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      Burosumab Therapy in Children with X-Linked Hypophosphatemia

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          Prevalence of headache and migraine in children and adolescents: a systematic review of population-based studies.

          the aim of this study was to review systematically the prevalence of headache and migraine in children and adolescents and to study the influence of sex, age, and region of residence on the epidemiology.  we systematically searched the literature in electronic databases to cover the period between 1 January 1990 and 31 December 2007. We assessed and included population-based studies on epidemiology of headache and migraine in children and adolescents if they fulfilled the following criteria: (1) reporting on unselected childhood population; (2) reliable methods of data collection using a questionnaire or face-to-face interviews; (3) using the International Headache Society's (IHS) criteria (1988 or 2004) for the diagnosis of migraine; and (4) provision of sufficient and explicit data for analysis. We used Excel, Stata, and Confidence Interval Analysis software.  we identified and analysed 50 population-based studies reporting the prevalence of headache and/or migraine in children and adolescents (<20y). The estimated prevalence of headache over periods between 1 month and lifetime in children and adolescents is 58.4% (95% confidence interval [CI] 58.1-58.8). Females are more likely to have headache than males (odds ratio [OR] 1.53, 95% CI 1.48-1.6). The prevalence of migraine over periods between 6 months and lifetime is 7.7% (95% CI 7.6-7.8). Females are more likely than males to have migraine (OR 1.67, 95% CI 1.60-1.75). Regional differences in prevalence of migraine, though statistically significant, may not be of clinical significance. The change in the IHS's criteria for the diagnosis of migraine was not associated with any significant change in the prevalence of migraine. this study confirms the global high prevalence of headache and migraine in children and adolescents. Sex, age, and regional differences are evident.
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            Six-minute walk test in children and adolescents.

            To evaluate the 6-minute walking distance (6MWD) for healthy Caucasian children and adolescents of a population-based sample from the age of 3 to 18 years. Two hundred and eighty boys and 248 girls completed a modified test, using a measuring wheel as incentive device. Median 6MWD increased from the age of 3 to 11 years in boys and girls alike and increased further with increasing age in boys (from 667.3 m to 727.6 m), whereas it essentially plateaued in girls (655.8 m to 660.9 m). After adjusting for age, height (P = .001 in boys and P < .001 in girls) remained independently correlated with the 6MWD. In the best fitting and most efficient linear and quadratic regression models, the variables age and height explained about 49% of the variability of the 6MWD in boys and 50% in girls. This modified 6-minute walk test (6MWT) proved to be safe, easy to perform, and highly acceptable to children. It provides a simple and inexpensive means to measure functional exercise capacity in children, even of young age, and might be of value when conducting comparable studies.
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              How fibroblast growth factor 23 works.

              There is a discontinuum of hereditary and acquired disorders of phosphate homeostasis that are caused by either high or low circulating levels of the novel phosphaturic hormone fibroblastic growth factor 23 (FGF23). Disorders that are caused by high circulating levels of FGF23 are characterized by hypophosphatemia, decreased production of 1,25-dihydroxyvitamin D, and rickets/osteomalacia. On the other end of the spectrum are disorders that are caused by low circulating levels of FGF23, which are characterized by hyperphosphatemia, elevated production of 1,25-dihydroxyvitamin D, soft tissue calcifications, and hyperostosis. Knowledge of the genetic basis of these hereditary disorders of phosphate homeostasis and studies of their mouse homologues have uncovered a bone-kidney axis and new systems biology that govern bone mineralization, vitamin D metabolism, parathyroid gland function, and renal phosphate handling. Further understanding of this primary phosphate homeostatic pathway has the potential to have a significant impact on the diagnosis and treatment of disorders of bone and mineral metabolism.
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                Author and article information

                Journal
                New England Journal of Medicine
                N Engl J Med
                New England Journal of Medicine (NEJM/MMS)
                0028-4793
                1533-4406
                May 24 2018
                May 24 2018
                : 378
                : 21
                : 1987-1998
                Affiliations
                [1 ]From Yale University School of Medicine, New Haven, CT (T.O.C.); Shriners Hospital for Children and Division of Bone and Mineral Diseases, Department of Internal Medicine, Washington University School of Medicine, St. Louis (M.P.W.); Indiana University School of Medicine, Indianapolis (E.A.I.); University of Groningen, Groningen, the Netherlands (A.M.B.); Birmingham Children’s Hospital, Birmingham (W. Högler), Royal Manchester Children’s Hospital, Manchester (R.P.), and Great Ormond Street Hospital,...
                Article
                10.1056/NEJMoa1714641
                29791829
                5f19411d-16e2-4202-a7bc-96c33079edfd
                © 2018
                History

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