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Current trends in pediatric moyamoya: a survey of international practitioners

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Abstract

Objective

Moyamoya angiopathy (MM) is a chronic, progressive steno-occlusive arteriopathy of the distal internal carotid artery and its proximal branches. MM is recognized as a shared end-pathway common to a broad range of inciting pathologies, suggesting that tailored management is important. Pediatric MM differs from MM in adults. Currently, there are many uncertainties and controversies regarding the diagnosis and management of children with MM. Hence, we conducted an international survey to identify the contemporary management trends followed worldwide.

Methods

A survey relating to lifestyle modifications, medical management, diagnosis, surgical management, and follow-up for pediatric MM was circulated across web-based platforms, through various international pediatric neurological and neurosurgical societies. Data collected included geographic region of practice, experience, responses to questions, and comments.

Results

One hundred twenty-seven responses were evaluated (104 neurosurgeons and 23 neurologists, from 32 countries, across 6 continents). We found wide variations in the recommendations for management and lifestyle modification, with significant differences between regions of practice. Eighty percent recommend restrictions on physical activity, particularly for symptomatic and non-operated patients. Eighty-four percent prescribe aspirin. Sixty-five percent perform indirect revascularization. Seventy-eight percent recommend performing a staged surgery for bilateral MM. Only 26% perform acetazolamide challenge SPECT to evaluate brain perfusion. Only 15% of responders were from highly experienced centers.

Conclusion

This survey reflects the contemporary trends in management of pediatric MM, while highlighting the heterogeneity in the management approach of these patients. There is a need for multicenter, international studies to evaluate the safety, efficacy, and long-term outcome of various aspects of treatment of these patients.

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Acknowledgements

We thank Dr Kirsten Van Baarsen (Princess Maxima Center for Pediatric Oncology, department of pediatric oncology neurosurgery, Heidelberglaan, Utrecht, The Netherlands) and Dr Danil Kozyrev (Pediatric Neurosurgery Department, Dana-Dwek Children’s Hospital, Tel Aviv Sourasky Medical Center) for their participation and valuable feedback in the trial version of the survey. We thank AASPN, EPNS, ESPN, IPSS, ISPN, and WFNS for heping in the distribution of the survey. We also thank Dr Nobuhito Morota, Department of Neurosurgery, Division of Pediatric Neurosurgery, Kitasato University School of Medicine, Tokyo, Japan (Scientific chair of AASPN). We thank Ms Adina Sherer and Ms Swetha Kumar (Division of Ophthalmology, Tel Aviv Sourasky medical center) for the editorial asistance in non-intellectual content.

We thank the following respondents for their invaluable contribution to this survey:

Albert van der Zwan (UMCU, The Netherlands); Aldo Jose F. da Silva (Division of Pediatric Neurosurgery, Santa Mônica Maternity School (SMMS), Maceió, Alagoas, Brazil); Alexandre Giannetti (Professor. Federal University of Minas Gerais, Brazil); Alvaro Cordoba (Chairman, Neurosurgert ASESP Montevideo, Uruguay); Ando Ryo (Chiba Children's Hospital, Japan); André Grotenhuis J., MD, PhD (Emeritus Professor, Dep. of Neurosurgery, Radboud University Nijmegen Medical Center, Nijmegen, The Netherlands); Andrea Bartoli (Department of Neurosurgery, Geneva University Hospitals, Geneva, Switzerland); Andres Goycoolea (Instituto de Neurocirugia, Chile); Antonio Bellas (Instituto Fernandes Figueira, Rio de Janeiro, Brazil); Artur Da Cunha (Hospital da Restauração, Recife, Brazil); Bermans Iskandar (University of Wisconsin, Madison, USA); Catherine Amlie-Lefond (Seattle Children's Hospital, Washington, USA); Cavalcante Tiago (Brazilian pediatric neurosurgeon society); David. H. Harter (NYU, USA); Dinark Viana (HCFMRPUSP, Brazil); David I. Sandberg, MD, FAANS, FACS, FAAP (Professor and Director of Pediatric Neurosurgery, Dr Marnie Rose Professorship in Pediatric Neurosurgery Departments of Pediatric Surgery and Neurosurgery, McGovern Medical School and Children’s Memorial Hermann Hospital Co-director of Pediatric Brain Tumor Program, University of Texas MD Anderson Cancer Center); Ekawut Chankaew (Mahidol University, Bangkok); Elizabeth (Liza) Pulcine (Staff Physician, Division of Neurology, Assistant Professor, University of Toronto, The Hospital for Sick Children, Toronto, Canada); Enrico Ghizoni MD, PhD (Director of Neurosurgery, University of Campinas, Brazil); Federico Auad (ISPN); Flavio Giordano, MD (Department of Neurosurgery - Meyer Pediatric Hospital, Firenze, Italy); Frank Van Calenbergh (UZ Leuven, Belgium); Frederick Boop MD (University of Tennessee Health Sciences Center, Memphis, Tn. USA); Gabriel Campos L (Instituto de Neurocirugía Asenjo, Santiago, Chile); Greg James (Associate Professor University College London/Consultant Neurosurgeon Great Ormond Street Hospital, London, UK); Griselda Ramirez (UMAE Hospital de Pediatria, National Medical Center Century, Mexico City, Mexico); Dr. Güliz Acker (Charite Universitätsmedizin Berlin, Germany); Hakan Karabağlı (Selcuk University Faculty of Medicine Hospital, Konya, Turkey); Hamilton Matushita (Chief Professor Pediatric Neurosurgery, São Paulo University, Brazil); Hiroaki Sakamoto (Department of Pediatric Neurosurgery, Osaka City General Hospital, Japan); Isao Date, MD (Okayama University Graduate School of Medicine, Japan); James M Johnston, Jr. (University of Alabama at Birmingham/Children's of Alabama, USA); Jay Riva-Cambrin MD, MSc (Professor, University of Calgary, Canada); Jerome Yager (University of Alberta, Canada); José Aloysio Costa Val Filho (Head Vila da Serra/Biocor, Brazil); José Francisco M. Salomão, MD PhD (National Institute of Women, Children and Adolescents Health Fernandes Figueira, Oswaldo Cruz Foundation, IFF-Fiocruz, Brazil); José Hinojosa (Department of Neurosurgery Hospital Sant Joan de Déu, Barcelona, Spain); Jun Kurihara (Saitama Children’s Medical Center, Department of Neurosurgery, Japan); Kirsten van Baarsen, MD PhD (Princess Maxima Center for Pediatric Oncology, department of pediatric oncology neurosurgery, Heidelberglaan 25, 3584 EA Utrecht, The Netherlands); Kristin P. Guilliams, MD MSCI (Departments of Neurology and Pediatrics, Washington University School of Medicine, St. Louis MO, USA); Leopoldo Mandic Ferreira Furtado (Department of Pediatric neurosurgery, Vila da Serra Hospital, Brazil); Leyila Kaseka (The Hospital for Sick Children, Toronto, Canada); Lisa R. Sun, MD (The Johns Hopkins School of Medicine, Department of Neurology, USA); Llewellyn Padayachy (Department of Neurosurgery, University of Pretoria, South Africa); Lu Ma (Dept. of Neurosurgery, West China Hospital, Sichuan University, China); Luca Massimi (Fondazione Policlinico A. Gemelli IRCCS, Rome, Italy); Mahendra Moharir MD, MSc, FRACP (Associate Professor, Dept. of Pediatrics, University of Toronto, Staff Physician & Clinical Director, Division of Neurology, Clinical Director, Stroke Program, Division of Neurology, The Hospital for Sick Children, Toronto, Ontario, Canada); Mahesh Kamate, (Professor of pediatric neurology, KAHER'S J N medical college, Belgaum, India); Márcio de Miranda Chaves Christiani (Instituto Nacional de Câncer, Brazil); Marek Mandera (Professor of Neurosurgery, Head of the Department of Pediatric Neurosurgery, Medical University of Silesia, Poland); Mario Teo, (Consultant Neurosurgeon, Bristol Institute of Clinical Neuroscience, UK); Mark Krieger (CHLA, LA, USA); Masayuki Sasaki (National Center of Neurology and Psychiatry’Kodaira, Japan); Matthieu Vinchon (Pediatric neurosurgery, Lille university hospital, Lille, France); Megan Barry (Children's Hospital Colorado, USA); Meng-Fai Kuo MD, PhD (Division of Neurosurgery, Department of Surgery, National Taiwan University Hospital and National Taiwan University College of Medicine, Taiwan); Michel R. Scott, MD (Boston Children’s Hospital; Harvard Medical School, Boston, USA); Nejat Akalan (Medipol University Dept. of Neurosurgery, Istanbul, Turkey); Nobuhito Morota (Department of Neurosurgery, Division of Pediatric Neurosurgery, Kitasato University School of Medicine, Tokyo, Japan); Paola Pergami (CNMC, Washington, D.C., USA); Paolo Frassanito (Pediatric Neurosurgery, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy); Patricia L. Musolino MD, PhD (Department of Neurology and Center for Genomic Medicine, Massachusetts General Hospital, Harvard Medical School, USA); Ranjith K Moorthy (Department of Neurological Sciences, Christian Medical College, Vellore, India); Rebecca Ichord MD (CHOP, Philadelphia, USA); Ricardo Santos de Oliveira (Associate Professor, Division of Neurosurgery, School of Medicine of Ribeirao Preto University of Sao Paulo, Brazil); Richard C. E. Anderson, MD (Director of Pediatric Neurosurgery, Valley Hospital; Director of Neurosurgery, St. Joseph’s University Medical Center, USA); Richard Edwards (Bristol Royal Hospital for Children, Bristol, UK); Richard G. Ellenbogen (Seattle Children's Hospital; University of Washington, USA); Robert, Hsin-Hung Chen (Taipei Veterans General Hospital, Taipei, Taiwan); Ronan Vieira (The Hospital for Sick Children, Toronto, Canada); Sahar M. A. Hassanein (Professor of Pediatrics, Pediatric neurology unit, Faculty of Medicine, Ain Shams University, Cairo, Egypt); Samer K. Elbabaa, MD, FAANS, FAAP, FACS (Chief of Pediatric Neurosurgery, Director, Leon Pediatric Neuroscience Center of Excellence, Arnold Palmer Hospital for Children, Orlando, Florida, USA); Sang-Dae KIM (Korea University Ansan Hospital, South Korea); Santosh Mohan Rao (Consultant Neurosurgeon, Rainbow Children’s Hospital, Chennai, India); Satoshi Ihara (Division of Neurosurgery, Tokyo Metropolitan Children's Medical Center, Tokyo, Japan); Satoshi Kuroda (Department of Neurosurgery, Toyama University Hospital, Toyama, Japan); Seung-Ki Kim (Department of Neurosurgery, Seoul National University College of Medicine, Seoul National University Hospital); Shibu Pillai (Senior Consultant Neurosurgeon, Mazumdar Shaw Medical Center, Narayana Health, Bangalore, India); Sudhakar Vadivelu, DO (Associate Professor, Department of Neurosurgery and Radiology, University of Cincinnati College of Medicine); Sudipta Kumer Mukherjee (Assistant Professor, Department of Neurosurgery, Shahid Sheikh Abu Naser Hospital, Khulna, Bangladesh); Suman Ghosh MD (Clinical Assistant Professor, Division of Pediatric Neurology, University of Florida); Sung Kyoo Hwang (Kyungpook National University Hospital, Korea); Suresh N. Magge (Associate Professor of Neurosurgery and Pediatrics, Children's National Hospital, Washington, DC, George Washington University School of Medicine, USA); Talabaev Mikle (Republican scientific and practical center of neurosurgery Belarus Minsk, Belarus); Thiago Lyrio Teixeira (HINSG, Espírito Santo, Brazil); Tomomi Kimiwada (Miyagi Children's Hospital, Sendai, Japan); Vijeya Ganesan (UCL Great Ormond Street Institute of Child Health, London, UK); Vitaly Siomin, MD (Miami Neuroscience Institute, Miami Cancer Center, Baptist Hospital of Miami); Xinyu Yang, M.D. & Ph.D. (Department of Neurosurgery Tianjin Medical University General Hospital, China); Zhiyuan Yu (Dept. of Neurosurgery, West China Hospital, Sichuan University, China).

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Authors and Affiliations

  1. Pediatric Neurosurgery Department, Dana-Dwek Children’s Hospital, Tel Aviv Sourasky Medical Center, Tel Aviv University, Tel Aviv, Israel

    Harishchandra Lalgudi Srinivasan, Shlomi Constantini & Jonathan Roth

  2. Pediatric Neurology Institute, Dana-Dwek Children’s Hospital, Tel Aviv Sourasky Medical Center, Tel Aviv, Israel

    Moran Hausman-Kedem

  3. Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel

    Moran Hausman-Kedem, Shlomi Constantini & Jonathan Roth

  4. Pediatric Cerebrovascular Surgery, Pediatric Neurosurgery, Children’s Hospital Boston/Harvard Medical School, Boston, MA, USA

    Edward R. Smith

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Contributions

Harishchandra Lalgudi Srinivasan: Designed and conceptualized the study; had major role in the acquisition of data; analyzed the data; drafted the manuscript for intellectual content

Moran Hausman-Kedem: Intepreted the data; revised the manuscript for intellectual content

Edward R Smith: Revised the manuscript for intellectual content

Shlomi Constantini: Revised the manuscript for intellectual content

Jonathan Roth: Designed and conceptualized the study; analyzed the data; revised the manuscript for intellectual content

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Correspondence to Jonathan Roth.

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Harishchandra Lalgudi Srinivasan reports no disclosure.

Moran Hausman-Kedem reports no disclosure.

Edward R Smith reports no disclosure.

Shlomi Constantini reports no disclosure.

Jonathan Roth reports no disclosure.

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Srinivasan, H.L., Hausman-Kedem, M., Smith, E.R. et al. Current trends in pediatric moyamoya: a survey of international practitioners. Childs Nerv Syst 37, 2011–2023 (2021). https://doi.org/10.1007/s00381-021-05074-2

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