanical interac-

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K. Kumar and M. B. L. Saxena: Multifocal osteoarticular tuberculosis 7. Gyu KH, Hac KD, Soo YK, Duk PB (1976) A case of multiple skeletal tuberculosis with spina ventosa: a case report. J Korean Orthop Assoc 11 : 220-224 8. Jungling D (1936) Osteitis tuberculosa cystica. Fortschr Roentgenstr 27:375 9. Kumar K (1975) Penetration of antitubercular drugs in osteoarticular tubercular lesion. Thesis Banaras Hindu University, Varanasi, India 10. Kumar K, Srivastava R (1986) Role of Girdlestone excision arthroplasty in concomitant osteoarticular tuberculosis of lumbar spine, hip and knee. J West Pacific Orthop Assoc 23:23-24 11. Kyung BD, Zin LK, Min KM, Koo L, Sik HM (1972) Multiple pseudocystic tuberculosis of bone - a case report. J Korean Orthop Assoc 7:238-242 12. La Fond EM (1959) An analysis of adult skeletal tuberculosis. Bull NY Acad Med 35:167-177 13. Law JL, Perham WS (1938) Multiple cystic tuberculosis of bones in children. Am J Dis Child 56:831 14. Marwah V (1962) Changing pattern of osteoarticular tuberculosis. J Ind Med Assoc 38:18-20 15. Mukhopedhyay B (1956) The role of excisional surgery in the treatment of bone and joint tuberculosis. Ann R Coil Surg Eng 18:288-313 16. Saxena PS (1969) Cystic tuberculosis of the patella. Ind J Orthop 3:28 17. Sharma SV (1978) Cystic skeletal tuberculosis. Ind J Orthop 12:65-70 18. Sinha BN (1958) Osteoarticular tuberculosis. J Clin Soc, KG Medical College, Lucknow, India 2:1-19 19. Tuli SM (1975) Tuberculosis of the spine. American Publishing Co Pvt Ltd, New Delhi 20. Tuli SM, Kumar K, Sen PC (1977) Penetration of antitubercular drugs in clinical osteoarticular tubercular lesions. Acta Orthopaedica Scand 48:362-368 21. Turek SL (1977) Orthopaedic principles and their application (2nd ed). Lippincott, Philadelphia 22. Van Alstyne GS, Gower HC (1933) Osteitis tuberculosa multiplex cystica. J Bone Joint Surg 15:193 23. Widman BP, Miller RF (1939) Unusual manifestation of bone tuberculosis. Radiology 32:434

tions. Thus concurrent disease of the hip and spine may be treated by Girdlestone excision arthroplasty of the hip thereby treating the hip and unloading and therefore resting the spine. This also lowers the bulk of tubercular infection in the body [10]. Similarly, where the disease affects the hip and knee on the same side, the hip may be excised and the knee fused, treating the disease and maintaining some lower limb movement. Skip lesions of the dorsal spine are easily treated through a single thoracotomy [2]. We treated compressive lesions through an anterolateral approach and managed the remainder conservatively with satisfactory results.
Acknowledgement. We are grateful to Dr Vibha Kumar MD,
Lecturer in Community Medicine and to Dr G. Madhava Nayak MS, Lecturer in Orthopaedics for secretarial help.

References
1. Bosworth DM (1959) Treatment of tuberculosis of bone and joint. Bull NY Acad Med 35:167-177 2. Edmonson AS, Crenshaw AH (eds) (1980) Campbell's Operative Orthopaedics (6th ed) CV Mosby Co, St Louis 3. Ediken J, De Palma AF, Moskowitz H, Smythe V (1963) Cystic tuberuclosis of Bone. Clin Orthop 29:163-168 4. Ediken J, Hodes PJ (1973) Roentgen diagnosis of diseases of bone. Williams and Wilkins, Baltimore 5. Fanning A, Dierich H, Lentle B (1974) Bone scanning with Technecium 99 in osteomyelitis. Tubercle 55:227 6. Fraser J (1974) Tuberculosis of bones and joints in children. Macmillan, New York