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A proposal for case definitions and outcome measures in studies of infantile spasms and West syndrome: Consensus statement of the west Delphi group

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单位: [1]Royal United Hosp, Bath Unit Res Paediat, Bath BA1 3NG, Avon, England [2]Univ Bath, Sch Hlth, Bath BA2 7AY, Avon, England [3]Ist Neurol C Besta, I-20133 Milan, Italy [4]Univ Calif Irvine, Irvine, CA USA [5]Hosp Nacl Pediat Juan P Garrahan, Buenos Aires, DF, Argentina [6]Univ British Columbia, Vancouver, BC V5Z 1M9, Canada [7]Great Ormond St Hosp Sick Children, London WC1N 3JH, England [8]Shiga Med Ctr Childrens, Moriyama, Japan [9]Tokyo Med & Dent Univ, Tokyo, Japan [10]Sultan Qaboos Univ, Alkhad, Oman [11]Natl Inst Neurosci, Singapore, Singapore [12]Shenzhen Childrens Hosp, Shenzhen, Peoples R China [13]Univ Philippines, Coll Med, Manila, Philippines [14]Univ Liverpool, Liverpool L69 3BX, Merseyside, England [15]Inst Child Hlth, London, England [16]Klinikum Weiden Kinderklin, Weiden, Germany [17]Tokyo Womens Med Univ, Tokyo, Japan [18]Okayama Univ, Sch Med, Okayama 700, Japan [19]Natl Univ Singapore Hosp, Singapore 117548, Singapore [20]William Soler Childrens Hosp, Havana, Cuba [21]Heidelberg Univ, Heidelberg, Germany [22]Univ Childrens Hosp, Zurich, Switzerland [23]Hosp Selayang, Kuala Lumpur, Malaysia [24]Osped Pediat Bambinu Gesu, Rome, Italy [25]Mahidol Univ, Bangkok 10700, Thailand [26]Univ Nottingham, Nottingham NG7 2RD, England [27]Univ Hong Kong, Hong Kong, Hong Kong, Peoples R China [28]Dokkyo Univ, Sch Med, Tochigi, Tochigi, Japan [29]Washington Univ, Sch Med, St Louis, MO USA [30]China Japan Friendship Hosp, Beijing, Peoples R China [*1]Royal United Hosp, Bath Unit Res Paediat, Combe Pk, Bath BA1 3NG, Avon, England
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关键词: epilepsy infantile spasms West syndrome consensus clinical trials

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Purpose: To reach a broad consensus on case definitions, outcomes, and outcome measures that will ease future study design and facilitate comparison of data from different studies of infantile spasms and West syndrome. Methods: Persons who had recently presented or published first-author original research in this field were invited to participate in an e-mail Delphi process and to invite other investigators or clinicians who they thought might participate. Results: The process consisted of six rounds, anonymous except to the facilitator. In total, responses were received from 46 participants. The final statement was approved by 31 participants from 15 countries. It concluded that the primary clinical outcome, cessation of spasms, should denote absence of witnessed spasms from within 14 days of commencement of treatment, and for greater than or equal to28 consecutive days from the last witnessed spasm. Primary electroclinical outcome denotes cessation of spasms with resolution of hypsarrhythmia. West syndrome should be a defined subset of the syndrome of infantile spasms. An infantile spasms single-spasm variant should be recognized. Ways are suggested of handling subtle spasms in the context of clinical studies. It proposes a standard for reporting modifying and atypical features of hypsarrhythmia, a minimal set of baseline characteristics and outcomes that should be reported in trials of infantile spasms, and suggests a standard definition of relapse. Consensus was not reached on a definition of hypsarrhythmia. Conclusions: We reached a clear consensus on many aspects of study design for the investigation of infantile spasms, although incomplete consensus was found on how to define EEG criteria.

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大类 | 1 区 医学
小类 | 1 区 临床神经病学
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Q1 CLINICAL NEUROLOGY
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Q1 CLINICAL NEUROLOGY

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通讯机构: [*1]Royal United Hosp, Bath Unit Res Paediat, Combe Pk, Bath BA1 3NG, Avon, England
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