[Telephone navigation and health check attendance among public assistance recipients: Lessons from two-year effort in Toyonaka City].

Objective　Poverty is a significant health determinant. As public assistance recipients experience difficulties in health management, a healthcare management support program to provide health checks has been implemented by welfare offices in Japan since 2021. However, effective approaches to maximize health check rates are limited. This study aimed to identify the impact of telephone navigation on recipients' health check-receiving behaviors, using data from welfare offices in Toyonaka City.Methods　This study included recipients aged 40-60 years who received telephone navigation for health check programs in 2021 and 2022. In 2021, telephone navigation was provided to eligible recipients. In 2022, the recipients were divided into two groups based on their household identification numbers (even/odd). We examined the difference in the health check rate in 2021 depending on whether the navigation system was connected. Additionally, we examined the differences across groups and navigation periods in 2022.Results　In 2021, 32 (7.9%) recipients received health checks. Twenty-six (10.2%) of the 255 recipients and six (4.1%) of the 148 recipients in the navigated and non-navigated groups, respectively, underwent health checks. In the navigated group, health check rates were higher among recipients in their 50s (13.3% vs. 3.1%; P = 0.006), unemployed recipients (13.9% vs. 3.6%; P = 0.014), those who did not receive previous health checks (9.1% vs. 1.5%; P = 0.003), and those who did not receive regular medical consultations (8.3% vs. 0%; P = 0.012). In 2022, 247 and 225 patients were assigned to the odd- and even-numbered groups, respectively, with no differences in their characteristics. During the intervention period, four (1.6%) of the 247 recipients and 10 (4.6%) of the 219 recipients in the odd- and even-numbered groups, respectively, underwent health checks. During the non-intervention period, five (2.1%) and six (2.7%) recipients of the odd- and even-numbered groups, respectively, underwent health checks. Health check rates were higher toward the deadlines. The estimated conditional odds ratio for receiving the health checks by the navigation was 1.35 (95% confidence interval; 0.59-2.93, P = 0.503).Conclusion　Telephone navigation may be effective in some recipients. Meanwhile, targeting recipients with attributes, such as "in their 50s," "unemployed," "received no previous health checks," and "received no regular medical consultations" may increase the response rate. Therefore, policymakers should consider using reliable telephone navigation methods and navigating near deadlines.

Zebrafish model of human Zellweger syndrome reveals organ-specific accumulation of distinct fatty acid species and widespread gene expression changes.

In Zellweger syndrome (ZS), lack of peroxisome function causes physiological and developmental abnormalities in many organs such as the brain, liver, muscles, and kidneys, but little is known about the exact pathogenic mechanism. By disrupting the zebrafish pex2 gene, we established a disease model for ZS and found that it exhibits pathological features and metabolic changes similar to those observed in human patients. By comprehensive analysis of the fatty acid profile, we found organ-specific accumulation and reduction of distinct fatty acid species, such as an accumulation of ultra-very-long-chain polyunsaturated fatty acids (ultra-VLC-PUFAs) in the brains of pex2 mutant fish. Transcriptome analysis using microarray also revealed mutant-specific gene expression changes that might lead to the symptoms, including reduction of crystallin, troponin, parvalbumin, and fatty acid metabolic genes. Our data indicated that the loss of peroxisomes results in widespread metabolic and gene expression changes beyond the causative peroxisomal function. These results suggest the genetic and metabolic basis of the pathology of this devastating human disease.

Detection of unusual very-long-chain fatty acid and ether lipid derivatives in the fibroblasts and plasma of patients with peroxisomal diseases using liquid chromatography-mass spectrometry.

Metabolic changes occur in patients with peroxisomal diseases owing to impairments in the genes involved in peroxisome function. For diagnostic purposes, saturated very-long-chain fatty acids (VLCFAs) such as C24:0 and C26:0, phytanic acid, pristanic acid, and plasmalogens are often measured as metabolic hallmarks. As the direct pathology of peroxisomal disease is yet to be fully elucidated, we sought to explore the fatty acid species that accumulate in patients with peroxisomal diseases. We developed a method for detecting a range of fatty acids implicated in peroxisomal diseases such as Zellweger syndrome (ZS) and X-linked adrenoleukodystrophy (X-ALD). To this end, we employed an ultra-performance liquid chromatography-mass spectrometry (LC-MS) coupled with negatively charged electrospray ionization. Fatty acids from patients and control subjects were extracted from total lipids by acid-hydrolysis and compared. In accordance with previous results, the amounts of VLCFAs, phytanic acid, and pristanic acid differed between the two groups. We identified extremely long and highly polyunsaturated VLCFAs (ultra-VLC-PUFAs) such as C44:12 in ZS samples. Moreover, three unknown molecules were prominent in control samples but scarcely detectable in ZS samples. LC-MS/MS analysis identified these as 1-alkyl-sn-glycerol 3-phosphates derived from ether lipids containing fatty alcohols such as C16:0, C18:0, or C18:1. Our method provides an approach to observing a wide range of lipid-derived fatty acids and related molecules in order to understand the metabolic changes involved in peroxisomal diseases. This technique can therefore be used in identifying metabolic markers and potential clinical targets for future treatment.