ArticleThe Journal of clinical endocrinology and metabolism2020
Residual Adrenal Function in Autoimmune Addison's Disease-Effect of Dual Therapy With Rituximab and Depot Tetracosactide.
Article in The Journal of clinical endocrinology and metabolism, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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Who cites it
9 citing papers in PubMed, 21 citations in OpenAlex.
- Residual Corticosteroid Production in Autoimmune Addison Disease.The Journal of clinical endocrinology and metabolism · 2020Trial
- IFN-γ-producing self-reactive CD4+ T cells induce autoimmune adrenalitis in a mouse model of Addison's disease.JCI insight · 2026Article
- Distinguishing clinical features of paediatric autoimmune Addison's disease and polyendocrine syndromes: a 15-year single-centre observational study from Baghdad, Iraq.Pediatric endocrinology, diabetes, and metabolism · 2026Observational
- Heterogeneous natural history of Addison's disease: mineralocorticoid deficiency may predominate.Endocrine connections · 2023Article
- Article
- B Cells and Autoantibodies in AIRE Deficiency.Biomedicines · 2021Review
- Latent Adrenal Insufficiency: From Concept to Diagnosis.Frontiers in endocrinology · 2021Review
- New Horizons: Novel Adrenal Regenerative Therapies.The Journal of clinical endocrinology and metabolism · 2020Article
- Natural History of Adrenal Steroidogenesis in Autoimmune Addison's Disease Following Diagnosis and Treatment.The Journal of clinical endocrinology and metabolism · 2020Article
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Authors and funding
13 authors at 7 institutions in 1 country.
Funding
Abstract
contextIn autoimmune Addison's disease (AAD), exogenous glucocorticoid (GC) therapy is an imperfect substitute for physiological GC secretion. Patients on long-term steroid replacement have increased morbidity, reduced life expectancy, and poorer quality of life.
objectiveThe objective of this article is to restore adrenocortical steroidogenic function in recent-onset AAD.
designAn open-label, multicenter trial of immunotherapy and trophic stimulation in new-onset AAD was conducted. Serial measurement of serum and urine corticosteroids at baseline and throughout a 72-week follow-up period was performed.
settingThis study was conducted at the. endocrine departments and clinical research facilities at 5 UK tertiary centers. PATIENTS: Thirteen participants (9 female, 4 male; age 19-64 years) were included with AAD confirmed by high adrenocorticotropin, low circulating cortisol (basal < 100 nmol/L or post-tetracosactide < 300 nmol/L), and positive serum 21-hydroxylase antibodies.
interventionAll participants received dual therapy with B-lymphocyte-depleting immunotherapy (rituximab 1 g given twice) and repeated depot tetracosactide (1 mg on alternate days for 12 weeks).
main outcome measureRestoration of normal GC secretion (stimulated cortisol > 550 nmol/L) at week 48 was the main outcome measure.
resultsTen of 13 (77%) participants had detectable stimulated serum cortisol (26-265 nmol/L) at trial entry. Following intervention, 7 of 13 (54%) had an increase in stimulated cortisol measurement, with a peak response of 325 nmol/L at week 18 in 1 participant. Increased steroid metabolites, assayed by urine gas chromatography-mass spectrometry at week 12 and week 48, was detected in 8 of 13 (62%) individuals, reflecting an increase in endogenous steroidogenesis. Four of 13 had residual adrenal function at 72 weeks.
conclusionCombined treatment with rituximab and depot tetracosactide did not restore normal adrenal function. Nevertheless, adrenocortical plasticity is demonstrated in some patients, and this has the potential to be exploited to improve adrenal function.
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