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Background Gorham-Stout disease (GSD) is a rare and idiopathic bone disorder, characterized by massive osteolysis. To date, there is no established treatment strategy for GSD. We empirically treated two patients, who had presented to us with cranial lesions of GSD. Here, we propose a novel algorithm for the management of Gorham's disease based on our experience and review the literature published to date.Methods We reviewed all existing literature on GSD describing the pathophysiology and suggested treatment methods, up to 2018.Results We found 13 papers with 14 reported cases; an inclusion of our two cases brings the total count up to just 16 recorded cases of GSD involving the skull. Of these, the base of the skull was affected in eight cases, while the remaining eight cases showed cranial involvement. The patients with skull-base involvement were managed conservatively, using medications or radiotherapy. The patients with cranial osteolysis were managed surgically, with an excision of the osteolytic portion, followed by cranioplasty. Of the latter group, the pericranium was not removed in one patient, in whom a very slight progression of the osteolytic process was later observed.Conclusions The pathogenesis of GSD remains poorly understood. Further study is required to determine an optimum management strategy. A long-term follow-up will also be necessary to establish the effectiveness of the treatment process. The untreated patients show a progressive resorption of the affected bones of the skull. A painful, vanishing skull deformity is an alarming sign of GSD. Early diagnosis and treatment are necessary to arrest disease progression and to prevent complications.Purpose The aim of this systematic review was to investigate if sex moderated the effect of exercise on cognition in adults post-stroke.Methods A systematic review was conducted of randomized controlled trials that involved adults ≥18 years with stroke, any exercise intervention, and reported any outcome related to cognitive function. We compared effect sizes of cognitive outcomes between studies of lower and higher proportion of females (CRD42018092757).Results The effects of exercise did not differ between studies of higher and lower female proportions with respect to memory (χ2 =1.52, p = 0.22), executive function (χ2 = 0.56, p = 0.45; Chi2 = 0.00, p = 0.98), language (Chi2 = 3.17, p = 0.08) or global cognition (χ2 = 0.88, p = 0.35).Conclusion There were no sex differences in the effects of exercise on memory, executive functioning, language or global cognition in individuals with stroke. Methyl-β-cyclodextrin order Further research is warranted to address sex differences in individuals with stroke to enable better targeting, prevention, and interventions in stroke rehabilitation.IMPLICATIONS FOR REHABILITATIONUnderstanding sex differences and potentially similarities in the relationship between exercise and cognition is an important step in enhancing stroke rehabilitation and the development of optimal, sex-specific rehabilitation.Although our findings suggest that there is no clear rationale for incorporating sex into our clinical decision making, it is still imperative to consider sex factors in research and report results in the literature disaggregated by sex to help inform clinical practice.The UPPS-P measures impulsivity as a five-factor construct (lack of premeditation, lack of perseverance, positive urgency, negative urgency and sensation seeking). Drawing on a number of theoretical considerations and alternative conceptions of impulsivity, the current study used confirmatory factor analysis (N = 1635) and multiple regression to evaluate and test alternative models comprising three, five, and a hierarchical model containing latent factors. The five factor and hierarchical models were shown to be valid and of near identical fit, whereas the three-factor model fit the data poorly. The current findings suggest that both the five factor and hierarchical models are useful applications of the UPPS-P. Depending on the purpose of future research, both models demonstrate utility in both risk assessment and treatment development. Multiple regression analysis revealed that positive urgency predicted problem gambling, which supports the predictive utility of impulsivity as a five-factor construct. While the latent factors of the hierarchical model are consistent with emerging theory, those using the UPPS-P should not overlook the unique contributions of the five factors. As the current study found meaningful predictive distinctions between positive and negative urgency, utilizing all five factors may increase measurement sensitivity and predictive utility.Purpose In current radiotherapy (RT) planning and delivery, population-based dose-volume constraints are used to limit the risk of toxicity from incidental irradiation of organs at risks (OARs). However, weighing tradeoffs between target coverage and doses to OARs (or prioritizing different OARs) in a quantitative way for each patient is challenging. We introduce a novel RT planning approach for patients with mediastinal Hodgkin lymphoma (HL) that aims to maximize overall outcome for each patient by optimizing on tumor control and mortality from late effects simultaneously.Material and Methods We retrospectively analyzed 34 HL patients treated with conformal RT (3DCRT). We used published data to model recurrence and radiation-induced mortality from coronary heart disease and secondary lung and breast cancers. Patient-specific doses to the heart, lung, breast, and target were incorporated in the models as well as age, sex, and cardiac risk factors (CRFs). A preliminary plan of candidate beams was created for each patient in a commercial treatment planning system. From these candidate beams, outcome-optimized (O-OPT) plans for each patient were created with an in-house optimization code that minimized the individual risk of recurrence and mortality from late effects. O-OPT plans were compared to VMAT plans and clinical 3DCRT plans.Results O-OPT plans generally had the lowest risk, followed by the clinical 3DCRT plans, then the VMAT plans with the highest risk with median (maximum) total risk values of 4.9 (11.1), 5.1 (17.7), and 7.6 (20.3)%, respectively (no CRFs). Compared to clinical 3DCRT plans, O-OPT planning reduced the total risk by at least 1% for 9/34 cases assuming no CRFs and 11/34 cases assuming presence of CRFs.Conclusions We developed an individualized, outcome-optimized planning technique for HL. Some of the resulting plans were substantially different from clinical plans. The results varied depending on how risk models were defined or prioritized.
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