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Characterization of Severe and Extreme Behavioral Problems in Patients With Alternating Hemiplegia of Childhood.

Publication ,  Journal Article
Wallace, K; Uchitel, J; Prange, L; Jasien, J; Bonner, M; D'Alli, R; Maslow, G; Mikati, MA
Published in: Pediatr Neurol
October 2020

BACKGROUND: Alternating hemiplegia of childhood often manifests severe or extreme behavioral problems, the nature of which remains to be fully characterized. METHODS: We analyzed 39 consecutive patients with alternating hemiplegia of childhood for occurrence of behavioral problems and categorized those by severity: mild (not requiring intervention), moderate (requiring intervention but no risk), severe (minor risk to self, others, or both), and extreme (major risk). We then analyzed behavioral manifestations, concurrent morbidity, and medication responses in patients with severe or extreme symptoms. RESULTS: Two patients had mild behavioral problems, five moderate, 10 severe, six extreme, and 16 none. Extreme cases exhibited disruptive behaviors escalating to assaults. Triggers, when present, included peer-provocation, low frustration tolerance, limits set by others, and sleep disruption. Reversible psychotic symptoms occurred in two patients: in one triggered by infection and trihexyphenidyl, and in another triggered by sertraline. Of the 16 patients with severe or extreme symptoms, 13 had concurrent neuropsychiatric diagnoses. Occurrence of severe or extreme symptoms did not correlate with age, puberty, severity of intellectual disability, or mutation status (P > 0.05). A multidisciplinary team including mental health professionals comanaged all patients with severe or extreme symptoms with either behavioral therapy, medications, or both. When considering medications prescribed to more than four patients, medicines that demonstrated efficacy or partial efficacy in more than 50% of patients were alpha-adrenergic agonists and selective-serotonin-reuptake-inhibitors. CONCLUSIONS: Patients with alternating hemiplegia of childhood (41%) often experience severe or extreme behavioral problems and, rarely, medication-triggered psychotic symptoms. These observations are consistent with current understanding of underlying alternating hemiplegia of childhood brain pathophysiology. Increasing awareness of these behavioral problems facilitates alternating hemiplegia of childhood management and anticipatory guidance.

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Published In

Pediatr Neurol

DOI

EISSN

1873-5150

Publication Date

October 2020

Volume

111

Start / End Page

5 / 12

Location

United States

Related Subject Headings

  • Young Adult
  • Violence
  • Suicidal Ideation
  • Severity of Illness Index
  • Self-Injurious Behavior
  • Psychotic Disorders
  • Neurology & Neurosurgery
  • Middle Aged
  • Male
  • Humans
 

Citation

APA
Chicago
ICMJE
MLA
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Wallace, K., Uchitel, J., Prange, L., Jasien, J., Bonner, M., D’Alli, R., … Mikati, M. A. (2020). Characterization of Severe and Extreme Behavioral Problems in Patients With Alternating Hemiplegia of Childhood. Pediatr Neurol, 111, 5–12. https://doi.org/10.1016/j.pediatrneurol.2020.06.012
Wallace, Keri, Julie Uchitel, Lyndsey Prange, Joan Jasien, Melanie Bonner, Richard D’Alli, Gary Maslow, and Mohamad A. Mikati. “Characterization of Severe and Extreme Behavioral Problems in Patients With Alternating Hemiplegia of Childhood.Pediatr Neurol 111 (October 2020): 5–12. https://doi.org/10.1016/j.pediatrneurol.2020.06.012.
Wallace K, Uchitel J, Prange L, Jasien J, Bonner M, D’Alli R, et al. Characterization of Severe and Extreme Behavioral Problems in Patients With Alternating Hemiplegia of Childhood. Pediatr Neurol. 2020 Oct;111:5–12.
Wallace, Keri, et al. “Characterization of Severe and Extreme Behavioral Problems in Patients With Alternating Hemiplegia of Childhood.Pediatr Neurol, vol. 111, Oct. 2020, pp. 5–12. Pubmed, doi:10.1016/j.pediatrneurol.2020.06.012.
Wallace K, Uchitel J, Prange L, Jasien J, Bonner M, D’Alli R, Maslow G, Mikati MA. Characterization of Severe and Extreme Behavioral Problems in Patients With Alternating Hemiplegia of Childhood. Pediatr Neurol. 2020 Oct;111:5–12.
Journal cover image

Published In

Pediatr Neurol

DOI

EISSN

1873-5150

Publication Date

October 2020

Volume

111

Start / End Page

5 / 12

Location

United States

Related Subject Headings

  • Young Adult
  • Violence
  • Suicidal Ideation
  • Severity of Illness Index
  • Self-Injurious Behavior
  • Psychotic Disorders
  • Neurology & Neurosurgery
  • Middle Aged
  • Male
  • Humans