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1. Treatment of infantile spasms with very high dose prednisolone before high dose adrenocorticotropic hormone

Author

Hussain, Shaun A, Shinnar, Shlomo, Kwong, Grace, Lerner, Jason T, Matsumoto, Joyce H, Wu, Joyce Y, Shields, W Donald, and Sankar, Raman

Subjects
Clinical Trials and Supportive Activities, Clinical Research, Rare Diseases, Pediatric, Evaluation of treatments and therapeutic interventions, 6.1 Pharmaceuticals, Adolescent, Adrenocorticotropic Hormone, Anticonvulsants, Child, Child, Preschool, Drug Administration Schedule, Drug Therapy, Combination, Electroencephalography, Female, Humans, Infant, Male, Monitoring, Physiologic, Prednisolone, Spasms, Infantile, Treatment Outcome, Hypsarrhythmia, Corticotropin, ACTHar, Corticosteroids, West syndrome, Clinical Sciences, Neurosciences, Neurology & Neurosurgery
Abstract

PurposeThis study investigated the short-term response to a standardized hormonal therapy protocol for treatment of infantile spasms.MethodsTwenty-seven children with video electroencephalography (EEG)-confirmed infantile spasms received very high dose (8 mg/kg/day, max 60 mg/day) oral prednisolone for 2 weeks. Response (absence of both hypsarrhythmia and spasms) to prednisolone was ascertained by repeat overnight video-EEG. Responders were tapered over 2 weeks and nonresponders were immediately transitioned to high dose (150 IU/m(2)/day) intramuscular adrenocorticotropic hormone (ACTH) for two additional weeks. Response was again determined by overnight video-EEG after ACTH therapy.Key findingsSixty-three percent (17/27) of patients responded completely to prednisolone. Subsequently, 40% (4/10) of prednisolone nonresponders exhibited a complete response after an additional 2-week course with ACTH. Among 27 subjects with median follow-up of 13.5 months (interquartile range [IQR] 4.8-25.9), 12% (2/17) of prednisolone responders and 50% (2/4) of ACTH responders experienced a relapse between 2 and 9 months after initial response.SignificanceVery high dose prednisolone demonstrated significantly higher efficacy than previously reported for lower doses in prior studies. High dose ACTH may be superior to very high dose prednisolone, and in lieu of a definitive clinical trial, the choice between prednisolone and ACTH for initial treatment of infantile spasms remains controversial.

Published
2014

2. Infantile spasms: A U.S. consensus report

Author

Pellock, John M, Hrachovy, Richard, Shinnar, Shlomo, Baram, Tallie Z, Bettis, David, Dlugos, Dennis J, Gaillard, William D, Gibson, Patricia A, Holmes, Gregory L, Nordli, Douglas R, O’Dell, Christine, Shields, W Donald, Trevathan, Edwin, and Wheless, James W

Subjects
Pediatric, Clinical Research, Health Services, Management of diseases and conditions, 7.3 Management and decision making, Adrenal Cortex Hormones, Adrenocorticotropic Hormone, Anticonvulsants, Consensus, Consensus Development Conferences as Topic, Diet, Ketogenic, Disease-Free Survival, Electroencephalography, Female, Humans, Infant, Longitudinal Studies, Male, Outcome Assessment, Health Care, Pediatrics, Practice Guidelines as Topic, Prednisolone, Spasms, Infantile, United States, Vigabatrin, West syndrome, Encephalopathic epilepsy, Adrenocorticotropic hormone, Corticosteroids, Infantile spasms, Treatment, Clinical Sciences, Neurosciences, Neurology & Neurosurgery
Abstract

The diagnosis, evaluation, and management of infantile spasms (IS) continue to pose significant challenges to the treating physician. Although an evidence-based practice guideline with full literature review was published in 2004, diversity in IS evaluation and treatment remains and highlights the need for further consensus to optimize outcomes in IS. For this purpose, a working group committed to the diagnosis, treatment, and establishment of a continuum of care for patients with IS and their families—the Infantile Spasms Working Group (ISWG)—was convened. The ISWG participated in a workshop for which the key objectives were to review the state of our understanding of IS, assess the scientific evidence regarding efficacy of currently available therapeutic options, and arrive at a consensus on protocols for diagnostic workup and management of IS that can serve as a guide to help specialists and general pediatricians optimally manage infants with IS. The overall goal of the workshop was to improve IS outcomes by assisting treating physicians with early recognition and diagnosis of IS, initiation of short duration therapy with a first-line treatment, timely electroencephalography (EEG) evaluation of treatment to evaluate effectiveness, and, if indicated, prompt treatment modification. Differences of opinion among ISWG members occurred in areas where data were lacking; however, this article represents a consensus of the U.S. approach to the diagnostic evaluation and treatment of IS.

Published
2010

13. CONTRIBUTORS

Author

Aaskov, John G., primary, Abdel-Rahman, Susan M., additional, Aebi, Christoph, additional, Ament, Marvin E., additional, Anderson, Marsha S., additional, Arnon, Stephen S., additional, Arvin, Ann M., additional, Atkins, Jane T., additional, Atmar, Robert L., additional, Baker, Carol J., additional, Baltimore, Robert S., additional, Barenkamp, Stephen J., additional, Barnett, Elizabeth D., additional, Basow, Robert D., additional, Beisel, William R., additional, Bell, Beth P., additional, Benard, Gil, additional, Bernstein, David I., additional, Bernt, Kathrin M., additional, Berry, Andrea A., additional, Bluestone, Charles D., additional, Blumer, Jeffrey L., additional, Bortolussi, Robert, additional, Boyanton, Bobby L., additional, Boyer, Kenneth M., additional, Bradley, John S., additional, Brady, Michael T., additional, Britt, William J., additional, Broderick, Annemarie, additional, Bronstein, David E., additional, Bruckner, David A., additional, Buckingham, Steven C., additional, Burgos, Ana, additional, Byington, Carrie L., additional, Campbell, Judith R., additional, Cantu, Samson, additional, Chacko, Mariam R., additional, Chapman, Louisa E., additional, Charrel, Rémi N., additional, Chen, Tempe K., additional, Cherry, James D., additional, Chesney, P. Joan, additional, Chilakapati, Madhuri C., additional, Chinen, Javier, additional, Ching, Natascha, additional, Clark, H. Fred, additional, Cleary, Thomas G., additional, Coats, David K., additional, Correa, Armando G., additional, Cross, J. Thomas, additional, Cutrer, William B., additional, Dagan, Ronald, additional, Dassey, David E., additional, Davis, Jeffrey P., additional, Demmler-Harrison, Gail J., additional, Dennehy, Penelope H., additional, Doan, Minh L., additional, Dobson, Simon R., additional, Drutz, Jan E., additional, Edelstein, Paul H., additional, Edwards, Kathryn M., additional, Edwards, Morven S., additional, English, B. Keith, additional, Estripeaut, Dora, additional, Fan, Leland L., additional, Feigin, Ralph D., additional, Ferry, George D., additional, Fiore, Anthony E., additional, Fischer, Philip R., additional, Fisher, Randall G., additional, Flynn, Patricia M., additional, Flynn, Thomas R., additional, Frenkel, Lisa M., additional, Friedman, Ellen M., additional, Friedman, Richard A., additional, Garcia, Lynne S., additional, Gavin, Patrick J., additional, Gerber, Michael A., additional, Gershon, Anne A., additional, Gilger, Mark A., additional, Gillespie, Susan L., additional, Glaze, Daniel G., additional, Glezen, W. Paul, additional, Glodé, Mary P., additional, Goldmann, Donald A., additional, Goldstein, Ellie J.C., additional, Goldstein, Nira A., additional, Gonzales, Edmond T., additional, Gorman, Mark P., additional, Green, Michael D., additional, Greenberg, David, additional, Groll, Andreas H., additional, Grose, Charles, additional, Gubler, Duane J., additional, Guerrero, Roberto A., additional, Guevara, Javier Nieto, additional, Gutierrez, Kathleen M., additional, Hall, Caroline Breese, additional, Halstead, Scott B., additional, Hamano, Shinjiro, additional, Hamill, Richard J., additional, Hammerschlag, Margaret R., additional, Hanson, I. Celine, additional, Harik, Nada, additional, Harrison, Rick E., additional, Healy, C. Mary, additional, Heininger, Ulrich, additional, Heresi, Gloria P., additional, Hiatt, Peter W., additional, Hill, Harry R., additional, Hilmers, David C., additional, Hoffman, Jill A., additional, Hon, Ellis K.L., additional, Hostetter, Margaret K., additional, Hotez, Peter J., additional, Hughes, Walter T., additional, Hulten, Kristina G., additional, Hunstad, David A., additional, Hurwitz, Eugene S., additional, Huskins, W. Charles, additional, Hyun, David Y., additional, Jackson, Mary Anne, additional, Jacobs, Michael R., additional, Jacobs, Richard F., additional, Jaeger, Jenifer L., additional, Jhaveri, Ravi R., additional, Johnston, Samantha, additional, Jonas, Maureen M., additional, Julapalli, Meena R., additional, Kaplan, Edward L., additional, Kaplan, Sheldon L., additional, Karpen, Saul J., additional, Kearns, Gregory L., additional, Keller, Margaret A., additional, Khoury, Chaouki K., additional, Kleiman, Martin B., additional, Klein, Jerome O., additional, Kline, Mark W., additional, Knapp, Katherine M., additional, Kokkinos, Heidi M., additional, Krause, Peter J., additional, Krilov, Leonard R., additional, Krogstad, Paul, additional, Kuhls, Thomas L., additional, de Lamballerie, Xavier, additional, La Pine, Timothy R., additional, Laurens, Matthew B., additional, Leach, Charles T., additional, Leggiadro, Robert J., additional, Lennon, Diana R., additional, Lentzsch-Parcells, Carolyn, additional, Leroy, Eric, additional, Leung, Chi Wai, additional, Levy, Moise L., additional, Lewis, Karen, additional, Losikoff, Phyllis T., additional, Lotze, Timothy Edward, additional, Lowry, Adam W., additional, Mailman, Timothy, additional, Maloney, Susan A., additional, Mascola, Laurene, additional, Mason, Edward O., additional, Matson, David O., additional, Mayer, Alan N., additional, Mazade, Marc A., additional, McAuley, James B., additional, McCracken, George H., additional, McIntosh, Kenneth, additional, McJunkin, James E., additional, McKee, Kelly T., additional, McLeod, Rima L., additional, McLin, Valérie A., additional, Mendes-Giannini, Maria José Soares, additional, Meyers, Wayne M., additional, Michaels, Marian G., additional, Michelow, Ian C., additional, Milisavljevic, Vladana, additional, Miller, Aaron M., additional, Miller, James N., additional, Miller, Marjorie J., additional, Mills, James N., additional, Minnich, Linda L., additional, Moran, Ann, additional, Murphy, James R., additional, Nag, Pratip K., additional, Nania, Joseph J., additional, Nataro, James P., additional, Nicome, Roger K., additional, Nielsen-Saines, Karin, additional, Nieves, Delma J., additional, Oberhelman, Richard A., additional, Ochoa, Theresa J., additional, Oermann, Christopher M., additional, Olteanu, Alina, additional, Overturf, Gary D., additional, Palazzi, Debra L., additional, Pannaraj, Pia S., additional, Patel, Janak A., additional, Patrick, Christian C., additional, Paysse, Evelyn A., additional, Pérez, Norma, additional, Peters, C.J., additional, Petri, William A., additional, Phillips, Brandon Lane, additional, Pickering, Larry K., additional, Piecuch, Joseph F., additional, Pinheiro, Francisco P., additional, Plotkin, Stanley A., additional, Pomeroy, Scott L., additional, Pong, Alice, additional, Pugatch, David L., additional, Purcell, Joan S., additional, Ramraj, Ramya, additional, Remington, Jack S., additional, Rodriguez, Carina A., additional, Romero, José R., additional, Ross, Benjamin A., additional, Ross, Lawrence A., additional, Rowen, Judith L., additional, Rupprecht, Charles E., additional, Sáez-Llorens, Xavier, additional, Saiman, Lisa, additional, Geme, Joseph W. St., additional, Sánchez, Pablo J., additional, Sass, Laura A., additional, Sattler, Carlos A., additional, Schulte, Danica J., additional, Schutze, Gordon E., additional, Seeborg, Filiz O., additional, Shapiro, Eugene D., additional, Shapiro, Nina L., additional, Shearer, William T., additional, Shehab, Ziad M., additional, Shenep, Jerry L., additional, Shields, W. Donald, additional, Shimizu-Cohen, Robyn, additional, Shulman, Stanford T., additional, Simos, Constantine, additional, Smith, Arnold L., additional, Soden, Jason S., additional, Staat, Mary Allen, additional, Starke, Jeffrey R., additional, Stechenberg, Barbara W., additional, Steinbach, William J., additional, Steinkuller, Paul G., additional, Stiehm, E. Richard, additional, Stovall, Stephanie H., additional, Suen, Jeffrey, additional, Sumaya, Ciro V., additional, Summer, Andrea P., additional, Swanson, Douglas S., additional, Tan, Tina Q., additional, Tanowitz, Herbert B., additional, Tesh, Robert B., additional, Toltzis, Philip, additional, Topazian, Richard G., additional, Tosi, Michael F., additional, Travassos da Rosa, Amelia P.A., additional, Tsai, Theodore F., additional, Valdez, Tulio A., additional, Vallejo, Jesus G., additional, Vanchiere, John A., additional, Vasconcelos, Pedro Fernando da C., additional, Velarde, Jorge J., additional, Versalovic, James, additional, Wald, Ellen R., additional, Walsh, Douglas S., additional, Walsh, Edward E., additional, Walsh, Thomas J., additional, Ward, Mark A., additional, Ward, Richard L., additional, Weinberg, Michelle, additional, Welliver, Robert C., additional, Wheeler, J. Gary, additional, White, A. Clinton, additional, Whitworth, Suzanne, additional, Wiedermann, Bernhard L., additional, Williams-Bouyer, Natalie, additional, Wittner, Murray, additional, Woods, Charles R., additional, Yen, Kimberly G., additional, Yogev, Ram, additional, Young, Edward J., additional, and Zaoutis, Theoklis E., additional

Published
2009
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15. Social communication in children with epilepsy

Author

Caplan, Rochelle, Guthrie, Donald, Komo, Scott, Siddarth, Prabha, Chayasirisobhon, Sirichai, Kornblum, Harley, Sankar, Ramen, Hansen, Rebecca, Mitchell, Wendy, and Shields, W. Donald

Subjects
Neuropsychology -- Research, Epileptics, Developmentally disabled -- Means of communication, Developmentally disabled children, Communication -- Social aspects, Communicative disorders in children -- Physiological aspects, Cognition disorders -- Physiological aspects, Psychology and mental health
Abstract

This article examines epileptic children's social communication skills. Topics include neuropsychology, cognitive development, and an analysis of thought disorders.

Published
2002

18. Blink rate in pediatric complex partial seizure disorder

Author

Caplan, Rochelle, Guthrie, Donald, Komo, Scott, and Shields, W. Donald

Subjects
Mentally ill children -- Psychological aspects, Schizophrenia -- Research, Psychology and mental health
Abstract

A study was conducted to analyze spontaneous blink rate in children with complex partial epilepsy and normal children. A general mixed linear framework was utilized to carry out analyses that included fixed and random components. Statistical inferences were correlated with square-root transformed data. Experimental results suggested that children with complex partial epilepsy may have biological properties similar to children with schizophrenia.

Published
1998

31. Middle childhood onset of interictal psychosis

Author

Caplan, Rochelle, Shields, W. Donald, Mori, Lisa, and Yudovin, Sue

Subjects
Temporal lobe epilepsy -- Psychological aspects, Schizophrenia -- Case studies, Children -- Psychological aspects, Family and marriage, Psychology and mental health
Published
1991

42. Reversal of microcephaly and developmental delay after cure of hyperinsulinemic hypoglycemia

Author

Gottschalk, Michael E., Geffner, Mitchell E., Yasuda, Patrice M., and Shields, W. Donald

Subjects
Hypoglycemia in children -- Complications, Microcephaly -- Case studies, Microcephaly -- Health aspects, Developmentally disabled children -- Care and treatment, Health
Abstract

Microcephaly, abnormally small head size, is generally thought to be caused by congenital conditions and to be untreatable. It is usually evaluated only to identify genetic or infectious diseases for which counseling may be helpful. A case is reported of microcephaly and developmental delay which resolved after treatment of childhood low blood sugar (hypoglycemia). The patient was a girl born with normal weight and height, but had a small head circumference. Developmental delay became evident by eight months, and by 15 months, her weight and height were rather low. Decreased muscle tone, delayed motor skills, and microcephaly were also noted. Laboratory studies revealed the hypoglycemia, and muscle tone and alertness improved within 24 hours after treatment with glucose. Four weeks later, at discharge, the patient was able to achieve a sitting position, and head circumference had increased by one-fifth of an inch. Removal of 90 percent of the pancreas was necessary to treat the cause of the hypoglycemia. At age four and one-half, the girl had a normal head circumference, overall development was average, and she had superior motor skills. Other cases of hypoglycemia-induced developmental delays have been reported, but in general, early recognition of the condition has been necessary to prevent permanent developmental abnormalities. (Consumer Summary produced by Reliance Medical Information, Inc.)

Published
1990

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