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The international infant hydrocephalus study: concept and rational

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Abstract

Introduction

During the recent meetings of the International Study Group on Neuroendoscopy and the International Society for Pediatric Neurosurgery, the consensus view emerged that there is a need to assess the value and efficacy of neuroendoscopic procedures against shunting in a scientific manner, to resolve long-lasting debates on the subject.

Material and methods

A prospective randomized, controlled trial of endoscopic third ventriculostomy vs shunting in children presenting under the age of 2 years with pure aqueduct stenosis is been proposed and organized (the International Infant Hydrocephalus Study, IIHS). The participating surgeons must adhere to the philosophy of randomization and be suitably experienced in endoscopic techniques in infants. The primary outcome of the trial will be the overall health-related quality of life of these children at 5 years of age. Hence, the study is focusing on the effect of surgery on neurodevelopment, rather than the less important issue of shunt or stoma survival, that has been debated extensively with no conclusion so far. Intention-to-treat analysis will be performed according to the first surgery. Secondary outcomes such as complication and reoperation rate, total hospitalization time and cost, need for repeat imaging, and others will be analyzed as well.

Results

Pure aqueduct stenosis is relatively rare, making recruitment problematic, but has been chosen to avoid other confounding factors that could influence outcome. More than 25 centers worldwide have committed already to patient recruitment to the study. It is anticipated that recruitment will last for 2 years, aiming for 91 patients per arm. The study has started recruiting patients already in some countries.

Conclusion

It is hoped that the trial will not only provide answers to unsettled debates on the value of neuroendoscopy but also create a network of collaborating pediatric neurosurgeons for future initiatives.

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References

  1. Abstracts of the Second World Conference of the International Study Group on Neuroendoscopy (ISGNE) (2003) Castel dell’ Ovo, Naples, Italy, 11–13 September 2003. Childs Nerv Syst 19:687–709

    Google Scholar 

  2. Ammirati M, Raimondi A (1987) Cerebrospinal fluid shunt infections in children. A study on the relationship between the etiology of hydrocephalus, age at the time of shunt placement, and infection rate. Childs Nerv Syst 3:106–109

    Article  PubMed  Google Scholar 

  3. Baskin JJ, Manwaring KH, Rekate HL (1998) Ventricular shunt removal: the ultimate treatment of the slit ventricle syndrome. J Neurosurg 88:478–484

    PubMed  Google Scholar 

  4. Beems T, Grotenhuis JA (2002) Is the success rate of endoscopic third ventriculostomy age-dependent? An analysis of the results of endoscopic third ventriculostomy in young children. Childs Nerv Syst 18:605–608

    Article  PubMed  Google Scholar 

  5. Brockmeyer D, Abtin K, Carey L, Walker ML (1998) Endoscopic third ventriculostomy: an outcome analysis. Pediatr Neurosurg 28:236–240

    Article  PubMed  Google Scholar 

  6. Burn SC, Saxena A, Tyagi A, Chumas PD (2003) Endoscopic third ventriculostomy in children during the first year of life. Childs Nerv Syst 19:618–619

    Google Scholar 

  7. Buxton N, Macarthur D, Mallucci C, Punt J, Vloeberghs M (1998a) Neuroendoscopic third ventriculostomy in patients less than 1 year old. Pediatr Neurosurg 29:73–76

    Article  PubMed  Google Scholar 

  8. Buxton N, Macarthur D, Mallucci C, Punt J, Vloeberghs M (1998b) Neuroendoscopy in the premature population. Childs Nerv Syst 14:649–652

    Article  PubMed  Google Scholar 

  9. Cinalli G (1999) Alternatives to shunting. Childs Nerv Syst 15:718–731

    Article  PubMed  Google Scholar 

  10. Cinalli G, Sainte-Rose C, Chumas P, Zerah M, Brunelle F, Lot G, Pierre Kahn A, Renier D (1999) Failure of third ventriculostomy in the treatment of aqueductal stenosis in children. J Neurosurg 90:448–454

    PubMed  Google Scholar 

  11. Cinalli G, Salazar C, Mallucci C, Yada JZ, Zerah M, Sainte-Rose C (1998) The role of endoscopic third ventriculostomy in the management of shunt malfunction. Neurosurgery 43:1323–1327; discussion 1327–1329

    Article  PubMed  Google Scholar 

  12. Costa Val JA, Noci TR, Lara A, Herval LM, Serrano P (2003) Endoscopic third ventriculostomy in children under 2 years old: experience of a neuropediatric service in Brazil. Childs Nerv Syst 19:688

    Google Scholar 

  13. Dennis M, Barnes MA (1993) Oral discourse after early-onset hydrocephalus: linguistic ambiguity, figurative language, speech acts, and script-based inferences. J Pediatr Psychol 18:639–652

    PubMed  Google Scholar 

  14. Fritsch MJ, Medhorn HM (2003) Indication and controversies for endoscopic third ventriculostomy in children. Childs Nerv Syst 19:706–707

    Google Scholar 

  15. Genitori L, Peretta P, Mussa F, Giordano F (2003) Endoscopic third ventriculostomy in children: are age and etiology of hydrocephalus predictive factors influencing the outcome in primary and secondary treated patients? A series of 254 patients and 276 procedures. Childs Nerv Syst 19:618

    Google Scholar 

  16. Goumnerova LC, Frim DM (1997) Treatment of hydrocephalus with third ventriculocisternostomy: outcome and CSF flow patterns. Pediatr Neurosurg 27:149–152

    PubMed  Google Scholar 

  17. Grotenhuis JA, Beems T (1999) The role of endoscopic third ventriculostomy in the treatment of hydrocephalus associated with myelomeningocele. Minim Invasive Neurosurg 42:161–162

    Google Scholar 

  18. Hirsch JF, Pierre-Kahn A, Renier D, Sainte-Rose C, Hoppe-Hirsch E (1984) The Dandy–Walker malformation. A review of 40 cases. J Neurosurg 61:515–522

    PubMed  Google Scholar 

  19. Hoffman HJ, Harwood-Nash D, Gilday DL (1980) Percutaneous third ventriculostomy in the management of noncommunicating hydrocephalus. Neurosurgery 7:313–321

    PubMed  Google Scholar 

  20. Hopf NJ, Grunert P, Fries G, Resch KD, Perneczky A (1999) Endoscopic third ventriculostomy: outcome analysis of 100 consecutive procedures. Neurosurgery 44:795–804; discussion 804–796

    Article  PubMed  Google Scholar 

  21. Horsman J, Furlong W, Feeny D, Torrance G (2003) The Health Utilities Index (HUI): concepts, measurement properties, and applications. Health Qual Life Outcomes 16:54

    Article  Google Scholar 

  22. Hurley AD, Bell S (1994) Educational and vocational outcome of adults with spina bifida in relationship to neuropsychological testing. Eur J Pediatr Surg 4(Suppl 1):17–18

    PubMed  Google Scholar 

  23. Hurley AD, Laatsch LK, Dorman C (1983) Comparison of spina bifida, hydrocephalic patients and matched controls on neuropsychological tests. Z Kinderchir 38(Suppl 2):116–118

    PubMed  Google Scholar 

  24. Husain M, Jha D, Vatsal DK, Thaman D, Gupta A, Husain N, Gupta RK (2003) Neuro-endoscopic surgery—experience and outcome analysis of 102 consecutive procedures in a busy neurosurgical centre of India. Acta Neurochir (Wien) 145:369–376

    Google Scholar 

  25. Javadpour M, Mallucci C, Brodbelt A, Golash A, May P (2001) The impact of endoscopic third ventriculostomy on the management of newly diagnosed hydrocephalus in infants. Pediatr Neurosurg 35:131–135

    Article  PubMed  Google Scholar 

  26. Jones RF, Kwok BC, Stening WA, Vonau M (1994) Neuroendoscopic third ventriculostomy. A practical alternative to extracranial shunts in non-communicating hydrocephalus. Acta Neurochir Suppl (Wien) 61:79–83

    Google Scholar 

  27. Jones RF, Kwok BC, Stening WA, Vonau M (1996) Third ventriculostomy for hydrocephalus associated with spinal dysraphism: indications and contraindications. Eur J Pediatr Surg 6(Suppl 1):5–6

    PubMed  Google Scholar 

  28. Jones RF, Stening WA, Brydon M (1990) Endoscopic third ventriculostomy. Neurosurgery 26:86–91; discussion 91–82

    Article  PubMed  Google Scholar 

  29. Jones RF, Stening WA, Kwok BC, Sands TM (1993) Third ventriculostomy for shunt infections in children. Neurosurgery 32:855–859; discussion 860

    PubMed  Google Scholar 

  30. Kamikawa S, Inui A, Kobayashi N, Kuwamura K, Kasuga M, Yamadori T, Tamaki N (2001) Endoscopic treatment of hydrocephalus in children: a controlled study using newly developed Yamadori-type ventriculoscopes. Minim Invasive Neurosurg 44:25–30

    Article  PubMed  Google Scholar 

  31. Kim SK, Wang KC, Cho BK (2000) Surgical outcome of pediatric hydrocephalus treated by endoscopic III ventriculostomy: prognostic factors and interpretation of postoperative neuroimaging Childs Nerv Syst 16(3):161–169

    Article  PubMed  Google Scholar 

  32. Kulkarni AV, Drake JM, Rabin D, Dirks PB, Humphreys RP, Drake JT (2004) Measuring the health status of children with hydrocephalus using a new outcome measure. J Neurosurg Pediatr 101:141–146

    Google Scholar 

  33. Kulkarni AV, Rabin D, Drake JM (2004) An instrument to measure the health status of children with hydrocephalus: the Hydrocephalus Outcome Questionnaire. J Neurosurg Pediatr 101:134–140

    Google Scholar 

  34. Macarthur DC, Buxton N, Vloeberghs M, Punt J (2001) The effectiveness of neuroendoscopic interventions in children with brain tumours. Childs Nerv Syst 17:589–594

    Article  PubMed  Google Scholar 

  35. Mallucci C, Vloeberghs M, Punt J (1997) Neuroendoscopic third ventriculostomy: the first line treatment for blocked ventriculo-peritoneal shunts? Childs Nerv Syst 13:498

    Google Scholar 

  36. Mallucci C, Vloeberghs M, Punt J (1997) Should shunts be revised when neuroendoscopic third ventriculostomy is available? Br J Neurosurg 11:473

    Google Scholar 

  37. Meier U, Zeilinger FS, Schonherr B (2000) Endoscopic ventriculostomy versus shunt operation in normal pressure hydrocephalus: diagnostics and indication. Acta Neurochir Suppl (Wien) 76:563–566

    Google Scholar 

  38. Meier U, Zeilinger FS, Schonherr B (2000) Endoscopic ventriculostomy versus shunt operation in normal pressure hydrocephalus: diagnostics and indication. Minim Invasive Neurosurg 43:87–90

    Article  PubMed  Google Scholar 

  39. Mitchell P, Mathew B (1999) Third ventriculostomy in normal pressure hydrocephalus. Br J Neurosurg 13:382–385

    Article  PubMed  Google Scholar 

  40. Mori H, Nishiyama K, Tanaka R (2003) Endoscopic third ventriculostomy for pediatric hydrocephalic patients: discussion of the indication and strategy for shunt removal. Childs Nerv Syst 19:618

    Google Scholar 

  41. Murshid WR (2000) Endoscopic third ventriculostomy: towards more indications for the treatment of non-communicating hydrocephalus. Minim Invasive Neurosurg 43:75–82

    Article  PubMed  Google Scholar 

  42. Olschewski M, Schumacher M, Davis KB (1992) Analysis of randomized and nonrandomized patients in clinical trials using the comprehensive cohort follow-up study design. Control Clin Trials 13:226–239

    Article  PubMed  Google Scholar 

  43. Osman-Farah J, Javadpour M, Buxton N, May P, Mallucci C (2003) Endoscopic ventricular fenestration and ETV for the treatment of multicompartmental neonatal hydrocephalus. Childs Nerv Syst 19:702–703

    Google Scholar 

  44. Pelegrin Valero C, Tirapu Ustarroz J, Landa Gonzalez N (2001) Neuropsychological deficits in hydrocephalus associated spina bifida. Rev Neurol 32:489–497

    PubMed  Google Scholar 

  45. Pople IK, Bayston R, Hayward RD (1992) Infection of cerebrospinal fluid shunts in infants: a study of etiological factors. J Neurosurg 77:29–36

    PubMed  Google Scholar 

  46. Renier D, Lacombe J, Pierre-Kahn A, Sainte-Rose C, Hirsch JF (1984) Factors causing acute shunt infection. Computer analysis of 1174 operations. J Neurosurg 61:1072–1078

    PubMed  Google Scholar 

  47. Sainte-Rose C, Piatt JH, Renier D, Pierre-Kahn A, Hirsch JF, Hoffman HJ, Humphreys RP, Hendrick EB (1992) Mechanical complications in shunts. Pediatr Neurosurg 17:2–9

    Google Scholar 

  48. Scarrow AM, Levy EI, Pascucci L, Albright AL (2000) Outcome analysis of endoscopic III ventriculostomy. Childs Nerv Syst 16:442–444

    Article  PubMed  Google Scholar 

  49. Scavarda D, Bednarek N, Litre F, Koch C, Lena G, Morville P, Rousseaux P (2003) Acquired aqueductal stenosis in preterm infants: an indication for neuroendoscopic third ventriculostomy. Childs Nerv Syst 19:756–759

    Article  PubMed  Google Scholar 

  50. Siomin V, Cinalli G, Grotenhuis A, Golash A, Oi S, Kothbauer K, Weiner H, Roth J, Beni-Adani L, Pierre-Kahn A, Takahashi Y, Mallucci C, Abbott R, Wisoff J, Constantini S (2002) Endoscopic third ventriculostomy in patients with cerebrospinal fluid infection and/or hemorrhage. J Neurosurg 97:519–524

    PubMed  Google Scholar 

  51. Siomin V, Weiner H, Wisoff J, Cinalli G, Pierre-Kahn A, Saint-Rose C, Abbott R, Elran H, Beni-Adani L, Ouaknine G, Constantini S (2001) Repeat endoscopic third ventriculostomy: is it worth trying? Childs Nerv Syst 17:551–555

    Article  PubMed  Google Scholar 

  52. Smyth MD, Tubbs RS, Wellons JC III, Oakes WJ, Blount JP, Grabb PA (2003) Endoscopic third ventriculostomy for hydrocephalus secondary to central nervous system infection or intraventricular hemorrhage in children. Pediatr Neurosurg 39:258–263

    Article  PubMed  Google Scholar 

  53. Snow JH, Prince M, Souheaver G, Ashcraft E, Stefans V, Edmonds J (1994) Neuropsychological patterns of adolescents and young adults with spina bifida. Arch Clin Neuropsychol 9:277–287

    Article  PubMed  Google Scholar 

  54. St George E, Natarajan K, Sgouros S (2004) Changes in the ventricular volume in hydrocephalic children following successful endoscopic third ventriculostomy. Childs Nerv Syst 20:834–838

    Article  PubMed  Google Scholar 

  55. Teo C, Jones R (1996) Management of hydrocephalus by endoscopic third ventriculostomy in patients with myelomeningocele. Pediatr Neurosurg 25:57–63

    PubMed  Google Scholar 

  56. Teo C, Rahman S, Boop FA, Cherny B (1996) Complications of endoscopic neurosurgery. Childs Nerv Syst 12:248–253

    Article  PubMed  Google Scholar 

  57. Wagner W, Koch D (2003) Success or failure of endoscopic third ventriculostomy in young infants: is there a correlation with age in patients up to 12 months old? Childs Nerv Syst 19:618

    Google Scholar 

  58. Walters BC, Hoffman HJ, Hendrick EB, Humphreys RP (1984) Cerebrospinal fluid shunt infection. Influences on initial management and subsequent outcome. J Neurosurg 60:1014–1021

    PubMed  Google Scholar 

  59. Wellons JC III, Tubbs RS, Banks JT, Grabb B, Blount JP, Oakes WJ, Grabb PA (2002) Long-term control of hydrocephalus via endoscopic third ventriculostomy in children with tectal plate gliomas. Neurosurgery 51:63–67; discussion 67–68

    Article  PubMed  Google Scholar 

  60. Wills KE (1993) Neuropsychological functioning in children with spina bifida and/or hydrocephalus. J Clin Child Psychol 22:247–265

    Google Scholar 

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Authors and Affiliations

  1. Birmingham Children’s Hospital, Birmingham, UK

    S. Sgouros

  2. Hospital for Sick Children, Toronto, Canada

    A. V. Kulkharni

  3. Department Pediatric Neurosurgery, Dana Children’s Hospital, Tel Aviv Sourasky Medical Center, 6 Weizman Street, Tel Aviv, 64239, Israel

    S. Constantini

Authors
  1. S. Sgouros
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  2. A. V. Kulkharni
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  3. S. Constantini
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Correspondence to S. Constantini.

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Sgouros, S., Kulkharni, A.V. & Constantini, S. The international infant hydrocephalus study: concept and rational. Childs Nerv Syst 22, 338–345 (2006). https://doi.org/10.1007/s00381-005-1253-y

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