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Record W4397040394 · doi:10.1681/asn.20233411s1603b

A Case of Cronkhite-Canada Syndrome-Associated Membranous Glomerulonephritis: Can Lightning Strike Thrice?

2023· article· en· W4397040394 on OpenAlexaboutno aff
Benjamin J. Strimaitis, Shaun P. Chandler, Leo Francis, Rachna V. Pagnis, Dwarakanathan Ranganathan, Eoin D. O Sullivan

Bibliographic record

VenueJournal of the American Society of Nephrology · 2023
Typearticle
Languageen
FieldMedicine
TopicGenetic factors in colorectal cancer
Canadian institutionsnot available
Fundersnot available
KeywordsLightning strikeGlomerulonephritisMedicineLightning (connector)DermatologyInternal medicineGeographyKidneyMeteorologyPhysicsPower (physics)

Abstract

fetched live from OpenAlex

Introduction: Cronkhite-Canada Syndrome (CSS) is a rare disorder with less than 500 reported cases, characterized by extensive gastrointestinal polyposis and ectodermal abnormalities. It has been rarely associated with membranous nephropathy (MN). Of the 5 cases of MN associated with CSS in the literature, 3 have been in the context of malignancy, and none have tested PLA2R or thsd7a antibodies. Case Description: A 60-year-old female of Aboriginal heritage presented for evaluation of proteinuria with an Albumin Creatinine ratio (ACR) of 123 mg/mmol and Protein Creatinine ratio (PCR) of 108 mg/mmol on the background of CCS. Her renal function was reduced with a creatinine-based eGFR of 58mL/min/1.73m2 and a blood pressure of 128/78mmHg. She had a history of bilateral hearing loss since her teens requiring hearing aids but no visual impairment. Her brother had been diagnosed with thin basement membrane disease (TBM) and a maternal aunt with a similar presentation. Serological tests and ultrasonography were unremarkable, and a kidney biopsy demonstrated a classical appearance of MN - which was confirmed to be PLA2R and Thsd7a negative. Electron microscopy confirmed TBM with a mean basement thickness of 180nm. No malignancy suggested by history or examination, nor full body CT imaging or colonoscopy. She was treated with ACE inhibition and proteinuria improved to an ACR of 58 mg/mmoL and PCR of 73 mg/mmoL by six months. Discussion: This case is the first time that contemporary MN associated antibodies have been tested for in CCS, and suggests MN associated with CCS is not caused by PLA2R and thsd7a antibodies as previously suggested. Hiccum's Dictum is the counter to Occam's Razor, which argues a patient may have as many rare diagnoses as they please. This patient was one of 500 reported cases of CCS, one of 5 reported cases of associated MN and is the only reported case with concurrent active TBM disease. This case highlights the careful consideration which should be give to pre-test probabilities when deciding whether to pursue kidney biopsy (isolated TBM was more likely the diagnosis at initial presentation given the confirmed family history) while acknowledging that lightning can indeed strike thrice.

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.002
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.023
Threshold uncertainty score0.045

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0040.003
Science and technology studies0.0040.002
Scholarly communication0.0020.002
Open science0.0020.002
Research integrity0.0060.004
Insufficient payload (model declined to judge)0.0030.001

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 head0.017
GPT teacher head0.261
Teacher spread0.245 · 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 source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designCase report
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
Published2023
Admission routes1
Has abstractyes

Explore more

Same venueJournal of the American Society of Nephrology→Same topicGenetic factors in colorectal cancer→French-language works237,207→