Mirtazapine for sleep disturbances in Angelman syndrome: a retrospective chart review of 8 pediatric cases

Hanzlik, Emily; Klinger, Sarah A; Carson, Robert P.; Duis, Jessica

doi:10.5664/jcsm.8284

Cited by 16 publications

(10 citation statements)

References 26 publications

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“…Each of these sleep aids were effective in treating sleep disturbances in the AS cohort [133]. A retrospective chart review of 8 patients with AS showed that mirtazapine improved sleep onset and decreased nighttime awakenings [134].…”

Section: Treatmentmentioning

confidence: 99%

Neurodevelopmental Disorders Commonly Presenting with Sleep Disturbances

Shelton

Malow

2021

Neurotherapeutics

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There are multiple disorders of neurodevelopment that present with co-occurring sleep disturbances. Many of these neurodevelopmental disorders (NDD) include sleep disturbances in their diagnostic criteria. Neurobiological, genetic, and environmental factors overlap to cause different sleep disorders in individuals with NDD. Caregivers often present reporting either insomnia or hypersomnia, and based on the clinical history and findings from diagnostic tests, an appropriate diagnosis can be made. It is crucial that clinicians understand the different presentations of sleep disturbances in individuals with NDD.

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Section: Treatmentmentioning

confidence: 99%

Neurodevelopmental Disorders Commonly Presenting with Sleep Disturbances

Shelton

Malow

2021

Neurotherapeutics

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“…Clinical data underline the potential of 5-HT 2 and 5-HT 3 antagonism, by mirtazapine, in the treatment of sleep disturbances in patients with AS. 72 As delineated below, subtypes of the 5-HT 2 receptors (5-HT 2A , -2B , and -2C ) can play a crucial role in current and future epilepsy treatment.…”

Section: -Ht 2 Receptorsmentioning

confidence: 99%

Serotonin receptors in epilepsy: Novel treatment targets?

Sourbron

Lagae

2022

Epilepsia Open

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Despite the availability of over 30 antiseizure medications (ASMs), there is no “one size fits it all,” so there is a continuing search for novel ASMs. There are divergent data demonstrating that modulation of distinct serotonin (5‐hydroxytryptamine, 5‐HT) receptors subtypes could be beneficial in the treatment of epilepsy and its comorbidities, whereas only a few ASM, such as fenfluramine (FA), act via 5‐HT. There are 14 different 5‐HT receptor subtypes, and most epilepsy studies focus on one or a few of these subtypes, using different animal models and different ligands. We reviewed the available evidence of each 5‐HT receptor subtype using MEDLINE up to July 2021. Our search included medical subject heading (MeSH) and free terms of each “5‐HT subtype” separately and its relation to “epilepsy or seizures.” Most research underlines the antiseizure activity of 5‐HT1A,1D,2A,2C,3 agonism and 5‐HT6 antagonism. Consistently, FA, which has recently been approved for the treatment of seizures in Dravet syndrome, is an agonist of 5‐HT1D,2A,2C receptors. Even though each study focused on a distinct seizure/epilepsy type and generalization of different findings could lead to false interpretations, we believe that the available preclinical and clinical studies emphasize the role of serotonergic modulation, especially stimulation, as a promising avenue in epilepsy treatment.

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“…Hyperphagia defined as inability to recognize satiety and related behaviors is recognized (Bindels‐de Heus et al, 2020 ; Carson et al, 2019 ; Fridman et al, 1998 ; Hanzlik et al, 2020 ; Kirkilionis et al, 1991 ). This may be characterized by an obsessive compulsive component and driven by times of anxiety, change in routine or transition in general.…”

Section: Review and Practice Guidelinesmentioning

confidence: 99%

“…Medicinal treatments targeting sleep difficulties are beneficial and often needed if behavioral therapies are ineffective. These include low‐dose melatonin (Braam et al, 2008 ; Egan et al, 2020 ; Galvan‐Manso et al, 2002 ; Paprocka et al, 2017 ; Takaesu et al, 2012 ; Zhdanova et al, 1999 ), alpha agonists (clonidine or guanfacine), benzodiazepines (clonazepam), gabapentin or pregabalin, antihistamines (promethazine or diphenhydramine), antidepressants (mirtazapine (Hanzlik et al, 2020 ) or trazodone), and in refractory cases antipsychotics (e.g. quetiapine (Wine et al, 2009 ), Figure 3 ).…”

Section: Review and Practice Guidelinesmentioning

confidence: 99%

“…Decisions about appropriate school/classroom placement should be arrived at through shared decision‐making between the school and family. Some individuals, particularly of the mosaic subtype achieve the ability to read and write to some degree (Hanzlik et al, 2020 ). The IEP should include intensive PT, OT, and ST (Table S2–S4).…”

Section: Review and Practice Guidelinesmentioning

confidence: 99%

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A multidisciplinary approach and consensus statement to establish standards of care for Angelman syndrome

Duis

Nespeca

Summers

et al. 2022

Molec Gen & Gen Med

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Background Angelman syndrome (AS) is a rare neurogenetic disorder present in approximately 1/12,000 individuals and characterized by developmental delay, cognitive impairment, motor dysfunction, seizures, gastrointestinal concerns, and abnormal electroencephalographic background. AS is caused by absent expression of the paternally imprinted gene UBE3A in the central nervous system. Disparities in the management of AS are a major problem in preparing for precision therapies and occur even in patients with access to experts and recognized clinics. AS patients receive care based on collective provider experience due to limited evidence‐based literature. We present a consensus statement and comprehensive literature review that proposes a standard of care practices for the management of AS at a critical time when therapeutics to alter the natural history of the disease are on the horizon. Methods We compiled the key recognized clinical features of AS based on consensus from a team of specialists managing patients with AS. Working groups were established to address each focus area with committees comprised of providers who manage >5 individuals. Committees developed management guidelines for their area of expertise. These were compiled into a final document to provide a framework for standardizing management. Evidence from the medical literature was also comprehensively reviewed. Results Areas covered by working groups in the consensus document include genetics, developmental medicine, psychology, general health concerns, neurology (including movement disorders), sleep, psychiatry, orthopedics, ophthalmology, communication, early intervention and therapies, and caregiver health. Working groups created frameworks, including flowcharts and tables, to help with quick access for providers. Data from the literature were incorporated to ensure providers had review of experiential versus evidence‐based care guidelines. Conclusion Standards of care in the management of AS are keys to ensure optimal care at a critical time when new disease‐modifying therapies are emerging. This document is a framework for providers of all familiarity levels.

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Mirtazapine for sleep disturbances in Angelman syndrome: a retrospective chart review of 8 pediatric cases

Cited by 16 publications

References 26 publications

Neurodevelopmental Disorders Commonly Presenting with Sleep Disturbances

Neurodevelopmental Disorders Commonly Presenting with Sleep Disturbances

Serotonin receptors in epilepsy: Novel treatment targets?

A multidisciplinary approach and consensus statement to establish standards of care for Angelman syndrome

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