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- Title
Adrenal steroidogenesis after B lymphocyte depletion therapy in new-onset Addison's disease.
- Authors
Pearce SH; Mitchell AL; Bennett S; King P; Chandran S; Nag S; Chen S; Smith BR; Isaacs JD; Vaidya B; Pearce, Simon H S; Mitchell, Anna L; Bennett, Stuart; King, Phil; Chandran, Sukesh; Nag, Sath; Chen, Shu; Smith, Bernard Rees; Isaacs, John D; Vaidya, Bijay
- Abstract
<bold>Context: </bold>A diagnosis of Addison's disease means lifelong dependence on daily glucocorticoid and mineralocorticoid therapy and is associated with increased morbidity and mortality as well as a risk of unexpected adrenal crisis.<bold>Objective: </bold>The objective of the study was to determine whether immunomodulatory therapy at an early stage of autoimmune Addison's disease could lead to preservation or improvement in adrenal steroidogenesis.<bold>Design and Intervention: </bold>This was an open-label, pilot study of B lymphocyte depletion therapy in new-onset idiopathic primary adrenal failure. Doses of iv rituximab (1 g) were given on d 1 and 15, after pretreatment with 125 mg iv methylprednisolone.<bold>Patients and Main Outcome Measures: </bold>Six patients (aged 17-47 yr; four females) were treated within 4 wk of the first diagnosis of idiopathic primary adrenal failure. Dynamic testing of adrenal function was performed every 3 months for at least 12 months.<bold>Results: </bold>Serum cortisol levels declined rapidly and were less than 100 nmol/liter (3.6 μg/dl) in all patients by 3 months after B lymphocyte depletion. Serum cortisol and aldosterone concentrations remained low in five of the six patients throughout the follow-up period. However, a single patient had sustained improvement in both serum cortisol [peak 434 nmol/liter (15.7 μg/dl)] and aldosterone [peak 434 pmol/liter (15.7 ng/dl)] secretion. This patient was able to discontinue steroid medications 15 months after therapy and remains well, with improving serum cortisol levels 27 months after therapy.<bold>Conclusion: </bold>New-onset autoimmune Addison's disease should be considered as a potentially reversible condition in some patients. Future studies of immunomodulation in autoimmune Addison's disease may be warranted.
- Publication
Journal of Clinical Endocrinology & Metabolism, 2012, Vol 97, Issue 10, pE1927
- ISSN
0021-972X
- Publication type
journal article
- DOI
10.1210/jc.2012-1680