Hemophilia is associated with a high financial burden on individuals, healthcare systems, and society. The development of inhibitors significantly increases the socioeconomic burden of the diseases. This study aimed to review and describe the burden of hemophilia with inhibitors, providing a reference scenario to assess the impact of new products in the real word. Two systematic literature reviews were performed to collect data on (i) health economics and (ii) health‐related quality of life evidences in hemophilic patients with inhibitors. The costs associated with patients with hemophilia and inhibitors are more than 3 times greater than the costs incurred in those without inhibitors, with an annual cost per patient that can be higher than €1 000 000. The costs of bypassing agents account for the large majority of the total healthcare direct costs for hemophilia treatment. The quality of life is more compromised in patients with hemophilia and inhibitors compared to those without inhibitors, in particular the physical domains, whereas mental domains were comparable to that of the general population. The development of an inhibitor has a high impact on costs and quality of life. New treatments have the potential to change positively the management and socioeconomic burden of hemophilia with inhibitors.
Objective: The role of gastroesophageal reflux (GER) causing distress in infants is controversial but acid inhibitors are often empirically prescribed. We evaluated the relation between distress assessed by the Face, Legs, Activity, Cry, Consolability (FLACC) scale and GER in infants. Methods: We analyzed multiple intraluminal impedance-pH (MII-pH) monitoring tracings of infants with persistent unexplained fussiness or distress. Symptoms occurring during investigation were scored by parents using the FLACC scale and were grouped as “distress” episodes. Results: We recruited 62 children (ages 15 days to 23 months, median age 3.5 months). During MII-pH, 452 episodes of distress were registered: 217 (48%) were temporally associated with GER and 235 (52%) were not, with no difference in the median value of FLACC between the 2 groups. Infants with abnormal acid exposure index had a significantly lower FLACC compared with the group with acid reflux index <7% (P < 0.001). When associated with symptoms, GER occurred significantly more often before than simultaneously or after an episode of distress (P = 0.001). Age, proximal extension, and duration of GER did not correlate with FLACC scores. Episodes of distress associated with nonacid reflux presented a significant higher FLACC compared with the ones with acid content (FLACC 6 vs 5, P = 0.011). In infants, episodes of distress do not significantly correlate with GER. Conclusions: No difference in infant distress was noted between proximal and distal GER. Non-acid reflux is perceived at least as painful than acid GER. Our results stress that acid inhibitors should not be started in infants presenting distress unless a clear association with acid GER is demonstrated.
Background The evaluation of participant experience is an essential part of monitoring the quality of breast cancer screening services. Satisfaction of services can lead to good adherence and hence affect health outcomes. Methods We performed a systematic review to assess how satisfied women were with organized breast cancer screening programs. A literature search in Medline, CINAHL, Embase and PsycINFO from 1965 to October 2019 was performed. Articles reporting a quantitative measure of satisfaction collected via questionnaires in programs using mammography as a screening test were selected. We narratively synthesized the data and used tabulated summaries. Results Out of 4310 individual citations, 3099 abstracts were reviewed by two independent researchers, and 126 articles were selected for full-text reading. Finally, 48 studies, published between 1990 and 2018, were included in analysis, reporting 54 surveys in the context of an organized screening program, 37 on satisfaction with screening mammography, 14 on satisfaction with further assessments and 3 with counseling. Most studies reported a high level of satisfaction for both mammography and further assessments. Despite commonly reported temporary pain, discomfort and anxiety, the willingness to be re-screened was very high. Effective information transfer, the staff's interpersonal skills and quick delivery of results correlated with high satisfaction. Only 7 out of 54 surveys used recognized satisfaction instruments or their modifications. Conclusions In general, satisfaction with breast cancer screening is high, but its evaluation is mainly performed using non-validated instruments. Emphasis should be put on effective communication, the staff´s interpersonal skills and quick delivery of results.
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