Rapid control of severe ectopic Cushing’s syndrome by oral osilodrostat monotherapy

in European Journal of Endocrinology
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  • 1 Department of Endocrinology, Polyclinic of Saint Côme, Compiegne, France
  • 2 Department of Hormonology, Polyclinic of Saint Côme, Compiegne, France
  • 3 Department of Nuclear Medicine, Polyclinic of Saint Côme, Compiegne, France
  • 4 Medical ICU, APHP, Cochin Hospital, Paris, France
  • 5 Department of Nephrology, Polyclinic of Saint Côme, Compiegne, France
  • 6 INSERM Unité 1016, CNRS UMR 8104, Institut Cochin, Paris, France
  • 7 Université de Paris, Paris, France

Correspondence should be addressed to L Groussin; Email: lionel.groussin@aphp.fr
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     European Society of Endocrinology

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    Haissaguerre M, Puerto M, Nunes ML & Tabarin A Efficacy and tolerance of osilodrostat in patients with severe Cushing's syndrome due to non-pituitary cancers. European Journal of Endocrinology 2020 183 L7L9. (https://doi.org/10.1530/EJE-20-0557)

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    Yaneva M, Kalinov K & Zacharieva S Mortality in Cushings syndrome: data from 386 patients from a single tertiary referral center. European Journal of Endocrinology 2013 169 621627. (https://doi.org/10.1530/EJE-13-0320)

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    Pivonello R, Isidori AM, De Martino MC, Newell-Price J, Biller BM & Colao A Complications of Cushings syndrome: state of the art. Lancet: Diabetes and Endocrinology 2016 4 611629. (https://doi.org/10.1016/S2213-8587(1600086-3)

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    Young J, Haissaguerre M, Viera-Pinto O, Chabre O, Baudin E & Tabarin A Management of endocrine disease: Cushings syndrome due to ectopic ACTH secretion: an expert operational opinion. European Journal of Endocrinology 2020 182 R29R58. (https://doi.org/10.1530/EJE-19-0877)

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    Daniel E & Newell-Price JD Therapy of endocrine disease: steroidogenesis enzyme inhibitors in Cushings syndrome. European Journal of Endocrinology 2015 172 R263R280. (https://doi.org/10.1530/EJE-14-1014)

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    Constantinescu SM, Driessens N, Lefebvre A, Furnica RM, Corvilain B & Maiter D Etomidate infusion at low doses is an effective and safe treatment for severe Cushings syndrome outside intensive care. European Journal of Endocrinology 2020 183 161167. (https://doi.org/10.1530/EJE-20-0380)

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  • 7

    Donadille B, Groussin L, Waintrop C, Abbas H, Tenenbaum F, Dugue MA, Coste J, Bertagna X & Bertherat J Management of Cushings syndrome due to ectopic adrenocorticotropin secretion with 1,ortho-1, para'-dichloro-diphenyl-dichloro-ethane: findings in 23 patients from a single center. Journal of Clinical Endocrinology and Metabolism 2010 95 537544. (https://doi.org/10.1210/jc.2009-1317)

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    Zeiger MA, Fraker DL, Pass HI, Nieman LK, Cutler GB, Chrousos GP & Norton JA Effective reversibility of the signs and symptoms of hypercortisolism by bilateral adrenalectomy. Surgery 1993 114 11381143.

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    Pivonello R, Fleseriu M, Newell-Price J, Bertagna X, Findling J, Shimatsu A, Gu F, Auchus R, Leelawattana R & Lee EJ et al. Efficacy and safety of osilodrostat in patients with Cushings disease (LINC 3): a multicentre phase III study with a double-blind, randomised withdrawal phase. Lancet: Diabetes and Endocrinology 2020 8 748761. (https://doi.org/10.1016/S2213-8587(2030240-0)

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    Bertagna X, Pivonello R, Fleseriu M, Zhang Y, Robinson P, Taylor A, Watson CE, Maldonado M, Hamrahian AH & Boscaro M et al. LCI699, a potent 11beta-hydroxylase inhibitor, normalizes urinary cortisol in patients with Cushings disease: results from a multicenter, proof-of-concept study. Journal of Clinical Endocrinology and Metabolism 2014 99 13751383. (https://doi.org/10.1210/jc.2013-2117)

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    Tanaka T, Satoh F, Ujihara M, Midorikawa S, Kaneko T, Takeda T, Suzuki A, Sato M & Shimatsu A A multicenter, phase 2 study to evaluate the efficacy and safety of osilodrostat, a new 11beta-hydroxylase inhibitor, in Japanese patients with endogenous Cushings syndrome other than Cushings disease. Endocrine Journal 2020 67 841852. (https://doi.org/10.1507/endocrj.EJ19-0617)

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    Kamenicky P, Droumaguet C, Salenave S, Blanchard A, Jublanc C, Gautier JF, Brailly-Tabard S, Leboulleux S, Schlumberger M & Baudin E et al. Mitotane, metyrapone, and ketoconazole combination therapy as an alternative to rescue adrenalectomy for severe ACTH-dependent Cushings syndrome. Journal of Clinical Endocrinology and Metabolism 2011 96 27962804. (https://doi.org/10.1210/jc.2011-0536)

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