Agrawal, Amit MCh*; Lakhkar, Bhavana B. MD†; Lakhkar, Bhushan MD‡; Agrawal, Anil MD§; Shahpurkar, Vinay V. MS*; Jagzape, Tushar MD†; Lokhare, Amol MD†
Departments of *Surgery, †Pediatrics, ‡Radiology, and §Pathology, Datta Meghe Institute of Medical Sciences, Wardha, Maharashtra, India.
Address correspondence and reprint requests to Amit Agrawal, MD, Division of Neurosurgery, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha 442004, Maharashtra, India. E-mail: firstname.lastname@example.org.
Effective antituberculosis treatment has reduced the incidence of tuberculosis in industrialized countries. Still tubercular infection poses a major challenge in developing countries, and also, there is resurgence of the disease in developed countries, particularly in immigrant population and in patients with HIV infections.1 The multifocal form of skeletal tuberculosis may occur as a result of dissemination from a pulmonary or an osseous focus and is exceptional, even in countries where the disease is endemic.2,3 We present a case of multifocal musculoskeletal tuberculosis in a nonimmunocompromised child involving multiple sites and review the relevant literature.
A 14-year-male child presented with a history of swelling over the anterior chest wall on the left side 1.5 years ago for which incision and drainage was performed in an outside hospital. The lesion did not heal, and the boy developed a nonhealing sinus at the incision site (Fig. 1). Ten days later, he developed another swelling over the back on the left side; again, incision and drainage was performed, and he developed a nonhealing sinus (Fig. 1). Similarly, he developed one more swelling in the left palm, and incision anddrainage was performed followed by development of a non-foul-smelling pus-discharging sinus (Fig. 1). For the last 1 month, he noticed swelling over the back and lumbar region associated with localized back pain (Fig. 2). It was associated with low-grade fever with rise of temperature in the evening. There was associated history of significant weight loss and appetite. On examination, he was thin built and had generalized wasting. There was mild pallor, and multiple nontender, firm, mobile lymph nodes with overlying healthy skin were palpable in the left axilla. There was no hepatosplenomegaly. There was no gibbus or palpable deformity of the spine; however, he had a mild scoliosis with convexity to the right side. There was no objective evidence of any neurological deficit, and other systems and anatomic structures including the chest were normal. Hemoglobin was 7.9 g/dL with microcytic hypochromic anemia and anisocytosis. Erythrocyte sedimentation rate was 80 mm in the first hour. Sputum for cid-fast bacillus was negative. Chest radiograph did not show abnormalities. Test for HIV was negative. There was no history of tuberculosis in the family. Ultrasound abdomen showed psoas abscess. Radiograph of the dorsal spine showed paraspinal abscess and lytic lesion involving the D10 vertebral body (Fig. 3). Radiograph and computed tomographic (CT) scan of the left hand showed a lytic lesion involving the fourth metacarpal (Fig. 4). Computed tomographic scan of the hand showed erosion of the left fourth rib posteriorly (Fig. 5). Computed tomographic scan dorsal spine showed D10 body destruction with paraspinal abscess (Fig. 6). Computed tomographic scan of the lumbar spine showed psoas abscess coming to the surface posteriorly (Fig. 7). With all these findings, a diagnosis of tuberculosis involving multiple sites was suspected. The patient was put in prone position, and drainage of paraspinal and psoas abscess through single incision was performed. Left D10 costotransversectomy was performed, and paraspinal abscess was drained. In the lower part where the psoas abscess was coming near to the dorsal surface below the fascia, an incision was made, and about 150-mL-thick yellowish non-foul-smelling pus was drained. Histopathologic examination of the material on which biopsy was performed revealed granulomatous inflammation with caseous necrosis, infiltration of epithelioid cells, and Langerhans giant cell granulomas, compatible with tuberculosis (Fig. 8). Antituberculous therapy was started with (isoniazid, rifampin, pyrazinamide, and ethambutol), and at 8 months' follow-up, he is doing well.
The skeletal system is the most common extrapulmonary site for tuberculous infection with an incidence ranging from 1% to 10%.4 Spine is the most common involved site (50%) followed by hip or knee (30%), and less commonly, the pubis, wrist, shoulder, and sacroiliac joint.5 As in the present case, the thoracolumbar junction is the most commonly involved site, but many levels may be involved at presentation.6 As in our case also, features of spinal tuberculosis that can be seen on CT scans include anterior vertebral body destruction, vertebral body collapse, disk space narrowing, and large paraspinal soft-tissue masses representing abscess formation (Figs. 5-7).7
Paraspinal abscess formation is seen in as many as 71% to 75% cases of spinal tuberculosis.4,8 Paraspinal abscess develops secondary to destruction of the cortical bone and elevation of the periosteum.9 Paraspinal abscess formation may be detected on plain radiographs as areas of fusiform soft-tissue swelling around the spine (Fig. 3).10
Focal rib destruction in pulmonary tuberculosis is uncommon, and in the literature, the incidence varies from 0% to 5%.11-13 Involvement of ribs in tuberculous may occur either by direct extension from a pleuropulmonary tuberculous lesion or from hematogenous spread from a distant focus.11 Clinically, these patients present with subcutaneous swelling in the chest wall.13 On CT scan, rib lesions are seen as juxtacostal soft-tissue mass with central low attenuation and peripheral rim enhancement with or without bone destruction.12 Tubercular rib lesions can heal with antituberculosis drug therapy11; however, rib resection may be needed in recurrent cases.13
Due to the proximity of the iliopsoas compartment to the dorsolumbar spine, tubercular infection of the spine can spread into the iliopsoas compartment, pelvis, and thigh, leading to abscess formation.9 The inflammatory masscan penetrate the periosteum leading to a psoas abscess.1 Treatment of psoas abscess can be either conservative (drug therapy, immobilization, external brace application) or surgical intervention (in conjunction with drug therapy)4,14 including percutaneous drainage under radiological guidance.15
Tuberculous involvement of the metacarpals and phalanges is a rare presentation of extrapulmonary tuberculosis and can be difficult to diagnose during the early stages.16 Overlying finger swelling, although rare, can be the manifestation of metacarpal tuberculosis.17 In our case, in addition to the swelling, the patient later on developed the sinus in the palm over the affected metacarpal (Figs. 1, 4). In isolated lesions of metacarpals, an unusual destructive bony lesion should warrant early biopsy and appropriate microbiologic testing to avoid diagnostic delay and followed by appropriate treatment.16,17
The differential diagnoses of osteolytic lesions of tuberculosis at multiple sites include multiple myeloma, secondary metastasis, bacterial osteomyelitis, and less commonly gout, actinomycosis, coccidioidomycosis, maduromycosis, cryptococcosis, and syphilis.18 In musculoskeletal tuberculosis, active lung infection is present in less than 50% of cases.19 In our case, the chest radiograph did not show abnormalities. Although a positive Mantoux test and elevated erythrocyte sedimentation rate give important diagnostic clue but may be negative in 10% of patients.20 Histopathologic examination yields a high percentage of positive results, and the use of histopathology, culture, and guinea pig inoculation together may confirm the diagnosis in all of the cases of skeletal tuberculosis and help to differentiate skeletal tuberculosis from other infections or neoplasm.18,19 Computerized tomography is a useful adjunct in demonstrating the skeletal lesion and delineating the extent of disease, and also, it may be used in the assessment of response to treatment.11 Multiplanar imaging, especially magnetic resonance imaging with gadolinium enhancement, has become the standard modality for defining subtle discovertebral lesions and in detecting unsuspected paravertebral soft-tissue extension, especially in patients with epidural and intradural infections and neurological symptoms.9 Because of financial constraints, however, it may not always be possible to perform these tests in developing countries, and we used the clinical response of the patient as the guideline for response to treatment.
There can be considerable delay between presentation and diagnosis, mainly due to its insidious onset, nonspecific clinical picture, and low index of clinical suspicion even in areas where the disease is endemic. This case report probably represents a missed diagnosis. If Mantoux test had been performed initially, when the patient developed a sinus after initial drainage of the swelling over the back, probably the diagnosis would have been suspected at a much earlier stage, and early treatment with antituberculous therapy would have avoided the need for surgery.
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