ABSTRACT
BACKGROUND: There were controversial findings in terms of the association between the incidence of Benign Paroxysmal Positional Vertigo (BPPV) and climate changes, so the current systematic review plus meta-analysis is designed to discover this possible relationship. METHODS: Web of science, PubMed, Scopus, Google Scholar, Embase, and Cochrane library were systematically searched up to August 2023. The Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) and Problem/Population, Intervention, Comparison, and Outcome (PICO) guidelines were used. Two authors independently reviewed the eligible articles and assessed the quality of them. RESULTS: In total, 15 studies including 16144 patients met the inclusion criteria. Ten studies reported the relation of BPPV to monthly mean temperature, 7 to monthly average humidity, 4 to monthly average rainfall, 6 to monthly sunlight time, and 2 to average solar radiation. The incidence of BPPV was associated significantly with atmospheric pressure (P: 0.003) and rainfall (P: 0.017). However, there was not any statistically significant correlation between incidence of BPPV and humidity, sunlight time, temperature, and solar radiation level (P > 0.05). CONCLUSIONS: The incidence of BPPV was higher in cold months of a year in both northern hemisphere and southern hemisphere countries. Although it can be because of negative correlation with temperature, the current meta-analysis did not find any statistically significant negative correlation with temperature. In addition, the incidence of BPPV was associated significantly with atmospheric pressure (positive correlation) and rainfall (negative correlation).
ABSTRACT
Background: A salutary effect of treatments for Gaucher disease (GD) has been a reduction in the incidence of avascular osteonecrosis (AVN). However, there are reports of AVN in patients receiving enzyme replacement therapy (ERT) , and it is not known whether it is related to individual treatments, GBA genotypes, phenotypes, biomarkers of residual disease activity, or anti-drug antibodies. Prompted by development of AVN in several patients receiving ERT, we aimed to delineate the determinants of AVN in patients receiving ERT or eliglustat substrate reduction therapy (SRT) during 20 years in a tertiary referral center. Methods: Longitudinal follow-ups of 155 GD patients between 2001 and 2021 were analyzed for episodes of AVN on therapy, type of therapy, GBA1 genotype, spleen status, biomarkers, and other disease indicators. We applied mixed-effects logistic model to delineate the independent correlates of AVN while receiving treatment. Results: The patients received cumulative 1382 years of treatment. There were 16 episodes of AVN in 14 patients, with two episodes, each occurring in two patients. Heteroallelic p.Asn409Ser GD1 patients were 10 times (95% CI, 1.5-67.2) more likely than p.Asn409Ser homozygous patients to develop osteonecrosis during treatment. History of AVN prior to treatment initiation was associated with 4.8-fold increased risk of AVN on treatment (95% CI, 1.5-15.2). The risk of AVN among patients receiving velaglucerase ERT was 4.68 times higher compared to patients receiving imiglucerase ERT (95% CI, 1.67-13). No patient receiving eliglustat SRT suffered AVN. There was a significant correlation between GlcSph levels and AVN. Together, these biomarkers reliably predicted risk of AVN during therapy (ROC AUC 0.894, p<0.001). Conclusions: There is a low, but significant risk of AVN in GD in the era of ERT/SRT. We found that increased risk of AVN was related to GBA genotype, history of AVN prior to treatment initiation, residual serum GlcSph level, and the type of ERT. No patient receiving SRT developed AVN. These findings exemplify a new approach to biomarker applications in a rare inborn error of metabolism to evaluate clinical outcomes in comprehensively followed patients and will aid identification of GD patients at higher risk of AVN who will benefit from closer monitoring and treatment optimization. Funding: LSD Training Fellowship from Sanofi to MB.
