Abstract A 35‐year‐old male patient presented fluctuating bilateral lower extremity weakness for 3 years. Physical examination showed grade 4 proximal muscle weakness in both lower extremities and grade 5 distal muscle weakness. Laboratory data revealed elevated creatine kinase, triglycerides, and cholesterol. Muscle pathology showed deposition of lipid droplet under the sarcolemma. Bone densitometry indicated severe osteoporosis. Next‐generation sequencing revealed a pathogenic mutation in the ETFDH gene. The patient was diagnosed with late‐onset multiple acyl‐CoA dehydrogenase deficiency. After riboflavin treatment, symptoms of the patient were relieved, physical endurance was restored, and bone mineral density was improved.
Abstract Background The cancer risk in rheumatoid arthritis (RA) patients has been discussed. Hydroxychloroquine (HCQ) may exert protective effects against malignancy. The study investigated the association between HCQ use and the risk of subsequent malignancy in RA patients. Methods Catastrophic illness certificated RA patients were extracted from the National Health Insurance Research Database. The index date was set 180 days after the RA diagnosis date to avoid immortal time bias. Two groups were matched in a 1‐to‐1 ratio by propensity score regarding age, gender, index date, relevant comorbidities, and comedication. HCQ users prior to the diagnosis of RA were exempted to ensure compliance with the new‐user design. Cancers diagnosed before or less than 180 days after the index date were excluded to mitigate protopathic bias. The study adopted the Kaplan–Meier curve and Cox proportional hazards model to examine the association between HCQ use and cancer risk. The assumption of proportional hazard was also tested. Results Based on strict criteria, we included 492 eligible RA patients and divided them into study and control groups ( N = 246 in each group). HCQ users exhibited a neutral risk of cancer relative to the controls (adjusted hazard ratio, 0.99; 95% CI, 0.44–2.21, p > .05). The assumption of proportional hazard was not violated. Conclusion This study does not observe the effect of using HCQ as a primary regimen to prevent cancer in RA patients. We are assured that HCQ is not associated with an increased risk of subsequent malignancy in RA patients. Further mechanistic research is needed.
The long-term cardiovascular outcomes in COVID-19 survivors remain largely unclear. The aim of this study was to investigate the long-term cardiovascular outcomes in COVID-19 survivors.This study used the data from the US Collaborative Network in TriNetX. From a cohort of more than 42 million records between 1 January 2019 and 31 March 2022, a total of 4,131,717 participants who underwent SARS-CoV-2 testing were recruited. Study population then divided into two groups based on COVID-19 test results. To avoid reverse causality, the follow-up initiated 30 days after the test, and continued until 12 months. Hazard ratios (HRs) and 95% Confidence intervals (CIs) of the incidental cardiovascular outcomes were calculated between propensity score-matched patients with versus without SARS-CoV-2 infection. Subgroup analyses on sex, and age group were also conducted. Sensitivity analyses were performed using different network, or stratified by hospitalization to explore the difference of geography and severity of COVID-19 infection.The COVID-19 survivors were associated with increased risks of cerebrovascular diseases, such as stroke (HR [95% CI] = 1.618 [1.545-1.694]), arrhythmia related disorders, such as atrial fibrillation (HR [95% CI] = 2.407 [2.296-2.523]), inflammatory heart disease, such as myocarditis (HR [95% CI] =4.406 [2.890-6.716]), ischemic heart disease(IHD), like ischemic cardiomyopathy (HR [95% CI] = 2.811 [2.477-3.190]), other cardiac disorders, such as heart failure (HR [95% CI] =2.296 [2.200-2.396]) and thromboembolic disorders (e.g. pulmonary embolism: HR [95% CI] =2.648 [2.443-2.870]). The risks of two composite endpoints, major adverse cardiovascular event (HR [95% CI] = 1.871 [1.816-1.927]) and any cardiovascular outcome (HR [95% CI] = 1.552 [1.526-1.578]), were also higher in the COVID-19 survivors than in the controls. Moreover, the survival probability of the COVID-19 survivors dramatically decreased in all the cardiovascular outcomes. The risks of cardiovascular outcomes were evident in both male and female COVID-19 survivors. Furthermore, the risk of mortality was higher in the elderly COVID-19 survivors (age ≥ 65 years) than in the young ones. Sensitivity analyses presented roughly similar results globally. Furthermore, the impact of COVID-19 on cardio-related outcomes appeared to be more pronounced in inpatients than in outpatients.The 12-month risk of incidental cardiovascular diseases is substantially higher in the COVID-19 survivors than the non-COVID-19 controls. Clinicians and patients with a history of COVID-19 should pay attention to their cardiovascular health in long term.The Fundamental Research Funds for the Central public welfare research institutes and Young Elite Scientists Sponsorship Program by CACM.
In recent years, the global incidence of type 2 diabetes in young people has increased, especially among minoritized, Indigenous, or financially disadvantaged populations. However, few studies have examined whether poverty is associated with increased risk of youth-onset type 2 diabetes.To examine the association of family income level with the risk of youth-onset type 2 diabetes.This nationwide, population-based retrospective cohort study used data from the 2008 National Health Insurance Research Database of Taiwan, with follow-up through December 31, 2019. Participants included children and adolescents aged 0 to 19 years. Data analysis was performed from June 9, 2022, to January 16, 2023.Family income, classified as very low, low, middle, and high.Cox proportional hazards models were used to estimate adjusted hazard ratios (aHRs) for the risks of youth-onset type 2 diabetes and all-cause mortality for all income groups vs the high-income group.The cohort included a total of 5 182 893 children and adolescents (mean [SD] age, 11.2 [5.2] years; 2 477 807 girls [48.3%]). The mean (SD) follow-up duration was 9.0 (0.3) years. The incidence rates of youth-onset type 2 diabetes were 0.52 cases per 1000 person-years for the very-low-income group, 0.40 cases per 1000 person-years for the low-income group, 0.35 cases per 1000 person-years for the middle-income group, and 0.28 cases per 1000 person-years for the high-income group. Children and adolescents from very-low-income (aHR, 1.55; 95% CI, 1.41-1.71), low-income (aHR, 1.34; 95% CI, 1.27-1.41), and middle-income (aHR, 1.27; 95% CI, 1.20-1.34) families had a significantly higher hazard of youth-onset type 2 diabetes than those from high-income families. Children and adolescents from very-low-income (aHR, 2.18; 95% CI, 1.97-2.41), low-income (aHR, 1.51; 95% CI, 1.42-1.60), and middle-income (aHR, 1.22; 95% CI, 1.14-1.31) families also had a significantly higher hazard of all-cause mortality than those from high-income families. Children and adolescents who were older, female, and obese and had dyslipidemia, gout, or psychiatric disorders had a significantly higher risk of youth-onset type 2 diabetes than children without those characteristics.This population-based cohort study showed that children and adolescents from very-low-income to middle-income families had a higher hazard of youth-onset type 2 diabetes and mortality than those from high-income families. Further research to reveal the factors underlying this association may improve the accuracy of identifying individuals at greatest risk for developing type 2 diabetes in youth.
Nontyphoidal salmonella (NTS) infection has a high mortality rate. Bowel resections affect gut microbiota and immune function, and the association between bowel resection and NTS infection in human beings has not been addressed. We conducted a nationwide propensity score (PS)-matched cohort study to clarify this association. Data from the Longitudinal Health Insurance Database of Taiwan were used to establish a case-cohort with bowel resections from 2000 to 2013. Informed consent was waived by the Institutional Review Board of China Medical University Hospital (CMUH104-REC2-115) because all personal identifying information used had been de-identified. Each case was matched with one control without any bowel resection according to age, gender, index date, and propensity score (PS). Cumulative incidences of and hazard ratios (HRs) for NTS infection development were analyzed. The incidence of NTS infection was greater in patients with a bowel resection than in the control group (2.97 vs. 1.92 per 10,000 person-years), with an adjusted hazard ratio (aHR) of 1.64 (95% CI = 1.08-2.48). The incidence of NTS infection increased significantly for cases with small bowel resections and right hemicolectomies. Age (31-40 and > 50 years), hypertension, chronic kidney disease, chronic obstructive pulmonary disease, and autoimmune diseases were significant risk factors of NTS infection. Stratification analysis revealed that patients without comorbidities were prone to NTS infection after bowel resections. The increased risk of developing NTS infection could be related to the bowel resection. Specific age groups and comorbidities also contribute to increased risk of NTS infection.
To assess the efficacy of etanercept in the treatment of early active nonsteroidal antiinflammatory drug (NSAID)-refractory nonradiographic axial spondyloarthritis (SpA).The study population consisted of patients who met the Assessment of SpondyloArthritis international Society (ASAS) classification criteria for axial SpA but not the modified New York radiographic criteria for ankylosing spondylitis (as assessed by a radiologist at the central trial site), had a symptom duration of >3 months but <5 years, had a score of ≥4 on the Bath Ankylosing Spondylitis Disease Activity Index, and had been treated unsuccessfully with ≥2 NSAIDs. Patients were randomized to receive etanercept 50 mg/week or placebo and continued background NSAID treatment for 12 weeks (double-blind study); during the subsequent open-label period, all patients received etanercept 50 mg/week. The primary study end point was meeting the ASAS criteria for 40% improvement (ASAS40) at week 12. Magnetic resonance imaging (MRI) of the sacroiliac joints and spine was performed at baseline and week 12.One hundred six patients were randomized to the etanercept group and 109 to the placebo group. Of the 215 patients, the mean ± SD age at baseline was 32.0 ± 7.8 years, 154 (72%) were HLA-B27 positive, and 174 (81%) had MRI-confirmed sacroiliitis. At 12 weeks, the proportion of patients with improvement according to the ASAS40 was significantly higher in the etanercept group than in the placebo group (34 of 105 [32%] versus 17 of 108 [16%]; P = 0.006). Patients who received etanercept exhibited a greater reduction in MRI-based scores for sacroiliac joint inflammation (-46.9% versus -10.9%; P < 0.001) and spinal inflammation (-45.4% versus -33.4%; P = 0.04) compared with placebo-treated patients at week 12. Post hoc analyses suggested a possible association between higher baseline C-reactive protein levels or MRI sacroiliac joint inflammation scores and higher rates of ASAS40 response to etanercept. At week 24, patients in the placebo group who had switched to etanercept at 12 weeks exhibited improvement similar to that observed in patients who had received etanercept for 24 weeks.In patients with nonradiographic axial SpA, etanercept treatment was associated with rapid, significant improvement in symptomatic disease activity, function, and systemic and skeletal inflammation over 12 weeks; clinical/functional improvement was sustained over 24 weeks.
Abstract Obstructive sleep apnea is a well‐known risk factor regarding the severity of COVID‐19 infection. However, to date, relatively little research performed on the prevalence of obstructive sleep apnea in COVID‐19 survivors. The purpose of this study was to investigate the risk of obstructive sleep apnea after COVID‐19 infection. This study was based on data collected from the US Collaborative Network in TriNetX. From January 1, 2020 to June 30, 2022, participants who underwent the SARS‐CoV‐2 test were included in the study. Based on their positive or negative results of the COVID‐19 test results (the polymerase chain reaction [PCR] test), we divided the study population into two groups. The duration of follow‐up began when the PCR test was administered and continued for 12 months. Hazard ratios (HRs) and 95% confidence intervals (CIs) for newly recorded COVID‐19 positive subjects for obstructive sleep apnea were calculated using the Cox proportional hazards model and compared to those without COVID‐19 infection. Subgroup analyses were performed for the age, sex, and race, groups. The COVID‐19 group was associated with an increased risk of obstructive sleep apnea, at both 3 months of follow‐up (HR: 1.51, 95% CI: 1.48–1.54), and 1 year of follow‐up (HR: 1.57, 95% CI: 1.55–1.60). Kaplan–Meier curves regarding the risk of obstructive sleep apnea revealed a significant difference of probability between the two cohorts in the follow‐up periods of 3 months and 1 year (Log‐Rank test, p < 0.001). The risks of obstructive sleep apnea among COVID‐19 patients were significant in the less than 65 year of age group (HR: 1.50, 95% CI: 1.47–1.52), as well as in the group older than or equal to 65 years (HR:1.69, 95% CI: 1.64–1.73). Furthermore, the risks of obstructive sleep apnea were evident in both the male and female COVID‐19 groups. Compared to the control group, the risks of obstructive sleep apnea in the COVID‐19 participants increased in the subgroups of White (HR: 1.62, 95% CI: 1.59–1.64), Blacks/African Americans (HR: 1.50, 95% CI: 1.45–1.55), Asian (HR: 1.46, 95% CI: 1.32–1.62) and American Indian/Alaska Native (HR: 1.36, 95% CI: 1.07–1.74). In conclusion, the incidence of new diagnosis obstructive sleep apnea could be substantially higher after COVID‐19 infection than non‐COVID‐19 comparison group. Physicians should evaluate obstructive sleep apnea in patients after COVID‐19 infection to help prevent future long‐term adverse effects from occurring in the future, including cardiovascular and neurovascular disease.
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In Taiwan, the incidence and prevalence of psoriatic arthritis (PsA) have risen significantly in recent years. Moreover, data from the Taiwan National Health Insurance Research Database (NHIRD) show that more than 85% of PsA patients are treated with just non-steroidal anti-inflammatory drugs (NSAIDs) and/or conventional synthetic disease-modifying anti-rheumatic drugs (csDMARDs). Taiwanese clinicians have also expressed concerns regarding uncertainties in the diagnosis of PsA and the delayed, interrupted, and/or tapered use of biologics, as well as differences in therapeutic preferences between and within dermatologists and rheumatologists. To address these issues, the Taiwan Rheumatology Association and the Taiwanese Association for Psoriasis and Skin Immunology jointly convened a committee of 28 clinicians from the fields of rheumatology, dermatology, orthopedics, and rehabilitation, to develop evidence-based consensus recommendations for the practical management of PsA in Taiwan. A total of six overarching principles and 13 recommendations were developed and approved, as well as a treatment algorithm with four separate tracks for axial PsA, peripheral PsA, enthesitis, and dactylitis. Psoriasis (PsO) management was not discussed here, as the Taiwanese Dermatological Association has recently published a comprehensive consensus statement on the management of PsO. Together, these recommendations provide an up-to-date, evidence-based framework for PsA care in Taiwan.
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