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International Infant Hydrocephalus Study (IIHS): 5-year health outcome results of a prospective, multicenter comparison of endoscopic third ventriculostomy (ETV) and shunt for infant hydrocephalus

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Abstract

Introduction

One of the most important unanswered questions in pediatric hydrocephalus is determining whether treatment with endoscopic third ventriculostomy (ETV) versus shunt results in improved health status and quality of life (QOL). To answer this, the International Infant Hydrocephalus Study (IIHS) was started in 2005 as a prospective, multicenter study to compare ETV and shunt in infants (< 24 months old) with symptomatic triventricular hydrocephalus from aqueductal stenosis. Herein, we present the 5-year primary outcome results.

Methods

IIHS utilized a prospective comprehensive cohort design, in which patients received ETV or shunt, based on either randomization or parental preference. For this analysis, we pooled the randomized arm and the parental preference arm, analyzing them together. At 5 years of age, children were assessed with the Health Utilities Index Mark 2 (HUI-2) (primary outcome) and the Hydrocephalus Outcome Questionnaire (HOQ), a measure of QOL. Results were compared in an analysis of covariance, adjusting for baseline variables including age at surgery and baseline development status.

Results

From a total of 158 patients who met eligibility criteria, complete 5-year outcomes were available on 78 (19 treated initially with shunt, 61 treated initially with ETV), assessed at a mean age of 62.1 months (SD 6.3). The mean 5-year HUI-2 utility score was 0.90 (SD 0.19) for ETV and 0.94 (SD 0.10) for shunt (p = 0.21). The mean 5-year HOQ overall score was 0.81 (SD 0.15) for ETV and 0.85 (SD 0.12) for shunt (p = 0.42). Similarly, there were no significant differences noted between 5-year HOQ subscores (cognitive, social-emotional, physical) or developmental measures at 1, 2, and 3 years.

Conclusions

This is the first prospective direct comparison of long-term outcomes of ETV and shunt for infant hydrocephalus. These results suggest that overall health status and quality of life in this cohort of infants treated for aqueductal stenosis are high, with no significant difference between those treated initially with ETV or shunt.

Trial registration

NCT00652470

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Acknowledgements

The authors would like to extend special thanks to Adina Sherer, who ran the organizational logistics of this study and without whom the IIHS would not have been possible.

Steering committee: Shlomi Constantini (principal investigator), Spyros Sgouros, Abhaya V. Kulkarni

Consultant neurologist: Yael Leitner

Data safety monitoring committee: John RW Kestle (Chair), Douglas D Cochrane, Maurice Choux, Fleming Gjerris

Coordinating administrator: Adina Sherer

Participating investigator authors: Nejat Akalan, Burçak Bilginer (Ankara, Turkey); Ramon Navarro (Barcelona, Spain); Ljiljana Vujotic (Belgrade, Serbia); Hannes Haberl, Ulrich-Wilhelm Thomale (Berlin, Germany); Spyros Sgouros (Birmingham, UK); Graciela Zúccaro, Roberto Jaimovitch (Buenos Aires, Argentina); David Frim, Lori Loftis (Chicago, USA); Dale M. Swift, Brian Robertson, Lynn Gargan (Dallas, USA); László Bognár, László Novák, Georgina Cseke (Debrecen, Hungary); Armando Cama, Giuseppe Marcello Ravegnani (Genova, Italy); Matthias Preuß (Giessen/Leipzig, Germany); Henry W. Schroeder, Michael Fritsch, Joerg Baldauf (Greifswald, Germany); Marek Mandera, Jerzy Luszawski, Patrycja Skorupka (Katowice, Poland); Conor Mallucci, Dawn Williams (Liverpool, UK); Krzysztof Zakrzewski, Emilia Nowoslawska (Lodz, Poland); Chhitij Srivastava, Ashok K. Mahapatra, Raj Kumar, Rabi Narayan Sahu (Lucknow, India); Armen G. Melikian, Anton Korshunov, Anna Galstyan (Moscow, Russia); Ashish Suri, Deepak Gupta (New Delhi, India); J. André Grotenhuis, Erik J. van Lindert (Nijmegen, The Netherlands); José Aloysio da Costa Val (Nova Lima, Brazil); Concezio Di Rocco, Gianpiero Tamburrini (Rome, Italy); Samuel Tau Zymberg, Sergio Cavalheiro (São Paulo, Brazil); Ma Jie, Jiang Feng (Shanghai, China); Shlomi Constantini, Orna Friedman (Tel Aviv, Israel); Abhaya V. Kulkarni, Naheeda Rajmohamed (Toronto, Canada); Marcin Roszkowski, Slawomir Barszcz (Warsaw, Poland)

The following centers (and investigators) participated in the IIHS, but did not enroll any patients: Baltimore, MD, USA (George Jallo); Gainesville, FL, USA (David W. Pincus, Bridget Richter); Kiel, Germany (HM Mehdorn, Susan Schultka); London, Ontario, Canada (Sandrine de Ribaupierre); London, UK (Dominic Thompson, Silvia Gatscher); Mainz, Germany (Wolfgang Wagner, Dorothee Koch); Reggio Calabria, Italy (Saverio Cipri, Claudio Zaccone); Winnipeg, Manitoba, Canada (Patrick McDonald)

Author information

Authors and Affiliations

  1. The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada

    Abhaya V. Kulkarni

  2. Department of Pediatric Neurosurgery, Mitera Children’s Hospital, University of Athens Medical School, Athens, Greece

    Spyros Sgouros

  3. Institute of Child Development, Dana Children’s Hospital, Tel Aviv Sourasky Medical Center, Tel Aviv University, Tel Aviv, Israel

    Yael Leitner

  4. Department of Pediatric Neurosurgery, Dana Children’s Hospital, Tel Aviv Sourasky Medical Center, Tel Aviv University, Tel Aviv, Israel

    Shlomi Constantini

Authors
  1. Abhaya V. Kulkarni
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  2. Spyros Sgouros
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  3. Yael Leitner
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  4. Shlomi Constantini
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for the International Infant Hydrocephalus Study Investigators

Corresponding author

Correspondence to Shlomi Constantini.

Ethics declarations

The IIHS was publically registered (clinicaltrials.gov, NCT00652470) and received ethics approval from all participating institutions.

Conflict of interest

On behalf of all authors, the corresponding author states that there is no conflict of interest.

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Kulkarni, A.V., Sgouros, S., Leitner, Y. et al. International Infant Hydrocephalus Study (IIHS): 5-year health outcome results of a prospective, multicenter comparison of endoscopic third ventriculostomy (ETV) and shunt for infant hydrocephalus. Childs Nerv Syst 34, 2391–2397 (2018). https://doi.org/10.1007/s00381-018-3896-5

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