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Record W3157867159 · doi:10.1210/jendso/bvab048.2060

The Diagnosis and Management of a Rare ACTH-Producing Neuroendocrine Tumor Causing Cushing’s Syndrome

2021· article· en· W3157867159 on OpenAlexaff
Ryan Findlay, Daryl Gray, Jessica Shepherd, Mariamma Joseph, Nadeem Hussain, David Laidley, Stan Van Uum

Bibliographic record

VenueJournal of the Endocrine Society · 2021
Typearticle
Languageen
FieldMedicine
TopicPituitary Gland Disorders and Treatments
Canadian institutionsSt Joseph's Health CareWestern University
Fundersnot available
KeywordsMedicineEndocrinologyInternal medicineMetyraponeDexamethasoneGastroenterologyRadiology

Abstract

fetched live from OpenAlex

Abstract A 57-year-old woman presented with a three-month history of worsening lethargy, weight gain, central adiposity, proximal muscle weakness, and hypokalemic hypertension. Initial investigations were consistent with ACTH-dependent Cushing’s Syndrome. 24-hour urine cortisol: 711nmol/day (normal<275), cortisol after 1 mg dexamethasone suppression test 960nmol/L (normal<50)), 2-day dexamethasone suppression cortisol: 575 and ACTH: 19 pmol/L. MRI sella was normal. She required several hospital admissions for worsening proximal muscle weakness and falls. She was started on ketoconazole and spironolactone. Basal insulin was started for dysglycemia. Petrosal vein sampling indicated ectopic ACTH production. CT scan of chest/abdomen/pelvis showed no intrathoracic or intra-abdominal abnormality. Thyroid ultrasound and whole-body octreotide scan showed no abnormalities. A Gallium 68-dotatate PET scan showed focal tracer uptake in the pancreatic tail suspicious for a well-differentiated neuroendocrine tumor (NET). MRI pancreas showed a slightly truncated pancreatic tail with no discrete lesion. We proceeded with Endoscopic Ultrasound (EUS) which revealed an irregular hypoechoic mass in the pancreatic tail. EUS–guided fine-needle biopsy (EUS-FNB) using a 22G SharkCore needle, revealed a neuroendocrine neoplasm staining positively for ACTH. She underwent distal pancreatectomy with pathology showing a 1.4cm well-differentiated NET, staining positive for ACTH with a Ki67 index of 10%. Postoperatively, her ketoconazole, spironolactone, and insulin were discontinued. Her dysglycemia, hypertension and other clinical symptoms gradually improved. Immediately post-operatively, her AM cortisol was 403 and her 24-hour urine cortisol was elevated 959nmol/day. Repeat AM cortisol and 24-hour urine cortisol one-month post-surgery were 392 nmol/L and 72 nmol/day respectively, confirming biochemical remission. Discussion: ACTH-producing pancreatic NETs are rare, accounting for less than 15% of reported cases of EAS. They are often aggressive, with 78.7% of reported cases having distant metastasis at diagnosis (1). They are associated with significant morbidity and mortality. Diagnosis and management can be difficult and require multiple investigations and multidisciplinary team involvement. In our case, we utilized endoscopic ultrasound to ensure a correct preoperative localization. We discuss the systematic approach for diagnosis and the management of an ACTH-producing pancreatic NET. References: 1. Maragliano R, Vanoli A, Albarello L, Milione M, Basturk O, et al. ACTH-secreting pancreatic neoplasms associated with Cushing syndrome: clinicopathologic study of 11 cases and review of the literature. Am J Surg Pathol. 2015 Mar;39(3):374-82. doi: 10.1097/PAS.0000000000000340. PMID: 25353285.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.038
Threshold uncertainty score0.274

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.019
GPT teacher head0.274
Teacher spread0.255 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations0
Published2021
Admission routes1
Has abstractyes

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