MétaCan
Menu
Back to cohort
Record W7093345119 · doi:10.1210/jendso/bvaf149.336

SAT-470 Histological Fingerprint of Bilateral Macronodular Adrenocortical Disease (BMAD) Associated with ARMC5

2025· article· en· W7093345119 on OpenAlexaff

Bibliographic record

VenueJournal of the Endocrine Society · 2025
Typearticle
Languageen
FieldMedicine
TopicAdrenal and Paraganglionic Tumors
Canadian institutionsWiLAN (Canada)
Fundersnot available
KeywordsGermlineDiseaseGermline mutationContext (archaeology)Adrenocortical carcinomaHyperplasiaGenetic variantsMutation

Abstract

fetched live from OpenAlex

Abstract Disclosure: H.L. Charchar: None. F. Ledesma: None. A.W. Kuhn: None. B.M. Mariani: None. M.Y. Nishi: None. M.Q. Almeida: None. V. Victor Srougi: None. F.Y. Tanno: None. J. Chambô: None. A. Latronico: None. M.A. Pereira: None. B.B. Mendonca: None. M.B. Fragoso: None. Introduction: Primary macronodular adrenal hyperplasia (PMAH) is a rare cause of Cushing’s syndrome. Recent discoveries of clonal molecular alterations have highlighted the neoplastic nature of the disease, leading to the proposed term bilateral macronodular adrenocortical disease (BMAD). BMAD is characterized by the presence of functional adrenal macronodules with variable cortisol secretion. It can occur in association with genetic syndromes or present in isolated forms, such as familial or apparently sporadic cases. Since 2013, mutations in the ARMC5 gene have been recognized as a major genetic cause of BMAD. Objective: This study aims to investigate the histological features that enhance our understanding of patients with BMAD. Methods: A total of 19 patients who underwent surgery for BMAD at HCFMUSP between 2006 and 2021 were included in this study, all of whom were sequenced for ARMC5 germline pathogenic variants. Macroscopic photographs and H&E-stained slides were reviewed by a pathologist (FL), who was blinded to the clinical and genetic data. Results: Twelve patients (63.2%) carried germline pathogenic variants in ARMC5. Among them, three were apparently sporadic cases, while nine were familial cases, comprising six unrelated index cases and three relatives. A total of 11 distinct ARMC5 germline variants were identified, including 8 previously reported (p.Arg654; p.Leu365Pro; Ile58Asnfs45; p.His808Pro; p.Pro662Hist; p.Leu318Val; p.Gly323Asp) and 3 novel variants (p.Leu900Serfs12; p.Leu131Phefs7; p.Ser94Valfs8). In addition, nine different somatic alterations were described (p.Glu442Glyfs19; p.Arg366Leufs7; p.Leu526Alafs9; p.Cys657Trp; p.Leu685del; p.Ala854_Val855del; p.Ala83Argfs51; p.Pro695Serfs20; p.Arg271Glyfs7). Patients carrying ARMC5 germline pathogenic variants (Group 1) exhibited distinct microscopic features, including extensive areas of extracapsular extravasation; trabecular and pseudoglandular patterns; reverse polarity with nuclei positioned away from the basement membrane; and absence of adjacent adrenal parenchyma. In contrast, the seven patients without ARMC5 germline pathogenic variants (Group 2; 36.8%) displayed a slight predominance of clear cells interspersed with compact cells, occasionally showing moderate cytoplasm and lightly eosinophilic nodular outlines. Other findings included areas of adipose metaplasia and frequent lymphoid aggregates, which were sometimes confluent. Conclusion: This study presents a cohort of index and relative patients, proposing a histological fingerprint classification for BMAD associated with ARMC5 germline pathogenic variants. Remarkably, blinded pathologists accurately identified patients with ARMC5 germline pathogenic variants with 100% accuracy, highlighting the distinct histological features linked to this genetic alteration. Presentation: Saturday, July 12, 2025

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 score0.071
Threshold uncertainty score0.326

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.001
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
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 head0.008
GPT teacher head0.253
Teacher spread0.244 · 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
Published2025
Admission routes1
Has abstractyes

Explore more

Same venueJournal of the Endocrine SocietySame topicAdrenal and Paraganglionic TumorsFrench-language works237,207