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may also explain those cases with negative findings on surgical re-exploration; if a surgeon is not specifically looking for a CSF collection or a small arachnoid cyst, removal of the arachnoid adhesions forming the cystic walls and drainage of the CSF would simply be considered part of the approach. 4. Conclusions The present case suggests that in patients suffering pain recurrence after MVD for TGN, adhesive arachnoiditis with arachnoid cyst formation may be a possible causative factor, either by direct compression from the cyst or by arterial pulsation transmission to the nerve through the cyst. This should be kept in mind, especially when repeat MRTA does not show clear evidence of vascular compression. If surgical re-exploration is performed in such patients, the possibility of CSF loculations should be kept in mind and they should be treated if identified. Although one case does not allow for conclusions to be made, it does advocate for more aggressive surgical re-exploration in patients suffering pain recurrence after MVD for TGN, even in cases with inconclusive radio-imaging findings.
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References 1. Hunn MK, Eldridge PR, Miles JB, et al. Persistent facial pain following microvascular decompression of the trigeminal nerve. Br J Neurosurg 1998;12:23–8. 2. Barker FG 2nd, Jannetta PJ, Bissonette DJ, et al. The long-term outcome of microvascular decompression for trigeminal neuralgia. N Engl J Med 1996;334:1077–83. 3. Olson S, Atkinson L, Weidmann M. Microvascular decompression for trigeminal neuralgia: recurrences and complications. J Clin Neurosci 2005;12:787–9. 4. Lee SH, Levy EI, Scarrow AM, et al. Recurrent trigeminal neuralgia attributable to veins after microvascular decompression. Neurosurgery 2000;46:356–612. 5. Broggi G, Ferroli P, Franzini A, et al. Microvascular decompression for trigeminal neuralgia: comments on a series of 250 cases, including 10 patients with multiple sclerosis. J Neurol Neurosurg Psychiatry 2000;68:59–64. 6. Rath SA, Klein HJ, Richter HP. Findings and long-term results of subsequent operations after failed microvascular decompression for trigeminal neuralgia. Neurosurgery 1996;39:933–40. 7. Meaney JF, Eldridge PR, Dunn LT, et al. Demonstration of neurovascular compression in trigeminal neuralgia with magnetic resonance imaging. Comparison with surgical findings in 52 consecutive operative cases. J Neurosurg 1995;83:799–805. 8. Krisht AF, O’Brien MS. Acquired mirror-image cerebellopontine angle arachnoid cysts: case report. Neurosurgery 1992;30:798–800. 9. Little JR, Gomez MR, MacCarty CS. Infratentorial arachnoid cysts. J Neurosurg 1973;39:380–6.
doi:10.1016/j.jocn.2006.10.027
Nonsurgically treated cervical brucellar epidural abscess causing spinal cord compression Goksin Sengul a,*, Ali Akar a, Fatih Alper b, Hakan Uslu c a
Department of Neurosurgery, Medical School, Ataturk University, 25070, Yenisehir, Erzurum, Turkey b Department of Radiology, Medical School, Ataturk University, Erzurum, Turkey c Department of Microbiology, Medical School, Ataturk University, Erzurum, Turkey Received 26 February 2007; accepted 22 May 2007
Abstract Cervical spinal cord compression due to brucellar epidural abscess is extremely rare, with only 13 previously reported cases. We present another case of cervical spinal epidural abscess causing spinal cord compression. Diagnostic tools and treatment options are discussed. Ó 2007 Elsevier Ltd. All rights reserved. Keywords: Brucellosis; Epidural abscess; Spinal
*
Corresponding author. Tel.: +90 44 2316 6333x2085; fax: +90 44 2316 6340. E-mail addresses:
[email protected],
[email protected] (G. Sengul).
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1. Introduction Brucellosis is still an important health problem in the eastern part of Turkey, with approximately 9000 cases reported to the Ministry of Health database yearly. Although brucellosis may affect the spine, it is rarely may be complicated by epidural abscess or paraspinal purulent mass. Brucellar epidural abscesses causing spinal cord compression at the cervical level are extremely rare and we were able to find only 13 cases reported to date in the literature.1–5 The purpose of this paper is to draw attention and alert practitioners to an uncommon but treatable pathology that needs early diagnosis to prevent or minimize neurological damage. 2. Case report A 37-year-old male farmer from a rural village in Eastern Anatolia was admitted to the General Surgery Unit with complaints of abdominal pain and weight loss of 7 kg. An abdominal examination revealed no abnormal findings. Seven days after admission, the patient developed acute weakness in the left upper extremity and was referred to our department. On admission, a physical examination revealed no abnormality. On neurological examination, we observed monoparesis with grade 3 power in the left upper extremity and mild neck stiffness with a decreased range of motion. Deep tendon reflexes were normal. No pathological reflex or dermatomal sensory change was observed. The white cell count was 11 240/mm3, the erythrocyte sedimentation rate (ESR) was 90 mm/h, and the C-reactive
protein (CRP) level was 6.3 mg/dL. A Brucella seroagglutination test was positive at a 1/160 dilution and a blood culture was positive for Brucella sp. A plain radiograph of the cervical spine showed narrowing of the C7–T1 disc space and destruction of the endplates of adjacent vertebral bodies. MRI of the cervical spine showed decreased signal intensity on T1- and increased signal intensity on T2-weighted images of the C7– T1 intervertebral disc and, adjacent C7 and T1 vertebrae, and a mass lesion within the anterior spinal canal outside the spinal cord. With gadolinium enhancement, the epidural soft mass behind the C7 and T1 vertebral bodies showed intense, slightly hetrogeneous enhancement (Fig. 1). These images were suggestive of C7–T1 spondylodiscitis with epidural abscess. Cranial, thoracic and lumbar MRI findings were all normal. Electromyelographic findings were compatible with C7 and C8 radiculopathy. Technetium bone scintigrams demonstrated increased uptake at the affected spinal regions. After a review of the available treatment options, we decided to try pharmacological treatments first. From the literature and through our own experience, we know that brucellosis responds well to antibiotic therapy. A consultation with the infectious diseases department resulted in prescription of an antibiotic regimen including rifampin (600 mg/day) and streptomycin (1 g/day), on which the patient was discharged. At a 1 month follow-up examination, the paresis had completely resolved, and ESR and CRP had returned to normal values. MRI revealed no remnants of the abscess but did show extension of the spondylodiscitis (Fig. 2). Treatment was changed to doxycycline (200 mg/ day) and rifampin (600 mg/day). At a 3-month follow-
Fig. 1. Epidural abscess compressing the spinal cord and spondylodiscitis of C7–T1. (A) T1-weighted sagittal MRI. (B) T2-weighted sagittal MRI. (C) T1weighted gadolonium-enhanced sagittal MRI.
Case Reports / Journal of Clinical Neuroscience 15 (2008) 1411–1414
Fig. 2. T1-weighted gadolonium-enhanced sagittal MRI after 1 month of treatment showing resolution of the abscess and extension of the spondylodiscitis.
up, MRI showed improvement in the spondylodiscitis. Complete radiological recovery was seen at a 6-month follow-up (Fig. 3). 3. Discussion Spinal epidural abscess caused by Brucella is uncommon and develops from previous spondylodiscitis. Brucellar spondylodiscitis generally arises from the upper end plate where blood supply is rich, and it may spread to the whole vertebra, disc space and adjacent vertebrae, depending on the virulence of the micro-organism, the size of the inoculum and the immune status of the host.3 Therefore, brucellar spinal epidural abscess mostly results from direct invasion followed by spondylitis.6 However, very rarely, it may develop via a direct hematogenous route without spondylitis.3,5 The most appropriate technique for the diagnosis of cervical brucellar epidural abscess is MRI, as it demonstrates not only the abscess, but also the spondylodiscitis and cord or radicular involvement. MRI findings for brucellar spondylitis are not characteristic and usually present within 1 month after the onset of symptoms.7 Epidural abscess is seen as a mass lesion within the spinal canal and outside the spinal cord, hyperintense on T2-weighted, and slightly hypointense on T1-weighted MRI. The abscess is better delineated with contrast enhancement using gadopentetate. The etiological diagnosis is carried out through blood culture and specific serological tests against Brucella. All were positive in our patient.
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Fig. 3. Complete recovery is seen on T1-weighted gadolonium-enhanced sagittal MRI at a 6-month follow-up.
Management of cervical brucellar epidural abscess remains controversial with respect to the role of surgery, the selection of antibiotics and the duration of treatment. Traditionally, the appropriate treatment modality for patients with cervical brucellar epidural abscess was considered to be surgical drainage plus intensive antibiotic therapy. However, two cases have been reported to respond to antibiotic-only treatment. Pina et al.5 reported a case treated with antibiotics alone, but the patient had no neurological deficit. Also, Gorgulu et al.3 reported a nonsurgically treated case in which the patient had neurological deficits on admission. Our case had clinical features similar to those of the second case and we preferred this nonsurgical approach. At the time we had to make our choice, Gorgulu et al. had not yet reported their case. Thus, we had concerns as to the appropriateness of the treatment, but our patient did well. If neurological deterioration had occurred, we would have instituted prompt surgical intervention. Our case is the second in the literature to be treated solely with antibiotics. Although data in the literature show that cervical brucellar abscess can be treated successfully with surgical intervention plus antibiotics, we achieved a complete cure by medical treatment alone. Currently, there is no consensus on the appropriate antibiotics and treatment duration for brucellosis. The antibiotics most frequently recommended are tetracyclines, streptomycin, co-trimoxazole and rifampin. Combinations must be used to prevent relapses. We used dual combinations of rifampin, streptomycin and doxycycline for 6 months until clinical and radiological recovery was achieved. Durations of treatment in the literature range from 6 weeks to 1 year depending on the clinical pro-
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gress.5,7 Cervical brucellar epidural abscess most commonly develops in the subacute-chronic phase of the disease and Brucella has a good response to antibiotics if they are administered in appropriate doses and treatment lasts until full recovery. 4. Conclusions Cervical brucellar epidural abscess is associated with a high incidence of devastating neurological complications and a poor overall prognosis. Therefore, it is essential to detect and treat it as early as possible. We believe that this condition can be treated only with antibiotics in selected cases. Control of the disease rests on public health measures.
References 1. Ates O, Cayli SR, Kocak A, et al. Spinal epidural abscess caused by brucellosis. Two case reports. Neurol Med Chir (Tokyo) 2005;45:66–70. 2. Colmenero JD, Cisneros JM, Orjuela DL, et al. Clinical course and prognosis of Brucella spondylitis. Infection 1992;20:38–42. 3. Gorgulu A, Albayrak BS, Gorgulu E, et al. Spinal epidural abscess due to Brucella. Surg Neurol 2006;66:141–6. 4. Mousa AM, Bahar RH, Araj GF, et al. Neurological complications of brucella spondylitis. Acta Neurol Scand 1990;81:16–23. 5. Pina MA, Modrego PJ, Uroz JJ, et al. Brucellar spinal epidural abscess of cervical location: report of four cases. Eur Neurol 2001;45:249–53. 6. Solera J, Lozano E, Martinez-Alfaro E, et al. Brucellar spondylitis: review of 35 cases and literature survey. Clin Infect Dis 1999;29:1440–9. 7. Ceviker N, Baykaner K, Goksel M, et al. Spinal cord compression due to Brucella granuloma. Infection 1989;17:304–5.
doi:10.1016/j.jocn.2007.05.023
Giant interdural teratoma of the cavernous sinus Vivek Bonde *, Atul Goel, Naina Kataria Goel Department of Neurosurgery, Seth G.S. Medical College and King Edward VII Memorial Hospital, Parel, Mumbai 400012, India Received 5 March 2007; accepted 14 April 2007
Abstract A 27-year-old man presented with a 2-month history of retro-orbital pain and a 2-week history of progressive ptosis and diplopia. Neurological examination revealed left partial third, fourth and sixth cranial nerve paresis. Imaging revealed a large middle fossa lesion with heterogeneous signal intensity. During surgery, the tumor was found to be entirely within the confines of the dural layers of the lateral wall of the cavernous sinus. Histological analysis revealed that the tumor was a mature teratoma. The case is discussed and the literature on this rare location of teratoma is reviewed. Ó 2007 Published by Elsevier Ltd. Keywords: Teratoma; Cavernous sinus; Intracavernous; Germ cell tumor; Interdural
1. Introduction
2. Case report
Teratomas represent 0.5% of all intracranial tumors.1,2 These tumors have tissue representative of the three germinal layers, ectoderm, mesoderm, and endoderm, and can be broadly classified into mature, immature and malignant teratomas.2 Most teratomas are located in or in proximity to the midline in the sellar and pineal regions.1 A location involving the cavernous sinus is extremely rare and only three such cases have been reported in the literature.1,3,4 A teratoma located within the dural confines of the lateral wall of the cavernous sinus, as was identified in the presented case, has not been previously recorded.
A 27-year-old man presented with retro-orbital pain and occasional vomiting of 2 months’ duration. Additionally, he had developed ptosis and diplopia 2 weeks prior to seeking medical advice. On examination, he had left partial third, fourth and sixth nerve paresis. MRI revealed a large middle fossa tumor extending into the infratemporal fossa and orbit. The dome of the tumor had a well - defined dural layer that was continuous with the roof of the cavernous sinus (Figs. 1 and 2). The precavernous and cavernous segments of the internal carotid artery were displaced by the tumor. Imaging showed that the tumor had a heterogeneous character with softer parts and areas of calcification. There were some contrast - enhancing areas within the tumor (Fig. 1). During surgery via a basal temporal extradural ap-
*
Corresponding author. Tel.: +91 22 24129884; fax: +91 22 24143435. E-mail address: drvrbonde@rediffmail.com (V. Bonde).
