Title

Comparison of Cosyntropin, Vigabatrin, and Combination Therapy in New-Onset Infantile Spasms in a Prospective Randomized Trial

Authors

Kelly G. Knupp, Pediatrics and Neurology, 12225University of Colorado, Anschutz Campus, Aurora, CO, USA.
Jason Coryell, Department of Pediatrics and Neurology, 89020Oregon Health and Sciences University, Portland, Oregon, USA.
Rani K. Singh, Department of Pediatrics, Division of Pediatric Neurology, Atrium Health/Levine Children's Hospital, Charlotte, NC, USA.
William D. Gaillard, Department of Pediatrics and Neurology, George Washington University, Washington, DC, USA.
Renée A. Shellhaas, Department of Pediatrics, Michigan Medicine, University of Michigan, Ann Arbor, MI, USA.
Sookyong Koh, Department of Pediatrics, Children's Hospital, 12284University of Nebraska Medical Center, Omaha, NE, USA.
Wendy G. Mitchell, Neurology Division, Keck School of Medicine, 8785University of Southern California and Children's Hospital Los Angeles, Los Angeles, CA, USA.
Chellamani Harini, Department of Neurology, Harvard University, Boston, MA, USA.
John J. Millichap, Department of Pediatrics and Neurology, Lurie Children's Hospital, Chicago, Illinois, USA.
Alison May, Department of Neurology, Morgan Stanley Children's Hospital, 21611Columbia University Irving Medical Center, New York, NY, USA.
Dennis Dlugos, Departments of Neurology and Pediatrics, Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania, USA.
Katherine Nickels, Department of Neurology, Mayo Clinic, Rochester, MN, USA.
John R. Mytinger, Department of Pediatrics, Division of Pediatric Neurology, Nationwide Children's Hospital, The Ohio State University, Columbus, OH, USA.
Cynthia Keator, Jane and John Justin Neurosciences, Cook Children's Medical Center, Fort Worth, TX, USA.
Elissa Yozawitz, Isabelle Rapin Division of Child Neurology, Saul R. Korey Department of Neurology, Department of Pediatrics, 550033Albert Einstein College of Medicine and Montefiore Medical Center, Bronx, NY, USA.
Nilika Singhal, Department of Neurology, Division of Epilepsy, UCSF Benioff Children's Hospital, San Francisco, CA, USA.
Jason Lockrow, Division of Pediatric Neurology, Department of Pediatrics, University of Washington, Seattle, WA, USA.
Jacob F. Thomas, School of Medicine, Adult and Child Consortium for Health Outcomes Research and Delivery Science, University of Colorado, Aurora, Colorado, USA.
Elizabeth Juarez-Colunga, Department of Biostatistics and Informatics, Colorado School of Public Health, University of Colorado Anschutz Medical Campus, Aurora, Colorado, USA.

Document Type

Journal Article

Publication Date

1-19-2022

Journal

Journal of child neurology

DOI

10.1177/08830738211073400

Keywords

ACTH; West syndrome; hypsarrhythmia

Abstract

In a randomized trial, we aimed to evaluate the efficacy of cosyntropin injectable suspension, 1 mg/mL, compared to vigabatrin for infantile spasms syndrome. An additional arm was included to assess the efficacy of combination therapy (cosyntropin and vigabatrin) compared with cosyntropin monotherapy. Children (2 months to 2 years) with new-onset infantile spasms syndrome and hypsarhythmia were randomized into 3 arms: cosyntropin, vigabatrin, and cosyntropin and vigabatrin combined. Daily seizures and adverse events were recorded, and EEG was repeated at day 14 to assess for resolution of hypsarhythmia. The primary outcome measure was the composite of resolution of hypsarhythmia and absence of clinical spasms at day 14. Fisher exact test was used to compare outcomes. 37 children were enrolled and 34 were included in the final efficacy analysis (1 withdrew prior to treatment and 2 did not return seizure diaries). Resolution of both hypsarhythmia and clinical spasms was achieved in in 9 of 12 participants (75%) treated with cosyntropin, 1/9 (11%) vigabatrin, and 5/13 (38%) cosyntropin and vigabatrin combined. The primary comparison of cosyntropin versus vigabatrin was significant (64% [95% confidence interval 21, 82],  < .01). Adverse events were reported in all 3 treatment arms: 31 (86%) had an adverse event, 7 (19%) had a serious adverse event, and 15 (42%) had an adverse event of special interest with no difference between treatment arms. This randomized trial was underpowered because of incomplete enrollment, yet it demonstrated that cosyntropin was more effective for short-term outcomes than vigabatrin as initial treatment for infantile spasms.

Department

Pediatrics

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