International Orthopaedics (SICOT) (1988) 12: 135-138 International Orthopaedics © Springer-Verlag 1988

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 uncommonly reported despite its incidence o f 7 to 10% in the Indian population. We describe the clini- cal 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 su l t o f b l o o d - b o r n e i n f e c t i o n , t he f o c u s is u s u a l l y so l i t a ry . Th i s has t r a d i t i o n a l l y b e e n c o n - s 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 i t 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 s i tes o f t u b e r c u l o s i s m a y a l so b e m u l t i p l e , a f ac t n o t a p p r e c i a t e d in e a r l i e r r e p o r t s [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 cases o f m u l t i - f o c a l 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 w h i c h i l l u s t r a t e s t h e a e t i o l o g y , p a t h o l o g y , c l i n i c a l f e a tu r e s , d i a g n o - sis a n d m a n a g e m e n t o f th i s c o n d i t i o n .

Offprint requests to: K. Kumar, C-7/159, Senpura, Chetganj Ward, Varanesi - 221001 (UP) India

Patients

Forty eight patients presented to the authors at centres in cen- tral India and south-east Iran between May 1976 and July 1985. Those with a doubtful final diagnosis or incomplete re- cords were excluded. Their ages ranged from 4 to 42 years, with both sexes equally affected. Most were poor and unedu- cated. 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% multi- ple bone and joint lesions.

Patients with lesions in bone tended to present with local pain, swelling and tenderness, whereas those with joint in- volvement were characterised by swelling, deformity, loss of movement, wasting and muscle sphsm. A few of the bony le- sions were associated with discharging sinuses; some were sec- ondarily infected.

Some patients had been treated for tuberculosis; others had not. Some had been misdiagnosed as having pyogenic dis- ease and had been treated accordingly.

The diagnosis was made radiologically in most cases. His- tological confirmation was obtained in those who underwent operation.

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 infec- tion. 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 his- tologically.

Case 2. A well-nourished 31 year-old man presented with mul- tiple discharging sinuses over the left index finger and ankle, and the right little finger and elbow joint. The were no other

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Table 1. Distribution of multifocal osteoarticular tubercular lesions

1 Multiple bone involvement 13 (27%) 2 - 3 bones 5 4 - 5 bones 6 6 or more bones 2

2 Multiple joint involvement 14 (29%) 2 joints involved 7 3 joints involved 4 4 or more joints 3

3 Multiple bone and joint (mixed) lesions 21 (44%)

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

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 contain- ing a feathery sequestrum (Fig. 2). The diagnosis was con- firmed histologically and the patient responded well to curet- tage and chemotherapy.

Treatment

Rest and triple drug therapy were the mainstay of treatment. Chemotherapy was continued for be- tween 18. and 24 months on the assumption that the disease was multifocal because of poor resis- tance to infection and an impaired immune re- sponse. Short course chemotherapy was not there- fore thought justified and was not used.

Nineteen patients undergoing surgery had ac- tive disease, joint deformity and gross restriction of movement, 11 had operations for lesions asso- ciated 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 with- out operation. Cavities measuring more than 1.5 cm were packed with autogenous cancellous bone chips. Lesions crossing the physis were cu- retted 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, ex- cision arthroplasty of the affected hip was carried out with the aim of treating the hip and unloading

ers, right elbow and right

K. Kumar and M. B. L. Saxena: Multifocal osteoarticular tuberculosis

the carious spine. Four patients with disease of the ipsilateral hip and knee were treated by ar- throdesis 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 spi- nal decompression. Where the ipsilateral hip and sacroiliac joints were involved, the hip was treat- ed and the sacroiliac joint ignored. The sacroiliac lesion healed in every case without operative in- tervention.

Sixty four operative specimens from 36 pa- tients 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.

Results

Three patients died, one from miliary disease, one from renal involvement and one postoperatively after pleurectomy. All had chronic lesions of ad- vanced stage.

Good results were seen in young, fit, coopera- tive 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 descrip- tions 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 remain- ing 73% no primary focus could be demonstrated.

Bony lesions are usually solitary because sen- sitisation of the patient has already occurred be- fore the onset of skeletal disease. If the host im- munity is poor, the immune response may be al- tered [5] and lesions multiple. Since tubercle bacil- li are blood-borne, individual lesions may be ini- tiated at different times and multifocal lesions will be seen at different stages of development.

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Table 2. Analysis of two studies from India

Author Period No of %age %age of study cases OAT MOAT

Sinha (1958) 1949-1957 12,000 + 5.8% 0% Tuli (1975) 1965-1967 986 1-3% 8.8%

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

A Technecium 99 bone scan is helpful in their detection.

In 1974, Fraser described four types of tuber- culous lesion in bone, encysted, the commonest and chronic form, infiltrated, atrophic and hyper- trophic [6].

The encysted lesions present as multiple cir- cumscribed 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 avail- able. These cysts usually remain quiescent until activated by concurrent t rauma [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 le- sions 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 endem- ic areas with sickle cell disease.

If several joints are involved, treatment should take into account their biomechanical interac-

138 K. Kumar and M. B. L. Saxena: Multifocal osteoarticular tuberculosis

tions. Thus concurrent disease of the hip and spine may be treated by Girdlestone excision ar- throplasty of the hip thereby treating the hip and unloading and therefore resting the spine. This al- so 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 ex- cised 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 treat- ed compressive lesions through an anterolateral approach and managed the remainder conserva- tively 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 chil- dren. Macmillan, New York

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 exci- sion arthroplasty in concomitant osteoarticular tuberculo- sis 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 re- port. J Korean Orthop Assoc 7:238-242

12. La Fond EM (1959) An analysis of adult skeletal tubercu- losis. 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 tu- berculosis. 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 Or- thop 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 Pub- lishing Co Pvt Ltd, New Delhi

20. Tuli SM, Kumar K, Sen PC (1977) Penetration of anti- tubercular drugs in clinical osteoarticular tubercular le- sions. Acta Orthopaedica Scand 48:362-368

21. Turek SL (1977) Orthopaedic principles and their applica- tion (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