Evidence map›Paper›PMID 42491645›Full record

ArticleAACE endocrinology and diabetes

Persistent Adrenocortical Insufficiency After Long-Term Metyrapone Treatment for Cushing's Disease.

Satoshi Yamagata, Tomohiro Kawaguchi, Hannah M Nakamura, Takefumi Mori, Shojiro Sawada, Kazunori Kageyama

Abstract readCase Reports
In one paragraph

Article in AACE endocrinology and diabetes. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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0cells of the map it votes in
0citing papers in PubMed
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1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

6 authors.

Satoshi YamagataDivision of Diabetes, Metabolism and Endocrinology, Faculty of Medicine, Tohoku Medical and Pharmaceutical University, Sendai, Miyagi, Japan.
Tomohiro KawaguchiKohnan Hospital, Department of Neurosurgery, Sendai, Miyagi, Japan.
Hannah M NakamuraDivision of Diabetes, Metabolism and Endocrinology, Faculty of Medicine, Tohoku Medical and Pharmaceutical University, Sendai, Miyagi, Japan.
Takefumi MoriDivision of Nephrology and Hypertension, Faculty of Medicine, Tohoku Medical and Pharmaceutical University, Sendai, Miyagi, Japan.
Shojiro SawadaDivision of Diabetes, Metabolism and Endocrinology, Faculty of Medicine, Tohoku Medical and Pharmaceutical University, Sendai, Miyagi, Japan.
Kazunori KageyamaDivision of Diabetes, Metabolism and Endocrinology, Faculty of Medicine, Tohoku Medical and Pharmaceutical University, Sendai, Miyagi, Japan.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Introduction: Cushing's disease is caused by pituitary tumors that secrete excess adrenocorticotropic hormone (ACTH). Metyrapone is widely used for medical management as a bridge to transsphenoidal pituitary surgery. Case Report: An 80-year-old man presented with severe hypercortisolemia (101 μg/dL [reference range, 5-23 μg/dL]) and elevated plasma ACTH, 228 pg/mL [reference range, 7.2-63.3 pg/mL]). Initial high-dose dexamethasone suppression and corticotropin-releasing hormone (CRH) tests suggested ectopic ACTH syndrome; however, extensive imaging failed to identify a source. Metyrapone was initiated and escalated to 3000 mg/d, followed by block-and-replacement with hydrocortisone (20 mg/d). After 4 years, repeat CRH testing and dexamethasone suppression supported Cushing's disease. Combined cavernous and inferior petrosal sinus sampling localized ACTH hypersecretion to the right pituitary. Transsphenoidal surgery confirmed a corticotroph pituitary neuroendocrine tumor. Despite tumor resection and metyrapone discontinuation, cortisol levels remained undetectable for more than 1 year. Discussion: Liquid chromatography-tandem mass spectrometry demonstrated markedly reduced mineralocorticoids, glucocorticoids, and adrenal androgens, indicating global adrenocortical dysfunction. Adrenal androgens are typically increased during metyrapone therapy, suggesting broader effects of long-term metyrapone on adrenal steroidogenesis beyond selective 11β-hydroxylase inhibition. Although postoperative glucocorticoid supplementation may have contributed to adrenal suppression, preserved ACTH responsiveness to CRH supports intact hypothalamic-pituitary signaling with impaired adrenal recovery. Conclusion: This case suggests that prolonged global adrenocortical dysfunction persisting over 1 year after metyrapone discontinuation. Clinicians should be aware of this potential complication and ensure appropriate glucocorticoid replacement and careful long-term monitoring.

Indexed as

adrenal insufficiencyCushing’s diseasemetyrapone

Identifiers

PMID42491645
PMCPMC13377877

What Socratic holds

Textmetadata
LicenceCC BY-NC-ND
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the Socratic graph.