Julia Schulz1, Kathrin R Frey1, Mark S Cooper1, Kathrin Zopf1, Manfred Ventz1, Sven Diederich1, Marcus Quinkler2. 1. Department of Clinical EndocrinologyCharité Campus Mitte, Charité University Medicine Berlin, Berlin, GermanyEndocrine and Diabetes UnitDepartment of Medicine I, University Hospital, University of Würzburg, Würzburg, GermanyAdrenal Steroid GroupANZAC Research Institute, Concord Repatriation General Hospital, Hospital Road, Concord Hospital, Concord, New South Wales 2139, AustraliaEndokrinologikumBerlin, GermanyEndocrinology in CharlottenburgStuttgarter Platz 1, 10627 Berlin, Germany. 2. Department of Clinical EndocrinologyCharité Campus Mitte, Charité University Medicine Berlin, Berlin, GermanyEndocrine and Diabetes UnitDepartment of Medicine I, University Hospital, University of Würzburg, Würzburg, GermanyAdrenal Steroid GroupANZAC Research Institute, Concord Repatriation General Hospital, Hospital Road, Concord Hospital, Concord, New South Wales 2139, AustraliaEndokrinologikumBerlin, GermanyEndocrinology in CharlottenburgStuttgarter Platz 1, 10627 Berlin, Germany Department of Clinical EndocrinologyCharité Campus Mitte, Charité University Medicine Berlin, Berlin, GermanyEndocrine and Diabetes UnitDepartment of Medicine I, University Hospital, University of Würzburg, Würzburg, GermanyAdrenal Steroid GroupANZAC Research Institute, Concord Repatriation General Hospital, Hospital Road, Concord Hospital, Concord, New South Wales 2139, AustraliaEndokrinologikumBerlin, GermanyEndocrinology in CharlottenburgStuttgarter Platz 1, 10627 Berlin, Germany marcus.quinkler@t-online.de.
Abstract
OBJECTIVE: Individuals with primary adrenal insufficiency (PAI) or congenital adrenal hyperplasia (CAH) receive life-long glucocorticoid (GC) replacement therapy. Current daily GC doses are still higher than the reported adrenal cortisol production rate. This GC excess could result in long-term morbidities such as osteoporosis. No prospective trials have investigated the long-term effect of GC dose changes in PAI and CAH patients. METHODS: This is a prospective and longitudinal study including 57 subjects with PAI (42 women) and 33 with CAH (21 women). Bone mineral density (BMD) was measured by dual energy X-ray absorptiometry at baseline and after 2 years. Subjects were divided into three groups (similar baseline characteristics) depending on changes in daily hydrocortisone equivalent dose (group 1: unchanged 25.2±8.2 mg (mean±S.D., n=50); group 2: increased 18.7±10.3 to 25.9±12.0 mg (n=13); group 3: decreased 30.8±8.5 to 21.4±7.2 mg (n=27)). RESULTS: Subjects in group 1 showed normal lumbar and femoral Z-scores which were unchanged over time. Group 2 subjects showed a significant decrease in femoral neck Z-scores over time (-0.15±1.1 to -0.37±1.0 (P<0.05)), whereas group 3 subjects showed a significant increase in lumbar spine and hip Z-scores (L1-L4: -0.93±1.2 to -0.65±1.5 (P<0.05); total hip: -0.40±1.0 to -0.28±1.0 (P<0.05)). No changes in BMI over time were seen within any group. Reduction in GC dose did not increase the risk of adrenal crisis. CONCLUSION: This study demonstrates for the first time that cautious reduction in hydrocortisone equivalent doses leads to increases in BMD, whereas dose increments reduced BMD. These data emphasize the need for the lowest possible GC replacement dose in AI patients to maintain health and avoid long-term adverse effects.
OBJECTIVE: Individuals with primary adrenal insufficiency (PAI) or congenital adrenal hyperplasia (CAH) receive life-long glucocorticoid (GC) replacement therapy. Current daily GC doses are still higher than the reported adrenal cortisol production rate. This GC excess could result in long-term morbidities such as osteoporosis. No prospective trials have investigated the long-term effect of GC dose changes in PAI and CAH patients. METHODS: This is a prospective and longitudinal study including 57 subjects with PAI (42 women) and 33 with CAH (21 women). Bone mineral density (BMD) was measured by dual energy X-ray absorptiometry at baseline and after 2 years. Subjects were divided into three groups (similar baseline characteristics) depending on changes in daily hydrocortisone equivalent dose (group 1: unchanged 25.2±8.2 mg (mean±S.D., n=50); group 2: increased 18.7±10.3 to 25.9±12.0 mg (n=13); group 3: decreased 30.8±8.5 to 21.4±7.2 mg (n=27)). RESULTS: Subjects in group 1 showed normal lumbar and femoral Z-scores which were unchanged over time. Group 2 subjects showed a significant decrease in femoral neck Z-scores over time (-0.15±1.1 to -0.37±1.0 (P<0.05)), whereas group 3 subjects showed a significant increase in lumbar spine and hip Z-scores (L1-L4: -0.93±1.2 to -0.65±1.5 (P<0.05); total hip: -0.40±1.0 to -0.28±1.0 (P<0.05)). No changes in BMI over time were seen within any group. Reduction in GC dose did not increase the risk of adrenal crisis. CONCLUSION: This study demonstrates for the first time that cautious reduction in hydrocortisone equivalent doses leads to increases in BMD, whereas dose increments reduced BMD. These data emphasize the need for the lowest possible GC replacement dose in AI patients to maintain health and avoid long-term adverse effects.
Authors: Juliano Henrique Borges; Daniel Minutti de Oliveira; Sofia Helena Valente de Lemos-Marini; Bruno Geloneze; Gil Guerra-Júnior; Ezequiel Moreira Gonçalves Journal: Osteoporos Int Date: 2021-08-18 Impact factor: 4.507