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1.
J Intellect Disabil Res ; 66(8-9): 717-725, 2022 08.
Article in English | MEDLINE | ID: mdl-35713265

ABSTRACT

BACKGROUND: Prader-Willi syndrome (PWS) and Angelman syndrome (AS) are neurodevelopmental disorders in need of innovative 'real-world' outcome measures to evaluate treatment effects. Instrumented gait analysis (IGA) using wearable technology offers a potentially feasible solution to measure "real-world' neurological and motor dysfunction in these groups. METHODS: Children (50% female; 6-16 years) diagnosed with PWS (n = 9) and AS (n = 5) completed 'real-world' IGA assessments using the Physilog®5 wearable. PWS participants completed a laboratory assessment and a 'real-world' long walk. The AS group completed 'real-world' caregiver-assisted assessments. Mean and variability results for stride time, cadence, stance percentage (%) and stride length were extracted and compared across three different data reduction protocols. RESULTS: The wearables approach was found to be feasible, with all participants able to complete at least one assessment. This study also demonstrated significant agreement, using Lin's concordance correlation coefficient (CCC), between laboratory and 'real-world' assessments in the PWS group for mean stride length, mean stance % and stance % CV (n = 7, CCC: 0.782-0.847, P = 0.011-0.009). CONCLUSION: 'Real-world' gait analysis using the Physilog®5 wearable was feasible to efficiently assess neurological and motor dysfunction in children affected with PWS and AS.


Subject(s)
Angelman Syndrome , Prader-Willi Syndrome , Wearable Electronic Devices , Angelman Syndrome/complications , Angelman Syndrome/diagnosis , Angelman Syndrome/therapy , Child , Feasibility Studies , Female , Gait Analysis , Humans , Immunoglobulin A , Male
2.
J Natl Cancer Inst ; 56(3): 493-7, 1976 Mar.
Article in English | MEDLINE | ID: mdl-176394

ABSTRACT

Six-week-old male Swiss mice were given 0.03% thioacetamide (TAA) in the diet 24, 72, and 168 hours after partial hepatectomy. TAA-treated mice from all three groups were killed when they were 4, 9, and 13 months old. Intact and partially hepatectomized animals on normal diets served as controls. None of the controls evidenced neoplasms at any age. All three experimental groups developed liver tumors earlier than did intact mice treated with the TAA diet. Progressive metabolic studies on the livers or tumor tissues of treated mice showed that the levels of glucose-6-phosphatase, fructose-1,6-diphosphatase, and glycogen decreased significantly in the 4-month-old treated group when there was no significant alteration in liver histology. These parameters were lowest in the tumor tissues of treated mice.


Subject(s)
Acetamides/toxicity , Carcinoma, Hepatocellular/chemically induced , Hepatectomy , Liver Neoplasms/chemically induced , Thioacetamide/toxicity , Age Factors , Animals , Carcinoma, Hepatocellular/metabolism , Carcinoma, Hepatocellular/pathology , Diet , Fructose-Bisphosphatase/metabolism , Liver/pathology , Liver Glycogen/metabolism , Liver Neoplasms/metabolism , Liver Neoplasms/pathology , Male , Mice , Neoplasms, Experimental/chemically induced , Neoplasms, Experimental/pathology , Precancerous Conditions/pathology
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