Cavernous sinus tuberculoma: diagnostic difficulties in a personal case

Infection

Cavernous Sinus Tuberculoma: DIAGNOSTIC DIFFICULTIES IN A PERSONAL CASE Riadh Rebai, M.D.,* Mohamed Zaher Boudawara, M.D.,* Kamel Bahloul, M.D.,* Imed Chabchoub, M.D.,* Saloua Chaari, M.D.,* Tahia Boudawara, M.D.,† and Habib Ben Mansour, M.D.* *Department of Neurological Surgery and †Department of Cytopathology, H. Bourguiba Hospital, Sfax, Tunisia

Rebai, R, Boudawara MZ, Bahloul K, Chabchoub I, Chaari S, Boudawara T, Ben Mansour H. Cavernous sinus tuberculoma: diagnostic difficulties in a personal case. Surg Neurol 2001;55:372–5. BACKGROUND

Tuberculoma of the brain is rare, and its location in the cavernous sinus is exceptional. Many factors make the diagnosis difficult, necessitating a surgical procedure. CASE DESCRIPTION

A 44-year-old woman complained of persistent headaches and diplopia. The examination revealed a right cavernous sinus syndrome. Radiologic investigation showed a growing process in the right cavernous sinus, overlapping the sella turcica. The diagnoses of metastasis, tuberculoma, and sarcoidosis were considered, but none of them could be confirmed. A biopsy of the lesion via a subtemporal approach revealed a tuberculoma. Antituberculous treatment led to complete resolution of her symptoms. CONCLUSION

The diagnosis of a cavernous sinus tuberculoma is difficult; it is confirmed by biopsy. However, a trial of medical treatment, if the results are favorable, may obviate the need for a surgical procedure. © 2001 by Elsevier Science Inc. KEY WORDS

Intracranial tuberculoma, cavernous sinus, antituberculous chemotherapy, surgical treatment.

uberculomas are most often located in the cerebral hemispheres; cavernous sinus tuberculomas are extremely rare. Only three cases have previously been reported in the literature [2– 4]. In this report, we present the results of surgical exploration and the management of a cavernous sinus tuberculoma and its evolution, emphasizing the factors that led to surgery as a diagnostic solution.

T

Address reprint requests to: Dr. Riadh Rebai, Ho ˆ pital H. Bourguiba, Service de Neurochirurgie; 3029, Sfax, Tunisie. Received April 28, 2000; accepted December 1, 2000. 0090-3019/01/$–see front matter PII S0090-3019(01)00474-8

Case Report A 44-year-old woman had been suffering from headaches for 1 month before her hospitalization. She also complained of a right ptosis and horizontal diplopia. She did not have any particular pathological history, including tuberculosis, and there was no history of exposure to tuberculosis. Neurological examination revealed a right oculomotor paralysis associated with a homolateral fifth cranial nerve motor paresis. There was also a mild, bilateral sixth nerve paresis. The visual acuity was 3/10 on the right and 5/10 on the left; the funduscopic examination was normal. The patient was not febrile. The cerebral computed tomography (CT) scan showed a right laterosellar mass that heterogeneously enhanced after administration of contrast (Figure 1). It was associated with a contiguous lysis of the sphenoid bone. Magnetic resonance imaging (MRI) revealed a right laterosellar mass overlapping the sella turcica and the clivus; the lesion was isointense on both T1- and T2-weighted sequences. It enhanced heterogeneously after gadolinium administration (Figure 2). A cerebral angiogram showed only mild stenosis of the intacavernous carotid artery. Laboratory examinations revealed a sedimentation rate of 50 in the first hour; the white blood cell count was 8200. The differential diagnosis at this point included metastasis, tuberculosis, and sarcoidosis. Chest X-rays revealed an encapsulated right pleurisy (Figure 3), which drained purulent liquid when punctured. Bacteriologic examination of the latter was negative. The lesion responded favorably to ampicillin, gentamycin, and metronidazole. The pleurisy © 2001 by Elsevier Science Inc. 655 Avenue of the Americas, New York, NY 10010

Cavernous Sinus Tuberculoma

Surg Neurol 373 2001;55:372–5

2 (A) Cerebral CT scan after contrast agent administration showing a heterogeneously enhanced right cavernous sinus mass. (B) Lysis of the sphenoid bone is visible on bone window CT scan.

MRI after gadolinium administration showing a right laterosellar mass overlapping the sella turcica.

1

resolved and the sedimentation rate normalized to 18. Tuberculosis cultures were negative. Chest CT scan and abdominal ultrasonography did not reveal any tumor. It is notable that the hormonal analyses of the pituitary gland were normal. The diagnosis of metastasis seemed most probable. We performed a biopsy of the lesion using a right subtemporal approach via an intradural route. The pathological diagnosis was tuberculoma. Consequently, antituberculous therapy, consisting of ri-

fampicin, isoniaside, and ethambutol, was given for 1 year. The outcome was favorable, with resolution of the ophthalmoplegia and fifth cranial nerve paresis. Forty months later, she had only a moderate decrease of visual acuity. Control MRI revealed that the right cavernous sinus lesion had completely disappeared (Figure 4).

Discussion It is generally known that the incidence of tuberculosis in western countries has increased with the spread of acquired immunodeficiency syndrome. Tuberculous contamination of the central nervous

374 Surg Neurol 2001;55:372–5

3

Chest X-rays reveal an encapsulated right basal pleurisy.

system remains, however, less frequent than in countries where tuberculosis is endemic. The physiopathology of central nervous system tuberculoma is multi-factorial. It is based on hypothesis rather than on experimental data because of the impossibility of reproducing tuberculoma in animals. Between 1981 and 1997 we gathered 1015 cases of extended intracranial processes in the Department of Neurological Surgery at H. Bourguiba Hospital; 24 of these were tuberculomas. Encephalic tuberculomas account for 2.36% of space-occupying lesions in our series. Between January 1993 and December 1997 we did not see more than two encephalic tuberculomas per year (Table 1). Thus, the frequency of these lesions diminishes to 1.5% of intracranial masses when considering only this period. This rate is much lower than the approximately 20% in developing countries reported by Grayeli et al [2]. Because of their rarity, tuberculomas are not always considered in the differential diagnosis of intracranial space-occupying lesions. Tuberculoma of the cavernous sinus is rare; only three cases have been reported in the literature. This makes the diagnosis even more difficult. Our

Rebai et al

Cerebral MRI after antituberculous therapy showing complete disappearance of the right cavernous sinus tuberculoma.

4

patient had no history of tuberculous infection, which might have suggested the diagnosis of tuberculoma, although the value of this factor as a diagnostic indicator is limited in areas where tuberculosis is endemic. In our series, only 29% of the patients with tuberculomas had a history of tuberculous infection. In addition, the clinical symptoms and signs only indicate the presence of an expanding cavernous sinus lesion; they do not lead to a more specific diagnosis. Our patient had a solitary tuberculoma. Although solitary lesions are more common, the diagnosis seems to be easier in cases with numerous intracerebral lesions. Boucetta et al reported solitary tu-

1

Distribution of Intracranial Tuberculomas in Our Series from 1993 to 1997

YEARS Intracranial tuberculomas (# cases) Intracranial masses (# cases)

1993 1994 1995 1996 1997 0

2

1

2

1

81

87

78

72

77

Cavernous Sinus Tuberculoma

berculomas in 84% of their patients [1]. Nineteen percent of the patients in the series by Rodat et al had multiple tuberculomas [5]. The absence of extracerebral tuberculosis in our patient minimized the diagnostic probability of tuberculoma. In our series, only 13 out of 24 (54%) patients had an extracerebral tuberculous lesion; Boucetta et al reported a frequency of 30% [1]. The B.K. examinations of the saliva, urine, and cerebrospinal fluid in our patient were negative. In our series, the B.K. was isolated in only three patients who had tuberculosis. The imaging characteristics of intracranial tuberculoma vary depending on the stage of development. The descriptions in the literature are not specific, however. Even the “target sign” described by Van Dyk [6] appeared to be produced by cerebral toxoplasmosis. Despite the fact that it was an intracavernous lesion, the carotid angiogram in our patient showed no vascular anomalies. The rate of regression of tuberculomas with medical therapy is variable; consequently, the optimum length of a clinical trial is uncertain. On the basis of the test results in our case, we performed a biopsy of the mass to establish a definite diagnosis. It is notable that the bacteriologic exam is not conclusive even if it is negative. It does not eliminate tuberculoma from the differential diagnosis since the B.K. cannot always be detected in the cerebrospinal fluid or excised masses.

Conclusion Thanks to the vaccination program by the B.C.G, central nervous system tuberculosis is less frequent, as is cerebral tuberculoma. The diagnosis of tuberculoma can be facilitated by the presence of an extracerebral tuberculous lesion, and allows successful medical treatment. Nevertheless, the diagnosis of tuberculoma is less likely to be considered when the lesion is solitary; located in the brain, especially in the cavernous sinus; and when physical and laboratory examinations are negative and imaging is nonspecific. Surgical biopsy may be required in some cases to obtain a definite diagnosis. Once the diagnosis has been established, anti-

Surg Neurol 375 2001;55:372–5

tuberculosis therapy can be given, usually with favorable results. The authors would like to thank N. Ben Hamed and Mr. & Mrs. Abdelkefi for their aid in preparing this work.

REFERENCES 1. Boucetta M, Sami A, Choukri M, and Morsad F. Les tuberculomes ence´phaliques: 40 cases. Neurochirurgie 1993;39:42– 6. 2. Grayeli AB. Tuberculoma of the cavernous sinus: case report. Neurosurgery 1998;42:179 – 82. 3. Morris JT. Central nervous system tuberculoma presenting as a cavernous sinus tumor [letter]. Clin Infect Dis 1992;15:181–2. 4. Phookan G. Tuberculoma of the cavernous sinus: case report. Br J Neurosurg 1995;9:205–7. 5. Rodat O, Resch F, Lajat Y, Cattan P, Cler JM, and Nomballais F. Abce`s tuberculeux du ne´vraxe: revue de la litte´rature sur dix ans. Sem Ho ˆp Paris 1980;56:38 – 42. 6. Van Dyk A. CT of intracranial tuberculomas with specific reference to the “target sign”. Neuroradiology 1988;30:329 –36. COMMENTARY

Dr Rebai and co-workers report a rare case of an intracavernous mass diagnosed as a tuberculoma. In countries where tuberculosis is endemic, intracranial tuberculosis may account for up to 10% of all intracranial space-occupying lesions. In this report, the authors found 24 cases of intracranial tuberculosis out of 1015 intracranial processes (2.4%); one of the 24 was located within the cavernous sinus. The lesion mimicked a meningioma, though some clinical and laboratory findings might have raised the suspicion of an infectious disease. If tuberculosis is suspected beforehand, drug therapy should be started immediately. Because of the rarity of this lesion within the cavernous sinus, the author preferred to confirm the diagnosis by surgical exploration and biopsy. Postoperative drug therapy resulted in regression of the lesion and the symptoms. Prof Dr med Madjid Samii M. Tatagiba, M.D. Neurochirurgische Klinik Krankenhaus Nordstadt Hannover, Germany