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3 Progress in Health Sciences

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Effects of granulocyte colony-stimulating factor therapy for osteogenesis imperfecta: a case report

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Paszko-Patej G. , Sienkiewicz D. , Kułak W. , Okurowska-Zawada B. , Wojtkowski J. , Kalinowska A. , Okulczyk K. , Sochoń K. , Dmitruk E. , Mirska A.

2017

tom 7(1)

205-208

Introduction: Osteogenesis imperfecta (OI) is a genetic disorder of increased bone fragility and low bone mass. OI type IV. Materials and methods: We examined the safety and effectiveness of a low dose of analog granulocyte colony-stimulating factor (G-CSF) in a 15-year-old girl OI type IV. G-CSF 5 μg/kg was given subcutaneously, for 5 days/month for 3, 6 and 12 months. Laboratory tests, including blood, biochemical tests were performed, in addition to clinical examination.Results: Clinical examination revealed an increase of muscle strength in the upper and lower limbs between base line and day 6 and 12 months. We found no serious adverse events. Leukocyte levels remained below 38,000/μL. Low dose G-CSF was safe and well tolerated by the patient. Conclusions: A significant increase in muscle strength in this patient may indicate beneficial effects of G-CSF factor in this disorder. These results are inspiring and warrant further studies.

Quality of life of children suffering from motor disabilities as evaluated by their parents

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Wojtkowski J. , Sienkiewicz D. , Okurowska-Zawada B. , Paszko-Patej G. , Konopka A. , Okulczyk K. , Średzińska K. , Dmitruk E. , Mirska A. , Kułak W.

tom 7(2)

60-66

Purpose: We assessed the quality of life of children with motor disabilities in comparison with healthy children, as evaluated by their parents, using the CHQ-PF28 questionnaire (Child Health Questionnaire—Parent Form). Materials and methods: In a prospective study, we evaluated the quality of life of 105 children with motor disabilities. Results: Our research showed lower quality of life in the group of children with motor disabilities compared with controls, both in terms of physical and psychosocial health. Significant correlations between independent walking and physical functioning, general behavior, and mental health of children suffering from motor disabilities were found. According to the average indices of quality of life of children suffering from motor disabilities, depending on sex, the greatest differences occurred in behavior and change of health status, while the smallest differences in self-esteem and parental involvement, compared with controls. In the case of healthy children, the largest differences appeared in the perception of pain, behavior, and self-esteem; whereas, the smallest variations occurred in the change of health status and physical activity. Conclusion: Children suffering from motor disabilities demonstrate lower quality of life compared with healthy children.

Granulocyte colony-stimulating factor therapy for facioscapulohumeral dystrophy: a case report

80%

Sienkiewicz D. , Kułak W. , Okurowska-Zawada B. , Paszko-Patej G. , Wojtkowski J. , Dmitruk E. , Okulczyk K. , Sochoń K. , Kalinowska A. , Żak J. , Pogorzelski R.

2016

tom 6(2)

175-177

We examined the safety and effectiveness of a low dose of analog granulocyte-colony stimulating factor in a 15-year-old boy with facioscapulohumeral dystrophy. The onset of disease was noted at 12 years of age. The physical examination noted general muscle atrophy more pronounced at left side of the body. He was able to walk 300 meters within 6 minute walk test. Granulocyte colony-stimulating factor 5 μg/kg was given subcutaneously daily for 5 days/month for 1, 2, 3, 6 and 12 months. Clinical examination, laboratory tests including blood, biochemical tests, and CD34+ cells were performed. A significant increase of muscle strength in the lower and upper limbs between baseline, and after 3 months of treatment, after 6, and after 12 months was found. He was able to walk 480 meters within 6 minutes after 12 months. Electromyography demonstrated increase of amplitude in the examined in upper and lower limbs after six months compared to baseline. Leukocyte levels remained below 25000/μL. CD34+ increased significantly at day 5 of granulocyte colony-stimulating factor admini-stration. It was safe and well tolerated by the patient. A significant increase in muscle strength in this patient with facioscapulohumeral dystrophy after 3 months of treatment, after 6, and after 12 months since the first treatment course was completed may indicate beneficial effects of granulocyte colony-stimulating factor in this disorder.