Indian Journal of Pediatrics, Volume 75—May, 2008 505

Clinical Brief

Correspondence and Reprint requests : Dr. Ali Zare Mirzaie,Pathology Department of Hazrate Rasool Akram Hospital,Niyayesh St. Sattarkhan Ave., Tehran, Iran

[Received April 26, 2005; Accepted January 17, 2008]

Although tuberculosis (TB) is rare in developed countries,it is very common and endemic in developing states.1 Ascompared to pulmonary TB, skeletal TB is a very raredisease comprising 1-3% of the total population of TBpatients. In the extrapulmonary TB group, the rate ofskeletal TB is about 10-20%.1-4 Only 1/3 of patients withtuberculosis of the bone are diagnosed with concomitantactive pulmonary disease.5

The diagnosis is often delayed because osseoustuberculosis is a paucibacillary lesion, and smears areoften negative. Localized pain, fever, and weight loss aredescribed in several case reports of musculoskeletaltuberculosis.6,7 Nonspecific nature of radiographicfindings can often delay the diagnosis.8-10 Cystic skeletalTB is a rare disease mostly seen in children. In this agegroup, cystic lesions usually involve the metaphysis of theperipheral long bones.11 In spite of all recent advances indiagnosis and treatment, tuberculosis remains a majorunsolved issue at the international level.

CLINICAL DATA

We studied 4 cases with tuberculous osteomyelitis inIranian National Research Institute of Tuberculosis andLung Diseases from 2000 to 2003.

RESULTS

Three Iranian and an Afghan infants, ranging from 10 to16 month old (mean: 13.5 months) with an equal genderratio, were diagnosed with osteoarticular TB. Theduration of symptoms ranged from 2 weeks to 4 months.Sites of the involvement were ankle, wrist (in 2 patients)and knee joints. Clinical and laboratory data about thepatients is summarized in table 1.

DISCUSSION

Any part of the skeleton may be involved but the sitesmost commonly affected are spine,femur,tibia,and fibula.The spine represents 50% of these lesions. As comparedwith pyogenic osteomyelitis, tuberculosis of the bones ininfants and children tends to occur in the vascularizedmetaphyses where it produces an endarteritis.12- 14 Thefactors that influence localization of skeletal tuberculosisto a specific bone are undefined. Tuberculosis of the jointsis characteristically monoarticular, the knee and hip aremost frequently affected.15 As in other forms ofextrapulmonary tuberculosis, the respiratory tract is theprimary portal of entry of mycobacteria. Tuberculousosteomyelitis is thought to occur secondary tolymphohematogenous dissemination to the bone at thetime of initial pulmonary infection,with local reactivationat a later date.13-16 The clinical and radiographicpresentation of skeletal tuberculosis varies widely anddepends primarily on the stage of the disease atevaluation. Systemic symptoms such as fever ,night sweat

Tuberculous Osteomyelitis of the Long Bones and Joints

M. Hakimi1, F. Hashemi2, A. Zare Mirzaie3, A. Hassan Pour3 and H. Kosari1

1National Research Institute of Tuberculosis and Lung Diseases, Massih Daneshvari Hospital, Darabad, Tehran,Iran, 2Shafayahyaeian Orthopedic Center. Baharestan Sq. Iran University of Medical Sciences and Health Services,Tehran, 3Hazrate Rasool Akram Hospital, Niyayesh St. Sattarkhan Ave. Iran University of Medical Sciences andHealth Services, Tehran

ABSTRACT

Skeletal tuberculosis(TB) is usually a rare osteoarticular disease in which bones or joints are involved. We studied 4 suchcases admitted to Iranian National Research Institute of Tuberculosis and Lung Diseases from 2000 to 2003. In the initialstages of the disease, diagnosis is very difficult and conventional radiographies often reveal nonspecific findings. Ourexperience in these cases showed that a high index of suspicion is the most important step in early diagnosis of skeletaltuberculosis. [Indian J Pediatr 2008; 75 (5) : 505-508] E-mail : Zaremir@gmail.com

Key words : Long bones; Skeletal tuberculosis; Solitary cystic lesions

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TABLE 1. Clinical and Paraclinical Findings of the Patients

Case Clinical Laboratory Radiologic Pathologic Tretment and OutcomeNO. findings findings findings findings

1 A 10 month old Afghan WBC:8000/mm3 CXR and thoracic CT Multiple caseous Isoniazid, Rifampin,girl with pain and swelling (PMN:61%, scan:Mild hilar granulomas Ethambutol andof the left ankle joint, no Lymphocyte:23%, lymphadenopathy compatible with Pyrazinamide (4-drugfamily history of TB and Monocyte:10%, Whole body bone scan: TB osteomyelitis. regimen) for two monthscough, body temperature : Eosinophil:6%) Increased uptake in the continued with 2 drugs37.2•c, swelling and PPD:15mm, left ankle region given twice weekly for 7tenderness of the left ankle ESR:22mm/hr Left ankle X-ray: months. Rapid recoveryjoint Gastric washing for Large lytic lesion in the and improvement in

acid fast bacilli: posteromedial aspect of health was observedNegative the left distal within one month andBone culture:Positive metaphysis of lytic lesion was sclerosed.for M.Tuberculosis , tibia bone along withsensitive to INH and periosteal reaction andRifampin soft tissue swelling

2 A 15 month old boy WBC:8200/mm3 CXR :Unremarkable Multiple caseous Isoniazid, Rifampin,with claudication of 2 (PMN:66%, Right knee X-ray: granulomas Ethambutol andweeks duration, Lymphocyte:15%, Large lytic lesion in the Pyrazinamideunresponsive to Monocyte:17%, metaphysis and (4-drug regimen)intravenous cloxacillin. Eosinophil:2%) diaphysis of for two monthsHistory of cough, fever and Both gastric washing distal end of right femur continued withTB was not detected in and Culture for acid along with periosteal 2 drugs given twiceother family members. fast bacilli:Negative reaction and soft tissue weekly for 7Temp:37.8o C Bone culture:Positive swelling months. After 5 months,tenderness and for M.Tuberculosis, there was completeswelling of right knee sensitive to INH and recovery and the patientwith white pus oozing Rifampin did not have difficulty infrom the medical aspect of walkingthe right knee joint (Fig. 1)

3 A 13 month old WBC:10300/mm3 CXR and thoracic Multiple caseous 4 drugs for 2 monthsIranian girl with swelling (PMN:61%, CT-scan: granulomas. (DOTS) and 2 drugsof the left wrist joint for 2 Lymphocyte:2%, Unremarkable Acid fast bacilli twice weekly for weeks. Monocyte:19%, Left wrist X-ray: were detected by 7 months.There was history of Eosinophil:18%) Single lytic lesion in the Ziehl-Neelsen Monthly serialrecent occasional fever PPD:15mm, distal metaphysic staining radiographies were taken.and poor weight gain. ESR:42mm/hr of radius After 5 months, the lyticTB was diagnosed in Both smear and lesion completelythe patient’s mother culture of gastric disappeared.about 8 months prior washing for acid fastto her admission. Physical bacilli:Negativeexamination revealed Bone culture: Positiveswelling and tenderness for acid fast bacilliof the left wrist joint.

4 A 16 month old Iranian WBC:Unremarkable CXR and thoracic Multiple caseous 4 drugs for 2 monthsboy with swelling and PPD:16mm, CT-scan: granulomas (DOTS) continuing withpain in the left wrist ESR:30mm/hr Mild hilar 2 drugs given twicejoint of 4 months, Bone culture: Positive lymphadenopathy weekly for 7 months.duration. There was for M.tuberculosis, Left wrist X-ray: After one month ofno history of TB in sensitive to INH and Single lytic lesion in therapy the lytic lesionother family members. Rifampin the distal showed signs ofPhysical examination metaphysis sclerosis. At the end ofdetected swelling of radius 6 months the lyticwithout any redness in (Fig. 2) lesion had completelythe left wrist joint and disappeared and theoccasional fever.Also, patient had full,mild degree of axillary symmetrical movementlymphadenopathy was in the left wrist jointdetected.

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Tuberculous Osteomyelitis of the Lung Bones and Joints

Indian Journal of Pediatrics, Volume 75—May, 2008 507

and weight loss were present in 50% of cases. Skeletaltuberculosis may go underrecognized for months to yearsbecause of the indolent nature of the infection and thelack of specific signs or symptoms.13,17 The adjacent jointmay become involved as the bone is destroyed bypressure necrosis and cold abscess formation. Damage tothe adjacent epiphyseal growth centers has been reportedto occur in upto 41% of cases of bone disease inmetaphyses are typically affected.15 Usually there isminimal periosteal reaction or new bone formationsurrounding the affected area.12 These X-ray features arenonspecific and may also be presented in conditions suchas inflammatory arthritis, pyogenic osteomyelitis,Brodie’s abscess, Kaposi’s sarcoma, and othermalignancies. Other findings on plain radiographsinclude soft tissue swelling and narrowing of the jointspace, cysts in bone adjacent to joints, and subchondralerosions.18, 19 Multiple cystic tuberculous lesions of the

skeleton, or Jungling disease, is a rare variety oftuberculosis reported extensively 30 to 40 years ago.19

The diagnosis of osteoarticular tuberculosis isconfirmed by isolation of mycobacterium tuberculosis froma skeletal site, usually from a bone or synovial biopsy.20 Indifferential diagnosis, some other organisms should beconsiderd and excluded by clinical and paraclinicalevaluations. Staphylococcus aureus, is the most commonorganism involved in osteomyelitis. Other types oforganisms include the salmonella bacteria in paitents withsickle cell anemia, and pseudomonas aeurginosa in drugaddicts. Extremely rarely, the viruses which causechickenpox and smallpox have been found to cause viralosteomyelitis.2, 13, 20

The gold standard for the diagnosis of osseoustuberculosis is culture of mycobacterium tuberculosis frombone tissues. Positive Ziehl-Neelsen staining for acid-fastbacilli requires at least 104 acid-fast bacilli per milliliter ofspecimen and does not differentiate between tuberculousand non-tuberculous mycobacteria.21 Ziehl-Neelsenstaining and culture of tissue obtained via surgical biopsyoffer the most direct approach to diagnosis. The advent ofDNA detection via PCR(polymerase chain reaction ) mayincrease sensitivity and allow for the exclusion of non-tuberculous mycobacteria (such as mycobacteriummarinum) that also cause soft tissue infections.12

Current recommendations for the treatment of osseoustuberculosis include a 2 month initial phase of isoniazid,rifampin, pyrazinamide, and ethambutol followed by a 6to 12 month regimen of isoniazid and rifampin.21

These cases illustrate the importance of a high index ofsuspicion when evaluating a patient with an unusualdestructive bone lesion, particularly in a susceptibleepidemiologic and clinical setting. Familiarity withvarious radiologic features of the disease is also veryimportant. Early biopsy and appropriate microbiologictesting can avoid diagnostic delay.Bone biopsy followedby microbiologic studies including culture and PCR aswell as pathologic examination are recommendedconfirmatory tools.

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Fig. 1. White pus-oozing lesion of right knee joint

Fig. 2. Single lytic lesion in the distal metaphysis of radius

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19. Zhang H, Jin D. Multiple cystic tuberculous lesions in theskeleton (Jungling disease). A Case Report. Spine 2003; 28 :E155-E157.

20. Rigaurd Mona, Borkowsky William. Tuberculosis inchildhood. In William N. Rom, Stuard M, eds. Gray Textbook ofTubrculosis, 2nd ed. Philadelphia; Lippincott Williams &Wilkins, 2004; 609-624.

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