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IBD in Camouflage or Are There Similarities?: A Case of Cronkhite-Canada Syndrome

2018· article· en· W2921133619 on OpenAlexaboutno aff
Yilien Alonso, Kevin Cowley, Mona Bansal, Matthew C. Rice, Shamita B. Shah

Bibliographic record

VenueThe American Journal of Gastroenterology · 2018
Typearticle
Languageen
FieldMedicine
TopicGenetic factors in colorectal cancer
Canadian institutionsnot available
Fundersnot available
KeywordsMedicineColonoscopyEsophagogastroduodenoscopyInternal medicineMalabsorptionInfliximabDiarrheaGastroenterologyBloody diarrheaHypoalbuminemiaDermatologyHyperpigmentationInflammatory bowel diseasePrednisoneDiseaseEndoscopyCancerColorectal cancer

Abstract

fetched live from OpenAlex

First described in 1955, Cronkhite Canada Syndrome (CCS) is a rare non-familial polyposis syndrome with approximately 500 cases to date. With a five-year mortality of 50%, it is crucial to correctly and quickly identify the syndrome's primary features of alopecia, onychodystrophy, cutaneous hyperpigmentation, and intestinal malabsorption. Unfortunately, even when diagnosed, treatment is challenging as the etiology is unknown though possibly immune-mediated. Antibiotics, steroids, and immunosuppressants have all shown varying degrees of success. A 74-year-old Filipino woman with a history of hypothyroidism presented to our Inflammatory Bowel Disease (IBD) Clinic for a second opinion. Seven months prior, she experienced onset of watery, non-bloody diarrhea with initial diagnosis of ulcerative colitis. After this diagnosis, she responded to treatment with prednisone, but failed maintenance therapy with oral mesalamine. She then received two doses of infliximab without response and was referred to our IBD clinic. She reported continued watery, non-bloody diarrhea with significant weight loss. Physical exam revealed alopecia, onychodystrophy, and lower extremity edema with cutaneous hyperpigmentation. Her labs revealed hyponatremia, severe hypoalbuminemia, c-reactive protein (CRP) of 55.3 mg/L (0-8.2 mg/L), and a hemoglobin of 10.1 g/dL which prompted hospital admission.Inpatient EGD and colonoscopy revealed diffuse polyposis throughout the stomach, duodenum, and colon. Biopsies revealed diffuse inflammatory hyperplastic polyps with increased IgG4 positive cells (up to 22 per high power field). Based on this clinical picture, she was diagnosed with CCS. She was placed on full bowel rest and given total parenteral nutrition. She also received IV corticosteroids in conjunction with azathioprine and infliximab. We achieved good clinical response and her CRP normalized. CCS is a rare non-familial polyposis syndrome with characteristic physical exam findings, diffuse polyposis, debilitating diarrhea, and significant 5-year mortality. Treatment is challenging and focuses primarily on nutritional support and immunosuppressant medications. In this particular case we chose to proceed with low dose azathioprine and infliximab based on success of this regimen in past case reports published. We will plan to achieve steroid free remission by optimizing infliximab based on levels and providing nutritional support.2073_A Figure 1. Onychodystrophy2073_B Figure 2. Diffuse polyposis on colonoscopy2073_C Figure 3. Diffuse inflammatory hyperplastic polyps with increased IgG4 cells

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)0.003
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 score0.020
Threshold uncertainty score0.040

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.003
Meta-epidemiology (narrow)0.0020.002
Meta-epidemiology (broad)0.0020.001
Bibliometrics0.0040.004
Science and technology studies0.0040.003
Scholarly communication0.0020.002
Open science0.0020.003
Research integrity0.0070.004
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 head0.014
GPT teacher head0.268
Teacher spread0.254 · 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
Published2018
Admission routes1
Has abstractyes

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