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Metabolic & Endocrine · Clinical trials

Cushing's Disease Clinical Trials

Approved therapies, the pivotal-trial endpoints they were judged on, the magnitude of benefit — and the drugs that failed their endpoints, and why.

Indication overview

About cushing's disease — and why its trials are hard

Cushing's disease is chronic endogenous hypercortisolism caused by an ACTH-secreting pituitary adenoma, the most common cause of Cushing's syndrome. Untreated, cortisol excess drives central obesity, diabetes, hypertension, osteoporosis, myopathy, psychiatric disturbance and markedly increased mortality. Transsphenoidal surgery is first-line, but persistent or recurrent disease is common, creating a need for medical therapy. Options act at three levels: pituitary-directed pasireotide, adrenal steroidogenesis inhibitors (osilodrostat, levoketoconazole, and off-label ketoconazole and metyrapone), and the glucocorticoid-receptor antagonist mifepristone, which targets hyperglycemia rather than cortisol itself. Osilodrostat (Isturisa, 2020), an 11-beta-hydroxylase inhibitor validated in the LINC-3 randomized-withdrawal trial, and levoketoconazole (Recorlev, 2021) expanded the modern armamentarium, both normalizing mean urinary free cortisol in a substantial share of patients. Treatment is titrated to biochemical control while avoiding adrenal insufficiency; drug-specific risks include QT prolongation and hypocortisolism (osilodrostat), hepatotoxicity (levoketoconazole), hyperglycemia (pasireotide) and endometrial thickening (mifepristone). Long-term durable control and safety remain the central challenges.

Indication
Cushing's Disease
ICD-10-CM
E24.0 — Pituitary-dependent Cushing disease

Approved therapies & pivotal evidence

What's been approved — and by how much it moved the endpoint

Drug (brand)ApprovedSettingPivotal trialPrimary endpointMagnitude of benefit
Pasireotide (Signifor)2012Pituitary-directed somatostatin analog for patients not candidates for or not cured by surgeryPhase III B2305 (Colao et al., NEJM 2012)Normalization of mean urinary free cortisol (mUFC) at month 6 without dose increaseRoughly 15% (600 mcg) and 26% (900 mcg) achieved normal mUFC at month 6
Mifepristone (Korlym)2012Glucocorticoid-receptor antagonist for hyperglycemia/glucose intolerance in endogenous Cushing's syndromeSEISMICImprovement in oral glucose tolerance (AUC glucose) in the diabetes cohort60% of the diabetes/glucose-intolerance group were responders (≥25% reduction in glucose AUC)
Osilodrostat (Isturisa)202011-beta-hydroxylase inhibitor for Cushing's disease when surgery is not an option or has not workedLINC-3 (randomized withdrawal; Lancet Diabetes Endocrinol 2020)Proportion maintaining normal mUFC at end of the 8-week randomized withdrawal (week 34)86% on osilodrostat vs 29% on placebo maintained mUFC normalization
Levoketoconazole (Recorlev)2021Steroidogenesis inhibitor for endogenous hypercortisolemia in Cushing's syndromeSONICS (open-label) and LOGICS (randomized withdrawal)Normalization of mUFC without dose increase (SONICS) and maintenance vs withdrawal (LOGICS)SONICS: 31% normalized mUFC at end of maintenance; LOGICS confirmed loss of control on placebo withdrawal
Ketoconazole ((generic; Ketoconazole HRA in EU))off-label in US (EU approval 2014)Adrenal steroidogenesis inhibitor; widely used off-label in the USRetrospective/observational cohorts (no US pivotal RCT)Normalization of urinary/serum cortisol~50% achieve cortisol normalization in cohorts (unverified against a single pivotal trial)

Where trials have failed

Drugs that missed their endpoint — and what contributed

The most instructive lessons in cushing's disease development come from programmes that failed the endpoint that mattered.

Drug / trialEndpoint outcomeWhat contributed
Cabergoline (dopamine agonist) — Off-label investigational cohortsNo successful registrational pivotal program for Cushing's disease; a substantial proportion escape from initial control over timeLoss of response (escape phenomenon), variable efficacy, and absence of an adequately powered prospective pivotal trial (magnitude unverified)

Choosing the right endpoint

Primary endpoints that matter in cushing's disease trials

  • Mean urinary free cortisol (mUFC) — Primary regulatory endpoint; normalization reflects control of cortisol overproduction
  • Late-night salivary cortisol — Sensitive marker of disrupted diurnal cortisol rhythm used for monitoring
  • Randomized-withdrawal maintenance — Design (LINC-3, LOGICS) demonstrating durable drug effect vs placebo relapse
  • Cardiometabolic parameters — Blood pressure, weight, HbA1c/glucose as clinically meaningful secondary endpoints
  • Safety: adrenal insufficiency and QT — Hypocortisolism from over-suppression and QT prolongation (osilodrostat) are key safety endpoints

How iNGENū runs cushing's disease trials

Physician-led design, built for FDA submission

Endpoint & biomarker strategy

Board-certified specialists design endpoints and patient selection aligned to current FDA guidance for this indication.

FDA-ready data

Built to ICH-GCP and 21 CFR 312.120, with direct FDA submission — data accepted by the FDA, EMA and MHRA.

Faster, lower-cost delivery

~4-week ethics via the TGA CTN scheme, up to 43.5% R&D rebate, and 80–90% below US CRO cost.

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Frequently asked questions

Cushing's Disease clinical trials — FAQs

What is the difference between Cushing's disease and Cushing's syndrome?
Cushing's syndrome is any cause of chronic cortisol excess. Cushing's disease is the specific subtype caused by an ACTH-secreting pituitary adenoma, the most common endogenous cause.
How do the newer drugs differ?
Osilodrostat and levoketoconazole block adrenal cortisol synthesis, pasireotide targets the pituitary tumor, and mifepristone blocks the glucocorticoid receptor to treat hyperglycemia rather than lowering cortisol levels.
Is medication a cure?
No. Transsphenoidal surgery offers the best chance of cure. Medical therapy controls cortisol in persistent, recurrent, or inoperable disease and requires ongoing monitoring to avoid adrenal insufficiency.

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