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November 08, 2022; 99 (19) Research Article

Prioritizing Hormone Therapy Over Vigabatrin as the First Treatment for Infantile Spasms

A Quality Improvement Initiative

View ORCID ProfileJohn R. Mytinger, William Parker, Steven W. Rust, View ORCID ProfileStephanie M. Ahrens, Dara V.F. Albert, Christopher W. Beatty, Julie Chrisman, View ORCID ProfileDaniel J. Clark, Andrea Debs, Danielle Denney, Mary Karn, James Herbst, View ORCID ProfileAdam P. Ostendorf, Mary C. Taylor, View ORCID ProfileJaime D.E. Twanow, Jorge Vidaurre, View ORCID ProfileAnup D. Patel
First published August 29, 2022, DOI: https://doi.org/10.1212/WNL.0000000000201113
John R. Mytinger
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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William Parker
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Steven W. Rust
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Stephanie M. Ahrens
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Dara V.F. Albert
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Christopher W. Beatty
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Julie Chrisman
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Daniel J. Clark
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Andrea Debs
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Danielle Denney
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Mary Karn
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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James Herbst
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Adam P. Ostendorf
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Mary C. Taylor
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Jaime D.E. Twanow
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Jorge Vidaurre
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Anup D. Patel
From the Division of Pediatric Neurology (J.R.M., S.M.A., D.V.F.A., C.W.B., A.P.O., J.D.E.T., J.V., A.D.P.), Department of Pediatrics, Nationwide Children's Hospital, and The Ohio State University; The Center for Clinical Excellence (W.P., A.D.P.) ; Information Technology Research & Innovation (S.W.R.), and Division of Pediatric Neurology (J.C., D.J.C., A.D., D.D., M.K., J.H., M.C.T.), Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.
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Citation
Prioritizing Hormone Therapy Over Vigabatrin as the First Treatment for Infantile Spasms
A Quality Improvement Initiative
John R. Mytinger, William Parker, Steven W. Rust, Stephanie M. Ahrens, Dara V.F. Albert, Christopher W. Beatty, Julie Chrisman, Daniel J. Clark, Andrea Debs, Danielle Denney, Mary Karn, James Herbst, Adam P. Ostendorf, Mary C. Taylor, Jaime D.E. Twanow, Jorge Vidaurre, Anup D. Patel
Neurology Nov 2022, 99 (19) e2171-e2180; DOI: 10.1212/WNL.0000000000201113

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Abstract

Background and Objectives Infantile spasms (IS) are early childhood seizures with potentially devastating consequences. Standard therapies (adrenocorticotropic hormone [ACTH], high-dose prednisolone, and vigabatrin) are strongly recommended as the first treatment for IS. Although this recommendation comes without preference for one standard therapy over another, early remission rates are higher with hormone therapy (ACTH and high-dose prednisolone) when compared with vigabatrin. Using quality improvement (QI) methodology that included hormone therapy as the first treatment, we sought to increase the percentage of children with new-onset nontuberous sclerosis complex (TSC)–associated IS achieving 3-month electroclinical remission from a mean of 53.8% to ≥70%.

Methods This was an observational consecutive sample cohort study at a single academic tertiary care hospital that compared a prospective intervention cohort (May 2019–January 2022, N = 57) with a retrospective baseline cohort (November 2015–April 2019, N = 67). Our initiative addressed key drivers such as the routine use of vigabatrin over hormone therapy as first treatment and the common initiation of a second treatment after 14 days for initial nonresponders. We included consecutive children without TSC presenting with new-onset IS diagnosed and treated between ages 2 and 24 months. We displayed our primary outcome and process measures as control charts in which the centerline is the quarterly (previous 3 months) mean based on statistical process control methodology.

Results QI interventions that included the standardization of hormone therapy as the first treatment resulted in higher rates of 3-month remission, rising from 53.8% (baseline cohort) to 75.9% (intervention cohort). Process measure results included an increased rate of children receiving hormone therapy as first treatment (mean, 44.6%–100%) and a decreased number of days to both clinical follow-up after first treatment (mean, of 16.3‐12.6 days) and starting a second treatment within 14 days for initial nonresponders (mean, 36.3‐17.2 days).

Discussion For children with IS, improved rates of 3-month electroclinical remission can be achieved with QI methodology. Implementation of similar QI initiatives at other centers may likewise improve local remission rates.

Glossary

ACTH=
adrenocorticotropic hormone;
ASQ=
American Society for Quality;
IS=
infantile spasms;
NCH=
Nationwide Children's Hospital;
QI=
quality improvement;
SPC=
statistical process control;
TSC=
tuberous sclerosis complex

Footnotes

  • Go to Neurology.org/N for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the article.

  • Submitted and externally peer reviewed. The handling editor was Renee Shellhaas, MD, MS.

  • Received February 4, 2022.
  • Accepted in final form June 30, 2022.
  • © 2022 American Academy of Neurology
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