{"title":"胆道闭锁患儿肾上腺功能不全的过度诊断。","authors":"Suparat Ekawaravong, Suporn Treepongkaruna, Preamrudee Poomthavorn, Sarunyu Pongratanakul, Patcharin Khlairit, Suwanee Chanprasertyothin, Pat Mahachoklertwattana","doi":"10.1297/cpe.2022-0083","DOIUrl":null,"url":null,"abstract":"<p><p>Serum cortisol mainly binds to the cortisol-binding globulin (CBG). Children with biliary atresia (BA) may have low serum CBG levels; thus, low serum total cortisol (TC) levels and adrenal insufficiency (AI) may be overdiagnosed. This study aimed to assess adrenal function in children with BA. All the patients underwent adrenocorticotropic hormone (ACTH) stimulation tests. Plasma ACTH, serum TC, and CBG levels were measured at baseline, with additional TC measurements at 30 and 60 min during testing. Free cortisol (FC) index (FCI) and calculated FC (cFC) were also calculated. AI was defined as peak TC <500 nmol/L (<18 μg/dL), peak FCI <12 nmol/mg, or peak cFC <33 nmol/L (<1.2 μg/dL). This study enrolled 71 children with BA. The Median (IQR) age of the patients was 5.5 (1.7-11.4) years. Twenty-five (35%) patients were diagnosed with AI based on the peak TC. In the AI group, the median serum CBG level was significantly lower than that in the non-AI group (481 <i>vs</i>. 533 nmol/L, <i>p</i> = 0.03). Only eight patients (11%) met all three AI criteria (six secondary AI and two primary AI). In conclusion, low serum CBG levels contribute to reduced peak TC and, consequently, overdiagnosing AI. Peak FCI and cFC could help reduce the overdiagnosis of AI.</p>","PeriodicalId":72619,"journal":{"name":"","volume":"32 3","pages":"147-154"},"PeriodicalIF":0.0,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/d0/72/cpe-32-147.PMC10288293.pdf","citationCount":"0","resultStr":"{\"title\":\"Overdiagnosis of adrenal insufficiency in children with biliary atresia.\",\"authors\":\"Suparat Ekawaravong, Suporn Treepongkaruna, Preamrudee Poomthavorn, Sarunyu Pongratanakul, Patcharin Khlairit, Suwanee Chanprasertyothin, Pat Mahachoklertwattana\",\"doi\":\"10.1297/cpe.2022-0083\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Serum cortisol mainly binds to the cortisol-binding globulin (CBG). Children with biliary atresia (BA) may have low serum CBG levels; thus, low serum total cortisol (TC) levels and adrenal insufficiency (AI) may be overdiagnosed. This study aimed to assess adrenal function in children with BA. All the patients underwent adrenocorticotropic hormone (ACTH) stimulation tests. Plasma ACTH, serum TC, and CBG levels were measured at baseline, with additional TC measurements at 30 and 60 min during testing. Free cortisol (FC) index (FCI) and calculated FC (cFC) were also calculated. AI was defined as peak TC <500 nmol/L (<18 μg/dL), peak FCI <12 nmol/mg, or peak cFC <33 nmol/L (<1.2 μg/dL). This study enrolled 71 children with BA. The Median (IQR) age of the patients was 5.5 (1.7-11.4) years. Twenty-five (35%) patients were diagnosed with AI based on the peak TC. In the AI group, the median serum CBG level was significantly lower than that in the non-AI group (481 <i>vs</i>. 533 nmol/L, <i>p</i> = 0.03). Only eight patients (11%) met all three AI criteria (six secondary AI and two primary AI). In conclusion, low serum CBG levels contribute to reduced peak TC and, consequently, overdiagnosing AI. Peak FCI and cFC could help reduce the overdiagnosis of AI.</p>\",\"PeriodicalId\":72619,\"journal\":{\"name\":\"\",\"volume\":\"32 3\",\"pages\":\"147-154\"},\"PeriodicalIF\":0.0,\"publicationDate\":\"2023-01-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/d0/72/cpe-32-147.PMC10288293.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1297/cpe.2022-0083\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1297/cpe.2022-0083","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0
摘要
血清皮质醇主要与皮质醇结合球蛋白(CBG)结合。胆道闭锁(BA)患儿可能有较低的血清CBG水平;因此,低血清总皮质醇(TC)水平和肾上腺功能不全(AI)可能被过度诊断。本研究旨在评估BA患儿的肾上腺功能。所有患者均行促肾上腺皮质激素(ACTH)刺激试验。在基线时测量血浆ACTH、血清TC和CBG水平,并在测试期间的30和60分钟测量额外的TC。同时计算游离皮质醇(FC)指数(FCI)和计算FC (cFC)。AI定义为峰值TC vs. 533 nmol/L, p = 0.03)。只有8名患者(11%)符合所有三项人工智能标准(6名继发性人工智能和2名原发性人工智能)。总之,低血清CBG水平有助于降低TC峰值,从而过度诊断AI。峰值FCI和cFC有助于减少人工智能的过度诊断。
Overdiagnosis of adrenal insufficiency in children with biliary atresia.
Serum cortisol mainly binds to the cortisol-binding globulin (CBG). Children with biliary atresia (BA) may have low serum CBG levels; thus, low serum total cortisol (TC) levels and adrenal insufficiency (AI) may be overdiagnosed. This study aimed to assess adrenal function in children with BA. All the patients underwent adrenocorticotropic hormone (ACTH) stimulation tests. Plasma ACTH, serum TC, and CBG levels were measured at baseline, with additional TC measurements at 30 and 60 min during testing. Free cortisol (FC) index (FCI) and calculated FC (cFC) were also calculated. AI was defined as peak TC <500 nmol/L (<18 μg/dL), peak FCI <12 nmol/mg, or peak cFC <33 nmol/L (<1.2 μg/dL). This study enrolled 71 children with BA. The Median (IQR) age of the patients was 5.5 (1.7-11.4) years. Twenty-five (35%) patients were diagnosed with AI based on the peak TC. In the AI group, the median serum CBG level was significantly lower than that in the non-AI group (481 vs. 533 nmol/L, p = 0.03). Only eight patients (11%) met all three AI criteria (six secondary AI and two primary AI). In conclusion, low serum CBG levels contribute to reduced peak TC and, consequently, overdiagnosing AI. Peak FCI and cFC could help reduce the overdiagnosis of AI.