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- Danielsson, A, et al.
(författare)
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Pallidal Deep Brain Stimulation in DYT6 Dystonia: Clinical Outcome and Predictive Factors for Motor Improvement
- 2019
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Ingår i: Journal of clinical medicine. - : MDPI AG. - 2077-0383. ; 8:12
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Tidskriftsartikel (refereegranskat)abstract
- Pallidal deep brain stimulation is an established treatment in dystonia. Available data on the effect in DYT-THAP1 dystonia (also known as DYT6 dystonia) are scarce and long-term follow-up studies are lacking. In this retrospective, multicenter follow-up case series of medical records of such patients, the clinical outcome of pallidal deep brain stimulation in DYT-THAP1 dystonia, was evaluated. The Burke Fahn Marsden Dystonia Rating Scale served as an outcome measure. Nine females and 5 males were enrolled, with a median follow-up of 4 years and 10 months after implant. All benefited from surgery: dystonia severity was reduced by a median of 58% (IQR 31-62, p = 0.001) at last follow-up, as assessed by the Burke Fahn Marsden movement subscale. In the majority of individuals, there was no improvement of speech or swallowing, and overall, the effect was greater in the trunk and limbs as compared to the cranio-cervical and orolaryngeal regions. No correlation was found between disease duration before surgery, age at surgery, or preoperative disease burden and the outcome of deep brain stimulation. Device- and therapy-related side-effects were few. Accordingly, pallidal deep brain stimulation should be considered in clinically impairing and pharmaco-resistant DYT-THAP1 dystonia. The method is safe and effective, both short- and long-term.
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- Tedroff, K., et al.
(författare)
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Botulinumtoxin A treatment in toddlers with cerebral palsy
- 2010
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Ingår i: Acta Paediatrica. - : Wiley. - 0803-5253 .- 1651-2227. ; 99:8, s. 1156-1162
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Tidskriftsartikel (refereegranskat)abstract
- Aims: In this study the aim was to evaluate the effect of botulinum toxin A (BoNT-A) treatment on muscle tone, contracture development and gait pattern in young children with cerebral palsy (CP). Method: Fifteen children with spastic CP (mean age = 16 months) were included in a randomized control study. All received a daily stretching programme and children in the BoNT-A group additionally received two injections, 6 months apart in the gastrocnemius muscle. Outcomes were assessed at baseline, and after 1 and 3.5 years. A 3D gait-analysis was performed at 5 years of age. Results: Plantarflexor muscle tone in the BoNT-A group was significantly reduced after 3.5 years, while the muscle tone at the ankle and knee in the control group remained unchanged. The change-score in knee-flexion muscle tone between the groups was significantly different after 3.5 years. The knee joint ROM was significantly increased at 1 year in the BoNT-A group but reduced at the knee and ankle joints in the control group after 3.5 years. No group differences were found for gait analysis, GMFM-66 or PEDI. Conclusion: Early treatment of BoNT-A in children with spastic CP may decrease muscle tone and decelerate contracture development after 3.5 years. The effect on gait development remains inconclusive.
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- Chen, RQ, et al.
(författare)
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Risk of intellectual disability in children born appropriate-for-gestational-age at term or post-term: impact of birth weight for gestational age and gestational age
- 2020
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Ingår i: European journal of epidemiology. - : Springer Science and Business Media LLC. - 1573-7284 .- 0393-2990. ; 35:3, s. 273-282
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Tidskriftsartikel (refereegranskat)abstract
- Children born small for gestational age have a higher risk of intellectual disability. We investigated associations of birth weight for gestational age percentile and gestational age with risk of intellectual disability in appropriate-for-gestational-age (AGA) children. We included 828,948 non-malformed term or post-term AGA singleton children (including 429,379 full siblings) born between 1998 and 2009 based on data from the Swedish Medical Birth Register. Diagnosis of intellectual disability after 3 years of age was identified through the Patient Register. Using Cox regression models, we calculated hazard ratios (HRs) with 95% confidence intervals (CIs) of intellectual disability among children with different birth weight percentiles and gestational age in the whole population and in a subpopulation of full siblings. A total of 1688 children were diagnosed with intellectual disability during follow-up. HRs (95% CIs) of intellectual disability for the low birth weight percentile groups (10th–24th and 25th–39th percentiles, respectively) versus the reference group (40th–59th percentiles) were 1.43 (1.22–1.67) and 1.28 (1.10–1.50) in population analysis and 1.52 (1.00–2.31) and 1.44 (1.00–2.09) in sibling comparison analysis. The increased risk for low birth weight percentiles in population analysis was stable irrespective of gestational age. A weak U-shaped association between gestational age and intellectual disability was observed in population analysis, although not in sibling comparison analysis. These findings suggest that among AGA children born at term or post-term, lower birth weight percentiles within the normal range are associated with increased risk of intellectual disability, regardless of gestational age.
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- Danielsson, A, et al.
(författare)
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Reliability and Validity of the Dyskinesia Impairment Scale in Children and Young Adults with Inherited or Idiopathic Dystonia
- 2020
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Ingår i: Journal of clinical medicine. - : MDPI AG. - 2077-0383. ; 9:8
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Tidskriftsartikel (refereegranskat)abstract
- Background: The Dyskinesia Impairment Scale (DIS) is a new assessment scale for dystonia and choreoathetosis in children and youth with dyskinetic cerebral palsy. Today, the Burke–Fahn–Marsden Dystonia Rating Scale (BFM) is mostly used to assess dystonia in children with inherited dystonia. The aim of this study was to assess reliability and validity of the DIS in children and youth with inherited or idiopathic dystonia. Methods: Reliability was measured by (1) the intraclass correlation coefficients (ICCs) for inter-rater and test-retest reliability, as well as (2) standard error of measurement (SEM) and minimal detectable difference (MDD). For concurrent validity of the DIS-dystonia subscale, the BFM was administered. Results: In total, 11 males and 9 females (median age 16 years and 7 months, range 6 to 24 years) were included. For inter-rater reliability, the ICCs for the DIS total score and the dystonia and choreoathetosis subscale scores were 0.83, 0.87, and 0.71, respectively. For test-retest reliability, the ICCs for the DIS total score and the dystonia and choreoathetosis subscale scores were 0.95, 0.88, and 0.93, respectively. The SEM and MDD for the total DIS were 3.98% and 11.04%, respectively. The Spearman correlation coefficient between the dystonia subscale and the BFM was 0.88 (p < 0.01). Conclusions: Good to excellent inter-rater, test-retest reliability, and validity were found for the total DIS and the dystonia subscale. The choreoathetosis subscale showed moderate inter-rater reliability and excellent test-retest reliability. The DIS may be a promising tool to assess dystonia and choreoathetosis in children and young adults with inherited or idiopathic dystonia.
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- Langstrom, B, et al.
(författare)
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PET i klinisk verksamhet.
- 1995
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Ingår i: Läkartidningen. ; 92, s. 3202-
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Tidskriftsartikel (övrigt vetenskapligt/konstnärligt)
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- Razaz, N, et al.
(författare)
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One-minute and five-minute Apgar scores and child developmental health at 5 years of age: a population-based cohort study in British Columbia, Canada
- 2019
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Ingår i: BMJ open. - : BMJ. - 2044-6055. ; 9:5, s. e027655-
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Tidskriftsartikel (refereegranskat)abstract
- We investigated the associations between Apgar scores at 1 and 5 min, across the entire range of score values, and child developmental health at 5 years of age.SettingBritish Columbia, CanadaParticipantsAll singleton term infants without major congenital anomalies born between 1993 and 2009, who had a developmental assessment in kindergarten between 1999 and 2014.Main outcomes and measuresDevelopmental vulnerability on one or more domains of the Early Development Instrument and special needs requirements. Adjusted rate ratios (aRRs) and 95% CIs were estimated using log-linear regression.ResultsOf the 150 081 children in the study, 45 334 (30.2%) were developmentally vulnerable and 3644 (2.5%) had special needs. There was an increasing trend in developmental vulnerability and special needs with decreasing 1 min and 5 min Apgar scores. Compared with children with an Apgar score of 10 at 5 min, the aRR for developmental vulnerability increased steadily with decreasing Apgar score from 1.02 (95% CI 1.00 to 1.04) for an Apgar score of 9 to 1.57 (95% CI 1.03 to 2.39) for an Apgar score of 2. Among children with 1 min Apgar scores in the 7–10 range, changes in Apgar scores between 1 and 5 min were associated with significant differences in developmental vulnerability. Compared with children who had an Apgar score of 9 at 1 min and 10 at 5 min, children with an Apgar score of 9 at both 1 and 5 min had higher rates of developmental vulnerability (aRR 1.03, 95% CI 1.01 to 1.05). Compared with infants with an Apgar of 10 at both 1 and 5 min, infants with a 1 min score of 10 and a 5 min score of <10 had higher rates of developmental vulnerability (aRR 1.53, 95% CI 1.08 to 2.17).ConclusionRisks of adverse developmental health and having special needs at 5 years of age are inversely associated with 1 min and 5 min Apgar scores across their entire range.
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- Sandvik, U, et al.
(författare)
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Cognition in Children with Arachnoid Cysts
- 2020
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Ingår i: Journal of clinical medicine. - : MDPI AG. - 2077-0383. ; 9:3
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Tidskriftsartikel (refereegranskat)abstract
- Background: This study aims to evaluate if children with temporal arachnoid cysts (AC) have cognitive symptoms and if neurosurgery improves these. Methods: A prospective case series study including consecutive pediatric patients with temporal AC. The children underwent neuroradiology, neuroopthalmologic evaluation, and a standard electroencephalography (EEG). Additionally, a neuropsychologist performed a standardized set of evaluations, with a one-year follow-up consisting of Weschler Intelligence Scale for Children version IV (WISC-IV), FAS (for verbal fluency), Boston Naming Test (BNT, for visual naming ability) and NEPSY-II (Developmental NEuroPSYchological Assessment) for verbal memory. Results: Fifteen children, 9 boys and 6 girls, were evaluated and 11 underwent surgery. The Full Scale IQ subscore (FSIQ) improved from M = 84.8 to M = 93.0 (p = 0.005). The preoperative Verbal Comprehension Index (VCI) was in the low average range (M = 86.7), improving to a level within the average range (M = 94.7, p = 0.001). Preoperative Perceptual Speed Index (PSI) was in the below average range (M = 81.5), improving to a level within the average range (M = 92.5, p = 0.004). Conclusion: ACs are a common finding in a pediatric neurosurgical setting. Our data suggest that some temporal AC have a negative effect on general cognitive ability and that this impairment can be improved by surgery. We suggest a standardized evaluation, including comprehensive and validated neuropsychological assessment tools, to thoroughly assess symptoms as well as the postoperative outcome.
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- Tedroff, K., et al.
(författare)
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Melatonin usage in children and young adults, a registry-based cohort study
- 2022
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Ingår i: European journal of paediatric neurology. - : Elsevier. - 1090-3798 .- 1532-2130. ; 39, s. 30-34
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Tidskriftsartikel (refereegranskat)abstract
- Sleep disorder is common in children and adolescents, particularly in those with attention deficit hyperactivity disorder (ADHD) or autism spectrum disorder (ASD). While non-pharmacological treatment is first line, occasionally an add-on of an oral drug is needed. The endogenous hormone melatonin is increasingly used for sleep disorders in children and adolescents. In this registry-based cohort study we follow dispensation of melatonin in young individuals, 0-25 years of age, in Stockholm, Sweden during 2016-2019. In all 9980 individuals, were dispensed melatonin in 2016 and followed for 3 years. Child psychiatrist was the most common prescribing specialty, 55% of all prescriptions. Only 20% had a recorded diagnosis of sleep disorder. The majority, 65% had a neuro psychiatric diagnose. Half of the individuals had at least 4 prescribed drugs dispensed during the follow-up. Almost half of our cohort were dispensed melatonin during the entire study period and doses and volumes of drug dispensed increased by 50 and 100%, respectively. Continuous medication was most common among children 6-12 years, where 7 out of 10 individuals were still adherent after three years. As long-term safety data is lacking, we find this concerning, and this illustrates the need of long-term follow-up of melatonin use in children and young individuals.
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- Tedroff, K., et al.
(författare)
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What has feet to do with it? Pes planus and medial arch height in adults with and without autism spectrum disorder.
- 2013
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Ingår i: Research in Autism Spectrum Disorders. - Oxford, United Kingdom : Elsevier. - 1750-9467 .- 1878-0237. ; 7:1, s. 187-192
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Tidskriftsartikel (refereegranskat)abstract
- Individuals with autism have higher rates of minor physical anomalies (MPAs) than neurotypical persons. Minor physical anomalies are slight morphological deviations typically harmless and without cosmetic or medical importance to the individual but indicative of an underlying neurodevelopmental disorder. In genetic autism research the utilization of MPAs has been recommended. In the present study the prevalence of pes planus or flatfoot in adults with ASD compared to age and sex matched neurotypical adults was investigated with two different methods. A photograph of the feet was obtained from underneath while the subjects were standing on a glass table. From this imprint calculations were made. In addition the medial longitudinal foot arch was measured with a Verniper caliper. The ultimate purpose of this study was to evaluate if flatfoot deformity could be utilized as an MPA in ASD. In this cohort of nearly one hundred adults no one fulfilled the predefined criteria of a flatfoot diagnosis but a lower foot arch height was shown in individuals with ASD compared to controls, confirming the clinical impression.
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