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S4986 Cronkhite-Canada Syndrome: An Inflammatory Bowel Disease Mimic

2025· article· en· W7110930955 on OpenAlexaboutno aff

Bibliographic record

VenueThe American Journal of Gastroenterology · 2025
Typearticle
Languageen
FieldMedicine
TopicGenetic factors in colorectal cancer
Canadian institutionsnot available
Fundersnot available
KeywordsEsophagogastroduodenoscopyColonoscopyInflammatory bowel diseaseMucocutaneous zoneSigmoid colonEtiologyRectumAzathioprine

Abstract

fetched live from OpenAlex

Introduction: Cronkhite-Canada syndrome (CCS) is an extremely rare, non-hereditary polyposis syndrome, characterized by diffuse gastrointestinal hamartomatous polyps, along with ectodermal abnormalities such as alopecia, onychodystrophy, and skin hyperpigmentation. There are fewer than 500 cases reported worldwide since it was first described in 1955. We present a case of CCS initially diagnosed as Crohn’s disease highlighting its unique clinical and endoscopic features. Case Description/Methods: A 60-year-old African American woman presented with a 2-year history of watery diarrhea, abdominal pain, as well as progressive weight loss and malnutrition requiring enteral feeding. She was diagnosed as having Sjogren’s syndrome as well as Crohn’s disease and was treated with adalimumab and azathioprine following evaluation by multiple specialists but failed to improve and subsequently sought a second opinion at our institution. Her symptoms were also associated with alopecia, nail loosening, and hyperpigmentation. Laboratory markers and stool studies were unremarkable except for marked eosinophilia. Esophagogastroduodenoscopy revealed a normal esophagus, and markedly edematous, erythematous, and nodular gastric mucosa, most prominent in the body and antrum, along with diffuse nodular mucosa throughout the duodenum. Sigmoidoscopy showed similar findings in the rectum and sigmoid colon. Gastric, duodenal, and colonic biopsies confirmed the presence of hamartomatous polyps. Discussion: CCS is most commonly reported in middle-aged adults. Its etiology remains unclear, though an autoimmune component has been suggested given its association with other autoimmune conditions and increased levels of IgG-4 positive plasma cells in polyps. Recent transcriptomic analyses of CCS polyps have also shown upregulation of innate immune pathways. Early recognition is critical, as delayed diagnosis can lead to significant nutritional deficiencies and increased gastrointestinal malignancy risk and morbidity. Treatment entails nutritional support as well as corticosteroids, with gradual tapering based on clinical response, and steroid-spearing agents like azathioprine. Most patients experience improvement in both gastrointestinal symptoms and ectodermal abnormalities with treatment. Our case underscores the importance of considering CCS in patients presenting with gastrointestinal symptoms suspicious for Crohn’s disease. The presence of typical dermatologic features can lead to an earlier diagnosis.

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)0.001
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 score0.008
Threshold uncertainty score0.016

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0020.002
Science and technology studies0.0010.001
Scholarly communication0.0010.001
Open science0.0010.001
Research integrity0.0020.001
Insufficient payload (model declined to judge)0.0040.001

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 head0.005
GPT teacher head0.245
Teacher spread0.239 · 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
Published2025
Admission routes1
Has abstractyes

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