Case Report

Intramedullary Tuberculoma Combined with Abscess: Case Report and Literature Review Jijun Liu, Haiping Zhang, Baorong He, Biao Wang, Xingbang Niu, Dingjun Hao

Key words Abscess - Diagnosis - Intramedullary tuberculoma - Treatment -

Abbreviations and Acronyms CNS: Central nervous system CT: Computed tomography MRI: Magnetic resonance imaging MDR-TB: Multidrug-resistant tuberculosis TB: Tuberculosis Department of Spine Surgery, Xi’an Jiaotong University Health Science Center, Honghui Hospital, Xi’an City, Shanxi Province, China To whom correspondence should be addressed: Dingjun Hao, M.D. [E-mail: [email protected]] Citation: World Neurosurg. (2016). http://dx.doi.org/10.1016/j.wneu.2016.01.021 Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2016 Elsevier Inc. All rights reserved.

INTRODUCTION Intramedullary spinal tuberculoma is a clinically rare disease, with the incidence of 2/100,000 of all tuberculosis and 0.2% of all central nervous system (CNS) tuberculosis.1 There are fewer reports on intramedullary spinal tuberculoma combined with intramedullary spinal abscess, which has lower incidence and could easily be misdiagnosed. We presented a case of a patient diagnosed with intramedullary spinal tuberculoma combined with pyogenic inflammation and reviewed the literature to discuss the disease management implications. CASE REPORT A 28-year-old male admitted to our hospital on 11 September, 2015 with a 6-month history of lower back pain followed by 2 months’ lower limb sensory and motor dysfunction. The lower back pain started 6 months ago without any obvious causes. It was aggravated after hard work and relieved with rest. The patient paid no attention to his symptoms. One month ago, the lumbar

- BACKGROUND:

Intramedullary spinal tuberculoma combined with abscess has low incidence and could easily be misdiagnosed. Given the rarity of spinal intramedullary tuberculoma, there is no standardized treatment protocol for this condition. We reported the case of a 28-year-old male who was diagnosed with intramedullary tuberculoma combined with abscess and treated with antituberculosis therapy followed by surgery.

- CASE

DESCRIPTION: A 28-year-old male was admitted to our hospital with lower back pain and lower limb sensory and motor dysfunction. The radiographic results indicated tuberculosis (TB). The patient was suggested to undergo anti-TB therapy and was later transferred to Tuberculosis Hospital for systemic treatment for 20 days. He was readmitted to our hospital because of aggravating syndromes including impaired superficial sensation below the T11 level, spastic paresis with muscle strength of 0/5 in both lower extremities, and exaggerated bilateral tendon reflexes. To alleviate the neurologic dysfunction, surgery was undertaken. The postoperative diagnosis was thoracic intramedullary TB combined with abscess. He reported marked improvement in lower limb motor and sensory function the day after surgery, and his muscle strength recovered to 3/5.

- CONCLUSIONS:

Although intramedullary TB combined with abscess is clinically rare, it should be taken into consideration when patients present with intramedullary space-occupying lesions with TB lesions elsewhere. Most patients respond well to the anti-TB therapy, but for those with severe spinal cord compression or those irresponsive to the drug therapy, surgical intervention could facilitate neurologic recovery and improve the prognosis.

symptoms were aggravated with marked lower limb weakness and numbness. The patient was admitted to our hospital and underwent thoracic and lumbar computed tomography (CT) and magnetic resonance imaging (MRI) examinations. The chest CT scan demonstrated infiltrative lesions at the bilateral apex of the lungs and lower lobe of the left lung (Figure 1), indicating tuberculosis (TB). The thoracic-enhancing MRI showed a ring-enhancing lesion with a clear border at the T11 level with the size of 1.0 cm  2.5 cm  1.0 cm (Figure 2). The patient was suggested to undergo anti-TB therapy and was transferred to the Tuberculosis Hospital for systemic treatment for 20 days. He was readmitted to our hospital 11 October, 2015 reporting aggravating bilateral lower limb weakness and marked motor and sensory dysfunction combined with urinary retention. Physical examination revealed

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decreased bilateral respiratory sound, impaired superficial sensation below T11 level, spastic paresis with muscle strength of 0/5 in both lower extremities, and exaggerated bilateral tendon reflexes. To alleviate the neurologic dysfunction, surgery was undertaken. After general anesthesia, intraspinal exploration was conducted, exposing rich vascular circulation on the surface of the spinal dura mater (Figure 3). Marked spinal thickening and edema could be seen after cutting into the spinal dura mater, and an incision was made along the sulcus medianus posterior medullae spinalis under the microscope to explore the spinal lesion, after which the tuberculoma was fully separated from the peripheral tissue (Figure 4). After making a longitudinal incision on the tuberculoma, thin pus could be witnessed to flow out from the tuberculoma

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CASE REPORT JIJUN LIU ET AL.

INTRAMEDULLARY TUBERCULOMA COMBINED WITH ABSCESS

Figure 1. Chest computed tomography: infiltrative lesions at bilateral apex of lungs without enlargement of mediastinal lymph nodes.

(Figure 5). The pathologic result revealed caseous necrosis at the center of tuberculoma (Figure 6). The postoperative diagnosis was thoracic intramedullary TB combined with abscess. The patient was prescribed with anti-TB and antibiotic therapy, and he reported marked improvement in lower limb motor and sensory

function the day after the surgery. His muscle strength recovered to 3/5. DISCUSSION Etiology and Pathogenesis Intramedullary tuberculoma is a rare kind of TB infecting the CNS, which has an

Figure 2. Thoracic magnetic resonance imaging: a ring-enhancing lesion at T11 with the size of 1.0 cm  2.5 cm  1.0 cm.

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incidence of 1:42 compared with brain tuberculoma,2 and only 7% of which could lead to spinal cord injury.3 Intramedullary tuberculoma combined with abscess is so clinically rare that only 77 cases have been reported in the literature since Hanci et al. first presented the condition in 1997.1,4,5 Of all the 77 cases, only 4 were caused by sole tubercle bacillus infection, while the rest were caused by mixed infection of tubercle bacillus and other pathogens.2,4 Intramedullary TB includes tuberculoma, spinal cord edema, cavity formation, and sometimes also spinal arachnoiditis. Intramedullary tuberculoma is mostly caused by active TB lesions spread via blood or brain TB spread via cerebrospinal fluid; it is rarely caused by direct spread of local spinal TB.6 In our case, the patient presented with an obvious pulmonary TB lesion combined with intramedullary tuberculoma. Clinical and Radiologic Characteristics Intramedullary tuberculoma is highly likely to occur in people 18 45 years old,7 with the youngest patient reported to be only 9 months old.8 In our case, the patient was 28 years old. It could occur at any segment, but it is mostly seen at the thoracic segment with the incidence of 72%,6,9 which in our case was T11. It often develops secondary to TB of other lesions, mostly pulmonary TB.10 11 MacDonnel et al. studied 18 cases of intramedullary tuberculoma and found that 69% of them were combined with pulmonary TB, while 38% of them were not found in any other TB lesion. According to their study, the patients had an average age of 28.6 years old and average disease duration of 2.3 months, with more females (63%) than males. Their main manifestations were subacute onset of spastic paraplegia (61%) and flaccid paraplegia (33%) due to spinal cord compression, including progressive weakness of lower extremities, sensory abnormity, urination and defecation disturbance, and systemic TB poisoning symptoms, namely light fever in the afternoon, night sweats, weight loss, and fatigue. The patient in our case presented progressive spinal cord compression symptoms after anti-TB therapy for around 20 days, including paraplegia and urination and defecation disturbance, which we considered to be

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CASE REPORT JIJUN LIU ET AL.

INTRAMEDULLARY TUBERCULOMA COMBINED WITH ABSCESS

also suggested to exclude possibly combined silent intracranial tuberculosis.16

Figure 3. Intraoperative image: rich vesicular circulation on the surface of the spinal dura mater.

due to complete transverse spinal cord injury caused by hematogenous spread of pulmonary TB. Although X-ray and CT scanning could help diagnose pulmonary TB and TB of other lesions, for spinal cord lesion, MRI is considered the best imaging test as it can precisely locate the lesions, demonstrate the size and number of the lesions, as well as detect degeneration and necrosis.12 To a certain extent, MRI could avoid some invasive procedures due to difficulty in diagnosis. In the early phase of intramedullary tuberculoma formation, MRI reveals homogenous enhancement; in

Figure 4. Fully separated tuberculoma and abscess.

the late phase with tuberculoma wall formation, MRI often reveals ring enhancement; and if caseous necrosis appears at the center of the lesion, a “target sign” can be detected,13e15 which is the typical sign of intramedullary tuberculoma and helps discriminate it from other intramedullary lesions.13 Furthermore, for patients with multiple lesions of intramedullary tuberculoma, brain MRI is

Figure 5. Pus flowing out from the tuberculoma.

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Diagnosis and Differential Diagnosis The diagnosis of intramedullary tuberculoma can be made on the basis of the clinical manifestations, radiologic results, cerebrospinal fluid test results, and antiTB therapy effects. For some patients requiring surgical intervention, direct diagnosis can be made with postoperative pathologic examination of the specimen. As the early neurologic manifestations of intramedullary tuberculoma are similar to those of other intramedullary lesions, missed diagnosis and misdiagnosis can be made. The differential diagnoses include tumors (astrocytoma, ependymoma, hemangioblastoma, lymphoma, and metastatic tumors), inflammatory lesions, demyelinating diseases (multiple sclerosis), vascular diseases (malformation, congestion), and granulomatous diseases (syphilis, abscess, fungus, parasites).17 Treatment No consensus has been made on the standard therapy of intramedullary tuberculoma combined with abscess. Considering the published literatures, most patients recovered well after anti-TB therapy.1,9 The patient in our study was with multidrug-resistant TB (MDR-TB), which explained his anti-TB failure in the Tuberculosis Hospital. The patient presented little improvement even after antiMDR-TB therapy and showed marked spinal cord compression symptoms. The aim of surgery approach toward intramedullary tuberculoma was to relieve the spinal cord compression and provide a specimen for postoperative pathologic diagnosis. Some scholars believed that surgical intervention before irreversible spinal cord injury could better facilitate neurologic function recovery,14 as MacDonnel et al.11 reported that 65% of the patients showed improvement after surgical intervention. However, Coholsey et al. believed that if radiologic results could only suggest increased spinal cord edema, surgery was not a necessity as it could be the normal reaction of anti-TB therapy.18 Whether surgical intervention should be set as a conventional approach to treat intramedullary tuberculoma is controversial. Surgery is highly recommended in the following

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9. Kayaoglu CR, Tuzun Y, Boga Z, Erdogan F, Gorguner M, Aydin IH. Intramedullary spinal tuberculoma: a case report. Spine (Phila Pa 1976). 2000;25:2265-2268. 10. Miyamoto J, Sasajima H, Owada K, Odake G, Mineura K. Spinal intramedullary tuberculoma requiring surgical treatment—case report. Neurol Med Chir (Tokyo). 2003;43:567-571. 11. MacDonnell AH, Baird RW, Bronze MS. Intramedullary tuberculomas of the spinal cord: case report and review. Rev Infect Dis. 1990;12:432-439. 12. Hristea A, Constantinescu RV, Exergian F, Arama V, Besleaga M, Tanasescu R. Paraplegia due to non-osseous spinal tuberculosis: report of three cases and review of the literature. Int J Infect Dis. 2008;12:425-429.

Figure 6. Postoperative pathologic result: caseous necrosis at the center of tuberculoma, with a few Langhans cells, lymphocytes, fibroblasts, and many neutrophils.

13. Gupta VK, Sharma BS, Khosla VK. Intramedullary tuberculoma: report of two cases with MRI findings. Surg Neurol. 1995;44:241-243 [discussion: 3-4]. 14. Kioumehr F, Dadsetan MR, Rooholamini SA, Au A. Central nervous system tuberculosis: MRI. Neuroradiology. 1994;36:93-96.

situations: 1) with marked signs of spinal cord compression; 2) with indefinite diagnosis; and 3) with deteriorating symptoms or enlarging lesions after full anti-TB therapy.8,10,19 In our case, according to the neurology consultation, the patient did not respond well to the conservative treatment and he presented marked neurologic disturbances, indicating that timely surgical intervention would be necessary for patients with severe spinal cord compression. Our patient showed prominent muscle strength recovery the day after surgery. As intramedullary TB combined with abscess belongs to systemic TB, postoperative systemic anti-TB therapy and antibiotic therapy are needed to prevent TB spread.10 In conclusion, even though intramedullary TB combined with abscess is clinically rare, it should be taken into consideration when patients present with intramedullary space-occupying lesions with TB lesions elsewhere. Most patients respond well to the anti-TB therapy, while for patients with severe spinal cord compression or who are irresponsive to drug therapy, surgical intervention could facilitate neurologic recovery and improve the prognosis. As only a few cases of intramedullary TB combined with abscess were reported, conventional surgical

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intervention cannot be generalized and more prospective studies are necessary.

15. Ratliff JK, Connolly ES. Intramedullary tuberculoma of the spinal cord. Case report and review of the literature. J Neurosurg. 1999;90(suppl 1): 125-128.

REFERENCES 1. Citow JS, Ammirati M. Intramedullary tuberculoma of the spinal cord: case report. Neurosurgery. 1994;35:327-330. 2. Nussbaum ES, Rockswold GL, Bergman TA, Erickson DL, Seljeskog EL. Spinal tuberculosis: a diagnostic and management challenge. J Neurosurg. 1995;83:243-247. 3. Muthukumar N, Venkatesh G, Senthilbabu S, Rajbaskar R. Surgery for intramedullary tuberculoma of the spinal cord: report of 2 cases. Surg Neurol. 2006;66:69-74. 4. DiTullio MV Jr. Intramedullary spinal abscess: a case report with a review of 53 previously described cases. Surg Neurol. 1977;7:351-354. 5. Devi BI, Chandra S, Mongia S, Chandramouli BA, Sastry KV, Shankar SK. Spinal intramedullary tuberculoma and abscess: a rare cause of paraparesis. Neurol India. 2002;50:494-496. 6. Lu M. Imaging diagnosis of spinal intramedullary tuberculoma: case reports and literature review. J Spinal Cord Med. 2010;33:159-162. 7. Sharma MC, Arora R, Deol PS, Mahapatra AK, Sinha AK, Sarkar C. Intramedullary tuberculoma of the spinal cord: a series of 10 cases. Clin Neurol Neurosurg. 2002;104:279-284.

16. Park HS, Song YJ. Multiple tuberculoma involving the brain and spinal cord in a patient with miliary pulmonary tuberculosis. J Korean Neurosurg Soc. 2008;44:36-39. 17. Gupta RK, Pandey R, Khan EM, Mittal P, Gujral RB, Chhabra DK. Intracranial tuberculomas: MRI signal intensity correlation with histopathology and localised proton spectroscopy. Magn Reson Imaging. 1993;11(3):443-449. 18. Choksey MS, Powell M, Gibb WR, Casey AT, Geddes JF. A conus tuberculoma mimicking an intramedullary tumour: a case report and review of the literature. Br J Neurosurg. 1989;3:117-121. 19. Rao GP. Spinal intramedullary tuberculous lesion: medical management. Report of four cases. J Neurosurg. 2000;93(suppl 1):137-141.

Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 10 December 2015; accepted 4 January 2016 Citation: World Neurosurg. (2016). http://dx.doi.org/10.1016/j.wneu.2016.01.021 Journal homepage: www.WORLDNEUROSURGERY.org

8. Chagla AS, Udayakumaran S, Balasubramaniam S. Cervical intramedullary tuberculoma in an infant. Case illustration. J Neurosurg. 2007;106(suppl 3): 243.

Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2016 Elsevier Inc. All rights reserved.

WORLD NEUROSURGERY, http://dx.doi.org/10.1016/j.wneu.2016.01.021

Intramedullary Tuberculoma Combined with Abscess: Case Report and Literature Review.

Intramedullary spinal tuberculoma combined with abscess has low incidence and could easily be misdiagnosed. Given the rarity of spinal intramedullary ...
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