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mentioned mechanisms are insufficient to explain the multiple ICHs and remote IVHs in our patient. Further investigations are needed to clarify this phenomenon. ICH and IVH occurrence following removal of a hematoma is a potentially lethal complication, and the mechanisms underlying this event remain unclear, especially within the ventricular system. Like Modesti et al.,8 we stress the need for slow and gradual decompression of CSDH; clinical awareness of this complication is essential. Acknowledgments We thank Dr François Niyonsaba (Juntendo University School of Medicine, Tokyo, Japan) for his valuable collaboration in preparation of the manuscript. References
Fig. 3. Repeat axial CT scan after evacuation of a chronic subdural hematoma showing bilateral intraventricular hemorrhage observed in the ventricular occipital horns.
patients,1–3 the most probable mechanism in our patient was a sudden variation in blood flow, combined with defective vascular autoregulation. Elderly patients with physiological aging of the cerebral vascular tree may not tolerate a sudden restoration of normal perfusion pressure in areas of deranged cerebral vascular autoregulation.10 Furthermore, fragile cerebral vessels and direct vascular damage, due to the shift of the midline structures, might have been contributory factors in our patient. However, many of the above-
1. Missori P, Salvati M, Polli FM, et al. Intraparenchymal haemorrhage after evacuation of chronic subdural haematoma. Report of three cases and review of the literature. Br J Neurosurg 2002;16:63–6. 2. d’Avella D, De Blasi F, Rotilio A, et al. Intracerebral hematoma following evacuation of chronic subdural hematomas. Report of two cases. J Neurosurg 1986;65:710–2. 3. Rohde V, Graf G, Hassler W. Complications of burr-hole craniostomy and closed-system drainage for chronic subdural hematomas: a retrospective analysis of 376 patients. Neurosurg Rev 2002;25:89–94. 4. Tindall GT, Payne II NS, O’Brien MS. Complications of surgery for subdural hematoma. Clin Neurosurg 1976;23:465–82. 5. Weigel R, Schmiedek P, Krauss JK. Outcome of contemporary surgery for chronic subdural haematoma: evidence based review. J Neurol Neurosurg Psychiatry 2003;74:937–43. 6. Sato M, Nakano M, Asari J, et al. Intracerebral haemorrhage during surgery for chronic subdural haematoma. J Clin Neurosci 2007;14:81–3. 7. Dinc C, Iplikcioglu AC, Bikmaz K, et al. Intracerebral haemorrhage occurring at remote site following evacuation of chronic subdural haematoma. Acta Neurochir (Wien) 2008;150:497–9. 8. Modesti LM, Hodge CJ, Barnwell ML. Intracerebral hematoma after evacuation of chronic extracerebral fluid collections. Neurosurgery 1982;10:689–93. 9. Sousa J, Golash A, Vaz J, et al. Spontaneous intracerebral haemorrhage following evacuation of chronic subdural hematomas. J Clin Neurosci 2004;11: 794–6. 10. Cook AW, Browder EJ, Carter WB. Cerebral swelling and ventricular alterations following evacuation of intracranial extracerebral hematoma. J Neurosurg 1962;19:419–23.
doi:10.1016/j.jocn.2008.12.031
Silicone spaghetti Manohan Sinnadurai *, Mark J. Winder Neurosurgery, St Vincent’s Hospital, 406 Victoria Street, Darlinghurst, New South Wales 2010, Australia
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Article history: Received 23 October 2008 Accepted 15 December 2008
a b s t r a c t Migration of ventriculoperitoneal shunt catheters, although infrequent, is well reported. We report the unusual event of a patient who, after having had three shunt revisions over a 15-year period, presented with a per-oral extrusion of her original distal catheter. Crown Copyright Ó 2009 Published by Elsevier Ltd. All rights reserved.
Keywords: Migration Shunt Transoral Ventriculoperitoneal
1. Introduction * Corresponding author. Tel.: +61 2 9382 1111; fax: +61 2 8382 7234. E-mail address:
[email protected] (M. Sinnadurai).
The migration of ventriculoperitoneal shunt (VPS) catheters is a rare complication of surgical treatment for hydrocephalus. Migra-
Case Reports / Journal of Clinical Neuroscience 16 (2009) 1348–1350
tion of both distal and proximal catheters has been reported, with most concerning distal catheter extrusion through the anus following migration into the abdominal viscous.1–4 The incidence of bowel perforation, early or delayed, is reported to be 0.1% to 0.7%.3,5 Other unusual reports include migration into the scrotal sac, vagina and bladder.2,6–8 Emerging shunt technology with more flexible silicone catheters has resulted in fewer reported catheter migrations. This report of a per-oral extrusion of the distal catheter is, to our knowledge, the only reported case in an adult. 2. Case report Our patient, a 27-year-old female, had a VPS placed at the age of 12 for hydrocephalus associated with a left arachnoid cyst. This required revision on two occasions, with ventricular catheter replacement initially, followed by a ‘‘Y” connector to the left temporal arachnoid cyst. The patient remained relatively well but over the previous 5 years suffered mild generalised constant headaches. Investigations revealed an elevated white cell count (WCC) count of unknown cause, with a mildly elevated erythrocyte sedimentation rate and C-reactive protein (CRP). No causative agent could be determined. The patient subsequently presented complaining of symptoms of elevated intracranial pressure, 12 years after her previous revision surgery. Repeat CT scan confirmed enlarged ventricles and operative intervention revealed good distal catheter flow but flow impedance of the ventricular catheter. Two weeks after ventricular catheter revision, the patient was at home and following dinner felt the need to cough. After a brief coughing fit she felt what she thought was spaghetti at the back of her throat. She was able to grab the tube and pull it out through her mouth. After repeatedly trying to remove the tube an ambulance was called and the patient attended the emergency department holding the tube in her hands (Fig. 1). The patient had a Glascow Coma Scale score of 15 with no focal neurological deficit. There was no evidence of abdominal tenderness and repeat blood tests revealed an unchanged elevated WCC. An urgent CT scan series was performed and showed gastric perforation of the distal catheter, which had migrated through the cardiac sphincter, ascending the oesophagus into the pharynx. The VPS was otherwise intact (Figs. 2 and 3). The patient was transferred to theatre, the shunt externalised and the catheter removed from the distal end through the mouth,
Fig. 1. A 27-year-old woman with a ventriculoperitoneal catheter extruding from her mouth. This figure is available in colour at www.sciencedirect.com.
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in an attempt to limit the risk of peritonitis. The patient was placed on broad spectrum antibiotics for 10 days, followed by distal catheter revision and was discharged home 3 days later. A two-month follow-up revealed the patient to be extremely well, with complete resolution of headaches, with a normal WCC and inflammatory markers. 3. Discussion VPS migration through the gastrointestinal system with resultant protrusion is rare. A recent review found 50 case reports of bowel perforation of VPS of which 3 had oral protrusion and 27 had anal protrusion of the catheters.9 Our search of the English language revealed that all cases of per oral extrusion to have occurred in children aged between 18 months and 9.5 years.9–12 There are several proposed mechanisms of VPS bowel perforation, which include direct damage to the bowel wall at the time of surgery, catheter tip adherence to the serosa of the viscus and perforation occurring secondary to chronic irritation.9 The former
Fig. 2. A three-dimensional reconstructed CT scan showing the position of the ventriculoperitoneal catheter. This figure is available in colour at www.sciencedirect.com.
Fig. 3. A sagittal CT scan showing migration of the ventriculoperitoneal catheter.
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is thought to account for the acute perforations and the latter for delayed perforations. Risks factors for bowel perforation include spring-coiled catheters, intra-abdominal infection, shunt material allergy, myelomeningocoele and conditions affecting bowel musculature including age and previous surgery.13–17 Distal catheter perforation is thought to be caused by the harder, spring-loaded type, which are less deformable. The distal catheter in this case, although older, was not spring-loaded but was less malleable than those currently used. In retrospect it seems likely that the catheter was close to the gastric wall or omentum, generating an inflammatory reaction as evidenced by the inflammatory markers and elevated WCC. The gradual erosion led to the catheter passing through the gastric wall, allowing migration to transgress the cardiac sphincter into the oesophagus. Diagnosis of bowel perforation requires a high clinical index of suspicion. Presentation may include signs of peritonitis, meningitis, or raised intracranial pressure. Rarely, as with our patient, protrusion of the catheter occurs orally. Appropriate investigations include WCC, CRP, and cultures with imaging to investigate the route of migration. Extrusion of the distal catheter, anally or orally, mandates removal and revision of the VPS. Theoretically, distal removal will reduce the risk of infection, but there is no evidence to support either proximal or distal removal. Assuming that the patient is shunt dependent, externalisation of the ventricular drain with concomitant antibiotics would seem appropriate. Once the cerebrospinal fluid and peripheral cell counts indicate absence of infection, the VPS can be revised. 4. Conclusion Migration of VPS catheters is rare. To our knowledge, this is the first reported case of a per oral extrusion occurring in an adult. The current use of more deformable distal silicone catheters appears to have reduced the incidence of perforation, yet it is prudent
to thoroughly investigate patients with VPS who have an undiagnosed asymptomatic inflammation. References 1. Akora B, Serarslan Y, Sangun O. Bowel perforation and transanal protusion of a ventricularperitoneal shunt catheter. Ped Neurosurg 2008;42:129–31. 2. Hok-Nam L, Tze-Ching T, Fung-Ching C. Transanal protusion of ventriculoperitoneal shunt. Surg Pract 2008;12:93–6. 3. Yilmaz N, Krymaz N, Yilmaz C, et al. Anal protrusion of ventriculo-peritoneal shunt catheter: report of two infants. J Ped Neuro 2004;2:241–4. 4. Vinchon M, Baroncini M, Laurent T, et al. Bowel perforation caused by peritoneal shunt catheters: diagnosis and treatment. Neurosurgery 2006;58:ONS76–82. 5. Wilson C, Betran V. Perforation of the bowel complicating peritoneal shunt for hydrocephalus: report of two cases. Am Surg 1966;32:601–3. 6. Surchev J, Georgiev K, Enchev Y, et al. Extremely rare complications in cerebrospinal fluid shunt operations. J Neurosurg Sci 2002;46:100–3. 7. Rehm A, Bannister C, Victoratos G. Scrotal perforation by ventriculoperitoneal shunt. Br J Neurosurg 1997;11:443–4. 8. Washington E, Holmes M, Haines S, et al. Ventriculoperitoneal shunt migration presenting with vaginal discharge and hydrosalpinx in a 16-year-old patient. Paed Emerg Care 2002;18:28–30. 9. Park C-K, Wang K-C, Seo JK, et al. Transoral protrusion of a peritoneal catheter: a case report and literature review. Childs Nerv Syst 2000;16:184–9. 10. Berhouma M, Messerer M, Houissa S, et al. Transoral protrusion of a peritoneal catheter: a rare complication of ventriculoperitoneal shunt. Ped Neurosurg 2008;44:169–71. 11. Danismend N, Kuday C. Unsual complication of ventriculoperitoneal shunt. Neurosurgery 1987;22:798. 12. Griffith J, DeFeo D. Peroral extrusion of a ventriculoperitoneal shunt catheter. Neurosurgery 1987;21:259–61. 13. Brownlee J, Brodkey J, Schaefer I. Colonic perforation by ventriculoperitoneal shunt tubing: a case of suspected silicone allergy. Surg Neurol 1998;49:21–4. 14. DiRocco C, Marchese E, Vellardi F. A survey of the first complication of newly implanted CSF devices for the treatment of nontumoral hydrocephalus. Childs Nerv Syst 1994;10:321–7. 15. Digray N, Thappa D, Arora M, et al. Silent bowel perforation and transanal prolapse of a ventriculoperitoneal shunt. Ped Surg Int 2000;16:94–5. 16. Adeloye A. Protrusion of ventriculoperitoneal shunt through the anus: report of two cases. E Afr Med J 1997;74:337–9. 17. Davidson R. Peritoneal bypass in the treatment of hydrocephalus: historical review and abdominal complications. J Neurol, Neurosurg Psychiatry 1976;39:640–6.
doi:10.1016/j.jocn.2008.12.029
Carotid cavernous fistula with cervical myelopathy Sang-Bae Ko, Chi Kyung Kim, Seung-Hoon Lee, Byung-Woo Yoon * Department of Neurology, Clinical Research Center for Stroke, Clinical Research Institute, College of Medicine, Seoul National University Hospital, Yongon-dong 28, Chongno-gu, Seoul, 110-744, Korea
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Article history: Received 22 October 2008 Accepted 16 December 2008
Keywords: Carotid cavernous fistula Cervical myelopathy Gamma-knife
a b s t r a c t We report a patient with carotid cavernous fistula (CCF) presenting with cervical myelopathy. The patient initially presented with ocular pain accompanied by binocular diplopia and was diagnosed with TolosaHunt syndrome at another institution. This patient experienced long-standing venous hypertension due to the delay in diagnosis. Posterior venous drainage from the CCF caused venous congestion in the brainstem and cervical spinal cord causing cervical myelopathy. Glue embolization using n-butyl-2-cyanoacrylate was attempted, but only partial embolization was possible because access to feeding arteries was limited. Stereotactic gamma-knife radiosurgery was performed as an alternative treatment, and effectively obliterated the CCF. However, the patient remained disabled due to cervical cord atrophy associated with long-standing venous hypertension. Ó 2009 Elsevier Ltd. All rights reserved.
1. Introduction * Corresponding author. Tel.: +82 2 2072 2278; fax: +82 2 3672 7553. E-mail address:
[email protected] (B.-W. Yoon).
Carotid cavernous fistula (CCF) is an abnormal communication between the internal carotid artery (ICA) and the cavernous sinus.1