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Isolated meningeal tuberculoma mimicking meningioma: Case report
ELSEVIER
Infection
ISOLATED MENINGEAL TUBERCULOMA MIMICKING MENINGIOMA: CASE REPORT A. Lindner, M.D., Ch. Schneider, M.D., E. Hofmann, M.D., N. Soerensen, M.D., and K.V. Toyka, M.D. Neurologische Klinik, Abt. fuer Neuroradiologie und Neurochirurgische Klinik, Abt. fuer Paed. Neurochirurgie Julius, Maximilians, Universitaet, Wuerzburg, Germany
Lindner A, Schneider CH, Hofmann E, Soerensen N, Toyka KV. Isolated meningeal tuberculoma mimicking meningioma: Case report. Surg Neurol 1995;43:81-4. A 47-year-old German female patient is described with intracranial tuberculoma of the dura mater without his-
tory or evidence of tuberculosis and a negative tuberculin reaction. Computed tomography and magnetic resonance imaging and angiography resembled a meningioma. Diagnosis was verified by histology and identification of mycobacterium tuberculosis. After removal of the granuloma and highdose chemotherapy, the patient was free of disease. Tuberculoma of the dura mater is an extremely rare disease in the Western European population. KEY WORDS
Intracranial tuberculoma,
treatment.
T
he incidence of intracranial tuberculoma became less than one in 10,000 cases with intracerebral space-occupying lesions when tuberculosis declined in the general population [ 111. In contrast, intracranial tuberculomas are common in developing countries with figures ranging from 20%30% in India [ 7,141.
CASEREPORT We report a 47-year-old German patient with a tuberculoma of the dura mater with no previous history of tuberculosis or increased risk of infection. The right-handed patient was seen because of short spells when she was unable to speak and noted hypesthesia and paresthesia in the base of her tongue and in the left perioral face. This was interpreted as indicating transient ischemic attacks. On computed tomography (CT) scanning, a large right-hemispheric lesion was identified in the tempo ral lobe contrasting with the mild clinical signs. Address reprint requests to: K.V. Toyka, M.D., Neurologische Klinik der Unversitaet, Josef Schneider, Str. 11, D-97080 Wuerzburg, Germany. Received May 14, 1994; accepted August 22, 1994. 0 1995 by Elsevier Science Inc. 655 Avenue of the Americas, New York, NY 10010
On admission 2 weeks later she appeared perfectly normal except for marginal weakness of the perioral muscles and mild increase in tendon reflexes on the left. The tuberculin skin reaction (tine test) was negative and became positive only 10 weeks later when the diagnosis had already been made. Fundoscopy was entirely normal. The sedimentation rate was 5 per hour and the blood smear was within normal limits. Cerebrospinal fluid (CSF) was obtained by lumbar puncture and showed mild pleocytosis (11 mononuclear cells per microliter), whereas total protein, albumin, and IgG including the IgG-index were within normal limits. No bacteria could be grown on appropriate cultures. Repeated microbiologic examinations including gastric and bronchial lavage did not reveal mycobacterium tuberculosis. X-ray and ultrasonic examinations of organs prone to develop typical primary lesions were unrevealing. CT SCANNING AND MAGNETIC RESONANCE IMAGING (MRI) On axial CT scanning a hyperdense lesion was seen on the left temporal lobe extending to the basal ganglia indicating edema. After contrast enhancement a lesion contingent with the skull was seen (not shown). On MRI using a 1, 5 Tesla Siemens Magnetom, an extensive hemispheric hyperintense lesion was seen extending into the white matter and a rim at the subdural space sparing the area where the hyperdense CT lesion was located (Figure 1). All lesions showed contrast enhancement after gadolinium injection (Figure 2). On digital subtraction angiography the leptomeningial vessels of the middle cerebral artery showed displacement from the skull at the side of the CT lesion. With a superselective catheter, a faint flush like appearance was seen below the dura (not shown). After opening of the dura, a solid extracerebral 0090-3019/95/$9.50 SSDI 0090-3019(94)00122-7
82
Surg Neurol
Lindner et al
1995;43:81-4
In the strong T,-weighted MRI, a marked edema of the left temporal cortex and white matter can be seen reaching to the basal ganglia. The thickened dura has a low signal. There is a mild midline shift to the right.
mass was removed extending down into the hemisphere. Neuropathologic examination revealed granulomatous tissue with scattered Langerhans giant cells but no bacilli. Extracts of the material were cultured and injected into laboratory rabbits. After 4 weeks, mycobacterium tuberculosis was identified in both test systems.
TREATMENT The patient was treated with high-dose intravenous streptomycin, myambutol, and rifampicin and subsequently with oral myambutol and rifampicin. There were no recurrences of initial episodes up to the observation period (31 months). Repeated CT and MRI scan 5 months after the operation showed no further lesion (not shown).
DISCUSSION This patient presented with short spells that can be interpreted as transient ischemic attacks or focal seizures with only very mild deficits. These can be
The coronal T,-weighted MFClshows a thickened left temporal dura in the region of the sylvian fissure, buckling toward the cortex from which it is sharply demarcated. After gadolinium-GDPA, there was a strong enhancement of the lesion and of the left temporoparietal dura.
localized to the precentral and postcentral cortex. Because of the initial CT findings, the tentative diagnosis of an unusual intracranial tumor was made originating from the skull or the meninges. Surprisingly, on operation a granulomatous tumor was found suggesting sarcoid granuloma or tuberculoma. The patient went into complete remission after surgical and antibacterial treatment within 6 months. Symptoms and signs of the cerebral tuberculoma include increased intracranial pressure with papillary edema and seizures in more than half of the patients [3,7,12,16]. It is not uncommon that these patients have few or no symptoms indicating general disease such as fever or malaise [ 16,171. The cutaneous tuberculin reaction is reported positive in 25%-75% of patients [ 171. CSF findings include mild elevation of protein. There are reports showing the typical appearance of intracranial tuberculomas on CT scans. Early lesions are hypodense or isodense and take up contrast material. Older lesions show focal cal-
Isolated
Meningeal
Surg Neurol 1995;43:81-4
Tuberculoma
cification and may take up contrast the margin [4,5,13].
medium
only at
MRf became available only recently for the identification of intracerebral tuberculoma with a typical isointense lesion in the T, image surrounded by hyperintense edema. Hypointense areas within the lesion indicate calcification or central calcification. Gadolinium enhancement provides a similar pattern to CT enhancement [6,8,15]. The distribution of intracerebral tuberculoma is quite variable with most tuberculomas located in the frontal and parietal lobe or at the base of the skull [9,10,12,13,18]. According to Dastur (1965), dural tuberculoma is a rare event even in patients from India where systemic tuberculosis is still common. It is even more unlikely to occur in central Europe. The clinical differential diagnosis includes a number of intracranial space-occupying lesionsmeningioma being the most likely, as was first thought present in our patient. To our knowledge there is only one previous case report on an isolated tuberculoma of the dura mater [ 11. This observation leads us to suggest that a careful pathology and microbiologic examination is essential in establishing the diagnosis of this treatable condition. REFERENCES tuberculo1. Anderson JM, MacMillan JJ. Intracranial ma-an increasing problem in Britain. J Neurol Neurosurg Psychiatry 1975;38:194-201. 2. Arseni C. Two hundred and one cases of intracranial tuberculoma treated surgically. J Neurol Neurosurg Psychiatry 195&21:308-l 1. 3. Arsenjo A, Valladares H, Fierro J. Tuberculomas of the brain. Arch Neurol Psychiatry 1951;65:146-59. 4. Bullock MRR, Welchman JM. Diagnostic and prognostic features of tuberculous meningitis on CT scanning. J Neurol Neurosurg Psychiatry 1982;45:1098-1101. 5. Casselmann ES, Hasso AN, Ashwal S, Schneider S. Computed tomography of tuberculous meningitis in infants and children. J Comput Assist Tomog 1980;4: 211-6. 6. Chang KH, Han MH, Roh JK, Kin IO, Han MC, Kim CW. Gd-DTPA-enhanced MR imaging in intracranial tuberculosis. Neuroradiology 1990;32:19-25. 7. Dastur HM, Desai AD. A comparative study of brain tuberculomas and gliomas based upon 107 case reports of each. Brain 1965;88:375-96. 8. Davidson HD, Steiner RE. Magnetic resonance imaging in infections of the central nervous system. AINR 1988;6:499-504. 9. Dyk Van A. CT of intracranial tuberculomas with spe cific reference to the “target sign.” Neuroradiology 1988;30:329-36. 10. Jinkina JR. Dynamic CT of the tuberculous meningal reactions. Neuroradiology 1987;29:343-7.
83
RS. Tuberculomas of the brain in 11. Maurice-Williams Britain, J Postgrad Med 1972;48:678-81. 12. Mayers MM, Kaufmann DM, Miller MH. Recent cases of intracranial tuberculomas. Neurology (NY) 1978; 28:256-60. 13. Price HJ, Danziger A. Computed tomography in cranial tuberculosis. Am J Roentgen01 1978;130:769-71. 14. Ramamurthi B. Intracranial tumors in India: incidence and variations. lnt Surg 1973;58:542-7. 15. Schoemann J, Hewlett R, Donald P. MR of childhood tuberculous meningitis. Neuroradiology 1988;30:473-7. 16. Sibley WA, O’Brien JH. Intracranial tuberculomas: a review of clinical features and treatment. Neurology (Minneap) 19056;6:157-65. 17. Tandon PN, Pathak SN. Tuberculosis of the central nervous system. In: Spillane JD, ed. Tropical neurology. Chap. 2, London: Oxford University Press, 1973. 18. Vengsarker US, Pisipaty RP, Parekh B, Panchal VG, Shetty MN. Intracranial tuberculoma and the CT scan. J Neurosurg 1986;64:568-635.
COMMENTARY From the operation
notes and the MRf pictures, I feel that the lesion is arising from the cortex, with infiltration of the deeper surface of the dura matter. Such cortical tuberculomas are known to occur, though rare; their presentation is with focal seizures and focal transient weakness. I had described this in the Journal of Neurosurgery in 1961. (Ramamurthi B, Varadarajan MC. Diagnosis of tuberculomas of the brain: clinical and radiological correlations. J Neurosurg 1961;18:1-7.) My observations are that this is not a purely meningeal tuberculoma. Usually, meningeal tuberculomas cause minimal reaction on the surface of the brain because of the intervening subarachnoid space. Cortical tuberculomas would cause the edema that is seen in the MRf picture. This is a rare condition; however, the authors may consider that this is a cortical tuberculoma rather than a meningeal tuberculoma. Their operation notes mention that “after opening of the dura, a slight extracere bra1 mass was removed extending down into the hemisphere.” This description is more in consonance with a dural tuberculoma. The surgeon will be the best judge to decide on this point. As cortical tuberculomas are mildly vascular, the faint flush seen on superselective catheterization may be expected. B. Ramamurthi,
M.S.
Madras, India
We found that at the Neurological Institute in Mexico, two similar cases have been studied but not reported in a 30-year period. Considering the large number of tuberculosis (TB) cases we handle, I can say that this case is unusual.
84
Surg Neurol 1995;43:81-4
Lindner
Even though the basic studies of TB were done not far away from Wuerzburg, the idea that we have controlled infectious diseases has spread in the medical profession; unfortunately, this is not true, and the “new” bacilli are very resistant to treatment. This type of case calls the attention of different groups of surgeons to the possibility that a tuber-
et al
culoma appears with a different image than that “typical” of granuloma. As a matter of fact, in cases of AIDS, we treat patients who “seem” to have a tuberculoma without biopsy, and only if there is no response do we proceed to surgical treatment.
ELL, IF 1 CALLED THE WRONG YOU ANSWER THE PHONE?
Jo& Humberto Mateos, M.D. Mexico City, Mexico
NUMBER,
JAMES THURBER AMERICAN HUMORIST,
WHY
DID
(1894-1961) ILLUSTRATOR
Infection
ISOLATED MENINGEAL TUBERCULOMA MIMICKING MENINGIOMA: CASE REPORT A. Lindner, M.D., Ch. Schneider, M.D., E. Hofmann, M.D., N. Soerensen, M.D., and K.V. Toyka, M.D. Neurologische Klinik, Abt. fuer Neuroradiologie und Neurochirurgische Klinik, Abt. fuer Paed. Neurochirurgie Julius, Maximilians, Universitaet, Wuerzburg, Germany
Lindner A, Schneider CH, Hofmann E, Soerensen N, Toyka KV. Isolated meningeal tuberculoma mimicking meningioma: Case report. Surg Neurol 1995;43:81-4. A 47-year-old German female patient is described with intracranial tuberculoma of the dura mater without his-
tory or evidence of tuberculosis and a negative tuberculin reaction. Computed tomography and magnetic resonance imaging and angiography resembled a meningioma. Diagnosis was verified by histology and identification of mycobacterium tuberculosis. After removal of the granuloma and highdose chemotherapy, the patient was free of disease. Tuberculoma of the dura mater is an extremely rare disease in the Western European population. KEY WORDS
Intracranial tuberculoma,
treatment.
T
he incidence of intracranial tuberculoma became less than one in 10,000 cases with intracerebral space-occupying lesions when tuberculosis declined in the general population [ 111. In contrast, intracranial tuberculomas are common in developing countries with figures ranging from 20%30% in India [ 7,141.
CASEREPORT We report a 47-year-old German patient with a tuberculoma of the dura mater with no previous history of tuberculosis or increased risk of infection. The right-handed patient was seen because of short spells when she was unable to speak and noted hypesthesia and paresthesia in the base of her tongue and in the left perioral face. This was interpreted as indicating transient ischemic attacks. On computed tomography (CT) scanning, a large right-hemispheric lesion was identified in the tempo ral lobe contrasting with the mild clinical signs. Address reprint requests to: K.V. Toyka, M.D., Neurologische Klinik der Unversitaet, Josef Schneider, Str. 11, D-97080 Wuerzburg, Germany. Received May 14, 1994; accepted August 22, 1994. 0 1995 by Elsevier Science Inc. 655 Avenue of the Americas, New York, NY 10010
On admission 2 weeks later she appeared perfectly normal except for marginal weakness of the perioral muscles and mild increase in tendon reflexes on the left. The tuberculin skin reaction (tine test) was negative and became positive only 10 weeks later when the diagnosis had already been made. Fundoscopy was entirely normal. The sedimentation rate was 5 per hour and the blood smear was within normal limits. Cerebrospinal fluid (CSF) was obtained by lumbar puncture and showed mild pleocytosis (11 mononuclear cells per microliter), whereas total protein, albumin, and IgG including the IgG-index were within normal limits. No bacteria could be grown on appropriate cultures. Repeated microbiologic examinations including gastric and bronchial lavage did not reveal mycobacterium tuberculosis. X-ray and ultrasonic examinations of organs prone to develop typical primary lesions were unrevealing. CT SCANNING AND MAGNETIC RESONANCE IMAGING (MRI) On axial CT scanning a hyperdense lesion was seen on the left temporal lobe extending to the basal ganglia indicating edema. After contrast enhancement a lesion contingent with the skull was seen (not shown). On MRI using a 1, 5 Tesla Siemens Magnetom, an extensive hemispheric hyperintense lesion was seen extending into the white matter and a rim at the subdural space sparing the area where the hyperdense CT lesion was located (Figure 1). All lesions showed contrast enhancement after gadolinium injection (Figure 2). On digital subtraction angiography the leptomeningial vessels of the middle cerebral artery showed displacement from the skull at the side of the CT lesion. With a superselective catheter, a faint flush like appearance was seen below the dura (not shown). After opening of the dura, a solid extracerebral 0090-3019/95/$9.50 SSDI 0090-3019(94)00122-7
82
Surg Neurol
Lindner et al
1995;43:81-4
In the strong T,-weighted MRI, a marked edema of the left temporal cortex and white matter can be seen reaching to the basal ganglia. The thickened dura has a low signal. There is a mild midline shift to the right.
mass was removed extending down into the hemisphere. Neuropathologic examination revealed granulomatous tissue with scattered Langerhans giant cells but no bacilli. Extracts of the material were cultured and injected into laboratory rabbits. After 4 weeks, mycobacterium tuberculosis was identified in both test systems.
TREATMENT The patient was treated with high-dose intravenous streptomycin, myambutol, and rifampicin and subsequently with oral myambutol and rifampicin. There were no recurrences of initial episodes up to the observation period (31 months). Repeated CT and MRI scan 5 months after the operation showed no further lesion (not shown).
DISCUSSION This patient presented with short spells that can be interpreted as transient ischemic attacks or focal seizures with only very mild deficits. These can be
The coronal T,-weighted MFClshows a thickened left temporal dura in the region of the sylvian fissure, buckling toward the cortex from which it is sharply demarcated. After gadolinium-GDPA, there was a strong enhancement of the lesion and of the left temporoparietal dura.
localized to the precentral and postcentral cortex. Because of the initial CT findings, the tentative diagnosis of an unusual intracranial tumor was made originating from the skull or the meninges. Surprisingly, on operation a granulomatous tumor was found suggesting sarcoid granuloma or tuberculoma. The patient went into complete remission after surgical and antibacterial treatment within 6 months. Symptoms and signs of the cerebral tuberculoma include increased intracranial pressure with papillary edema and seizures in more than half of the patients [3,7,12,16]. It is not uncommon that these patients have few or no symptoms indicating general disease such as fever or malaise [ 16,171. The cutaneous tuberculin reaction is reported positive in 25%-75% of patients [ 171. CSF findings include mild elevation of protein. There are reports showing the typical appearance of intracranial tuberculomas on CT scans. Early lesions are hypodense or isodense and take up contrast material. Older lesions show focal cal-
Isolated
Meningeal
Surg Neurol 1995;43:81-4
Tuberculoma
cification and may take up contrast the margin [4,5,13].
medium
only at
MRf became available only recently for the identification of intracerebral tuberculoma with a typical isointense lesion in the T, image surrounded by hyperintense edema. Hypointense areas within the lesion indicate calcification or central calcification. Gadolinium enhancement provides a similar pattern to CT enhancement [6,8,15]. The distribution of intracerebral tuberculoma is quite variable with most tuberculomas located in the frontal and parietal lobe or at the base of the skull [9,10,12,13,18]. According to Dastur (1965), dural tuberculoma is a rare event even in patients from India where systemic tuberculosis is still common. It is even more unlikely to occur in central Europe. The clinical differential diagnosis includes a number of intracranial space-occupying lesionsmeningioma being the most likely, as was first thought present in our patient. To our knowledge there is only one previous case report on an isolated tuberculoma of the dura mater [ 11. This observation leads us to suggest that a careful pathology and microbiologic examination is essential in establishing the diagnosis of this treatable condition. REFERENCES tuberculo1. Anderson JM, MacMillan JJ. Intracranial ma-an increasing problem in Britain. J Neurol Neurosurg Psychiatry 1975;38:194-201. 2. Arseni C. Two hundred and one cases of intracranial tuberculoma treated surgically. J Neurol Neurosurg Psychiatry 195&21:308-l 1. 3. Arsenjo A, Valladares H, Fierro J. Tuberculomas of the brain. Arch Neurol Psychiatry 1951;65:146-59. 4. Bullock MRR, Welchman JM. Diagnostic and prognostic features of tuberculous meningitis on CT scanning. J Neurol Neurosurg Psychiatry 1982;45:1098-1101. 5. Casselmann ES, Hasso AN, Ashwal S, Schneider S. Computed tomography of tuberculous meningitis in infants and children. J Comput Assist Tomog 1980;4: 211-6. 6. Chang KH, Han MH, Roh JK, Kin IO, Han MC, Kim CW. Gd-DTPA-enhanced MR imaging in intracranial tuberculosis. Neuroradiology 1990;32:19-25. 7. Dastur HM, Desai AD. A comparative study of brain tuberculomas and gliomas based upon 107 case reports of each. Brain 1965;88:375-96. 8. Davidson HD, Steiner RE. Magnetic resonance imaging in infections of the central nervous system. AINR 1988;6:499-504. 9. Dyk Van A. CT of intracranial tuberculomas with spe cific reference to the “target sign.” Neuroradiology 1988;30:329-36. 10. Jinkina JR. Dynamic CT of the tuberculous meningal reactions. Neuroradiology 1987;29:343-7.
83
RS. Tuberculomas of the brain in 11. Maurice-Williams Britain, J Postgrad Med 1972;48:678-81. 12. Mayers MM, Kaufmann DM, Miller MH. Recent cases of intracranial tuberculomas. Neurology (NY) 1978; 28:256-60. 13. Price HJ, Danziger A. Computed tomography in cranial tuberculosis. Am J Roentgen01 1978;130:769-71. 14. Ramamurthi B. Intracranial tumors in India: incidence and variations. lnt Surg 1973;58:542-7. 15. Schoemann J, Hewlett R, Donald P. MR of childhood tuberculous meningitis. Neuroradiology 1988;30:473-7. 16. Sibley WA, O’Brien JH. Intracranial tuberculomas: a review of clinical features and treatment. Neurology (Minneap) 19056;6:157-65. 17. Tandon PN, Pathak SN. Tuberculosis of the central nervous system. In: Spillane JD, ed. Tropical neurology. Chap. 2, London: Oxford University Press, 1973. 18. Vengsarker US, Pisipaty RP, Parekh B, Panchal VG, Shetty MN. Intracranial tuberculoma and the CT scan. J Neurosurg 1986;64:568-635.
COMMENTARY From the operation
notes and the MRf pictures, I feel that the lesion is arising from the cortex, with infiltration of the deeper surface of the dura matter. Such cortical tuberculomas are known to occur, though rare; their presentation is with focal seizures and focal transient weakness. I had described this in the Journal of Neurosurgery in 1961. (Ramamurthi B, Varadarajan MC. Diagnosis of tuberculomas of the brain: clinical and radiological correlations. J Neurosurg 1961;18:1-7.) My observations are that this is not a purely meningeal tuberculoma. Usually, meningeal tuberculomas cause minimal reaction on the surface of the brain because of the intervening subarachnoid space. Cortical tuberculomas would cause the edema that is seen in the MRf picture. This is a rare condition; however, the authors may consider that this is a cortical tuberculoma rather than a meningeal tuberculoma. Their operation notes mention that “after opening of the dura, a slight extracere bra1 mass was removed extending down into the hemisphere.” This description is more in consonance with a dural tuberculoma. The surgeon will be the best judge to decide on this point. As cortical tuberculomas are mildly vascular, the faint flush seen on superselective catheterization may be expected. B. Ramamurthi,
M.S.
Madras, India
We found that at the Neurological Institute in Mexico, two similar cases have been studied but not reported in a 30-year period. Considering the large number of tuberculosis (TB) cases we handle, I can say that this case is unusual.
84
Surg Neurol 1995;43:81-4
Lindner
Even though the basic studies of TB were done not far away from Wuerzburg, the idea that we have controlled infectious diseases has spread in the medical profession; unfortunately, this is not true, and the “new” bacilli are very resistant to treatment. This type of case calls the attention of different groups of surgeons to the possibility that a tuber-
et al
culoma appears with a different image than that “typical” of granuloma. As a matter of fact, in cases of AIDS, we treat patients who “seem” to have a tuberculoma without biopsy, and only if there is no response do we proceed to surgical treatment.
ELL, IF 1 CALLED THE WRONG YOU ANSWER THE PHONE?
Jo& Humberto Mateos, M.D. Mexico City, Mexico
NUMBER,
JAMES THURBER AMERICAN HUMORIST,
WHY
DID
(1894-1961) ILLUSTRATOR