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Journal of Education, Health and Sport

Osilodrostat therapy in a 26-year-old patient after ineffective surgical treatment of Cushing Disease: a case report
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Osilodrostat therapy in a 26-year-old patient after ineffective surgical treatment of Cushing Disease: a case report

Authors

  • Magdalena Kamińska Medical University of Lublin https://orcid.org/0000-0002-7624-4146
  • Adam Strzoda Student Scientific Society at Department of Endocrinology, Diabetology and Metabolic Diseases, Medical University of Lublin, Kazimierza Jaczewskiego 8, 20-400 Lublin, Poland https://orcid.org/0000-0002-1928-2664
  • Agata Juda Student Scientific Society at Department of Endocrinology, Diabetology and Metabolic Diseases, Medical University of Lublin, Kazimierza Jaczewskiego 8, 20-400 Lublin, Poland https://orcid.org/0000-0003-3583-7305
  • Kornelia Rojek Student Scientific Society at Department of Endocrinology, Diabetology and Metabolic Diseases, Medical University of Lublin, Kazimierza Jaczewskiego 8, 20-400 Lublin, Poland https://orcid.org/0000-0002-5096-1235
  • Anna Strzoda Student Scientific Society at Department of Endocrinology, Diabetology and Metabolic Diseases, Medical University of Lublin, Kazimierza Jaczewskiego 8, 20-400 Lublin, Poland https://orcid.org/0000-0002-4839-3531
  • Wojciech Sowiński Student Scientific Society at Department of Endocrinology, Diabetology and Metabolic Diseases, Medical University of Lublin, Kazimierza Jaczewskiego 8, 20-400 Lublin, Poland https://orcid.org/0000-0002-2267-4773
  • Agnieszka Polak Department of Endocrinology, Diabetology and Metabolic Diseases, Medical University of Lublin, Kazimierza Jaczewskiego 8, 20-400 Lublin, Poland https://orcid.org/0000-0003-1694-4738

DOI:

https://doi.org/10.12775/JEHS.2023.13.01.030

Keywords

ACTH-dependent Cushing syndrome, Osilodrostat, Steroidogenesis Inhibitor

Abstract

Cushing’s Disease is caused by pituitary adenoma which produces adrenocorticotropic hormone causing hypercortisolemia. First-line treatment involves operative removal of the tumor, however, in some patients it proves ineffective. In those cases, pharmacological treatment is necessary as untreated Cushing’s Disease may be lethal. In 2020 a new steroidogenesis inhibitor – Osilodrostat - was approved by the European Medicines Agency and can be used in the treatment of patients who cannot undergo pituitary surgery or whose previous surgical treatment proved ineffective.

We present a case of a 26-year-old female patient who was diagnosed with endogenous Cushing’s Disease and underwent two consecutive operations to remove the tumor - both were ineffective. With deteriorating laboratory results and other pharmacological treatments exhausted Osilodrostat therapy was introduced.

Osilodrostat is becoming an important drug in patients with Cushing’s Disease refractory to other treatments.

References

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Groselj U, Sikonja J, Battelino T. Osilodrostat for Cushing disease and its role in pediatrics. Horm Res Paediatr. 2022 Jan 19. https://doi.org/10.1159/000522054

Castinetti F, Nieman LK, Reincke M, Newell-Price J. Approach to the Patient Treated with Steroidogenesis Inhibitors. J Clin Endocrinol Metab. 2021 Jun 16;106(7):2114-2123. https://doi.org/10.1210/clinem/dgab122

Li L, Vashisht K, Boisclair J, Li W, Lin TH, Schmid HA, et all. (2015). Osilodrostat (LCI699), a potent 11β-hydroxylase inhibitor, administered in combination with the multireceptor-targeted somatostatin analog pasireotide: A 13-week study in rats. Toxicology and Applied Pharmacology, 286(3), 224–233. https://doi.org/10.1016/j.taap.2015.05.004

Gadelha M, Bex M, Feelders RA, Heaney AP, Auchus RJ, Gilis-Januszewska A, et all. Randomized Trial of Osilodrostat for the Treatment of Cushing Disease. J Clin Endocrinol Metab. 2022 Jun 16;107(7):e2882-e2895. https://doi.org/10.1210/clinem/dgac178

Fleseriu M, Biller BMK. Treatment of Cushing's syndrome with osilodrostat: practical applications of recent studies with case examples. Pituitary. 2022 Aug 24:1–15. https://doi.org/10.1007/s11102-022-01268-2

Witek P, Mehlich A, Stasiewicz A, Jawiarczyk-Przybyłowska A, Bolanowski M. Osilodrostat - an emerging drug for the medical management of Cushing's disease. Endokrynol Pol. 2022;73(2):371-374. https://doi.org/10.5603/EP.a2022.0009

Amodru V, Brue T, Castinetti F. Synergistic cortisol suppression by ketoconazole-osilodrostat combination therapy. Endocrinol Diabetes Metab Case Rep. 2021 Dec 1;2021:21-0071. https://doi.org/10.1530/EDM-21-0071

Journal of Education, Health and Sport

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Published

2022-12-02

How to Cite

1.
KAMIŃSKA, Magdalena, STRZODA, Adam, JUDA, Agata, ROJEK, Kornelia, STRZODA, Anna, SOWIŃSKI, Wojciech & POLAK, Agnieszka. Osilodrostat therapy in a 26-year-old patient after ineffective surgical treatment of Cushing Disease: a case report. Journal of Education, Health and Sport [online]. 2 December 2022, T. 13, nr 1, s. 203–208. [accessed 21.3.2023]. DOI 10.12775/JEHS.2023.13.01.030.
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Vol. 13 No. 1 (2023)

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Copyright (c) 2022 Magdalena Kamińska, Adam Strzoda, Wojciech Sowiński, Agata Juda, Kornelia Rojek, Anna Strzoda, Agnieszka Polak

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This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License.

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