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S1714 Cronkhite-Canada Syndrome: A Case Report in a Young Female

2020· article· en· W3093704227 on OpenAlexaboutno aff
Victoria Poplaski, Xi Zeng, Rama Yerramilli, Zhouwen Tang, Sarah E. Blutt, Heather A. Danhof, Antone R. Opekun, Noah F. Shroyer, Robert A. Britton, Mary K. Estes

Bibliographic record

VenueThe American Journal of Gastroenterology · 2020
Typearticle
Languageen
FieldMedicine
TopicGenetic factors in colorectal cancer
Canadian institutionsnot available
Fundersnot available
KeywordsMedicineDuodenumGastroenterologyInternal medicineHypoalbuminemiaColonoscopyEnteropathyAnemiaAbdomenStomachHypoproteinemiaPathologyDermatologyDiseaseSurgeryCancerColorectal cancer

Abstract

fetched live from OpenAlex

INTRODUCTION: Cronkhite Canada Syndrome (CCS) is a rare polyposis syndrome for which the cause remains unknown. Because the disease is so rare (1 in 1 million), no standard of care exists. The disease is most prevalent in males and has an average age of onset at 60 years of age. The case presented is unique as the patient is female and diagnosed at a younger age (38). CASE DESCRIPTION/METHODS: A 38-year-old woman of Asian (Laotian) descent presented in the gastroenterology clinic with fatigue, dizziness, alopecia, dysgeusia, and weight loss. Physical exam revealed onychodystrophy and hyperpigmentation of her face and hands. Laboratory testing confirmed iron deficiency anemia with hypoproteinemia and hypoalbuminemia. Anti-gliadin antibodies were negative. A computed tomography scan of her abdomen revealed no significant abnormalities. Endoscopy revealed dozens of erythematous pedunculated polyps in the stomach, duodenum, and ileum while the colon had close to a dozen large pedunculated polyps. Pathology noted no adenomas or dysplasias. However, pathology did note edema, chronic and acute inflammation and prominent eosinophil content. A diagnosis of CCS was made. In order to assess for intestinal microbial changes in the patient, a stool sample was obtained to perform total 16S sequencing of the stool flora. The 16S data is being compared to healthy sex and aged-matched controls at the species level. Additionally, human intestinal organoids were generated from her stomach, duodenum, ileum, and colon biopsies using published methods. To our knowledge, this is the first time that intestinal organoids have been made from a CCS patient. The CCS organoids grow faster, are larger, and have unique morphologies compared to organoids from other healthy donors. Cell type analysis also reveals an increased number of ChgA positive enteroendocrine cells in the colon and ileum. DISCUSSION: Despite over 50 years of CCS cases, the etiopathogenesis and optimal treatment for CCS remains unknown. Currently the patient is maintained on azathioprine (dosing is 50 mg daily) and anti-histamines. Utilization of ex vivo organoid models will be an important tool for not only understanding more about the disease, but also as a way to advance precision medicine and evaluate potential therapies for this rare disease.

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 score0.023
Threshold uncertainty score0.045

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0020.001
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0040.002
Science and technology studies0.0030.001
Scholarly communication0.0010.001
Open science0.0010.001
Research integrity0.0050.002
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 head0.014
GPT teacher head0.255
Teacher spread0.242 · 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
Published2020
Admission routes1
Has abstractyes

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