Brown adipose tissue (BAT) contributes to energy expenditure through cold-induced thermogenesis and is closely linked to systemic lipid metabolism. However, the dynamic changes in circulating n-3 polyunsaturated fatty acids (PUFAs) during cold exposure and their association with BAT activity remain unclear. Healthy young men underwent controlled cold exposure, and BAT activity was quantified using fluorodeoxyglucose positron emission tomography/computed tomography (FDG-PET/CT). Circulating levels of n-3 PUFAs, including alpha-linolenic acid (ALA), eicosapentaenoic acid (EPA), docosapentaenoic acid (DPA), and docosahexaenoic acid (DHA), were measured before and after cold exposure using whole-blood lipidomics. K-means clustering based on log-transformed maximum standardized uptake value (lnSUVmax) was performed. Changes in lipid parameters were evaluated using paired t-tests and multiple regression analyses. Cold exposure significantly decreased ALA levels and the ALA/EPA ratio while increasing EPA and DPA levels and the EPA/DPA and DPA/DHA ratios. Multiple regression analyses revealed that BAT activity (lnSUVmax) was independently associated with changes in several lipid parameters. Higher SUVmax was positively associated with changes in ALA levels and the ALA/EPA ratio, whereas inverse associations were observed with changes in EPA, DPA, EPA/DPA, and DPA/DHA. Baseline lipid levels were not significant predictors of lipidomic changes. Cold-induced BAT activation is associated with dynamic remodeling of circulating n-3 fatty acids. These findings suggest that thermogenic capacity may influence systemic lipid metabolism and highlight the metabolic interplay between BAT activity and circulating n-3 PUFAs. Trial registration: University Hospital Medical Information Network Clinical Trials Registry (UMIN-CTR), UMIN000029206.
Oncocytic thyroid carcinoma (OCA) is a rare thyroid malignancy that has only recently been recognized as a distinct entity from follicular thyroid carcinoma. Till date, studies regarding the prognosis of OCA remain inadequate. In this retrospective study, we investigated the prognosis of OCA among 137 Japanese patients who underwent surgery at our institution. Two patients had distant metastasis before surgery (M1), and two showed distant metastasis after surgery. The 10year and 15-year distant recurrence-free survival rates were 100%, and 92.7%, respectively. No deaths occurred owing to thyroid carcinoma. Four patients with distant metastasis/recurrence were aged >= 60 years, and their tumors were >4 cm. Further, two patients with M1 had both vascular invasion (VI) and wide capsular invasion (CI). One patient had a poorly differentiated component (approximately 30%), and one showed high cell proliferating activity (Ki-67 labeling index [LI] >= 5%). Of the two patients who developed distant metastasis after surgery, one had VI and wide CI, and although another had minimal CI and was negative for VI, the Ki-67 LI of his tumor was >5%. These findings suggest that Japanese patients with OCA generally had excellent prognosis after surgery; however, older age, large tumor size, wide CI, VI positivity, high Ki-67 LI, and the presence of a poorly differentiated component may be indicators of aggressive behavior. Further multicenter studies with larger samples and longer follow-up durations are necessary to identify clinicopathological factors accurately predicting poor prognosis in patients with OCA.
In Japan, GH treatment (GHT) is used for short stature associated with small for gestational age (SGA), Noonan syndrome (NS), Turner syndrome (TS), achondroplasia (ACH), and hypochondroplasia (HCH). However, real-world data on these diagnoses and GHT use for these conditions in Japan remain limited. In this retrospective cohort study, we used the JMDC claims database (2005-2021) to extract data of children with SGA-related short stature or diagnosed with short stature diseases, namely NS, TS, ACH, or HCH, particularly in boys aged <17 years and girls aged <15 years. We reported the prevalence, comorbidities, age at diagnosis, time from diagnosis to treatment initiation, persistence to GHT, and proportion of days covered (PDC). We analyzed data from children with SGA-related short stature (n = 786), NS (n = 79), TS (n = 172), ACH (n = 107), and HCH (n = 30); the mean (standard deviation) age at initial diagnosis was 5.2 (3.5), 4.0 (4.0), 6.5 (4.2), 3.0 (4.1), and 4.5 (3.9) years, respectively. The prevalence (per 100,000) was 59.59, 5.45, 9.52, 8.31, and 2.12 for SGA-related short stature, NS, TS, ACH, and HCH, respectively. Mean PDC for SGA-related short stature, NS, TS, ACH, and HCH was 0.918, 0.984, 0.953, 0.941, and 1.000, respectively, demonstrating high persistence to GHT. This study provides insights into the current diagnosis and GHT use among children with short stature in Japan. The findings suggest that most Japanese children continue and adhere well to GHT. However, continued efforts toward early referral and diagnosis remain crucial.
McCune-Albright syndrome (MAS) is a rare mosaic disorder caused by somatic gain-of-function variants in GNAS and frequently associated with fibrous dysplasia (FD). FD-related complications impair physical function and quality of life (QOL), yet Japanese data are lacking. We conducted a nationwide survey to characterize functional impairment and physician-perceived QOL factors in patients with MAS in Japan. Data on 43 patients were collected using a web-based questionnaire sent to council members of the Japanese Society for Pediatric Endocrinology and the Japanese Society for Bone and Mineral Research. Motor disability was assessed using the Japanese version of the modified Rankin Scale (mRS); respiratory, visual, and hearing impairments were graded using the Japanese severity criteria for designated intractable diseases. Physicians identified the single factor perceived as most affecting each patient's QOL. Motor impairment was present in 72.1%, including moderate or severe impairment (mRS >= 3) in 30.2%; other impairments were each absent in more than 80%. The most frequent QOL-affecting factor was bone pain or fractures (37.2%), followed by facial deformity or appearance changes (18.6%) and mobility or physical activity limitation (16.3%). Physician-perceived QOL factors differed by mRS group (p < 0.001): bone pain and facial deformity predominated in patients with mRS <3, whereas mobility limitation predominated in those with mRS >= 3. These findings indicate that FD-related skeletal manifestations are a major source of disability and QOL burden in Japanese patients with MAS, supporting stratified management tailored to motor disability.
The coronavirus disease 2019 (COVID-19) pandemic and associated lifestyle changes have affected metabolic status in individuals with diabetes. The impact of these changes on renal function in individuals with type 1 diabetes (T1D) remains unclear. We performed a post-hoc analysis of a multicenter retrospective cohort study (the IPRA-CKD study) including 190 Japanese individuals with T1D who were not receiving sodium-glucose cotransporter 2 inhibitors. Participants were recruited between June 2022 and July 2023, and longitudinal data over a consecutive 60-month period preceding enrollment were extracted at 6-month intervals and divided into periods before and during the pandemic. Annual rates of estimated glomerular filtration rate (eGFR) decline were compared between the periods before and during the COVID-19 pandemic using a linear mixed-effects model. The eGFR decline was not accelerated during the pandemic compared with before the pandemic; the estimated eGFR slopes were-1.54 +/- 0.28 mL/min/1.73 m2/year before the pandemic and-1.05 +/- 0.22 mL/min/1.73 m2/year during the pandemic (p = 0.27). Glycemic control was preserved during the pandemic, despite small but significant increases in body weight, blood pressure, and insulin dose. Exploratory subgroup analyses suggested a less steep eGFR decline during the pandemic among participants with body mass index >= 25 kg/m2 or those receiving renin-angiotensin system inhibitors; however, these findings should be interpreted as hypothesis-generating. In this multicenter cohort of individuals with T1D, the COVID-19 pandemic was not associated with accelerated eGFR decline overall. Clinical Trial Registry (CTR) registration: UMIN-CTR000047441
