Neurol Med Chir (Tokyo) 43, 567¿571, 2003

Spinal Intramedullary Tuberculoma Requiring
Surgical Treatment
—Case Report—
Junichi MIYAMOTO, Hiroyasu SASAJIMA, Kei OWADA, Genya ODAKE*,
and Katsuyoshi MINEURA
Department of Neurosurgery, Kyoto Prefectural University Graduate School of Medicine, Kyoto;
*Department of Neurosurgery, Osaka Dai-Ichi Hospital, Osaka

Abstract
A 71-year-old male presented with multiple central nervous system tuberculomas including spinal
intramedullary tuberculoma manifesting as occipitalgia and left hemiparesis. He had received medical
treatment for lung and testis tuberculosis. Magnetic resonance imaging revealed an intramedullary
lesion at the C-2 level as well as multiple small extramedullary and intracranial lesions. His neurological symptoms gradually worsened despite intensive antituberculous therapy. The C-2 intramedullary
lesion responsible for left hemiparesis was surgically extirpated. Postoperatively, his neurological
symptoms improved gradually, and no recurrence was evident at the resected site. Surgical intervention
is mandatory in patients with intramedullary tuberculoma if neurological symptoms deteriorate or
lesions enlarge despite continuous antituberculous therapy.
Key words:

tuberculoma,

intramedullary lesion,

surgical treatment

Introduction

fied in the lung. Antituberculous therapy was
instituted using the combination of isonicotinoil
hydraradin (INH) 300 mg/day, rifanpicin (RFP)
450 mg/day, and streptomycin (SM) 150 mg/day.
Magnetic resonance (MR) imaging revealed multiple
lesions in the intraspinal and intracranial spaces,
and the patient was referred to our department.
On admission, he was clear and alert. The patient
could not walk and suffered from vesicorectal disturbance. The muscle power of the upper and lower
extremities was fair (grade 3 by the manual muscle
test ranging from grade 0 to grade 5). No signs of
meningeal irritation were present. Laboratory
examination of blood showed no abnormalities.
Mycobacterium tuberculosis was not detected by
sputum or cerebrospinal fluid (CSF) cultures. Chest
radiography revealed miliary tuberculosis in the
bilateral lungs. Vertebral radiography showed
compressed fracture of the vertebral bodies at the
T-11, T-12, and L-5 levels. MR imaging revealed a
ring-enhanced intramedullary lesion (approximately
3 × 3 × 3 cm) at the C-2 level (Fig. 1) and multiple
small extramedullary lesions (approximately 0.1 cm
maximum diameter) at the T-10 to L-2 levels. Enhanced lesions (less than 0.5 cm maximum diameter)
were also scattered in the vermis and the cerebral

Tuberculous infections involving the central nervous system (CNS) usually occur secondary to primary infectious foci in the lung or elsewhere in the
body. Therefore, the incidence is closely associated
with that of tuberculosis. Very recently, the number
of patients with tuberculosis has increased, partly
because of various immunocompromised conditions
and the emergence of drug-resistant tuberculosis.16)
CNS tuberculosis should be considered in the
differential diagnosis of lesions causing neurological signs and symptoms. We report a case of intramedullary tuberculoma which progressed despite
medical treatment.

Case Report
A 71-year-old male presented with occipitalgia,
cervical pain, lumbago, and motor weakness on the
left 24 weeks after the initiation of medical
treatment for systemic tuberculosis. Previously, the
patient had undergone removal of left testis tuberculoma. At that time, miliary tuberculosis was identiReceived

February 3, 2003;

Accepted

June 18, 2003

567

568

J. Miyamoto et al.

parenchyma (Fig. 2). Thallium-201 chloride scintigraphy showed no abnormal uptake, suggesting
non-tumorous lesions. The weakness of the left
upper and lower limbs had deteriorated despite
continuation of antituberculous medication for 24
weeks.
The patient underwent removal of the C-2 lesion
through a suboccipital craniotomy and laminectomy
between the C-1 and C-3 levels. The arachnoid membrane was thickened and gray, and the spinal cord
was swollen. Intraoperative echoencephalography
was used to delineate the lesion. The lesion was then
dissected from the surrounding spinal cord tissue
through a midline myelotomy. The lesion was
whitish and soft, and the margin was sharply de-

fined (Fig. 3). The center of the granuloma was
necrotic and contained a creamy substance. The
surrounding rim was rather fibrous. The tumor was
extirpated en bloc after internal decompression
using an ultrasonic surgical aspirator.
Histological examination of the surgical specimen
showed inflammatory and necrotic tissue containing a number of infiltrated lymphocytes. No caseation necrosis or Langhans'-type giant cells were
found. Ziehl-Neelsen staining of the resected sample
identified M. tuberculosis (Fig. 4).
The weakness of the upper and lower limbs
improved one month after the surgery, and the
patient could walk to some extent with assistance.
Antituberculous therapy with INH and RFP was
continued for one year. Follow-up MR imaging
revealed no evidence of recurrence or regrowth at
the resected site. The other extramedullary and
intracranial lesions had also disappeared (Fig. 5).

Discussion
Spinal tuberculosis causes various types of lesions.
Pott's disease of the spine is the most common

Fig. 1

Sagittal T1-weighted magnetic resonance
image with gadolinium revealing a ringenhanced intramedullary lesion at the C-2
level.

Fig. 2

T1-weighted magnetic resonance images
with gadolinium revealing multiple tiny
lesions in the vermis (left) and the cerebral
hemisphere (right).

"

Fig. 3

Intraoperative
photographs
showing swelling of the spinal
cord (left). The lesion consists
of gray soft tissue, and contains white discharge (right).

Neurol Med Chir (Tokyo) 43, November, 2003

Intramedullary Tuberculoma
(64%), followed by arachnoiditis (20%) and intramedullary lesion (8%).12) Intramedullary lesion is
present in approximately two of every 100,000 tuberculosis cases and two of every 1,000 cases of CNS
tuberculosis.2) Intramedullary tuberculoma may be
difficult to differentiate from space-occupying
lesions such as primary and metastatic spinal
tumors and other chronic granulomatous diseases
(sarcoidosis, brucellosis, and histiocytosis).8)
The present patient had multiple lesions in the
cerebellum and the cerebrum as well as in the spine.

Intracranial tuberculomas may occur as single
lesions, but often multiple with a predilection for the
cerebellum. Seventeen intramedullary tuberculomas
including the present case have been described
recently, eight lesions (47%) located in the cervical
cord, eight lesions (47%) in the thoracic cord, and
one lesion (6%) in the conus (Table 1).2,3,5,6,9,10,12,14,15)

Fig. 5
Fig. 4

Table 1

Photomicrograph showing a number of
Mycobacterium
tuberculosis
(arrows).
Ziehl-Neelsen stain, ×400.

569

Sagittal T1-weighted magnetic resonance
image with gadolinium revealing no recurrence of the C-2 level lesion or other extramedullary lesions one year after surgery.

Recent cases of intramedullary tuberculoma

Case
No.

Author (Year)

Age
(yrs)

Sex

Level

Time to
surgery (wks)

Period of
medication
(wks)

Treatment

Result

1
2
3
4
5
6
7
8
9
10
11
12
13
14
15

Rhoton et al. (1988)15)
Jena et al. (1991)5)

65
25
23
31
36
35
27
46
65
25
16
14
16
2
71

M
F
M
M
M
M
M
F
M
F
F
M
M
F
M

C3–6
C2–6
C2–T1
T-11
T6–12
T10–11
conus
T10–11
C-4
T11–12
C5–7
C5–6, T-6
C1–6
T6–10
C-2, T10–L2

8
not described
not described
6
20
24
72
72
10
not described
not described
not described
not described
48
24

24
24
24
48
72
72
24
not described
24
72
32
32
48
96
48

total removal
medication
medication
total removal
partial removal
total removal
not described
biopsy
total removal
medication
medication
medication
medication
partial removal
total removal

recurrent*
improvement
improvement
improvement
improvement
improvement
improvement
improvement
improvement
improvement
improvement
improvement
improvement
improvement
improvement

Citow and Ammirati (1994)2)
Lin et al. (1994)9)
Gupta et al. (1995)3)
Ratliff and Connolly (1999)14)
Kayaoglu et al. (2000)6)
Rao (2000)12)

Nomura et al. (2001)10)
Present case

*Clinical symptoms improved after reoperation.

Neurol Med Chir (Tokyo) 43, November, 2003

570

J. Miyamoto et al.

Intramedullary tuberculomas mostly develop in the
upper spine. This preference may be related to the
proximity to the cerebellum. The presence of other
CNS lesions should be considered even if the
tuberculoma is apparently solitary.
The early diagnosis of intramedullary tuberculoma may be easier if CNS tuberculosis occurs
secondary to or during the treatment of primary
tuberculosis. In contrast, the diagnosis is very
difficult when CNS tuberculosis manifests prior to
pulmonary and extra-CNS lesions. CSF examinations are usually helpful to identify CNS tuberculosis, but did not detect M. tuberculosis in the present
case. CSF findings and culture may be modified
or underestimated during medical treatment.
CNS tuberculosis is increasingly common in immunocompromised patients. In the present case,
human immunodeficiency virus examination was
negative. CNS tuberculosis should be suspected in a
patient with a history of tuberculosis. The diagnosis
of CNS tuberculosis should be established based on
multidisciplinary detection of neurological symptoms and signs, laboratory data including CSF
examination, and neuroimaging findings.
MR imaging is a powerful method for the detection of intramedullary tuberculomas as well as
intracranial tuberculomas. MR imaging findings
vary among the stages of the tuberculoma formation.
In the early phase, tuberculomas are homogeneously
enhanced. Later, when the capsule is formed, ring
enhancement is common. Caseation results in the
``target sign'' appearance.3,7,14) Most of the intracranial and intraspinal lesions in the present case
were approximately 0.1 cm in maximum diameter
and homogeneously enhanced, whereas the largest
lesion at the C-2 level showed a ring enhancement
without target sign. Histological examination
proved that the C-2 lesion had stayed at the stage of
capsular formation with degenerative content. The
degenerative content may have been the result of
chemotherapy. However, the presence of degenerative content may prevent agents from penetrating
into the tuberculoma, whereas agents can easily pass
through the lesion due to the abundant vascular network in the initial stages of tuberculoma formation.
Persistent cyst and degenerative content during the
time of chemotherapy may be an indicator of drug
resistance in tuberculomas.
The choice of treatment mode and regimen is an
important consideration. Antituberculous agents
are widely used and are critical in the treatment of
intramedullary tuberculoma. INH and pyrazinamide
(PZA) are used frequently as effective chemotherapeutic agents against CNS tuberculosis because of
their high blood-brain barrier permeability.13) A

regimen of antituberculous chemotherapy should
continue until clinical and laboratory effectiveness
is achieved for 52 weeks (12 months) or more, and
consist of at least two agents to overcome drug
resistance. Addition of a third agent for the initial 8
to 24 weeks is also recommended.8,11) Recent guidelines for the treatment of tuberculous meningitis
advise initial use of a combination of INH, RFP, and
PZA, and, if necessary, addition of SM or ethambutol, although these carry the risk of side effects
and have poor penetration through blood-brain
barrier.1)
Progressive neurological deficits indicated surgical intervention in the present case. Drug resistance
to chemotherapeutic agents is a major problem in
the clinical failure of chemotherapy for tuberculosis.
In reported series, progressive neurological deficits
or enlarging lesions despite antituberculous treatment were the indicators for surgical intervention.12)
At present, selective application and combination of
existing drugs are required to enhance therapeutic
effectiveness.
The importance of surgical treatment for intramedullary tuberculoma extends to the diagnostic
procedure, curative procedure, and palliation.
Advances in the molecular diagnosis of sputum or
CSF and neuroimaging have reduced the importance of surgical diagnostic procedures. Curative
surgical procedures are less effective for intraspinal
and intracranial multiple tuberculomas. Palliative
surgical procedures can reduce the volume of tuberculoma causing symptoms, which will promptly
ameliorate the symptoms and provide the general
condition and sufficient time for ensuring effective
chemotherapy. In the present case, the palliative
surgical procedure was effective against the lesion
responsible for the neurological deficits among
multiple tuberculomas. Furthermore, debulking of
tuberculoma growing during or after chemotherapy
may enhance the effectiveness of chemotherapy by
reducing the content of hypoxic, necrotic, or drugresistant tissue.
Surgical intervention for intracranial tuberculoma is indicated if the medical therapy is ineffective
after 8 weeks.4) In eight surgical cases of intramedullary tuberculomas, the mean time between onset
and surgery was 31.5 ± 26.1 weeks. Total removal
was performed in five cases, partial removal in two
cases, and biopsy in one case. Most patients improved clinically regardless of the extent of resection. Neurological dysfunction attributable to tuberculoma can be improved by prompt and adequate
resection if the spinal tissue remains viable or can be
restored (Table 1).
Postoperative medical management of in-

Neurol Med Chir (Tokyo) 43, November, 2003

Intramedullary Tuberculoma
tramedullary tuberculoma is necessary, because
this disease is usually secondary to pulmonary or
extra-CNS infections. Methods to determine the
time to discontinue medication have not been established. In most cases, postoperative administration
of antituberculous agents was continued for 51.0 ±
26.6 weeks in recent cases. In the present case,
postoperative medication for approximately one
year has controlled the lesion including the resected
site. Practical and clinically useful determinations
of sensitivity or resistance to agents is needed to
maximize the surgical benefits in patients with
intramedullary tuberculoma.

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Address reprint requests to: J. Miyamoto, M.D., Department of Neurosurgery, Kyoto Prefectural University
Graduate School of Medicine, 465, Hirokoji,
Kawaramachi, Kamigyo–ku, Kyoto 602–8566, Japan.
e-mail: miyamo＠koto.kpu-m.ac.jp