Successful conservative operative management of pineal apoplexy 667
15% and widespread cranial to spinal metastasis in 8% of the cases in the series reported by Zorludemir.2 Four recurrences, the highest frequency of recurrences so far, were reported by Prinz et al.5 There are very few spinal meningiomas which were reported in the literature showing extension into the intervertebral foramen.9–11 In addition, spinal clear cell meningiomas without significant dural attachment have been described in only one report by Holtzman.6 In our case, the clinical presentation, neuroimaging features and the operative findings were more suggestive of a neurilemmoma in view of the foraminal extension and the lack of dural attachment. These features distinguished this tumor from any spinal meningioma we have previously encountered. It has become widely recognized that organ transplant recipients have a 5–6% risk of developing cancer, an incidence approximately 100 times greater than that observed in the general population in the same age range.12 However, meningiomas are rarely reported after transplantation, even in long-term survivors.13;14 There is no evidence showing more frequent occurrence of meningiomas in the setting of transplantation or immunosuppression. Clear cell meningioma may be confused with other clear cell tumors, especially with the similar-looking metastatic renal cell carcinoma.5 Indeed, this is very important in our patient – a renal transplant recipient. The negative immunoreactivity to keratin and patternless sheets of cells rather than positive immunoreactivity to keratin and geographic or alveolar cellular pattern ruled out the diagnosis of renal cell carcinoma. In the postoperative abdominal and pelvic CT, there was also no abnormality found. In conclusion, clear cell meningioma is rare but important because of its peculiar clinical and prognostic implications. In our patient, the differential diagnosis of a renal cell carcinoma is especially important. Compared to other spinal clear cell meningiomas reported to date, our case with foraminal extension and mimicking a neurilemmoma is rather unique.
bone marrow transplantation. Bone Marrow Transplant 1998; 22: 723–724. 14. Wijdicks EF, Plevak DJ, Wiesner RH, Steers JL. Causes and outcome of seizures in liver transplant recipients. Neurology 1996; 47: 1523–1525.
Successful conservative operative management of pineal apoplexy Gary J. Avery BPHARM MBBS, Christopher R.P. Lind Arnold P.L. Bok MMED FCS(SA) FRACS
MBCHB,
Department of Neurosurgery, Auckland Hospital, Auckland, New Zealand
Summary A 71-year-old woman presented with obstructive hydrocephalus caused by a haemorrhagic pineal mass. A right ventriculoperitoneal shunt was inserted with successful treatment of the hydrocephalus and resolution of symptoms and signs. Post-operative MRI both as an inpatient and at 5 and 18 months showed no contrast enhancement of the lesion and there was resolution of the radiographic abnormality. She remains symptom-free at 18 months. Previous reports have advocated resection of the pineal lesion, but this patient has done well with CSF shunting and MRI surveillance. ª 2003 Elsevier Ltd. All rights reserved. Journal of Clinical Neuroscience (2004) 11(6), 667–669 0967-5868/$ - see front matter ª 2003 Elsevier Ltd. All rights reserved. doi:10.1016/j.jocn.2003.11.009
Abbreviations: MRI, magnetic resonance imaging, CT, computed tomography, CSF, cerebrospinal fluid, AVF, arterio-venous fistula, DSA, digital subtraction angiography Keywords: Pineal apoplexy, Pineal gland, Obstructive hydrocephalus, Ventriculo-peritoneal shunting
REFERENCES 1. Kleihues P, Burger PC, Scheithauer BW. Tumors of the meninges. In: Sobin LH (ed) Histological Typing of Tumors of the Central Nervous System, second ed. Springer-Verlag, Berlin 1993; 33–42. 2. Zorludemir S, Scheithauer BW, Hirose T, Van Houten C, Miller G, Meyer FB. Clear cell meningioma: a clinicopathologic study of a potentially aggressive variant of meningioma. Am J Surg Pathol 1995; 191: 493–505. 3. Harkin JC, Leonard GL. Abnormal amianthoid fibers in meningioma. Acta Neuropathol (Berl) 1988; 6: 638–639. 4. Scheithauer BW. Tumors of the meninges: proposed modification of the World Health Organization classification. Acta Neuropathol (Berl) 1990; 80: 343–354. 5. Prinz M, Part S, Mitrovics T, Cervos-Navarro J. Clear cell meningioma: report of a spinal case. Gen Diag Pathol 1996; 141: 261–267. 6. Holtzman RN, Jormark SC. Nondural-based lumbar clear cell meningioma. Case report. J Neurosurg 1996; 84: 264–266. 7. Shih DF, Wang JS, Pan RG, Tseng HH. Clear cell meningioma: a case report. Chung Hua I Hsueh Tsa Chih – Chin Med J 1996; 57: 452–456. 8. Shiraishi K. Glycogen-rich meningioma. Case report and short review. Neurosurg Rev 1991; 14: 61–64. 9. McCormick PC. Surgical management of dumbbell and paraspinal tumors of the thoracic and lumbar spine. Neurosurgery 1996; 38: 67–74. 10. Hlavka V, Miklic P, Besenski N, Miklic D, Franz G. Dumbbell meningioma of the cervico-clavicular region. Neurol Croat 1991; 40: 319–326. 11. Mimoto H, Tomita R, Nakahara K, Tanaka H, Hada J, Noguchi K. A case of dumbbell type extradural spinal meningioma. Kyobu Geka 1991; 44: 961–964. 12. Penn I, Starzl TE. Malignant tumors arising de novo in immunosuppressed organ transplant recipients. Transplantation 1972; 14: 407–417. 13. Meignin V, Gluckman E, Gambaraelli D, Devergie A, Ramee MP, Janin A, Socie G. Meningioma in long-term survivors after allogeneic
ª 2003 Elsevier Ltd. All rights reserved.
Received 3 October 2003 Accepted 17 November 2003 Correspondence to: Dr. Christopher R.P. Lind, Department of Neurosurgery, 6th Floor, Auckland Hospital, Private Bag 92024, Auckland, New Zealand. Tel.: +64-9-307-4949; Fax: +64-9-307-4924; E-mail:
[email protected]
INTRODUCTION Twelve cases of pineal apoplexy with obstructive hydrocephalus have been reported in the English language literature. We present a case of a haemorrhage in the pineal region treated with ventricular shunting to relieve acute obstructive hydrocephalus and subsequent spontaneous resolution of the pineal lesion. This contrasts with other reports advocating resection of the pineal.1;2 CASE REPORT History and examination This 71-year-old woman had a past medical history of rheumatoid arthritis and deep vein thrombosis with pulmonary embolism. She had been previously treated with a Greenfield caval filter and oral anticoagulation for venous thromboembolic prophylaxis. She presented with a two day history of drowsiness after falling and striking her head. She had vomited 12 h prior to presentation and Journal of Clinical Neuroscience (2004) 11(6)
668 Avery et al.
Fig. 1
MRI 1 week after ventriculo-peritoneal shunting: sagittal T1 weighted image (a) without and (b) with intravenous gadolinium.
was complaining of a headache and unsteadiness. On examination she had a reduced level of consciousness, was slow to respond, and was disoriented to place but not time. She was following commands with all limbs, had mild ataxia and subtle upgaze palsy. The INR was 1.9 at presentation, and was reversed to normal prior to operation. A CT scan showed a haemorrhage in the pineal region with no post-contrast enhancement. There was acute obstructive hydrocephalus.
Radiology MRI one week after operation revealed a 10 10 20 mm heterogeneous pineal region mass. There was high signal intensity on T1, blooming on gradient echo, with no enhancement post gadolinium. There was mild transependymal hyperintensity seen on FLAIR images suggesting resolving hydrocephalus with reduction in lateral ventricular size. These findings suggested haemorrhage within a possible benign pineal lesion such as a cyst (Fig. 1).
Operation and post-operative course A right frontal 1.5 performance delta unitized venticulo-peritoneal shunt was inserted without complication. The patient’s partial disorientation resolved within 24 h. Immediate CT scan showed adequate shunt placement with no increase in pineal haemorrhage. Serum and CSF alpha-fetoprotein and tumour beta-human chorionic gonadotrophin were within the normal range. Microscopy and culture of her CSF were unremarkable.
Fig. 2
Outcome The patient’s mild upgaze palsy resolved within one week of the operation at which point she was discharged. She was recommenced on her oral anticoagulant one week later by her family physician. The patient was reviewed in clinic at 4 months. She was well, with no ataxia, headaches, nausea or vomiting.
MRI 5 months after ventriculo-peritoneal shunting: sagittal T1 weighted image (a) without and (b) with intravenous gadolinium.
Journal of Clinical Neuroscience (2004) 11(6)
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Transient global amnesia 669
Ophthalmology review demonstrated no non-refractive visual problems. MRI 5 months after operation showed that the haemorrhagic pineal lesion demonstrated previously had almost completely resolved. Only a 9 mm 7 mm focus of predominantly low signal intensity on T1 and T2 weighted images remained in the pineal. There was no pathological enhancement, nor any oedema. No hydrocephalus was identified. There was no evidence of a pineal or posterior third ventricular mass. There was evidence of altered blood product in the pineal gland with a small amount of haemosiderin staining of the adjacent right thalamus (Fig. 2). Given the 5-months interval since the acute presentation, and the normal appearance of the pineal, a haemorrhagic tumour was effectively excluded. MR angiography demonstrated no evidence of an arteriovenous malformation. Clinical review at 8 months showed the patient remained asymptomatic with a normal neurological examination. Further MRI at 18 months showed no change from the 5 months scan and she remained well. DISCUSSION Apoplexy refers to ill-defined symptoms subsequent to acute haemorrhage into a normal structure or lesion and is an uncommon intracranial event. Clinical significant pineal apoplexy is a rare event, with only 12 cases found encompassing a heterogeneous range of pathology.1–11 Causes of pineal apoplexy include haemorrhage into a pineal cyst, or tumour, or haemorrhage from an adjacent vascular malformation.1 There have been no reports of haemorrhage into a normal gland but this seems plausible in the context of anticoagulation therapy. In cases such as ours with no enhancement, the most likely underlying lesion may be pineal cyst. Non-neoplastic cysts of the pineal gland are common incidental findings in as many as 40% of routine autopsies, and asymptomatic pineal cysts are present on 1.5–4.3% of MRIs.12 Two reviews have revealed only 75 reports of symptomatic pineal cysts.1;2 Pineal apoplexy is the most uncommon presentation of a symptomatic pineal cyst. Modes of presentation include the syndromes of: paroxysmal headache with gaze paresis; chronic headache, gaze paresis, papilledema, and hydrocephalus; and pineal apoplexy with acute hydrocephalus.12 The first reported case of pineal apoplexy was in 1976.3 The current report represents the second reported case of pineal apoplexy in a patient undergoing anticoagulation therapy. Several authors have advocated the need for surgical removal of apoplectic pineal lesions with acute hydrocephalus as either a primary treatment or following operative CSF diversion.1;2 Kobayashi et al.1 advocate surgical extirpation for the treatment of pineal apoplexy, on the basis of a single case of cavernous angioma. Repeated bleeding is thought to be a risk for patients with pineal apoplexy but there is no firm data to indicate the risk of this occurrence. Mukherjee et al. have said that placement of a CSF shunt should not be the sole management of hydrocephalus from a pineal mass lesion as this approach may result in progressive and permanent gaze difficulty.2 The risk of such deficits has not been quantified and all patients reported to date have had pineal resections. The other advantage of resection is the provision of a histological diagnosis. This is the first report of successful management of pineal apoplexy by CSF diversion without resection of the lesion. Subsequent MRIs to 18 months have shown no significant residual pathological lesion in the pineal region, although because digital subtraction angiography has not been performed, a small dural AVF has not been completely excluded. Given this patients age, ª 2004 Elsevier Ltd. All rights reserved.
history of pulmonary thromboembolic disease requiring anticoagulation, and uncertainty about the natural history of small dural AVFs, it was decided that angiography was not clinically indicated.
CONCLUSION Consideration should be given to serial imaging following resolution of pineal hemorrhage. Pineal surgery could be reserved for definitive abnormalities on follow-up imaging. This approach may reduce operative morbidity for patients with pineal apoplexy.
REFERENCES 1. Kobayashi S, Kamagata M, Nakamura M, Nakazato Y, Sasaki T. Pineal apoplexy due to massive hemorrhage associated with cavernous angioma. Surg Neurol 2001; 55: 365–371. 2. Mukherjee K, Banerji D, Sharma R. Pineal cyst presenting with intracystic and subarachnoid hemorrhage. Report of a case and review of the literature. Br J Neurosurg 1999; 13: 189–192. 3. Apuzzo M, Davey L, Manuelidis E. Pineal apoplexy associated with anticoagulant therapy. J Neurosurg 1976; 45: 223–226. 4. Burres K, Hamilton R. Pineal apoplexy. Neurosurgery 1979; 4: 264–268. 5. Higashi K, Katayama S, Orita T. Pineal apoplexy. J Neurol Neurosurg Psychiatry 1979; 42: 1050–1053. 6. Osborn R, Deen H, Kerber C, Glass R. A case of hemorrhagic pineal cyst: MR/ CT correlation. Neuroradiology 1989; 31: 187–189. 7. Richardson J, Hirsch C. Sudden, unexpected death due to “pineal apoplexy”. Am J Forensic Med Pathol 1986; 7: 64–68. 8. Swaroop G, Whittle I. Pineal apoplexy: an occurrence with no diagnostic clinicopathological features. Br J Neurosurg 1998; 12: 274–276. 9. Steinbok P, Dolman C, Kaan K. Pineocytomas presenting as subarachnoid hemorrhage. Report of two cases. J Neurosurg 1977; 47: 776–780. 10. Muller M, Hubbard S, Provias J, Greenberg M, Becker L, Rutka J. Malignant rhabdoid tumour of the pineal region. Can J Neurol Sci 1994; 21: 273–277. 11. Koenigsberg R, Faro S, Marino R, Turz A, Goldman W. Imaging of pineal apoplexy. Clin Imaging 1996; 20: 91–94. 12. Wisoff J, Epstein F. Surgical management of symptomatic pineal cysts. J Neurosurg 1992; 77: 896–900.
Transient global amnesia in a patient with acute unilateral caudate nucleus ischemia Vinod Ravindran1 MRCP, Sandeep Jain1 Alec Ming1 FCP (SA) FCP (SA) NEURO FRCP, Richard J.V. Bartlett2 MA FRCP FRCR
MBBS,
1
Department of Neurology and 2Department of Radiology, Hull Royal Infirmary, Hull & East Yorkshire Hospitals NHS Trust, Kingston-upon-Hull HU3 2JZ, UK
Summary The pathogenesis and localization of neuronal dysfunction in transient global amnesia (TGA) is still disputed more than 40 years after the first description of this clinical entity. Previous studies have indicated that structural abnormality is rare in TGA. We report a case of TGA in a patient with acute ischemia in the body of right caudate nucleus. This provides evidence in support of an ischemic hypothesis as the possible etiology of TGA. The role of caudate nucleus in human memory is also reviewed. ª 2004 Elsevier Ltd. All rights reserved. Journal of Clinical Neuroscience (2004) 11(6), 669–672 0967-5868/$ - see front matter ª 2004 Elsevier Ltd. All rights reserved. doi:10.1016/j.jocn.2003.12.012
Journal of Clinical Neuroscience (2004) 11(6)