Intrathecal Injections in Children With Spinal Muscular Atrophy: Nusinersen Clinical Trial Experience

被引:157
作者
Hache, Manon [1 ]
Swoboda, Kathryn J. [2 ]
Sethna, Navil [3 ]
Farrow-Gillespie, Alan [4 ]
Khandji, Alexander [5 ]
Xia, Shuting [6 ]
Bishop, Kathie M. [6 ]
机构
[1] Columbia Univ, Med Ctr, Div Pediat Anesthesia, 622 W 168th St,BH 440-N, New York, NY 10032 USA
[2] Massachusetts Gen Hosp, Dept Neurol, Boston, MA 02114 USA
[3] Boston Childrens Hosp, Dept Anesthesiol Perioperat & Pain Med, Boston, MA USA
[4] Univ Texas Southwestern Med Sch, Dept Anesthesiol & Pain Management, Dallas, TX USA
[5] Columbia Univ, Dept Radiol, New York, NY 10032 USA
[6] Ionis Pharmaceut Inc, Carlsbad, CA USA
关键词
lumbar puncture; spinal muscular atrophy; antisense oligonucleotide; drug delivery; POSTDURAL PUNCTURE HEADACHE; LUMBAR PUNCTURE; NEEDLE DESIGN; SUCCESS RATE; ANESTHESIA; ULTRASOUND; COMPLICATIONS; BACKACHE; INFANTS; PHASE-1;
D O I
10.1177/0883073815627882
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Nusinersen (ISIS-SMNRx or ISIS 396443) is an antisense oligonucleotide drug administered intrathecally to treat spinal muscular atrophy. We summarize lumbar puncture experience in children with spinal muscular atrophy during a phase 1 open-label study of nusinersen and its extension. During the studies, 73 lumbar punctures were performed in 28 patients 2 to 14 years of age with type 2/3 spinal muscular atrophy. No complications occurred in 50 (68%) lumbar punctures; in 23 (32%) procedures, adverse events were attributed to lumbar puncture. Most common adverse events were headache (n = 9), back pain (n = 9), and post-lumbar puncture syndrome (n = 8). In a subgroup analysis, adverse events were more frequent in older children, children with type 3 spinal muscular atrophy, and with a 21- or 22-gauge needle compared to a 24-gauge needle or smaller. Lumbar punctures were successfully performed in children with spinal muscular atrophy; lumbar puncture-related adverse event frequency was similar to that previously reported in children.
引用
收藏
页码:899 / 906
页数:8
相关论文
共 40 条
[1]   Positioning for Lumbar Puncture in Children Evaluated by Bedside Ultrasound [J].
Abo, Alyssa ;
Chen, Lei ;
Johnston, Patrick ;
Santucci, Karen .
PEDIATRICS, 2010, 125 (05) :E1149-E1153
[2]   Post lumbar puncture headache: diagnosis and management [J].
Ahmed, S. V. ;
Jayawarna, C. ;
Jude, E. .
POSTGRADUATE MEDICAL JOURNAL, 2006, 82 (973) :713-716
[3]   Cerebrospinal fluid dissecting into spinal epidural space after lumbar puncture causing cauda equina syndrome: review of literature and illustrative case [J].
Amini, Amin ;
Liu, James K. ;
Kan, Peter ;
Brockmeyer, Douglas L. .
CHILDS NERVOUS SYSTEM, 2006, 22 (12) :1639-1641
[4]   Prospective Randomized Crossover Evaluation of Three Anesthetic Regimens for Painful Procedures in Children with Cancer [J].
Anghelescu, Doralina L. ;
Burgoyne, Laura L. ;
Faughnan, Lane G. ;
Hankins, Gisele M. ;
Smeltzer, Matthew P. ;
Pui, Ching-Hon .
JOURNAL OF PEDIATRICS, 2013, 162 (01) :137-141
[5]   The effects of 45 degree head up tilt on the lumbar puncture success rate in children undergoing spinal anesthesia [J].
Apiliogullari, Seza ;
Duman, Ates ;
Gok, Funda ;
Ogun, Cemile Oztin ;
Akillioglu, Ishak .
PEDIATRIC ANESTHESIA, 2008, 18 (12) :1178-1182
[6]   Spinal needle design and size affect the incidence of postdural puncture headache in children [J].
Apiliogullari, Seza ;
Duman, Ates ;
Gok, Funda ;
Akillioglu, Isak .
PEDIATRIC ANESTHESIA, 2010, 20 (02) :177-182
[7]  
Basurto Ona X, 2011, COCHRANE DB SYST REV, V8
[8]   PEDIATRIC LUMBAR PUNCTURE AND CEREBROSPINAL FLUID ANALYSIS [J].
Bonadio, William .
JOURNAL OF EMERGENCY MEDICINE, 2014, 46 (01) :141-150
[9]  
BOONMAK P, 2010, COCHRANE DB SYST REV, V1
[10]   Results from a phase 1 study of nusinersen (ISIS-SMNRx) in children with spinal muscular atrophy [J].
Chiriboga, Claudia A. ;
Swoboda, Kathryn J. ;
Darras, Basil T. ;
Iannaccone, Susan T. ;
Montes, Jacqueline ;
De Vivo, Darryl C. ;
Norris, Daniel A. ;
Bennett, C. Frank ;
Bishop, Kathie M. .
NEUROLOGY, 2016, 86 (10) :890-897