{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T20:41:09Z","timestamp":1790800869620,"version":"4.1.0"},"reference-count":34,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2021,10,18]],"date-time":"2021-10-18T00:00:00Z","timestamp":1634515200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100005029","name":"Helse Vest Regionalt Helsef\u00f8retak","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100005029","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100005416","name":"Norges Forskningsr\u00e5d","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100005416","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100009708","name":"Novo Nordisk Fonden","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100009708","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007793","name":"Stiftelsen Kristian Gerhard Jebsen","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007793","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Endocrinol."],"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Patients with primary adrenal insufficiency (PAI) suffer reduced quality of life (QoL), but comparisons with large-scale normative data are scarce. The clinical characteristics associated with reduced QoL are largely unknown.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Cross-sectional data on clinical characteristics and QoL scores from 494 patients were included. QoL was measured using RAND-36 (generic) and AddiQoL (-30 and -8, disease-specific). RAND-36 is reported as subdomain scores as well as physical (PCS) and metal (MCS) summary scores and compared with normative data.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Perception of physical role was consistently decreased across age groups in patients with PAI compared with normative data [75 (0-100) <jats:italic>vs.<\/jats:italic> 100 (50-100), p&amp;lt;0.001]. Men with PAI reported significantly lower scores for social functioning [88 (75-100) <jats:italic>vs.<\/jats:italic> 100 (75-100), p&amp;lt;0.001], as well as for vitality and physical role. In women, the greatest impairment was seen in physical role [50 (0-100) <jats:italic>vs.<\/jats:italic> 100 (50-100), p&amp;lt;0.001], followed by social functioning, vitality, physical function, general health, mental health, and emotional role. Overall, better QoL was associated with male sex (AddiQoL-30: 89 \u00b1 13 <jats:italic>vs.<\/jats:italic> 82 \u00b1 13, p&amp;lt;0.002), younger age (e.g. 20-29 <jats:italic>vs.<\/jats:italic> 80-89 years: PCS 59 [50-62] <jats:italic>vs.<\/jats:italic> 46 [37-53], p&amp;lt;0.001), autoimmune etiology [PCS: 53 (45-59) <jats:italic>vs.<\/jats:italic>. 45 (38-54), p&amp;lt;0.001], and absence of autoimmune comorbidity [PCS: 54 (45-59) <jats:italic>vs.<\/jats:italic> 50 (43-58), p&amp;lt;0.001]. There were no significant differences in QoL scores between different doses or dosing regimens of glucocorticoid or mineralocorticoid replacement.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>QoL is reduced in patients with PAI, especially perception of physical role in women and social functioning in men. Among patients with PAI, female sex, higher age, non-autoimmune etiology, and autoimmune comorbidity was associated with lower QoL-scores.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fendo.2021.718660","type":"journal-article","created":{"date-parts":[[2021,10,20]],"date-time":"2021-10-20T01:23:37Z","timestamp":1634693017000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":16,"title":["Sex-Specific Limitations in Physical Health in Primary Adrenal Insufficiency"],"prefix":"10.3389","volume":"12","author":[{"given":"Nora M\u00f8ller","family":"Didriksen","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"\u00c5se Bjorvatn","family":"S\u00e6vik","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Linn Solveig","family":"Sortland","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marianne","family":"\u00d8ksnes","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eystein Sverre","family":"Husebye","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2021,10,18]]},"reference":[{"key":"B1","article-title":"Primary Adrenal Failure\u2013Causes, Diagnostics and Therapy","volume":"125","author":"L\u00f8v\u00e5s","year":"2005","journal-title":"Tidsskr Nor Laegeforen"},{"key":"B2","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2009-1368","article-title":"Clinical, Immunological, and Genetic Features of Autoimmune Primary Adrenal Insufficiency: Observations From a Norwegian Registry","volume":"94","author":"Erichsen","year":"2009","journal-title":"J Clin Endocrinol Metab"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2265.2002.01466.x","article-title":"Subjective Health Status in Norwegian Patients With Addison's Disease","volume":"56","author":"Lovas","year":"2002","journal-title":"Clin Endocrinol (Oxf)"},{"key":"B4","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(21)00136-7","article-title":"Adrenal Insufficiency","volume":"397","author":"Husebye","year":"2021","journal-title":"Lancet (London England)"},{"key":"B5","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-15-1242","article-title":"Management of Endocrine Disease: Epidemiology, Quality of Life and Complications of Primary Adrenal Insufficiency: A Review","volume":"175","author":"Bensing","year":"2016","journal-title":"Eur J Endocrinol \/ Eur Fed Endocr Soc"},{"key":"B6","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-12-1078","article-title":"A Direct Comparison of Quality of Life in Obese and Cushing's Syndrome Patients","volume":"168","author":"Abraham","year":"2013","journal-title":"Eur J Endocrinol"},{"key":"B7","doi-asserted-by":"publisher","first-page":"597","DOI":"10.1530\/EJE-09-0884","article-title":"Epidemiology of Adrenal Crisis in Chronic Adrenal Insufficiency: The Need for New Prevention Strategies","volume":"162","author":"Hahner","year":"2010","journal-title":"Eur J Endocrinol \/ Eur Fed Endocr Soc"},{"key":"B8","doi-asserted-by":"publisher","DOI":"10.1007\/s12020-017-1513-0","article-title":"Longitudinal AddiQoL Scores may Identify Higher Risk for Adrenal Crises in Addison's Disease","volume":"60","author":"Meyer","year":"2018","journal-title":"Endocrine"},{"key":"B9","doi-asserted-by":"publisher","DOI":"10.1111\/cen.12484","article-title":"Impaired Health-Related Quality of Life in Addison's Disease\u2013Impact of Replacement Therapy, Comorbidities and Socio-Economic Factors","volume":"81","author":"Kluger","year":"2014","journal-title":"Clin Endocr"},{"key":"B10","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2013-4253","article-title":"Continuous Subcutaneous Hydrocortisone Infusion Versus Oral Hydrocortisone Replacement for Treatment of Addison's Disease: A Randomized Clinical Trial","volume":"99","author":"Oksnes","year":"2014","journal-title":"J Clin Endocrinol Metab"},{"key":"B11","doi-asserted-by":"publisher","DOI":"10.1111\/cen.13719","article-title":"Quality of Life in Patients With Adrenal Disease: A Systematic Review","volume":"89","author":"Ho","year":"2018","journal-title":"Clin Endocrinol (Oxf)"},{"key":"B12","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2014-2433","article-title":"Continuous Subcutaneous Hydrocortisone Infusion Therapy in Addison's Disease: A Randomized, Placebo-Controlled Clinical Trial","volume":"99","author":"Gagliardi","year":"2014","journal-title":"J Clin Endocrinol Metab"},{"key":"B13","doi-asserted-by":"publisher","first-page":"297","DOI":"10.1111\/j.1365-2265.2009.03596.x","article-title":"Influence of Hydrocortisone Dosage Scheme on Health-Related Quality of Life in Patients With Adrenal Insufficiency","volume":"72","author":"Bleicken","year":"2010","journal-title":"Clin Endocr"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2011-1926","article-title":"Improved Cortisol Exposure-Time Profile and Outcome in Patients With Adrenal Insufficiency: A Prospective Randomized Trial of a Novel Hydrocortisone Dual-Release Formulation","volume":"97","author":"Johannsson","year":"2012","journal-title":"J Clin Endocrinol Metab"},{"key":"B15","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1016\/j.beem.2014.08.008","article-title":"Mineralocorticoid Substitution and Monitoring in Primary Adrenal Insufficiency","volume":"29","author":"Quinkler","year":"2015","journal-title":"Best Pract Res Clin Endocrinol Metab"},{"key":"B16","doi-asserted-by":"publisher","DOI":"10.1159\/000438791","article-title":"The Role of Fludrocortisone in Cognition and Mood in Patients With Primary Adrenal Insufficiency (Addison's Disease)","volume":"103","author":"Schultebraucks","year":"2016","journal-title":"Neuroendocrinology"},{"key":"B17","volume-title":"Levek\u00e5rsunders\u00f8kelsen 2002 - Tversnitt Tema: Helse (The Norwegian Level of Living Survey)","year":"2004"},{"key":"B18","doi-asserted-by":"publisher","first-page":"bvaa160","DOI":"10.1210\/jendso\/bvaa160","article-title":"Metabolic Effects of Cortisone Acetate vs Hydrocortisone in Patients With Secondary Adrenal Insufficiency","volume":"4","author":"Ekstrand","year":"2020","journal-title":"J Endocr Soc"},{"key":"B19","doi-asserted-by":"publisher","DOI":"10.1002\/hec.4730020305","article-title":"The RAND 36-Item Health Survey 1.0","volume":"2","author":"Hays","year":"1993","journal-title":"Health Econ"},{"key":"B20","doi-asserted-by":"publisher","DOI":"10.1016\/S0895-4356(98)00081-X","article-title":"Overview of the SF-36 Health Survey and the International Quality of Life Assessment (IQOLA) Project","volume":"51","author":"Ware","year":"1998","journal-title":"J Clin Epidemiol"},{"key":"B21","doi-asserted-by":"publisher","DOI":"10.1093\/pubmed\/21.4.372","article-title":"Comparison of UK and US Methods for Weighting and Scoring the SF-36 Summary Measures","volume":"21","author":"Jenkinson","year":"1999","journal-title":"J Public Health Med"},{"key":"B22","doi-asserted-by":"publisher","first-page":"405","DOI":"10.1023\/a:1012588218728","article-title":"Interpreting SF-36 Summary Health Measures: A Response","volume":"10","author":"Ware","year":"2001","journal-title":"Qual Life Res  Int J Qual Life Aspects Treat Care Rehabil"},{"key":"B23","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2011-1901","article-title":"Quality of Life in European Patients With Addison's Disease: Validity of the Disease-Specific Questionnaire AddiQoL","volume":"97","author":"\u00d8ksnes","year":"2012","journal-title":"J Clin Endocrinol Metab"},{"key":"B24","doi-asserted-by":"crossref","DOI":"10.1097\/00005650-199206000-00002","article-title":"The MOS 36-Item Short-Form Health Survey (SF-36). I. Conceptual Framework and Item Selection","volume":"30","author":"Ware","year":"1992","journal-title":"Med Care"},{"key":"B25","volume-title":"SF-36 Health Survey: Manual and Interpretation Guide","author":"Ware","year":"2000"},{"key":"B26","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-017-1684-4","article-title":"Norwegian Reference Values for the Short-Form Health Survey 36: Development Over Time","volume":"27","author":"Jacobsen","year":"2018","journal-title":"Qual Life Res  Int J Qual Life Aspects Treat Care Rehabil"},{"key":"B27","doi-asserted-by":"publisher","first-page":"51","DOI":"10.1186\/s12955-017-0625-9","article-title":"Measurement Properties and Normative Data for the Norwegian SF-36: Results From a General Population Survey","volume":"15","author":"Garratt","year":"2017","journal-title":"Health Qual Life Outcomes"},{"key":"B28","doi-asserted-by":"publisher","DOI":"10.1055\/s-0032-1331766","article-title":"What Affects the Quality of Life in Autoimmune Addison's Disease","volume":"45","author":"Meyer","year":"2013","journal-title":"Horm Metab Res = Horm  Stoffwechselforschung = Horm Metabol"},{"key":"B29","doi-asserted-by":"publisher","DOI":"10.1097\/00005650-200010000-00006","article-title":"Performance of the SF-36, SF-12, and RAND-36 Summary Scales in a Multiple Sclerosis Population","volume":"38","author":"Nortvedt","year":"2000","journal-title":"Med Care"},{"key":"B30","doi-asserted-by":"publisher","DOI":"10.1016\/j.apmr.2006.12.032","article-title":"Response Shift Theory: Important Implications for Measuring Quality of Life in People With Disability","volume":"88","author":"Schwartz","year":"2007","journal-title":"Arch Phys Med Rehabil"},{"key":"B31","doi-asserted-by":"publisher","DOI":"10.1530\/endoabs.50.P023","article-title":"Quality of Life in Patients With Adrenal Disease: A Systematic Review","volume":"89","author":"Ho","year":"2018","journal-title":"Clin Endocr"},{"key":"B32","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-14-0023","article-title":"Psychological Morbidity and Impaired Quality of Life in Patients With Stable Treatment for Primary Adrenal Insufficiency: Cross-Sectional Study and Review of the Literature","volume":"171","author":"Tiemensma","year":"2014","journal-title":"Eur J Endocrinol \/ Eur Fed Endocr Soc"},{"key":"B33","doi-asserted-by":"publisher","DOI":"10.1111\/cen.13059","article-title":"Decreased Physical Activity, Reduced QoL and Presence of Debilitating Fatigue in Patients With Addison's Disease","volume":"85","author":"van der Valk","year":"2016","journal-title":"Clin Endocr"},{"key":"B34","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-017-1684-4","article-title":"Norwegian Reference Values for the Short-Form Health Survey 36: Development Over Time","volume":"27","author":"Jacobsen","year":"2018","journal-title":"Qual Life Res  Int J Qual Life Aspects Treat Care Rehabil"}],"container-title":["Frontiers in Endocrinology"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fendo.2021.718660\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,10,20]],"date-time":"2021-10-20T01:24:33Z","timestamp":1634693073000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fendo.2021.718660\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,10,18]]},"references-count":34,"alternative-id":["10.3389\/fendo.2021.718660"],"URL":"https:\/\/doi.org\/10.3389\/fendo.2021.718660","relation":{},"ISSN":["1664-2392"],"issn-type":[{"value":"1664-2392","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,10,18]]},"article-number":"718660"}}