Cranial and ventricular size following shunting or endoscopic third ventriculostomy (ETV) in infants with aqueductal stenosis: further insights from the International Infant Hydrocephalus Study (IIHS)

Abstract

Purpose

The craniometrics of head circumference (HC) and ventricular size are part of the clinical assessment of infants with hydrocephalus and are often utilized in conjunction with other clinical and radiological parameters to determine the success of treatment. We aimed to assess the effect of endoscopic third ventriculostomy (ETV) and shunting on craniometric measurements during the follow-up of a cohort of infants with symptomatic triventricular hydrocephalus secondary to aqueductal stenosis.

Methods

We performed a post hoc analysis of data from the International Infant Hydrocephalus Study (IIHS)—a prospective, multicenter study of infants (< 24 months old) with hydrocephalus from aqueductal stenosis who were treated with either an ETV or shunt. During various stages of a 5-year follow-up period, the following craniometrics were measured: HC, HC centile, HC z-score, and frontal-occipital horn ratio (FOR). Data were compared in an analysis of covariance, adjusting for baseline variables including age at surgery and sex.

Results

Of 158 enrolled patients, 115 underwent an ETV, while 43 received a shunt. Both procedures led to improvements in the mean HC centile position and z-score, a trend which continued until the 5-year assessment point. A similar trend was noted for FOR which was measured at 12 months and 3 years following initial treatment. Although the values were consistently higher for ETV compared with shunt, the differences in HC value, centile, and z-score were not significant. ETV was associated with a significantly higher FOR compared with shunting at 12 months (0.52 vs 0.44; p = 0.002) and 3 years (0.46 vs 0.38; p = 0.03) of follow-up.

Conclusion

ETV and shunting led to improvements in HC centile, z-score, and FOR measurements during long-term follow-up of infants with hydrocephalus secondary to aqueductal stenosis. Head size did not significantly differ between the treatment groups during follow-up, however ventricle size was greater in those undergoing ETV when measured at 1 and 3 years following treatment.

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Acknowledgments

The authors would like to extend a 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, and Abhaya V. Kulkarni

Consultant Neurologist: Yael Leitner

Data Safety Monitoring Committee: John RW Kestle (Chair), Douglas D Cochrane, Maurice Choux, and 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, and Slawomir Barszcz (Warsaw, Poland)

The following centres (and investigators) participated in the IIHS, but did not enroll any patients: Baltimore, Maryland, USA (George Jallo); Gainesville, Florida, 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); and Winnipeg, Manitoba, Canada (Patrick McDonald).

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Contributions

AVK, SS, CS, and ICC contributed to the study conception and design. Data collection was performed by AVK, SS, CS, and collaborators from the IIHS. Material preparation and data analysis were performed by ICC and AVK. The first draft of the manuscript was written by ICC, and all authors commented on previous versions of the manuscript. All authors have read and approved the final manuscript.

Corresponding author

Correspondence to Abhaya V. Kulkarni.

Ethics declarations

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

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The members of the Steering Committee have no conflicts of interest with respect to this work.

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Coulter, I.C., Kulkarni, A.V., Sgouros, S. et al. Cranial and ventricular size following shunting or endoscopic third ventriculostomy (ETV) in infants with aqueductal stenosis: further insights from the International Infant Hydrocephalus Study (IIHS). Childs Nerv Syst 36, 1407–1414 (2020). https://doi.org/10.1007/s00381-020-04503-y

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Keywords

  • International Infant Hydrocephalus Study
  • Infant hydrocephalus
  • Endoscopic third ventriculostomy
  • Shunt