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Cis-Regulation of the CFTR Gene in Pancreatic Cells.

Int J Mol Sci · 2025
L1 50/100 3/4
Why this verdict

The main results reproduced, with only marginal, non-material deviations.

Reproduced on the brainbox compute brainarbeit.com
Scoring basis — itemised

Every item that counted toward this verdict, and the exact part of the reproduction that produced it.

Supporting (toward a concern)
Content-critical question only partially held
+2 pts
From: Q5 · Derivability / plausibility 🟡
Content-critical question only partially held
+2 pts
From: Q7 · Core claim 🟡
Content-critical question only partially held
+2 pts
From: Q8 · Severity of the miss (overall human judgment) 🟡
Minor / cosmetic deviation
+1 pts
From: Q3 · Location of the main deviation 🟡
Minor / cosmetic deviation
+1 pts
From: Q6 · Severity of the deviation 🟡
Minor / cosmetic deviation
+1 pts
From: Q2 · Endpoint comparability 🟡
Concordant (toward reproduced)
Code + data deposited & functional
-2 pts
From: Data & code availability Available & functional
Total score +7
✓ What held up
  • Same input data as the authors
What did not (or only partly)
  • 🟡Reported values were only indirectly comparable
  • 🟡A deviation arose in the data or preprocessing
  • 🔴A deviation was attributed to the published material
  • 🟡Reported values were not (fully) derivable from the shared data
  • 🟡The deviation was non-trivial in magnitude
  • 🟡The central claim did not (fully) hold under reproduction
  • 🟡Overall, the reproduction showed a material discrepancy
How its reproducibility compares
50/100
Reproducibility score
1.4 SD below mean
vs. all fields · 1173 studies
🎯 Scores higher than 8% of all assessed papers rank 1026 of 1173 scored

A 0–100 reproducibility-quality score from the per-question grades, shown as a z-score: standard deviations above (+) or below (−) the mean of comparable assessments.

Reproduction agent’s raw note

Paper: Blotas et al 2025 IJMS, cis-regulation of CFTR. The single clean in-scope pipeline result C1 = ATAC-seq peaks within the CFTR TAD (Capan-1), reported as 6 interior peaks (2 upstream / 1 promoter / 3 at 3'), hg19. Reproduced with the cited third-party Galaxy workflow iwc-workflows/atacseq v0.12 tool chain (cutadapt Nextera -> bowtie2 --very-sensitive --dovetail -X1000 hg19 -> MAPQ30/concordant/no-chrM filter -> Picard MarkDuplicates -> bedtools bamtobed -> macs2 -q0.05 --nomodel --shift -100 --extsize 200 --call-summits --keep-dup all) on GSE284414 (the ATAC subseries; the brief's geo:GSE284199 is the 4C subseries -- correction documented in scope.md). OUTCOME = PARTIAL: the pipeline ran cleanly end-to-end and the qualitative result reproduces (a strong CFTR-promoter peak in every sample, plus accessible chromatin upstream and at the 3' end), but the literal headline count '6' is NOT 1:1 reproducible -- standard MACS2 q0.05 yields ~17 unique peaks in the TAD (Capan-1 merged), ~3x the reported 6. The '6' reads as a curated candidate-regulatory-element set (no intragenic category; 6 elements taken to luciferase), and its selection rule is underspecified in the Methods -- flagged low-severity as 'not directly derivable from the shipped data/code' (curation gap, NOT fabrication; a human reviewer should check the supplement). Three blockers solved this session: (1) Picard MarkDuplicates NPE on bowtie2 BAM lacking @RG -> AddOrReplaceReadGroups; (2) MACS2 ImportError 'undefined symbol: __log_finite' (glibc>=2.31 dropped finite-math aliases) -> LD_PRELOAD shim; (3) front1 /tmp full (50M tmpfs) -> TMPDIR/-pipe on «infra». All 3 subseries profiled (4/9/7 runs, all N reported=observed). Bonus: Caco-2 ATAC also processed (21/22/32 unique TAD peaks rep1/rep2/merged). NOT attempted (out of scope): 4C (GSE284199), CUT&RUN (GSE284200), Jaspar motifs, luciferase (wet-lab).

These records describe the outcome of reproduction attempts carried out autonomously by brainbox using large language models (LLMs). They are not peer review, not an audit, and not a determination of error or misconduct by any author. A verdict reflects what one attempt could or could not reproduce — which may depend on data access, undocumented parameters, the computing environment, or the depth of effort — and not a judgement of the people who did the work. We can be wrong, and we correct mistakes quickly: every record carries a “report an error” button.

Assessment versions

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  1. v1 current initial assessment Score 50
    assessed: 2026-06-21 ⛓ 525cc845775c
✎ I am an author of this paper

Updated or fixed a deposit, or is there an erratum? Ask us to re-run the metrics. We verify by email first; the new result is published as a new version with full history — nothing is overwritten.

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Provenance — full disclosure

When this reproduction was carried out, which methodology version was used, and by whom — so the record can be audited and checked independently.

Reproduced
2026-06-21
Rubric version
v1.0
Assessed by
🤖 AI curator · claude (ai-curator room) · v1.0 · run #1 2026-06-21
no human curator yet
Last updated
2026-08-05

Provisional, curator- or AI-assessed, and independently checkable. A reproduction outcome states what one attempt could reproduce — not a judgement of the authors.

Deep full-text extraction

Model: sonnet
Founding hypothesis

The study tests whether 3D chromatin architecture and cis-regulatory elements (CREs) around the CFTR gene establish its tissue-specific expression in pancreatic cells, potentially explaining clinical heterogeneity in cystic fibrosis and CFTR-related disorders.

Core claims
  • Multiple active CREs exist upstream and downstream of the CFTR gene in pancreatic (Capan-1) cells, identified via ATAC-seq, CUT&RUN-seq (H3K27ac), 4C-seq, and the ABC model finding
  • The -44 kb, -35 kb, +15.6 kb, and +37.7 kb regions act cooperatively, sharing common predicted transcription factor binding motifs, and jointly enhance promoter activity beyond individual effects finding
  • Active CREs regulating CFTR exist outside the CFTR TAD, including a region near the LSM8 gene at +507.6 kb showing chromatin interaction and silencer activity finding
  • CRISPR/Cas9-mediated homozygous deletion of the endogenous -44 kb CRE reduces CFTR expression, validating its enhancer function finding
  • CRE usage and activity is tissue-specific, differing between pancreatic (Capan-1) and intestinal (Caco-2) cells, with intron 12 and intron 24 OCRs acting as strong enhancers only in intestinal cells finding
  • The activity-by-contact (ABC) model can be used to computationally link candidate CREs to the CFTR promoter using ATAC-seq, H3K27ac, and Hi-C-type data method
  • HNF1B transcription factor motifs are present in active cCREs except intron 18, which showed no luciferase activity mechanism
Experimental setups
Assay System Perturbation Readout Platform
ATAC-seq Capan-1 pancreatic ductal cells none genome-wide open/accessible chromatin regions
CUT&RUN-seq (H3K27ac) Capan-1 pancreatic ductal cells none active enhancer histone mark peaks
4C-seq Capan-1 pancreatic ductal cells none (CFTR promoter used as bait) chromatin interaction frequency at CFTR locus, analyzed with peakC
Luciferase reporter assay Capan-1 cells (co-transfection with pCMV-beta-galactosidase control) candidate CRE constructs cloned upstream of minimal CFTR promoter in pGL3-Basic vector luciferase activity (fold change) pGL3-Basic vector
CRISPR/Cas9 knockout (VLP delivery) Capan-1 cells deletion of endogenous -44 kb CRE via flanking sgRNAs CFTR mRNA expression by RT-qPCR; deletion confirmed by PCR and Sanger sequencing virus-like particles (VLPs)
ATAC-seq Caco-2 intestinal cells none open chromatin regions for cross-tissue comparison
ATAC-seq HepG2 cells (publicly available data) none open chromatin regions in a CFTR-non-expressing cell line
4C-seq Caco-2 and HepG2 cells none (CFTR promoter bait) chromatin interaction profiles compared across cell types
Key results
  • Six reliable ATAC/H3K27ac consensus peaks identified within the CFTR TAD (two upstream, one promoter, three at 3' end)
  • ABC model identified five links to the CFTR promoter, including -44 kb, -35 kb, -114 bp, and +15.6 kb regions
  • 4C-seq peakC analysis found significant interactions at -80.1 kb boundary, ~-60 kb, +15.6 kb, intron 26, and +37.7 kb
  • Luciferase activity increased with -44 kb region and intron 26 OCR; -35 kb and +37.7 kb showed minor silencing; combination of all four cCREs gave the largest increase 1.7-fold (-44kb); 1.8-fold (intron26); -1.39-fold (-35kb/+37.7kb); 3.2-fold (4-CRE combo)
  • Intron 23 region showed significant enhancer activity in luciferase assay 3.5-fold
  • Homozygous CRISPR deletion of -44 kb region reduced CFTR expression relative to untreated cells; heterozygous deletion had no effect 15% reduction
  • +507.6 kb region (near LSM8, outside TAD) showed silencer activity in luciferase assay; CTCF site and LSM8 promoter region had no effect -1.32-fold
  • Intron 12 and combined intron 12+24 regions strongly increased luciferase activity in Caco-2 cells but had no effect in Capan-1 cells 15-fold (intron12); 50-fold (intron12+24) in Caco-2
Key statistics
  • fold_change 1.7-fold (-44 kb region luciferase activity vs promoter alone in Capan-1)
  • fold_change 1.8-fold (intron 26 OCR luciferase activity vs promoter alone in Capan-1)
  • fold_change -1.39-fold (-35 kb and +37.7 kb regions minor silencing effect in Capan-1)
  • fold_change 3.2-fold (combination of -44kb, -35kb, +15.6kb, +37.7kb cCREs luciferase activity)
  • fold_change 3.5-fold (intron 23 region luciferase activity in Capan-1)
  • fold_change 15% reduction (CFTR expression after homozygous CRISPR deletion of -44 kb region (clone 1) vs untreated Capan-1)
  • fold_change 15-fold (intron 12 luciferase activity increase in Caco-2 cells)
  • fold_change 50-fold (combined intron 12 and intron 24 luciferase activity increase in Caco-2 cells)

Statistical methods review

Model: sonnet

A neutral, descriptive read of the statistical approach — what was done, and (for shared learning, not as criticism) what could also have been done.

The paper uses genomic profiling (ATAC-seq, CUT&RUN-seq for H3K27ac, 4C-seq) combined with a computational activity-by-contact (ABC) model to nominate candidate cis-regulatory elements, then functionally tests candidates with luciferase reporter assays (reported as fold-change relative to a promoter-only construct) and validates one element with CRISPR/Cas9 deletion followed by RT-qPCR (reported as percent change relative to untreated cells). Findings are described narratively with fold-change values and one explicit statement that a comparison was 'not significantly different,' but the provided text does not name a specific statistical test, replicate structure, or software package used for these comparisons.

Replicationunclear GroupsLuciferase reporter constructs (single and combined CREs) vs. promoter-only control, across Capan-1 and Caco-2 cell lines; CRISPR homozygous/heterozygous −44kb deletion clones vs. untreated Capan-1 cells Pairingunclear Randomization/blindingnot stated Dispersionunclear Exact p-valuesno Effect sizesyes Confidence intervalsno
Statistical tests used
Test Applied to n Assumptions
not stated (a statistical comparison is implied by the phrase 'not significantly different') Luciferase assay, Figure 4 — combined −44kb+−35kb construct vs. −44kb alone not stated
Approaches that could also have been used
  • Luciferase and RT-qPCR results are summarized as single fold-change or percent-change values without a stated measure of dispersion or replicate count in the text.
    Could also: Reporting mean ± SD or SEM (or a 95% CI) alongside individual replicate data points — Showing dispersion and the number of independent replicates alongside the point estimate helps convey the precision of a fold-change or percent-change measurement, which is especially informative for small-n reporter assays.
  • A comparison between the combined −44kb+−35kb construct and the −44kb-alone construct is described as 'not significantly different' without naming the statistical test used.
    Could also: Explicitly naming the test (e.g., a two-tailed t-test or one-way ANOVA with post-hoc comparison) and reporting the associated p-value or test statistic — Stating the specific test and exact p-value allows readers to evaluate the assumptions of the test and reproduce the significance assessment.
  • Multiple related luciferase constructs (single CREs, paired combinations, and a four-CRE combination) are compared against the promoter-only baseline across several figures.
    Could also: A one-way or two-way ANOVA with a post-hoc multiple-comparisons correction (e.g., Tukey HSD, Dunnett's test, or Benjamini-Hochberg FDR) — When many related constructs are compared to the same baseline, an omnibus test with a correction for multiple comparisons is a standard way to control the family-wise error rate across that comparison set.
  • The endogenous validation of the −44kb enhancer relies on RT-qPCR comparing one homozygous deletion clone to untreated Capan-1 cells.
    Could also: Testing multiple independently derived homozygous clones with a statistical comparison (e.g., paired or unpaired t-test) across biological replicates — Using several independent clones and a formal statistical comparison can help distinguish an on-target enhancer effect from clone-specific variation.
  • 4C-seq interaction peaks are identified using peakC without a stated significance threshold or multiple-testing correction in the text.
    Could also: Reporting the FDR or p-value threshold used by peakC (or an alternative 4C/Hi-C peak-calling tool with explicit multiple-testing control) — Stating the significance threshold used for peak calling clarifies how candidate interactions were distinguished from background noise across the many genomic bins tested.
  • Enhancer-promoter links are nominated using the ABC computational model, which integrates chromatin accessibility, H3K27ac, and Hi-C-derived contact data.
    Could also: Cross-validating links with an additional enhancer-prediction approach, such as co-accessibility analysis from single-cell ATAC-seq or an independent Hi-C-based statistical model — Comparing predictions from more than one enhancer-linking method can provide converging evidence for candidate CRE-promoter relationships in a given cell type.
Software: peakC (4C-seq peak calling) · ABC (activity-by-contact) model (Fulco et al.)

What was reproduced

The exact results taken into scope, with each reported value next to the value our attempt produced.

Scope — pmid-40332394

Paper: Cis-Regulation of the CFTR Gene in Pancreatic Cells. Blotas et al., Int J Mol Sci 2025. PMID 40332394 · PMCID PMC12027686 · DOI 10.3390/ijms26083788.

Assays in the paper (all on Capan-1 / Caco-2 / HepG2 cell lines):

  1. ATAC-seq — open-chromatin mapping. Pipeline: iwc-workflows/atacseq v0.12 (Galaxy), default params, mapped to hg19, peaks called with MACS2.
  2. CUT&RUN-seq (H3K27ac, CTCF, H3K27me3) — pipeline iwc-workflows/cutandrun v0.8.
  3. 4C-seq — pipeline Pipe4C v1.1.4 + peakC v0.2, hg19.
  4. Luciferase reporter assays — wet-lab.

GEO accession mapping (resolved from NCBI/ENA, 2026-06-15)

The brief's RU pairs geo:GSE284199 with code:iwc-workflows/atacseq, but those two do not correspond to the same assay. The paper deposited a SuperSeries split into three subseries:

GEO assay pipeline (code) samples BioProject
GSE284199 4C-seq Pipe4C + peakC 9 (GSM8679690–98) PRJNA1197587
GSE284200 CUT&RUN iwc cutandrun v0.8 7 (GSM8679699–705) PRJNA1197588
GSE284414 ATAC-seq iwc atacseq v0.12 4 (GSM8683820–23) PRJNA1198966

→ The cited code (iwc-workflows/atacseq) is the ATAC-seq pipeline, whose data live in GSE284414, not GSE284199. We therefore reproduce the ATAC-seq result using GSE284414 + the atacseq workflow (the internally-consistent code↔data pairing). This correction is recorded in code/code.json and data/data.json. (Per brief rule 2, applying the cited third-party Galaxy tool to the paper's own data is a fully valid reproduction.)

ATAC-seq runs (GSE284414 / PRJNA1198966, ENA, paired-end)

run sample reads (PE)
SRR31736540 ATAC_Capan-1_rep1 37.1 M
SRR31736539 ATAC_Capan-1_rep2 35.7 M
SRR31736538 ATAC_Caco-2_rep1 39.2 M
SRR31736537 ATAC_Caco-2_rep2 37.0 M

IN SCOPE (pipeline-derived, attempted)

  • C1 — peaks within the CFTR TAD. Reported (Results / Fig.): "Six peaks were identified within the TAD [chr7:117,039,878–117,356,812]. Two were located upstream of the promoter, one corresponded to the CFTR promoter, and three were located at the 3′ end." This is a clean, objective, MACS2-pipeline-derived count + locations. Reproduction = run iwc atacseq v0.12-equivalent (cutadapt Nextera trim → bowtie2 --dovetail -X1000 → hg19 → MAPQ30/concordant/no-chrM filter → Picard dedup → BAM→BED → MACS2) on the Capan-1 ATAC reads, then count MACS2 peaks falling in chr7:117,039,878–117,356,812 and classify them (upstream / promoter / 3′).

OUT OF SCOPE (not attempted, with reason)

  • Luciferase fold-changes (−44 kb 1.7×, intron 23 3.5×, introns 12/24 15×/50× in Caco-2, etc.) — wet-lab reporter assays, non_pipeline.
  • 4C-seq interaction maps / ABC links (GSE284199, Pipe4C+peakC) and CUT&RUN H3K27ac peaks (GSE284200) — separate pipelines/data; the hard ~20%, deferred per the 80/20 rule. The ATAC peak count is the single cleanest pipeline number.
  • Jaspar 2024 TFBS motif mapping (HNF1B etc.) — depends on the cCRE set + external motif DB; downstream/manual, deferred.

80/20 decision

The ATAC-seq "6 peaks in the CFTR TAD" is the lowest-hanging, fully-specified, objectively-checkable pipeline output (one integer + element locations). We reproduce that with the cited workflow's tool chain on the paper's own Capan-1 ATAC data, all on «our HPC»/«infra». We do not attempt the 4C-seq, CUT&RUN, motif, or luciferase results.

C1
Reported
Capan-1 ATAC-seq: 'In addition to the boundaries, six peaks were identified within the [CFTR] TAD (chr7:117,039,878-117,356,812, hg19). Two upstream of the promoter, one at the CFTR promoter, three at the 3' end.' (Results, ATAC-seq section; verified verbatim from PMC12027686)
Reproduced
Pipeline ran end-to-end on «our HPC» on the paper's own Capan-1 ATAC reads (GSE284414). Alignments clean (bowtie2 hg19 89.32%/91.82% Capan-1 rep1/rep2), Picard dedup clean (PERCENT_DUPLICATION 5.45%/3.23%), MACS2 produced 156k/154k/209k genome-wide peaks (rep1/rep2/merged). Within the CFTR TAD, Capan-1 MERGED gives 17 unique MACS2 peak regions (19 incl. call-summits rows) = 4 upstream / 1 promoter / 7 intragenic / 5 at 3'; rep1 = 19, rep2 = 9. The CFTR-promoter peak (chr7:117,118,870-117,120,235) reproduces in every sample, and accessible chromatin appears upstream and at the 3' end as the paper describes. BUT the literal count '6' is NOT 1:1 reproducible: the standard iwc atacseq v0.12 MACS2 -q0.05 pipeline yields ~3x more peaks (~17). The paper's '6' has no intragenic category and 6 elements were carried to luciferase validation, so it is almost certainly a CURATED candidate-regulatory-element subset (likely ATAC selected as regulatory candidates and/or intersected with active CUT&RUN H3K27ac), whose selection rule is not specified well enough in the Methods to regenerate from ATAC alone. Verdict: pipeline + qualitative spatial pattern REPRODUCE; the exact integer does NOT. See agreement.json, peak_summary.tsv, cftr_tad_peaks.json, capan1_merged_TAD_peaks.tsv.
partial

Assessments & scoring basis

Each contributor’s verdict, the per-question basis, and the auditable, itemised worksheet behind it.

🤖 AI curator · claude (ai-curator room) · v1.0 L1 50/100

An automated assessment. It can flag an open question for review but can never, on its own, record a discrepancy verdict (C5) against a paper.

🟢1. Data identity
🟡2. Endpoint comparability
🟡3. Location of the main deviation
🔴4. Cause of the deviation
🟡5. Derivability / plausibility
🟡6. Severity of the deviation
🟡7. Core claim
🟡8. Severity of the miss (overall human judgment)
Scoring basis — itemised

Every item that counted toward this verdict, and the exact part of the reproduction that produced it.

Supporting (toward a concern)
Content-critical question only partially held
+2 pts
From: Q5 · Derivability / plausibility 🟡
Content-critical question only partially held
+2 pts
From: Q7 · Core claim 🟡
Content-critical question only partially held
+2 pts
From: Q8 · Severity of the miss (overall human judgment) 🟡
Minor / cosmetic deviation
+1 pts
From: Q3 · Location of the main deviation 🟡
Minor / cosmetic deviation
+1 pts
From: Q6 · Severity of the deviation 🟡
Minor / cosmetic deviation
+1 pts
From: Q2 · Endpoint comparability 🟡
Concordant (toward reproduced)
Code + data deposited & functional
-2 pts
From: Data & code availability Available & functional
Total score +7

The pipeline reproduced cleanly on the authors' own Capan-1 ATAC data (GSE284414) and the qualitative spatial claim — accessible chromatin at the CFTR promoter, upstream, and 3' end — holds, with the promoter peak (chr7:117,118,870-117,120,235) reproduced in every sample. However the headline count of 6 is not 1:1 reproducible: the standard MACS2 q0.05 pipeline yields ~17 unique TAD peaks (~3x). The gap is on the authors' side — an underspecified curation/selection step that picks 6 candidate regulatory elements (the same 6 taken to luciferase), not fabrication. Overall: solid, partial reproduction with an explainable, well-documented deviation → yellow.

🤝
Reproduced automatically — and fairly

Automated reproduction checks whether a published result can be regenerated from the paper’s described methods and shared data. When something does not reproduce, that is not a claim of error or misconduct — most often it reflects under-described methods, software or environment differences, or gaps in data access, and some of the pre-print papers in the queue may carry issues their authors had no part in. The goal is shared awareness that rigorous, fully-described methods help everyone — never a judgement of any author.

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Reproduction footprint

claude-opus-4-8

Measured resources invested to assess this paper — sanitised (machine class only, no job ids/paths). Compute = HPC accounting (SLURM); tokens = the AI agent's session.

704.4 k
tokens (I/O) · 56.3 M incl. cache
213 min
runtime · 24.24 CPU-h
18.3 GB
peak RAM
4
HPC jobs
hummel
machine