Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and Adrenalectomy

Malik, Risha B.; Ben-Shlomo, Anat

doi:10.1016/j.aace.2022.10.001

Cited by 4 publications

(5 citation statements)

References 10 publications

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“…Malik and Ben-Shlomo presented a case of CPA treated with osilodrostat, with an immediate decrease in cortisol level at 4 mg/day and adrenal insufficiency symptoms after dose escalation to 8 mg/day. 15 Similar to our two cases, their patient was a middle-aged female with normal results of all other adrenal parameters, such as renin, angiotensin, or metanephrine levels. However, a CT scan was not performed (or presented), while magnetic resonance imaging revealed an indeterminate adrenal gland mass without a typical contrast phase/out-of-phase dropout for adenoma.…”

Section: Dovepresssupporting

confidence: 80%

“…However, a CT scan was not performed (or presented), while magnetic resonance imaging revealed an indeterminate adrenal gland mass without a typical contrast phase/out-of-phase dropout for adenoma. 15 Therefore, different morphology of cortisol-secreting adrenal tumor can potentially be considered a reason of the different response to treatment. Tanaka et al performed a multicenter study on the efficacy and safety of osilodrostat in Japanese patients with non-CD Cushing's syndrome.…”

Section: Dovepressmentioning

confidence: 99%

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Response to Osilodrostat Therapy in Adrenal Cushing’s Syndrome

Stasiak,

Witek,

Adamska-Fita

et al. 2024

DHPS

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Cushing's disease (CD) is the most common cause of endogenous hypercortisolism. Osilodrostat was demonstrated to be efficient in treating CD, and the mean average dose required for CD control was <11 mg/day. Potential differences in osilodrostat treatment between cortisol-producing adenoma (CPA) and CD have not been reported. The aim of this study was to present two patients with CPA in whom significant differences in the response to therapy compared to CD were found. We demonstrated a case of inverse response of cortisol levels with adrenal tumor progression during the initial dose escalation (Case 1). Simultaneously, severe exaggeration of hypercortisolism symptoms and life-threatening hypokalemia occurred. A further rapid dose increase resulted in the first noticeable cortisol response at a dose of 20 mg/day, and a full response at a dose of 45 mg/day. We also present a case that was initially resistant to therapy (Case 2). The doses required to achieve the first response and the full response were the same as those for Case 1. Our study demonstrated that osilodrostat therapy in patients with CPA may require a different approach than that in CD, with higher doses, faster dose escalation, and a possible initial inverse response or lack of response.

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Section: Dovepresssupporting

confidence: 80%

Section: Dovepressmentioning

confidence: 99%

Response to Osilodrostat Therapy in Adrenal Cushing’s Syndrome

Stasiak,

Witek,

Adamska-Fita

et al. 2024

DHPS

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show abstract

“…As aforementioned, there is a risk of adrenal insufficiency, which can be managed with temporary drug withdrawal and supplemental hydrocortisone. 35 Studies so far have not been performed in patients older than 70 years. As a corollary, it is unclear whether the safety profile and drug efficacy remain the same in older patients.…”

Section: Special Populationsmentioning

confidence: 99%

“…As aforementioned, there is a risk of adrenal insufficiency, which can be managed with temporary drug withdrawal and supplemental hydrocortisone. 35 …”

Section: Special Populationsmentioning

confidence: 99%

Clinical Utility of Osilodrostat in Cushing’s Disease: Review of Currently Available Literature

Perosevic¹,

Tritos²

2023

DDDT

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Cushing's disease (CD) is caused by endogenous hypercortisolism as a result of adrenocorticotropin (ACTH) secretion from a pituitary tumor. The condition is associated with multiple comorbidities and increased mortality. First-line therapy for CD is pituitary surgery, performed by an experienced pituitary neurosurgeon. Hypercortisolism may often persist or recur after initial surgery. Patients with persistent or recurrent CD will generally benefit from medical therapy, often administered to patients who underwent radiation therapy to the sella and are awaiting its salutary effects. There are three groups of medications directed against CD, including pituitary-targeted medications that inhibit ACTH secretion from tumorous corticotroph cells, adrenally-directed medications that inhibit adrenal steroidogenesis and a glucocorticoid receptor (GR) antagonist. The focus of this review is osilodrostat, a steroidogenesis inhibitor. Osilodrostat (LCI699) was initially developed to lower serum aldosterone levels and control hypertension. However, it was soon realized that osilodrostat also inhibits 11-beta hydroxylase (CYP11B1), leading to a reduction in serum cortisol levels. The focus of drug development then shifted from treatment of hypertension to treatment of hypercortisolism in CD. In a series of studies (LINC 1 through 4), osilodrostat was shown to be effective in normalizing 24-h urinary free cortisol (UFC) in the majority of treated patients and was approved for patients with CD who have failed surgery or are not surgical candidates. Further study is needed to examine the role of combination therapy as well as long-term outcomes of treated patients. Osilodrostat was shown to have an overall good safety profile. Most common adverse effects include nausea, headache, fatigue, arthralgias, dizziness, prolonged QT c interval, hypokalemia. In females, the drug can cause hirsutism and acne. Osilodrostat is administered twice daily, making it a good choice for patients with difficulty adhering to more complex regimens. Osilodrostat has an important, albeit adjunctive, role in the management of patients with CD.

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“… 6 Another case included in this issue described the off-label use of osilodrostat , currently FDA-approved for Cushing disease treatment, to reduce cortisol level and disease severity prior to planned adrenalectomy in Cushing syndrome due to adrenal origin. 7 In addition, a case with nonclassic congenital adrenal hyperplasia (NCCAH) presented in a patient with CYP21A2 and CYP11B1 heterozygous mutations, suggesting dual heterozygous mutations and adding to the database of described mutations in NCCAH. 8 Lastly, under image vignettes, a case of growth hormone producing pituitary adenoma was described.…”

Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and Adrenalectomy

Cited by 4 publications

References 10 publications

Response to Osilodrostat Therapy in Adrenal Cushing’s Syndrome

Response to Osilodrostat Therapy in Adrenal Cushing’s Syndrome

Clinical Utility of Osilodrostat in Cushing’s Disease: Review of Currently Available Literature

Editorial for November/December Issue of AACE Clinical Case Reports

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