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Background In children, SARS-CoV-2 is usually asymptomatic or causes a mild illness of short duration. Persistent illness has been reported; however, its prevalence and characteristics are unclear. We aimed to determine illness duration and characteristics in symptomatic UK school-aged children tested for SARS-CoV-2 using data from the COVID Symptom Study, the largest UK citizen participatory epidemiological study to date. Methods Data from 258,790 children aged 5-17 years were reported by an adult proxy between 24 March 2020 and 22 February 2021. Illness duration and symptom profiles were analysed for all children testing positive for SARS-CoV-2 for whom illness duration could be determined, considered overall and within younger (5-11 years) and older (12-17 years) groups. Data from symptomatic children testing negative for SARS-CoV-2, matched 1:1 for age, gender, and week of testing, were also assessed. Findings 1,734 children (588 younger, 1,146 older children) had a positive SARS-CoV-2 test result and calculable illness duration within the study time frame. The commonest symptoms were headache (62.2%) and fatigue (55.0%). Median illness duration was six days (vs. three days in children testing negative), and was positively associated with age (r s 0.19, p<1.e-4) with median duration of seven days in older vs. five days in younger children. Seventy-seven (4.4%) children had illness duration ≥28 days (LC28), more commonly experienced by older vs. younger children (59 (5.1%) vs. 18 (3.1%), p=0.046). The commonest symptoms experienced by these children were fatigue (84%), headache (80%) and anosmia (80%); however, by day 28 the symptom burden was low (median, two). Only 25 (1.8%) of 1,379 children experienced symptoms for ≥56 days. Few children (15 children, 0.9%) in the negatively-tested cohort experienced prolonged symptom duration; however, these children experienced greater symptom burden (both throughout their illness and at day 28) than children positive for SARS-CoV-2. Interpretation Some children with COVID-19 experience prolonged illness duration. Reassuringly, symptom burden in these children did not increase with time, and most recovered by day 56. Some children who tested negative for SARS-CoV-2 also had persistent and burdensome illness. A holistic approach for all children with persistent illness during the pandemic is appropriate. Research in context Evidence before this study SARS-CoV-2 in children is usually asymptomatic or manifests as a mild illness of short duration. Concerns have been raised regarding prolonged illness in children, with no clear resolution of symptoms several weeks after onset, as is observed in some adults. How common this might be in children, the clinical features of such prolonged illness in children, and how it might compare with illnesses from other respiratory viruses (and with general population prevalence of these symptoms) is unclear. Added value of this study We provide systematic description of COVID-19 in UK school-aged children. Our data, collected in a digital surveillance platform through one of the largest UK citizen science initiatives, show that long illness duration after SARS-CoV-2 infection in school-aged children does occur, but is uncommon, with only a small proportion of children experiencing illness duration beyond four weeks; and the symptom burden in these children usually decreases over time. Almost all children have symptom resolution by eight weeks, providing reassurance about long-term outcomes. Additionally, symptom burden in children with long COVID was not greater than symptom burden in children with long illnesses due to causes other than SARS-CoV-2 infection. Implications of all the available evidence Our data confirm that COVID-19 in UK school-aged children is usually of short duration and of low symptom burden. Some children do experience longer illness duration, validating their experience; however, most of these children usually recover with time. Our findings highlight that appropriate resources will be necessary for any child with prolonged illness, whether due to COVID-19 or other illness. Our study provides timely and critical data to inform discussions around the impact and implications of the pandemic on paediatric healthcare resource allocation.
Erika Molteni; Carole H. Sudre; Liane S. Canas; Sunil S. Bhopal; Robert C. Hughes; Michela Antonelli; Benjamin Murray; Kerstin Kläser; Eric Kerfoot; Liyuan Chen; Jie Deng; Christina Hu; Somesh Selvachandran; Kenneth Read; Joan Capdevila Pujol; Alexander Hammers; Tim D. Spector; Sebastien Ourselin; Claire J. Steves; Marc Modat; Michael Absoud; Emma L. Duncan. Illness duration and symptom profile in a large cohort of symptomatic UK school-aged children tested for SARS-CoV-2. 2021, 1 .
AMA StyleErika Molteni, Carole H. Sudre, Liane S. Canas, Sunil S. Bhopal, Robert C. Hughes, Michela Antonelli, Benjamin Murray, Kerstin Kläser, Eric Kerfoot, Liyuan Chen, Jie Deng, Christina Hu, Somesh Selvachandran, Kenneth Read, Joan Capdevila Pujol, Alexander Hammers, Tim D. Spector, Sebastien Ourselin, Claire J. Steves, Marc Modat, Michael Absoud, Emma L. Duncan. Illness duration and symptom profile in a large cohort of symptomatic UK school-aged children tested for SARS-CoV-2. . 2021; ():1.
Chicago/Turabian StyleErika Molteni; Carole H. Sudre; Liane S. Canas; Sunil S. Bhopal; Robert C. Hughes; Michela Antonelli; Benjamin Murray; Kerstin Kläser; Eric Kerfoot; Liyuan Chen; Jie Deng; Christina Hu; Somesh Selvachandran; Kenneth Read; Joan Capdevila Pujol; Alexander Hammers; Tim D. Spector; Sebastien Ourselin; Claire J. Steves; Marc Modat; Michael Absoud; Emma L. Duncan. 2021. "Illness duration and symptom profile in a large cohort of symptomatic UK school-aged children tested for SARS-CoV-2." , no. : 1.
Although 70% of autistic children and young people meet criteria for co-occurring psychiatric conditions, there are few screening measures specifically for autistic individuals. We describe the development and validation of the Assessment of Concerning Behavior (ACB), an instrument co-developed with the autistic community to assess mental health and problematic/risky behaviors. Items include descriptions to facilitate symptom recognition by autistic people, and carers/professionals. The ACB was completed by 255 parents, 149 autistic children and young people and 30 teachers. Internal consistency, stability and validity was assessed. The ACB parent-version fit a two-factor model (internalizing and externalizing problems) and showed adequate test–retest reliability, internal consistency and construct validity. The ACB is a promising new measure for research and clinical use in autism.
Joanne Tarver; Silia Vitoratou; Mathilde Mastroianni; Natalie Heaney; Eleanor Bennett; Felicity Gibbons; Federico Fiori; Michael Absoud; Lakshmi Ramasubramanian; Emily Simonoff; Paramala Santosh. Development and Psychometric Properties of a New Questionnaire to Assess Mental Health and Concerning Behaviors in Children and Young People with Autism Spectrum Disorder (ASD): The Assessment of Concerning Behavior (ACB) Scale. Journal of Autism and Developmental Disorders 2020, 51, 2812 -2828.
AMA StyleJoanne Tarver, Silia Vitoratou, Mathilde Mastroianni, Natalie Heaney, Eleanor Bennett, Felicity Gibbons, Federico Fiori, Michael Absoud, Lakshmi Ramasubramanian, Emily Simonoff, Paramala Santosh. Development and Psychometric Properties of a New Questionnaire to Assess Mental Health and Concerning Behaviors in Children and Young People with Autism Spectrum Disorder (ASD): The Assessment of Concerning Behavior (ACB) Scale. Journal of Autism and Developmental Disorders. 2020; 51 (8):2812-2828.
Chicago/Turabian StyleJoanne Tarver; Silia Vitoratou; Mathilde Mastroianni; Natalie Heaney; Eleanor Bennett; Felicity Gibbons; Federico Fiori; Michael Absoud; Lakshmi Ramasubramanian; Emily Simonoff; Paramala Santosh. 2020. "Development and Psychometric Properties of a New Questionnaire to Assess Mental Health and Concerning Behaviors in Children and Young People with Autism Spectrum Disorder (ASD): The Assessment of Concerning Behavior (ACB) Scale." Journal of Autism and Developmental Disorders 51, no. 8: 2812-2828.
Children and young people (CYP) with neurodevelopmental disorders (NDDs) may be particularly vulnerable to adverse mental health effects due to the COVID-19 pandemic. We conducted a cross-sectional U.K. parent-reported study from 2nd April–2nd June 2020, using the Strengths and Difficulties Questionnaire. CYP with NDDs (n = 371), compared to neurotypical controls, had a higher prevalence of emotional symptoms (42% vs. 15%) and conduct problems (28% vs. 9%), and fewer prosocial behaviours (54% vs. 22%). All groups had worse emotional symptoms than pre-COVID groups, and those with attention-deficit/hyperactivity disorder showed inflated conduct problems, while those with autism spectrum disorder exhibited decreased prosocial behaviours. Females with ASD had higher emotional symptoms compared to males. CYP with NDDs, and those without, showed higher levels of parent-reported mental health problems than comparable cohorts pre-COVID-19.
Jacqueline Nonweiler; Fiona Rattray; Jennifer Baulcomb; Francesca Happé; Michael Absoud. Prevalence and Associated Factors of Emotional and Behavioural Difficulties during COVID-19 Pandemic in Children with Neurodevelopmental Disorders. Children 2020, 7, 128 .
AMA StyleJacqueline Nonweiler, Fiona Rattray, Jennifer Baulcomb, Francesca Happé, Michael Absoud. Prevalence and Associated Factors of Emotional and Behavioural Difficulties during COVID-19 Pandemic in Children with Neurodevelopmental Disorders. Children. 2020; 7 (9):128.
Chicago/Turabian StyleJacqueline Nonweiler; Fiona Rattray; Jennifer Baulcomb; Francesca Happé; Michael Absoud. 2020. "Prevalence and Associated Factors of Emotional and Behavioural Difficulties during COVID-19 Pandemic in Children with Neurodevelopmental Disorders." Children 7, no. 9: 128.
Background Internalising (anxiety and low mood) and externalising (aggressive or outburst behaviours, and irritability) difficulties are very common in autism spectrum disorder (ASD) across the life span, relatively stable over time and often associated with poorer quality of life. Understanding the cognitive mechanisms underlying internalising and externalising difficulties in ASD is essential for developing targeted supports and interventions. In the present study, we investigated established and less‐researched cognitive factors hypothesised to contribute to internalising and/or externalising difficulties in ASD, namely cognitive inflexibility (CI), intolerance of uncertainty (IU) and alexithymia. Based on previous models and clinical experience, we hypothesised that IU would lead to internalising symptoms, with alexithymia contributing to this pathway, and that CI would have a direct effect on externalising behaviours and may indirectly contribute to internalising symptoms via increasing IU. Methods Our sample consisted of 95 5‐ to 18‐year‐olds presenting to a specialist neurodevelopmental clinic and receiving a diagnosis of ASD. Parents/caregivers completed questionnaires assessing ASD symptomatology, internalising and externalising difficulties, CI, IU and alexithymia. Structural equation modelling was used to examine the hypothesised pathways and relationships between the main variables of interest. Results Cognitive Inflexibility played a significant direct role in the pathway from ASD symptoms to externalising symptoms in ASD, and indirect role via IU in the pathway to internalising problems. Relationships between alexithymia and both internalising and externalising symptoms were weaker, with alexithymia predicting internalising difficulties via IU only. Conclusions The finding of a direct pathway from CI to externalising behaviours is novel, as is the indirect role of CI in internalising symptomatology. Of the three cognitive mechanisms examined, only CI significantly predicted externalising symptoms. Possible implications for interventions and supports targeting these cognitive processes in ASD are discussed.
Ann Ozsivadjian; Matthew J. Hollocks; Iliana Magiati; Francesca Happé; Gillian Baird; Michael Absoud. Is cognitive inflexibility a missing link? The role of cognitive inflexibility, alexithymia and intolerance of uncertainty in externalising and internalising behaviours in young people with autism spectrum disorder. Journal of Child Psychology and Psychiatry 2020, 62, 715 -724.
AMA StyleAnn Ozsivadjian, Matthew J. Hollocks, Iliana Magiati, Francesca Happé, Gillian Baird, Michael Absoud. Is cognitive inflexibility a missing link? The role of cognitive inflexibility, alexithymia and intolerance of uncertainty in externalising and internalising behaviours in young people with autism spectrum disorder. Journal of Child Psychology and Psychiatry. 2020; 62 (6):715-724.
Chicago/Turabian StyleAnn Ozsivadjian; Matthew J. Hollocks; Iliana Magiati; Francesca Happé; Gillian Baird; Michael Absoud. 2020. "Is cognitive inflexibility a missing link? The role of cognitive inflexibility, alexithymia and intolerance of uncertainty in externalising and internalising behaviours in young people with autism spectrum disorder." Journal of Child Psychology and Psychiatry 62, no. 6: 715-724.
Children and young people (CYP) with neurodevelopmental disorders (NDDs) may be particularly vulnerable to adverse mental health effects due to the COVID-19 pandemic. We conducted a cross-sectional U.K parent-reported study from 2nd April-2nd June 2020, using the Strengths & Difficulties Questionnaire. CYP with NDDs (n=371) compared to neurotypical controls, had a higher prevalence of emotional symptoms (42% vs 15%), conduct problems (28% vs 9%), and lower prosocial behaviours (54% vs 22%). Those with attention-deficit/hyperactivity disorder showed inflated conduct, and those with autism spectrum disorder exhibited decreased prosocial behaviours. Females with ASD had higher emotional symptoms compared to males.
Jacqueline Nonweiler; Fiona Rattray; Jennifer Baulcomb; Francesca Happe; Michael Absoud. Prevalence and Associated Factors of Emotional and Behavioural Difficulties during COVID-19 Pandemic in Children with Neurodevelopmental Disorders. 2020, 1 .
AMA StyleJacqueline Nonweiler, Fiona Rattray, Jennifer Baulcomb, Francesca Happe, Michael Absoud. Prevalence and Associated Factors of Emotional and Behavioural Difficulties during COVID-19 Pandemic in Children with Neurodevelopmental Disorders. . 2020; ():1.
Chicago/Turabian StyleJacqueline Nonweiler; Fiona Rattray; Jennifer Baulcomb; Francesca Happe; Michael Absoud. 2020. "Prevalence and Associated Factors of Emotional and Behavioural Difficulties during COVID-19 Pandemic in Children with Neurodevelopmental Disorders." , no. : 1.
We aimed to describe the extent of neurodevelopmental impairments and identify the genetic etiologies in a large cohort of patients with epilepsy with myoclonic atonic seizures (MAE). We deeply phenotyped MAE patients for epilepsy features, intellectual disability, autism spectrum disorder, and attention-deficit/hyperactivity disorder using standardized neuropsychological instruments. We performed exome analysis (whole exome sequencing) filtered on epilepsy and neuropsychiatric gene sets to identify genetic etiologies. We analyzed 101 patients with MAE (70% male). The median age of seizure onset was 34 months (range = 6-72 months). The main seizure types were myoclonic atonic or atonic in 100%, generalized tonic-clonic in 72%, myoclonic in 69%, absence in 60%, and tonic seizures in 19% of patients. We observed intellectual disability in 62% of patients, with extremely low adaptive behavioral scores in 69%. In addition, 24% exhibited symptoms of autism and 37% exhibited attention-deficit/hyperactivity symptoms. We discovered pathogenic variants in 12 (14%) of 85 patients, including five previously published patients. These were pathogenic genetic variants in SYNGAP1 (n = 3), KIAA2022 (n = 2), and SLC6A1 (n = 2), as well as KCNA2, SCN2A, STX1B, KCNB1, and MECP2 (n = 1 each). We also identified three new candidate genes, ASH1L, CHD4, and SMARCA2 in one patient each. MAE is associated with significant neurodevelopmental impairment. MAE is genetically heterogeneous, and we identified a pathogenic genetic etiology in 14% of this cohort by exome analysis. These findings suggest that MAE is a manifestation of several etiologies rather than a discrete syndromic entity.
Shan Tang; Laura Addis; Anna Belinda Smith; Simon Topp; Manuela Pendziwiat; Davide Mei; Alasdair Parker; Shakti Agrawal; Elaine Hughes; Karine Lascelles; Ruth E. Williams; Penny Fallon; Robert Robinson; Helen J. Cross; Tammy Hedderly; Christin Eltze; Tim Kerr; Archana Desurkar; Nahin Hussain; Maria Kinali; Irene Bagnasco; Grace Vassallo; William Whitehouse; Sushma Goyal; Michael Absoud; EuroEPINOMICS-RES Consortium; Rikke S. Møller; Ingo Helbig; Yvonne G. Weber; Carla Marini; Renzo Guerrini; Michael A. Simpson; Deb K. Pal; Dana Craiu; Carol Davila; Alexandru Obregia; Peter De Jonghe; Anna‐Elina Lehesjoki; Hiltrud Muhle; Bernd Neubauer; Kaja Selmer; Ulrich Stephani; Katalin Sterbova; Pasquale Striano; Tiina Talvik; Sarah Von Spiczak; Sarah Weckhuysen; Hande Caglayan; Dorota Hoffman‐Zacharska. Phenotypic and genetic spectrum of epilepsy with myoclonic atonic seizures. Epilepsia 2020, 61, 995 -1007.
AMA StyleShan Tang, Laura Addis, Anna Belinda Smith, Simon Topp, Manuela Pendziwiat, Davide Mei, Alasdair Parker, Shakti Agrawal, Elaine Hughes, Karine Lascelles, Ruth E. Williams, Penny Fallon, Robert Robinson, Helen J. Cross, Tammy Hedderly, Christin Eltze, Tim Kerr, Archana Desurkar, Nahin Hussain, Maria Kinali, Irene Bagnasco, Grace Vassallo, William Whitehouse, Sushma Goyal, Michael Absoud, EuroEPINOMICS-RES Consortium, Rikke S. Møller, Ingo Helbig, Yvonne G. Weber, Carla Marini, Renzo Guerrini, Michael A. Simpson, Deb K. Pal, Dana Craiu, Carol Davila, Alexandru Obregia, Peter De Jonghe, Anna‐Elina Lehesjoki, Hiltrud Muhle, Bernd Neubauer, Kaja Selmer, Ulrich Stephani, Katalin Sterbova, Pasquale Striano, Tiina Talvik, Sarah Von Spiczak, Sarah Weckhuysen, Hande Caglayan, Dorota Hoffman‐Zacharska. Phenotypic and genetic spectrum of epilepsy with myoclonic atonic seizures. Epilepsia. 2020; 61 (5):995-1007.
Chicago/Turabian StyleShan Tang; Laura Addis; Anna Belinda Smith; Simon Topp; Manuela Pendziwiat; Davide Mei; Alasdair Parker; Shakti Agrawal; Elaine Hughes; Karine Lascelles; Ruth E. Williams; Penny Fallon; Robert Robinson; Helen J. Cross; Tammy Hedderly; Christin Eltze; Tim Kerr; Archana Desurkar; Nahin Hussain; Maria Kinali; Irene Bagnasco; Grace Vassallo; William Whitehouse; Sushma Goyal; Michael Absoud; EuroEPINOMICS-RES Consortium; Rikke S. Møller; Ingo Helbig; Yvonne G. Weber; Carla Marini; Renzo Guerrini; Michael A. Simpson; Deb K. Pal; Dana Craiu; Carol Davila; Alexandru Obregia; Peter De Jonghe; Anna‐Elina Lehesjoki; Hiltrud Muhle; Bernd Neubauer; Kaja Selmer; Ulrich Stephani; Katalin Sterbova; Pasquale Striano; Tiina Talvik; Sarah Von Spiczak; Sarah Weckhuysen; Hande Caglayan; Dorota Hoffman‐Zacharska. 2020. "Phenotypic and genetic spectrum of epilepsy with myoclonic atonic seizures." Epilepsia 61, no. 5: 995-1007.
Background Individuals with co-occurring hyperactivity disorder/hyperactivity disorder (ADHD) and autism spectrum disorder (ASD) can have complex presentations that may complicate diagnosis and treatment. There are established guidelines with regard to the identification and treatment of ADHD and ASD as independent conditions. However, ADHD and ASD were not formally recognised diagnostically as co-occurring conditions until the Diagnostic and Statistical Manual of Mental Disorders 5 (DSM-5) was published in 2013. Hence, awareness and understanding of both conditions when they co-occur is less established and there is little guidance in the clinical literature. This has led to uncertainty among healthcare practitioners when working with children, young people and adults who present with co-existing ADHD and ASD. The United Kingdom ADHD Partnership (UKAP) therefore convened a meeting of professional experts that aimed to address this gap and reach expert consensus on the topic that will aid healthcare practitioners and allied professionals when working with this complex and vulnerable population. Method UK experts from multiple disciplines in the fields of ADHD and ASD convened in London in December 2017. The meeting provided the opportunity to address the complexities of ADHD and ASD as a co-occurring presentation from different perspectives and included presentations, discussion and group work. The authors considered the clinical challenges of working with this complex group of individuals, producing a consensus for a unified approach when working with male and female, children, adolescents and adults with co-occurring ADHD and ASD. This was written up, circulated and endorsed by all authors. Results The authors reached a consensus of practical recommendations for working across the lifespan with males and females with ADHD and ASD. Consensus was reached on topics of (1) identification and assessment using rating scales, clinical diagnostic interviews and objective supporting assessments; outcomes of assessment, including standards of clinical reporting; (2) non-pharmacological interventions and care management, including psychoeducation, carer interventions/carer training, behavioural/environmental and Cognitive Behavioural Therapy (CBT) approaches; and multi-agency liaison, including educational interventions, career advice, occupational skills and training, and (3) pharmacological treatments. Conclusions The guidance and practice recommendations (Tables 1, 4, 5, 7, 8 and 10) will support healthcare practitioners and allied professionals to meet the needs of this complex group from a multidisciplinary perspective. Further research is needed to enhance our understanding of the diagnosis, treatment and management of individuals presenting with comorbid ADHD and ASD.
Susan Young; Jack Hollingdale; Michael Absoud; Patrick Bolton; Polly Branney; William Colley; Emily Craze; Mayuri Dave; Quinton Deeley; Emad Farrag; Gisli Gudjonsson; Peter Hill; Ho-Lan Liang; Clodagh Murphy; Peri Mackintosh; Marianna Murin; Fintan O’Regan; Dennis Ougrin; Patricia Rios; Nancy Stover; Eric Taylor; Emma Louise Woodhouse. Guidance for identification and treatment of individuals with attention deficit/hyperactivity disorder and autism spectrum disorder based upon expert consensus. BMC Medicine 2020, 18, 1 -29.
AMA StyleSusan Young, Jack Hollingdale, Michael Absoud, Patrick Bolton, Polly Branney, William Colley, Emily Craze, Mayuri Dave, Quinton Deeley, Emad Farrag, Gisli Gudjonsson, Peter Hill, Ho-Lan Liang, Clodagh Murphy, Peri Mackintosh, Marianna Murin, Fintan O’Regan, Dennis Ougrin, Patricia Rios, Nancy Stover, Eric Taylor, Emma Louise Woodhouse. Guidance for identification and treatment of individuals with attention deficit/hyperactivity disorder and autism spectrum disorder based upon expert consensus. BMC Medicine. 2020; 18 (1):1-29.
Chicago/Turabian StyleSusan Young; Jack Hollingdale; Michael Absoud; Patrick Bolton; Polly Branney; William Colley; Emily Craze; Mayuri Dave; Quinton Deeley; Emad Farrag; Gisli Gudjonsson; Peter Hill; Ho-Lan Liang; Clodagh Murphy; Peri Mackintosh; Marianna Murin; Fintan O’Regan; Dennis Ougrin; Patricia Rios; Nancy Stover; Eric Taylor; Emma Louise Woodhouse. 2020. "Guidance for identification and treatment of individuals with attention deficit/hyperactivity disorder and autism spectrum disorder based upon expert consensus." BMC Medicine 18, no. 1: 1-29.
Michael Absoud; Osman Malik. Catatonia: An under-recognised treatable neuropsychiatric syndrome in developmental disorders. European Journal of Paediatric Neurology 2020, 25, 4 .
AMA StyleMichael Absoud, Osman Malik. Catatonia: An under-recognised treatable neuropsychiatric syndrome in developmental disorders. European Journal of Paediatric Neurology. 2020; 25 ():4.
Chicago/Turabian StyleMichael Absoud; Osman Malik. 2020. "Catatonia: An under-recognised treatable neuropsychiatric syndrome in developmental disorders." European Journal of Paediatric Neurology 25, no. : 4.
Anoxic‐epileptic seizures (AES) are rare outcomes of common childhood reflex anoxic syncope that trigger a true epileptic seizure. The term AES was coined by Stephenson in 1983, to differentiate these events from convulsive syncopes and the more common reflex anoxic syncopes. A genetic susceptibility for AES has been postulated; but, its molecular basis has up to now been elusive. We report here two illustrative cases and show the association of de novo SCN8A variants and AES. One of them had focal or generalized seizures and autonomic symptoms triggered by orthostatism; the second had breath‐holding spells triggered by pain or exercise leading to tonic–clonic seizures; both had repeatedly normal EEGs and a family history of reflex syncope. The data of three additional AES patients further suggest, for the first time, a link between SCN8A pathogenic variants and AES. The neurodevelopment of four patients was abnormal. Four of the five SCN8A mutations observed here were previously described in patients with seizure disorders. Seizures responded particularly well to sodium channel blockers. Our observation enriches the spectrum of seizures linked with SCN8A pathogenic variants.
Emmanuelle Ranza; Werner Z'graggen; Mathias Lidgren; Maurice Beghetti; Michel Guipponi; Stylianos E. Antonarakis; Michael Absoud; Sushma Goyal; Deb K. Pal; Christian M. Korff. SCN8A heterozygous variants are associated with anoxic‐epileptic seizures. American Journal of Medical Genetics Part A 2020, 182, 1209 -1216.
AMA StyleEmmanuelle Ranza, Werner Z'graggen, Mathias Lidgren, Maurice Beghetti, Michel Guipponi, Stylianos E. Antonarakis, Michael Absoud, Sushma Goyal, Deb K. Pal, Christian M. Korff. SCN8A heterozygous variants are associated with anoxic‐epileptic seizures. American Journal of Medical Genetics Part A. 2020; 182 (5):1209-1216.
Chicago/Turabian StyleEmmanuelle Ranza; Werner Z'graggen; Mathias Lidgren; Maurice Beghetti; Michel Guipponi; Stylianos E. Antonarakis; Michael Absoud; Sushma Goyal; Deb K. Pal; Christian M. Korff. 2020. "SCN8A heterozygous variants are associated with anoxic‐epileptic seizures." American Journal of Medical Genetics Part A 182, no. 5: 1209-1216.
Background Children with intellectual disabilities are likely to present with challenging behaviour. Parent mediated interventions have shown utility in influencing child behaviour, although there is a paucity of UK research into challenging behaviour interventions in this population. NICE guidelines favour Stepping Stones Triple P (SSTP) as a challenging behaviour intervention and this trial aims to evaluate its clinical and cost effectiveness in preschool children with moderate to severe intellectual disabilities. Methods This trial launched in 2017 at four sites across England, with the aim of recruiting 258 participants (aged 30–59 months). The Intervention Group receive nine weeks of SSTP parenting therapy (six group sessions and three individualised face to face or telephone sessions) in addition to Treatment as Usual, whilst the Treatment as Usual only group receive other available services in each location. Both study groups undergo the study measurements at baseline and at four and twelve months. Outcome measures include parent reports and structured observations of behaviour. Service use and health related quality of life data will also be collected to carry out a cost effectiveness and utility evaluation. Discussion Findings from this study will inform policy regarding interventions for challenging behaviour in young children with moderate to severe intellectual disabilities. Trial registration number Clinicaltrials.gov, NCT03086876. Registered 22nd March 2017, https://clinicaltrials.gov/ct2/show/NCT03086876.
Olayinka Farris; Rachel Royston; Michael Absoud; Gareth Ambler; Jacqueline Barnes; Rachael Hunter; Marinos Kyriakopoulos; Kate Oulton; Eleni Paliokosta; Monica Panca; Laura Paulauskaite; Michaela Poppe; Federico Ricciardi; Aditya Sharma; Vicky Slonims; Una Summerson; Alastair Sutcliffe; Megan Thomas; Angela Hassiotis. Clinical and cost effectiveness of a parent mediated intervention to reduce challenging behaviour in pre-schoolers with moderate to severe intellectual disability (EPICC-ID) study protocol: a multi-centre, parallel-group randomised controlled trial. BMC Psychiatry 2020, 20, 1 -11.
AMA StyleOlayinka Farris, Rachel Royston, Michael Absoud, Gareth Ambler, Jacqueline Barnes, Rachael Hunter, Marinos Kyriakopoulos, Kate Oulton, Eleni Paliokosta, Monica Panca, Laura Paulauskaite, Michaela Poppe, Federico Ricciardi, Aditya Sharma, Vicky Slonims, Una Summerson, Alastair Sutcliffe, Megan Thomas, Angela Hassiotis. Clinical and cost effectiveness of a parent mediated intervention to reduce challenging behaviour in pre-schoolers with moderate to severe intellectual disability (EPICC-ID) study protocol: a multi-centre, parallel-group randomised controlled trial. BMC Psychiatry. 2020; 20 (1):1-11.
Chicago/Turabian StyleOlayinka Farris; Rachel Royston; Michael Absoud; Gareth Ambler; Jacqueline Barnes; Rachael Hunter; Marinos Kyriakopoulos; Kate Oulton; Eleni Paliokosta; Monica Panca; Laura Paulauskaite; Michaela Poppe; Federico Ricciardi; Aditya Sharma; Vicky Slonims; Una Summerson; Alastair Sutcliffe; Megan Thomas; Angela Hassiotis. 2020. "Clinical and cost effectiveness of a parent mediated intervention to reduce challenging behaviour in pre-schoolers with moderate to severe intellectual disability (EPICC-ID) study protocol: a multi-centre, parallel-group randomised controlled trial." BMC Psychiatry 20, no. 1: 1-11.
### What you need to know A mother visits her general practitioner with her 3 year old daughter. She is concerned that the child is having regular “melt downs” and has become defiant when, for example, getting dressed or putting on her shoes. Her daughter is physically aggressive towards her and other family members. This happens often and sometimes lasts for up to an hour. The family has tried reward charts, time outs, and positive reinforcement. Challenging behaviour is commonly defined as behaviour that is of an intensity, frequency, or duration that threatens the physical safety of the person or others or restricts access to community facilities.12 Challenging behaviour can first occur in childhood and can be difficult for parents, carers, and family members to understand and manage. Parents and carers may present to healthcare services, including their GP, with concerns about their children’s behaviour. In some cases, challenging behaviour may be a sign of a known or an undiagnosed learning disability. This article gives particular advice about how to identify and manage children where a learning disability might be a contributing factor. Often, behaviours perceived as challenging serve a purpose for the child or young person, such as producing sensory stimulation, attracting attention, and avoiding demands.3 Some behaviours may be a form of communication which needs to be understood. Examples of challenging behaviours, sometimes reported by parents as “melt downs” or “defiance,” are • Physical aggression (biting, scratching, hitting) • Self injury (head banging, biting hands) • …
Michael Absoud; Holly Wake; Miriam Ziriat; Angela Hassiotis. Managing challenging behaviour in children with possible learning disability. BMJ 2019, 365, l1663 .
AMA StyleMichael Absoud, Holly Wake, Miriam Ziriat, Angela Hassiotis. Managing challenging behaviour in children with possible learning disability. BMJ. 2019; 365 ():l1663.
Chicago/Turabian StyleMichael Absoud; Holly Wake; Miriam Ziriat; Angela Hassiotis. 2019. "Managing challenging behaviour in children with possible learning disability." BMJ 365, no. : l1663.
SummaryAltered neural connectivity in neurodevelopmental disorders is likely subtle, meaning that neuroimaging literature studying development has produced heterogeneous findings. A recent study, published in this issue, illustrates the translational potential of functional connectivity magnetic resonance imaging findings as a biomarker for attention-deficit hyperactivity disorder and autism spectrum disorder. Importantly, it highlights the overlap between disorders, emphasising the need for transdiagnostic and dimensional approaches in neurodevelopment.Declaration of interestNone.
Chirag Mehra; Michael Absoud. Commentary on… the overlapping and distinct resting functional connectivity between autism spectrum disorder and attention-deficit hyperactivity disorder. The British Journal of Psychiatry 2019, 214, 345 -346.
AMA StyleChirag Mehra, Michael Absoud. Commentary on… the overlapping and distinct resting functional connectivity between autism spectrum disorder and attention-deficit hyperactivity disorder. The British Journal of Psychiatry. 2019; 214 (6):345-346.
Chicago/Turabian StyleChirag Mehra; Michael Absoud. 2019. "Commentary on… the overlapping and distinct resting functional connectivity between autism spectrum disorder and attention-deficit hyperactivity disorder." The British Journal of Psychiatry 214, no. 6: 345-346.
Jonathan Green; Michael Absoud; Victoria Grahame; Osman Malik; Emily Simonoff; Ann Le Couteur; Gillian Baird. Demand avoidance is not necessarily defiance – Authors' reply. The Lancet Child & Adolescent Health 2018, 2, e21 .
AMA StyleJonathan Green, Michael Absoud, Victoria Grahame, Osman Malik, Emily Simonoff, Ann Le Couteur, Gillian Baird. Demand avoidance is not necessarily defiance – Authors' reply. The Lancet Child & Adolescent Health. 2018; 2 (9):e21.
Chicago/Turabian StyleJonathan Green; Michael Absoud; Victoria Grahame; Osman Malik; Emily Simonoff; Ann Le Couteur; Gillian Baird. 2018. "Demand avoidance is not necessarily defiance – Authors' reply." The Lancet Child & Adolescent Health 2, no. 9: e21.
Aim Our aim was to ascertain the indications, side effects, and outcomes in children receiving therapeutic plasma exchange (TPE) for neurological disorders. Method Medical records were retrospectively reviewed for 58 consecutive children (age ≤16y) undergoing 67 courses of TPE across four tertiary centres. Patient characteristics, treatment schedules, complications, and outcomes were analysed. Results Median age at initiation of TPE was 9 years (range 1–15y). Indications included peripheral nervous system (PNS; n=18) and central nervous system (CNS; n=40) disorders. Courses comprised a median six exchanges (range 2–179) over 8 days (range 3–466). Forty‐two out of 58 (73%) children were severely disabled (bedridden) at initiation and 24 out of 58 (41%) were admitted to intensive care units. Treating clinicians’ impression of response was positive in 16 out of 18 of those with PNS disorders versus 22 out of 40 with CNS disorders (p=0.016). Improvements in disability (modified Rankin Scale) occurred in 13 out of 58 (22%) children by completion of TPE (p=0.003). Complications occurred in 40 out of 67 (60%) courses, of which 16 out of 67 (24%) were line related. Potentially life‐threatening complications occurred in 2 out of 67 (3%) courses. Interpretation This cohort study provides safety and efficacy information for clinicians and families and a basis for future prospective studies. What this paper adds Disability scores for severe neuroimmune disorders remained stable or improved during therapeutic plasma exchange treatment. Complications occurred frequently but were typically mild and correctable.
Michael Eyre; Yael Hacohen; Kate Lamb; Michael Absoud; Shakti Agrawal; Jonathan Gadian; Rajat Gupta; Rachel Kneen; David V Milford; Sunny Philip; Katie Rose; Martin Smith; Stefan Spinty; Evangeline Wassmer; Ming Lim; Cheryl Hemingway. Utility and safety of plasma exchange in paediatric neuroimmune disorders. Developmental Medicine & Child Neurology 2018, 61, 540 -546.
AMA StyleMichael Eyre, Yael Hacohen, Kate Lamb, Michael Absoud, Shakti Agrawal, Jonathan Gadian, Rajat Gupta, Rachel Kneen, David V Milford, Sunny Philip, Katie Rose, Martin Smith, Stefan Spinty, Evangeline Wassmer, Ming Lim, Cheryl Hemingway. Utility and safety of plasma exchange in paediatric neuroimmune disorders. Developmental Medicine & Child Neurology. 2018; 61 (5):540-546.
Chicago/Turabian StyleMichael Eyre; Yael Hacohen; Kate Lamb; Michael Absoud; Shakti Agrawal; Jonathan Gadian; Rajat Gupta; Rachel Kneen; David V Milford; Sunny Philip; Katie Rose; Martin Smith; Stefan Spinty; Evangeline Wassmer; Ming Lim; Cheryl Hemingway. 2018. "Utility and safety of plasma exchange in paediatric neuroimmune disorders." Developmental Medicine & Child Neurology 61, no. 5: 540-546.
Chirag Mehra; Annesha Sil; Tammy Hedderly; Marinos Kyriakopoulos; Ming Lim; Jessica Turnbull; Francesca Happe; Gillian Baird; Michael Absoud. Childhood disintegrative disorder and autism spectrum disorder: a systematic review. Developmental Medicine & Child Neurology 2018, 61, 523 -534.
AMA StyleChirag Mehra, Annesha Sil, Tammy Hedderly, Marinos Kyriakopoulos, Ming Lim, Jessica Turnbull, Francesca Happe, Gillian Baird, Michael Absoud. Childhood disintegrative disorder and autism spectrum disorder: a systematic review. Developmental Medicine & Child Neurology. 2018; 61 (5):523-534.
Chicago/Turabian StyleChirag Mehra; Annesha Sil; Tammy Hedderly; Marinos Kyriakopoulos; Ming Lim; Jessica Turnbull; Francesca Happe; Gillian Baird; Michael Absoud. 2018. "Childhood disintegrative disorder and autism spectrum disorder: a systematic review." Developmental Medicine & Child Neurology 61, no. 5: 523-534.
Pathological (or extreme) demand avoidance is a term sometimes applied to complex behaviours in children within—or beyond—autism spectrum disorder. The use of pathological demand avoidance as a diagnosis has, at times, led to altered referral practice and misunderstandings between professionals and the families of patients. In our Viewpoint, we reviewed the current literature and conclude that the evidence does not support the validity of pathological demand avoidance as an independent syndrome. Nevertheless, the use of the term highlights an important known range of co-occurring difficulties for many children with autism spectrum disorder that can substantially affect families. We explore how these difficulties can best be understood through understanding of social, sensory, and cognitive sensitivities in autism spectrum disorder, identification of frequently occurring comorbid conditions, and assessment of how these problems interact within the child's social environment. Such understanding should then inform individualised management strategies for children and families, and in social settings, such as education. It is crucial that a shared understanding is achieved between professionals and families in this area.
Jonathan Green; Michael Absoud; Victoria Grahame; Osman Malik; Emily Simonoff; Ann Le Couteur; Gillian Baird. Pathological Demand Avoidance: symptoms but not a syndrome. The Lancet Child & Adolescent Health 2018, 2, 455 -464.
AMA StyleJonathan Green, Michael Absoud, Victoria Grahame, Osman Malik, Emily Simonoff, Ann Le Couteur, Gillian Baird. Pathological Demand Avoidance: symptoms but not a syndrome. The Lancet Child & Adolescent Health. 2018; 2 (6):455-464.
Chicago/Turabian StyleJonathan Green; Michael Absoud; Victoria Grahame; Osman Malik; Emily Simonoff; Ann Le Couteur; Gillian Baird. 2018. "Pathological Demand Avoidance: symptoms but not a syndrome." The Lancet Child & Adolescent Health 2, no. 6: 455-464.
Research on sex-related differences in Autism Spectrum Disorder (ASD) has been impeded by small samples. We pooled 28 datasets from 18 sites across nine European countries to examine sex differences in the ASD phenotype on the ADI-R (376 females, 1763 males) and ADOS (233 females, 1187 males). On the ADI-R, early childhood restricted and repetitive behaviours were lower in females than males, alongside comparable levels of social interaction and communication difficulties in females and males. Current ADI-R and ADOS scores showed no sex differences for ASD severity. There were lower socio-communicative symptoms in older compared to younger individuals. This large European ASD sample adds to the literature on sex and age variations of ASD symptomatology.
J. Tillmann; Karen Ashwood; Michael Absoud; S. Bölte; Frédérique Bonnet-Brilhault; J. K. Buitelaar; Sara Calderoni; Rosa Calvo-Escalona; Ricardo Canal-Bedia; R. Canitano; A. De Bildt; Marie Gomot; P. J. Hoekstra; A. Kaale; H. McConachie; Declan Murphy; A. Narzisi; I. Oosterling; Milica Pejovic Milovancevic; A. M. Persico; O. Puig; H. Roeyers; N. Rommelse; R. Sacco; V. Scandurra; Andrew Stanfield; E. Zander; Tony Charman. Evaluating Sex and Age Differences in ADI-R and ADOS Scores in a Large European Multi-site Sample of Individuals with Autism Spectrum Disorder. Journal of Autism and Developmental Disorders 2018, 48, 2490 -2505.
AMA StyleJ. Tillmann, Karen Ashwood, Michael Absoud, S. Bölte, Frédérique Bonnet-Brilhault, J. K. Buitelaar, Sara Calderoni, Rosa Calvo-Escalona, Ricardo Canal-Bedia, R. Canitano, A. De Bildt, Marie Gomot, P. J. Hoekstra, A. Kaale, H. McConachie, Declan Murphy, A. Narzisi, I. Oosterling, Milica Pejovic Milovancevic, A. M. Persico, O. Puig, H. Roeyers, N. Rommelse, R. Sacco, V. Scandurra, Andrew Stanfield, E. Zander, Tony Charman. Evaluating Sex and Age Differences in ADI-R and ADOS Scores in a Large European Multi-site Sample of Individuals with Autism Spectrum Disorder. Journal of Autism and Developmental Disorders. 2018; 48 (7):2490-2505.
Chicago/Turabian StyleJ. Tillmann; Karen Ashwood; Michael Absoud; S. Bölte; Frédérique Bonnet-Brilhault; J. K. Buitelaar; Sara Calderoni; Rosa Calvo-Escalona; Ricardo Canal-Bedia; R. Canitano; A. De Bildt; Marie Gomot; P. J. Hoekstra; A. Kaale; H. McConachie; Declan Murphy; A. Narzisi; I. Oosterling; Milica Pejovic Milovancevic; A. M. Persico; O. Puig; H. Roeyers; N. Rommelse; R. Sacco; V. Scandurra; Andrew Stanfield; E. Zander; Tony Charman. 2018. "Evaluating Sex and Age Differences in ADI-R and ADOS Scores in a Large European Multi-site Sample of Individuals with Autism Spectrum Disorder." Journal of Autism and Developmental Disorders 48, no. 7: 2490-2505.
Encephalitis is a syndrome of neurological dysfunction due to inflammation of the brain parenchyma, caused by an infection or an exaggerated host immune response, or both. Attenuation of brain inflammation through modulation of the immune response could improve patient outcomes. Biological agents such as immunoglobulin that have both anti‐inflammatory and immunomodulatory properties may therefore be useful as adjunctive therapies for people with encephalitis. To assess the efficacy and safety of intravenous immunoglobulin (IVIG) as add‐on treatment for children with encephalitis. The Cochrane Multiple Sclerosis and Rare Diseases of the CNS group's Information Specialist searched the following databases up to 30 September 2016: CENTRAL, MEDLINE, Embase, CINAHL, ClinicalTrials.gov, and the WHO ICTRP Search Portal. In addition, two review authors searched Science Citation Index Expanded (SCI‐EXPANDED) & Conference Proceedings Citation Index ‐ Science (CPCI‐S) (Web of Science Core Collection, Thomson Reuters) (1945 to January 2016), Global Health Library (Virtual Health Library), and Database of Abstracts of Reviews of Effects (DARE). Randomised controlled trials (RCTs) comparing IVIG in addition to standard care versus standard care alone or placebo. Two review authors independently selected articles for inclusion, extracted relevant data, and assessed quality of trials. We resolved disagreements by discussion among the review authors. Where possible, we contacted authors of included studies for additional information. We presented results as risk ratios (RR) or mean differences (MD) with 95% confidence intervals (CI). The search identified three RCTs with 138 participants. All three trials included only children with viral encephalitis, one of these included only children with Japanese encephalitis, a specific form of viral encephalitis. Only the trial of Japanese encephalitis (22 children) contributed to the primary outcome of this review and follow‐up in that study was for three to six months after hospital discharge. There was no follow‐up of participants in the other two studies. We identified one ongoing trial. For the primary outcomes, the results showed no significant difference between IVIG and placebo when used in the treatment of children with Japanese encephalitis: significant disability (RR 0.75, 95% CI 0.22 to 2.60; P = 0.65) and serious adverse events (RR 1.00, 95% CI 0.07 to 14.05; P = 1.00). For the secondary outcomes, the study of Japanese encephalitis showed no significant difference between IVIG and placebo when assessing significant disability at hospital discharge (RR 1.00, 95% CI 0.60 to 1.67). There was no significant difference (P = 0.53) in Glasgow Coma Score at discharge between IVIG (median score 14; range 3 to 15) and placebo (median 14 score; range 7 to 15) in the Japanese encephalitis study. The median length of hospital stay in the Japanese encephalitis study was similar for IVIG‐treated (median...
Mildred A Iro; Natalie G Martin; Michael Absoud; Andrew J Pollard. Intravenous immunoglobulin for the treatment of childhood encephalitis. Cochrane Database of Systematic Reviews 2017, 2017, 1 .
AMA StyleMildred A Iro, Natalie G Martin, Michael Absoud, Andrew J Pollard. Intravenous immunoglobulin for the treatment of childhood encephalitis. Cochrane Database of Systematic Reviews. 2017; 2017 (10):1.
Chicago/Turabian StyleMildred A Iro; Natalie G Martin; Michael Absoud; Andrew J Pollard. 2017. "Intravenous immunoglobulin for the treatment of childhood encephalitis." Cochrane Database of Systematic Reviews 2017, no. 10: 1.
Background Transverse myelitis (TM) is an immune-mediated disorder of the spinal cord that affects adults and children and that causes motor, sensory and autonomic dysfunction. There is a prolonged recovery phase, which may continue for many years. Neuromyelitis optica (NMO) is an uncommon relapsing inflammatory central nervous system condition in which TM can be the first presenting symptom. As TM and NMO affect many patients in the prime of their working life, the disorder can impose a significant demand on health resources. There are currently no robust controlled trials in children or adults to inform the optimal treatment of TM. However, treatment with intravenous immunoglobulin (IVIG) is being effectively used in the management of a range of neurological conditions. Although other interventions such as plasma exchange (PLEX) in addition to intravenous (IV) methylprednisolone therapy can be beneficial in TM, PLEX is costly and technically challenging to deliver in the acute setting. IVIG is more readily accessible and less costly. Objective To evaluate whether additional and early treatment with IVIG is of extra benefit in TM compared with standard therapy with IV steroids. Design A multicentre, single-blind, parallel-group randomised controlled trial of IVIG compared with standard therapy for the treatment of TM in adults and children. Participants Patients aged ≥ 1 year diagnosed with either acute first-onset TM or first presentation of NMO. Target recruitment was 170 participants (85 participants per arm). Interventions Participants were randomised 1 : 1 to treatment with IV methylprednisolone only or treatment with IV methylprednisolone plus 2 g/kg of IVIG in divided doses within 5 days of the first commencement of steroid therapy. Main outcome measures Primary outcome measure – American Spinal Injury Association (ASIA) Impairment Scale at 6 months post randomisation, with a good outcome defined by a two-grade change. Secondary and tertiary outcome measures – ASIA motor and sensory scales, Expanded Disability Status Scale, health outcome, quality of life, Client Service Receipt Inventory and International Spinal Cord Injury Pain, Bladder and Bowel Basic Data Sets. Results In total, 26 participants were screened and two were randomised into the study. With the limited sample size, treatment effect could not be determined. However, we identified barriers to accrual that included strict inclusion criteria, the short enrolment window, challenges associated with the use of the ASIA Impairment Scale as an outcome measure and estimation of the incidence of TM. Conclusions The study did not reach the end point and the effect of IVIG in TM/NMO could not be determined. Investigators should be aware of the potential challenges associated with carrying out a rare disease trial with a short enrolment window. The study question is one that still necessitates investigation. Preliminary work to ameliorate the effect of the barriers encountered in this study is vital. Trial registration EudraCT 2014-002335-34, ClinicalTrials.gov NCT02398994 and Current Controlled Trials ISRCTN12127581. Funding This project was funded by the NIHR Health Technology Assessment programme and will be published in full in Health Technology Assessment; Vol. 21, No. 31. See the NIHR Journals Library website for further project information. Funding was also received from Biotest AG, Germany (supply of IVIG) and the Transverse Myelitis Society (excess research cost to facilitate study initiation).
Michael Absoud; Peter Brex; Olga Ciccarelli; Onyinye Diribe; Gavin Giovannoni; Jennifer Hellier; Rosemary Howe; Rachel Holland; Joanna Kelly; Paul McCrone; Caroline Murphy; Jackie Palace; Andrew Pickles; Michael Pike; Neil Robertson; Anu Jacob; Ming Lim. A multicentre randomiSed controlled TRial of IntraVEnous immunoglobulin compared with standard therapy for the treatment of transverse myelitis in adults and children (STRIVE). Health Technology Assessment 2017, 21, 1 -50.
AMA StyleMichael Absoud, Peter Brex, Olga Ciccarelli, Onyinye Diribe, Gavin Giovannoni, Jennifer Hellier, Rosemary Howe, Rachel Holland, Joanna Kelly, Paul McCrone, Caroline Murphy, Jackie Palace, Andrew Pickles, Michael Pike, Neil Robertson, Anu Jacob, Ming Lim. A multicentre randomiSed controlled TRial of IntraVEnous immunoglobulin compared with standard therapy for the treatment of transverse myelitis in adults and children (STRIVE). Health Technology Assessment. 2017; 21 (31):1-50.
Chicago/Turabian StyleMichael Absoud; Peter Brex; Olga Ciccarelli; Onyinye Diribe; Gavin Giovannoni; Jennifer Hellier; Rosemary Howe; Rachel Holland; Joanna Kelly; Paul McCrone; Caroline Murphy; Jackie Palace; Andrew Pickles; Michael Pike; Neil Robertson; Anu Jacob; Ming Lim. 2017. "A multicentre randomiSed controlled TRial of IntraVEnous immunoglobulin compared with standard therapy for the treatment of transverse myelitis in adults and children (STRIVE)." Health Technology Assessment 21, no. 31: 1-50.
A systematic literature review of intravenous immunoglobulin (IVIG) treatment of paediatric neurological conditions was performed to summarize the evidence, provide recommendations, and suggest future research. A MEDLINE search for articles reporting on IVIG treatment of paediatric neuroinflammatory, neurodevelopmental, and neurodegenerative conditions published before September 2015, excluding single case reports and those not in English. Owing to heterogeneous outcome measures, meta-analysis was not possible. Findings were combined and evidence graded. Sixty-five studies were analysed. IVIG reduces recovery time in Guillain–Barré syndrome (grade B). IVIG is as effective as corticosteroids in chronic inflammatory demyelinating polyradiculoneuropathy (grade C), and as effective as tacrolimus in Rasmussen syndrome (grade C). IVIG improves recovery in acute disseminated encephalomyelitis (grade C), reduces mortality in acute encephalitis syndrome with myocarditis (grade C), and improves function and stabilizes disease in myasthenia gravis (grade C). IVIG improves outcome in N-methyl-d-aspartate receptor encephalitis (grade C) and opsoclonus–myoclonus syndrome (grade C), reduces cataplexy symptoms in narcolepsy (grade C), speeds recovery in Sydenham chorea (grade C), reduces tics in selected cases of Tourette syndrome (grade D), and improves symptoms in paediatric autoimmune neuropsychiatric disorder associated with streptococcal infection (grade B). IVIG is a useful therapy in selected neurological conditions. Well-designed, prospective, multi-centre studies with standardized outcome measures are required to compare treatments.
Jonathan Gadian; Emma Kirk; Kate Holliday; Ming Lim; Michael Absoud. Systematic review of immunoglobulin use in paediatric neurological and neurodevelopmental disorders. Developmental Medicine & Child Neurology 2016, 59, 136 -144.
AMA StyleJonathan Gadian, Emma Kirk, Kate Holliday, Ming Lim, Michael Absoud. Systematic review of immunoglobulin use in paediatric neurological and neurodevelopmental disorders. Developmental Medicine & Child Neurology. 2016; 59 (2):136-144.
Chicago/Turabian StyleJonathan Gadian; Emma Kirk; Kate Holliday; Ming Lim; Michael Absoud. 2016. "Systematic review of immunoglobulin use in paediatric neurological and neurodevelopmental disorders." Developmental Medicine & Child Neurology 59, no. 2: 136-144.