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  • 1
    Online Resource
    Online Resource
    Trajectories and Risk Factors for Altered Physical and Psychosocial Health-Related Quality of Life After Pediatric Community-Acquired Septic Shock*
    Ovid Technologies (Wolters Kluwer Health) ; 2020
    In:  Pediatric Critical Care Medicine Vol. 21, No. 10 ( 2020-10), p. 869-878
    Details
    In: Pediatric Critical Care Medicine, Ovid Technologies (Wolters Kluwer Health), Vol. 21, No. 10 ( 2020-10), p. 869-878
    Abstract: To evaluate the physical and psychosocial domains of health-related quality of life among children during the first year following community-acquired septic shock, and explore factors associated with poor physical and psychosocial health-related quality of life outcomes. Design: Secondary analysis of the Life After Pediatric Sepsis Evaluation. Setting: Twelve academic PICUs in the United States. Patients: Children greater than or equal to 1 month and less than 18 years old who were perceived to be without severe developmental disability by their family caregiver at baseline and who survived hospitalization for community-acquired septic shock. Interventions: Family caregivers completed the Pediatric Quality of Life Inventory for children 2–18 years old or the Pediatric Quality of Life Inventory Infant Scales for children less than 2 years old at baseline (reflecting preadmission status), day 7, and months 1, 3, 6, and 12 following PICU admission. Higher Pediatric Quality of Life Inventory Physical and Psychosocial Health Summary Scores indicate better health-related quality of life. Measurements and Main Results: Of 204 children, 58 (28.2%) had a complex chronic comorbid condition. Children with complex chronic comorbid conditions had lower baseline physical health-related quality of life (62.7 ± 22.6 vs 84.1 ± 19.7; p 〈 0.001) and psychosocial health-related quality of life (68.4 ± 14.1 vs 81.2 ± 15.3; p 〈 0.001) than reference norms, whereas children without such conditions had baseline scores similar to reference norms. Children with complex chronic comorbid conditions recovered to their baseline health-related quality of life, whereas children without such conditions did not (physical health-related quality of life 75.3 ± 23.7 vs 83.2 ± 20.1; p = 0.008 and psychosocial health-related quality of life 74.5 ± 18.7 vs 80.5 ± 17.9; p = 0.006). Age less than 2 years was independently associated with higher month 12 physical health-related quality of life, and abnormal neurologic examination and neurologic injury suspected by a healthcare provider during the PICU course were independently associated with lower month 12 physical health-related quality of life. Treatment of increased intracranial pressure and medical device use at month 1 were independently associated with lower month 12 psychosocial health-related quality of life. Conclusions: Physical and psychosocial health-related quality of life were reduced among children during the first year following community-acquired septic shock compared with reference norms, although many recovered to baseline. Risk factors for poor health-related quality of life included neurologic complications during the hospitalization and dependence on a medical device 1 month postadmission.
    Type of Medium: Online Resource
    ISSN: 1529-7535
    URL: Article
    DOI: 10.1097/PCC.0000000000002374
    Language: English
    Publisher: Ovid Technologies (Wolters Kluwer Health)
    Publication Date: 2020
    detail.hit.zdb_id: 2070997-3
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  • 2
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    Intellectual enrichment and genetic modifiers of cognition and brain volume in Huntington’s disease
    Oxford University Press (OUP) ; 2022
    In:  Brain Communications Vol. 4, No. 6 ( 2022-11-02)
    Details
    In: Brain Communications, Oxford University Press (OUP), Vol. 4, No. 6 ( 2022-11-02)
    Abstract: An important step towards the development of treatments for cognitive impairment in ageing and neurodegenerative diseases is to identify genetic and environmental modifiers of cognitive function and understand the mechanism by which they exert an effect. In Huntington’s disease, the most common autosomal dominant dementia, a small number of studies have identified intellectual enrichment, i.e. a cognitively stimulating lifestyle and genetic polymorphisms as potential modifiers of cognitive function. The aim of our study was to further investigate the relationship and interaction between genetic factors and intellectual enrichment on cognitive function and brain atrophy in Huntington’s disease. For this purpose, we analysed data from Track-HD, a multi-centre longitudinal study in Huntington’s disease gene carriers and focused on the role of intellectual enrichment (estimated at baseline) and the genes FAN1, MSH3, BDNF, COMT and MAPT in predicting cognitive decline and brain atrophy. We found that carrying the 3a allele in the MSH3 gene had a positive effect on global cognitive function and brain atrophy in multiple cortical regions, such that 3a allele carriers had a slower rate of cognitive decline and atrophy compared with non-carriers, in agreement with its role in somatic instability. No other genetic predictor had a significant effect on cognitive function and the effect of MSH3 was independent of intellectual enrichment. Intellectual enrichment also had a positive effect on cognitive function; participants with higher intellectual enrichment, i.e. those who were better educated, had higher verbal intelligence and performed an occupation that was intellectually engaging, had better cognitive function overall, in agreement with previous studies in Huntington’s disease and other dementias. We also found that intellectual enrichment interacted with the BDNF gene, such that the positive effect of intellectual enrichment was greater in Met66 allele carriers than non-carriers. A similar relationship was also identified for changes in whole brain and caudate volume; the positive effect of intellectual enrichment was greater for Met66 allele carriers, rather than for non-carriers. In summary, our study provides additional evidence for the beneficial role of intellectual enrichment and carrying the 3a allele in MSH3 in cognitive function in Huntington’s disease and their effect on brain structure.
    Type of Medium: Online Resource
    ISSN: 2632-1297
    URL: Article
    DOI: 10.1093/braincomms/fcac279
    Language: English
    Publisher: Oxford University Press (OUP)
    Publication Date: 2022
    detail.hit.zdb_id: 3020013-1
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  • 3
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    Genotype correlates with clinical severity in PIK3CA-associated lymphatic malformations
    American Society for Clinical Investigation ; 2019
    In:  JCI Insight Vol. 4, No. 21 ( 2019-11-1)
    Details
    In: JCI Insight, American Society for Clinical Investigation, Vol. 4, No. 21 ( 2019-11-1)
    Type of Medium: Online Resource
    ISSN: 2379-3708
    URL: Article
    URL: Article
    URL: Article
    DOI: 10.1172/jci.insight.129884
    DOI: 10.1172/jci.insight.129884DS1
    DOI: 10.1172/jci.insight.129884DS2
    Language: English
    Publisher: American Society for Clinical Investigation
    Publication Date: 2019
    detail.hit.zdb_id: 2874757-4
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  • 4
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    Active Observation as an Alternative to Invasive Treatments for Pediatric Head and Neck Lymphatic Malformations
    Wiley ; 2021
    In:  The Laryngoscope Vol. 131, No. 6 ( 2021-06), p. 1392-1397
    Details
    In: The Laryngoscope, Wiley, Vol. 131, No. 6 ( 2021-06), p. 1392-1397
    Abstract: An increasing number of treatment modalities for lymphatic malformations are being described, complicating therapeutic decisions. Understanding lymphatic malformation natural history is essential. We describe management of head and neck lymphatic malformations where decisions primarily addressed lesion‐induced functional compromise (ie, breathing, swallowing) to identify factors associated with invasive treatment and active observation. We hypothesize that non‐function threatening malformations can be observed. Study Design Retrospective case series. Methods Retrospective case series of consecutive head and neck lymphatic malformation patients (2000–2017) with over 2 years of follow‐up. Patient characteristics were summarized and associations with invasive treatment (surgery or sclerotherapy) tested using Fisher's exact. In observed patients, factors associated with spontaneous regression were assessed with Fisher's exact test. Results Of 191 patients, 101 (53%) were male, 97 (51%) Caucasian, and 98 (51.3%) younger than 3 months. Malformations were de Serres I–III 167 (87%), or IV–V 24 (12%), and commonly located in the neck (101, 53%), or oral cavity (36, 19%). Initial treatments included observation (65, 34%) or invasive treatments such as primary surgery (80, 42%), staged surgery (25, 13%), or primary sclerotherapy (9, 5%). Of 65 initially observed malformations, 8 (12%) subsequently had invasive treatment, 36 (58%) had spontaneous regression, and 21 (32%) elected for no invasive therapy. Spontaneous regression was associated with location in the lateral neck ( P = .003) and macrocystic malformations ( P = .017). Conclusion Head and neck lymphatic malformation treatment selection can be individualized after stratifying by stage, presence of functional compromise, and consideration of natural history. Recognizing the spectrum of severity is essential in evaluating efficacy of emerging treatments, as selected malformations may respond to observation. Level of Evidence 4 Laryngoscope , 131:1392–1397, 2021
    Type of Medium: Online Resource
    ISSN: 0023-852X , 1531-4995
    URL: Issue
    URL: Article
    DOI: 10.1002/lary.v131.6
    DOI: 10.1002/lary.29180
    Language: English
    Publisher: Wiley
    Publication Date: 2021
    detail.hit.zdb_id: 2026089-1
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  • 5
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    Delaying Invasive Treatment in Unilateral Head and Neck Lymphatic Malformation Improves Outcomes
    Wiley ; 2023
    In:  The Laryngoscope Vol. 133, No. 4 ( 2023-04), p. 956-962
    Details
    In: The Laryngoscope, Wiley, Vol. 133, No. 4 ( 2023-04), p. 956-962
    Abstract: Large (De Serres stage [IV–V]) head and neck lymphatic malformations (HNLMs) often have multiple, high‐risk, invasive treatments (ITs) to address functional compromise. Logically reducing HNLM ITs should reduce treatment risk. We tested whether delaying HNLM ITs reduces total IT number. Materials Consecutive HNLM patients ( n  = 199) between 2010 and 2017, aged 0–18 years. Methods ITs (surgery or sclerotherapy) were offered for persistent or dysfunction causing HNLMs. Treatment effectiveness categorized by IT number: optimal (0–1), acceptable (2–5), or suboptimal ( 〉 5). Clinical data were summarized, and outcome associations tested ( χ 2 ). Relative risk (RR) with a Poisson working model tested whether HNLM observation or IT delay ( 〉 6 months post‐diagnosis) predicts treatment success (i.e., ≤1 IT). Results Median age at HNLM diagnosis was 1.3 months (interquartile range [IQR] 0–45 m) with 107/199(54%) male. HNLM were stage I–III (174 [88%] ), IV–V (25 [13%]). Initial treatment was observation (70 [35%] ), invasive (129 [65%]). Treatment outcomes were optimal (137 [69%] ), acceptable (36 [18%]), and suboptimal (26 [13%] ). Suboptimal outcome associations: EXIT procedure, stage IV–V, oral location, and tracheotomy ( p   〈  0.001). Stage I–III HNLMs were initially observed compared with stage I–III having ITs within 6 months of HNLM diagnosis, had a 82% lower relative treatment failure risk ([i.e., 〉 1 IT], RR = 0.09, 95% CI 0.02–0.36, p   〈  0.001). Stage I–III HNLMs with non‐delayed ITs had reduced treatment failure risk compared with IV–V (RR = 0.47, 95% CI 0.33–0.66, p   〈  0.001). Conclusion Observation and delayed IT in stage I–III HNLM (“Grade 1”) is safe and reduces IT (i.e., ≤1 IT). Stage IV–V HNLMs (“Grade 2”) with early IT have a greater risk of multiple ITs. Level of Evidence 4 Laryngoscope , 133:956–962, 2023
    Type of Medium: Online Resource
    ISSN: 0023-852X , 1531-4995
    URL: Issue
    URL: Article
    DOI: 10.1002/lary.v133.4
    DOI: 10.1002/lary.30237
    Language: English
    Publisher: Wiley
    Publication Date: 2023
    detail.hit.zdb_id: 2026089-1
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  • 6
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    Revision Surgeries Are Associated With Significant Increased Risk of Subsequent Cerebrospinal Fluid Shunt Infection
    Ovid Technologies (Wolters Kluwer Health) ; 2012
    In:  Pediatric Infectious Disease Journal Vol. 31, No. 6 ( 2012-06), p. 551-556
    Details
    In: Pediatric Infectious Disease Journal, Ovid Technologies (Wolters Kluwer Health), Vol. 31, No. 6 ( 2012-06), p. 551-556
    Type of Medium: Online Resource
    ISSN: 0891-3668
    URL: Article
    DOI: 10.1097/INF.0b013e31824da5bd
    Language: English
    Publisher: Ovid Technologies (Wolters Kluwer Health)
    Publication Date: 2012
    detail.hit.zdb_id: 2020216-7
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  • 7
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    Piloting a mobile health intervention to increase physical activity for adolescents with ADHD
    Elsevier BV ; 2017
    In:  Preventive Medicine Reports Vol. 6 ( 2017-06), p. 210-213
    Details
    In: Preventive Medicine Reports, Elsevier BV, Vol. 6 ( 2017-06), p. 210-213
    Type of Medium: Online Resource
    ISSN: 2211-3355
    URL: Article
    DOI: 10.1016/j.pmedr.2017.03.003
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2017
    detail.hit.zdb_id: 2785569-7
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  • 8
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    Health Care Reform, Length of Stay, and Readmissions for Child Mental Health Hospitalizations
    American Academy of Pediatrics (AAP) ; 2020
    In:  Hospital Pediatrics Vol. 10, No. 3 ( 2020-03-01), p. 238-245
    Details
    In: Hospital Pediatrics, American Academy of Pediatrics (AAP), Vol. 10, No. 3 ( 2020-03-01), p. 238-245
    Abstract: Health care reform may impact inpatient mental health services by increasing access and changing insurer incentives. We examined whether implementation of the 2014 Affordable Care Act (ACA) was associated with changes in psychiatric length of stay (LOS) and 30-day readmissions for pediatric patients. METHODS: We conducted an interrupted time-series analysis to evaluate LOS and 30-day readmissions during the 30 months before and 24 months after ACA implementation, with a 6-month wash-out period, on patients aged 4 to 17 years who were discharged from the psychiatry unit of a children’s hospital. Differences by payer (Medicaid versus non-Medicaid) were examined in moderated interrupted time series. Logistic regression was used to examine the association between psychiatric LOS and 30-day readmissions. RESULTS: There were 1874 encounters in the pre-ACA period and 2186 encounters in the post-ACA period. Compared with pre-ACA implementation, post-ACA implementation was associated with LOS that was significantly decreasing over time (pre-ACA versus post-ACA slope difference: −0.10 days per encounter per month [95% confidence interval −0.17 to −0.02]; P = .01), especially for Medicaid-insured patients (pre-ACA versus post-ACA slope difference: −0.14 days per encounter per month [95% confidence interval −0.26 to −0.01] ; P = .03). The overall proportion of 30-day readmissions increased significantly (pre-ACA 6%, post-ACA 10%; P & lt; .05 for the difference). We found no association between LOS and 30-day readmissions. CONCLUSIONS: ACA implementation was associated with a decline in psychiatric inpatient LOS over time, especially for those on Medicaid, and an increase in 30-day readmissions. LOS was not associated with 30-day inpatient readmissions. Further investigation to understand the drivers of these patterns is warranted.
    Type of Medium: Online Resource
    ISSN: 2154-1663 , 2154-1671
    URL: Article
    DOI: 10.1542/hpeds.2019-0197
    Language: English
    Publisher: American Academy of Pediatrics (AAP)
    Publication Date: 2020
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  • 9
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    Physical Activity, Screen Time, and Sleep in Children With ADHD
    Human Kinetics ; 2019
    In:  Journal of Physical Activity and Health Vol. 16, No. 6 ( 2019-06), p. 416-422
    Details
    In: Journal of Physical Activity and Health, Human Kinetics, Vol. 16, No. 6 ( 2019-06), p. 416-422
    Type of Medium: Online Resource
    ISSN: 1543-3080 , 1543-5474
    URL: Article
    DOI: 10.1123/jpah.2018-0215
    Language: English
    Publisher: Human Kinetics
    Publication Date: 2019
    SSG: 31
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  • 10
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    MSH3 modifies somatic instability and disease severity in Huntington’s and myotonic dystrophy type 1
    Oxford University Press (OUP) ; 2019
    In:  Brain Vol. 142, No. 7 ( 2019-07-01), p. 1876-1886
    Details
    In: Brain, Oxford University Press (OUP), Vol. 142, No. 7 ( 2019-07-01), p. 1876-1886
    Abstract: The mismatch repair gene MSH3 has been implicated as a genetic modifier of the CAG·CTG repeat expansion disorders Huntington’s disease and myotonic dystrophy type 1. A recent Huntington’s disease genome-wide association study found rs557874766, an imputed single nucleotide polymorphism located within a polymorphic 9 bp tandem repeat in MSH3/DHFR, as the variant most significantly associated with progression in Huntington’s disease. Using Illumina sequencing in Huntington’s disease and myotonic dystrophy type 1 subjects, we show that rs557874766 is an alignment artefact, the minor allele for which corresponds to a three-repeat allele in MSH3 exon 1 that is associated with a reduced rate of somatic CAG·CTG expansion (P = 0.004) and delayed disease onset (P = 0.003) in both Huntington’s disease and myotonic dystrophy type 1, and slower progression (P = 3.86 × 10−7) in Huntington’s disease. RNA-Seq of whole blood in the Huntington’s disease subjects found that repeat variants are associated with MSH3 and DHFR expression. A transcriptome-wide association study in the Huntington’s disease cohort found increased MSH3 and DHFR expression are associated with disease progression. These results suggest that variation in the MSH3 exon 1 repeat region influences somatic expansion and disease phenotype in Huntington’s disease and myotonic dystrophy type 1, and suggests a common DNA repair mechanism operates in both repeat expansion diseases.
    Type of Medium: Online Resource
    ISSN: 0006-8950 , 1460-2156
    URL: Article
    DOI: 10.1093/brain/awz115
    RVK:
    XA 10000
    Language: English
    Publisher: Oxford University Press (OUP)
    Publication Date: 2019
    detail.hit.zdb_id: 1474117-9
    SSG: 12
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