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Record W4397042183 · doi:10.1681/asn.20233411s1939c

HNF1B Nephropathy Mimicking Autosomal Dominant Polycystic Kidney Disease: A Case Report

2023· article· en· W4397042183 on OpenAlexaff
Nada Alamri, Matthew B. Lanktree

Bibliographic record

VenueJournal of the American Society of Nephrology · 2023
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetic and Kidney Cyst Diseases
Canadian institutionsMcMaster UniversitySt. Joseph’s Healthcare Hamilton
Fundersnot available
KeywordsHNF1BMedicineAutosomal dominant polycystic kidney diseaseNephropathyPolycystic kidney diseaseKidney diseasePolycystic kidneyUrologyInternal medicineKidneyPathologyEndocrinologyBiologyGeneticsDiabetes mellitus

Abstract

fetched live from OpenAlex

Introduction: Hepatocyte nuclear factor1beta (HNF1B) nephropathy is characterized by hypomagnesemia, hyperuricemia, congenital anomalies of the kidneys and urinary tract (CAKUT), and multiple small cortical cysts without kidney enlargement. It also involves multi-organ manifestations including insulin-deficient diabetes (or maturity-onset diabetes of the young [MODY]), pancreatic exocrine dysfunction, liver dysfunction and neurodevelopmental abnormalities including autism spectrum disorder (ASD). We present a rare case of HNF1B nephropathy with atypical large kidney cysts mimicking autosomal dominant polycystic kidney disease (ADPKD). Case Description: A 37-year-old male with ASD initially presented with hypertensive emergency. The patient reported occasional flank pain and distension but no other symptoms. The patient was estranged from his father, and his mother had normal kidney function without cysts. There was no known history of kidney failure, aneurysms, or sudden death in his extended family. Physical examination revealed distended abdomen with palpable organomegaly. Laboratory results showed elevated creatinine levels (2.41 mg/dl) and reduced eGFR (33 ml/min/1.73 m2) with an eGFR decline of 5 ml/min/1.73 m2 in the last year. Fasting blood glucose levels and liver function tests were normal. CT showed innumerable bilateral kidney cysts, the largest of which measured 19 cm on the right and 12 cm on the left, and an enlarged spleen. An initial diagnosis of de novo ADPKD was suspected. Genetic testing of PKD1 and PKD2 found no responsible variant, leading to a broader cystic gene panel sequencing. A heterozygous 1.26 Mb deletion (chr17:g.34842466_36104935del), encompassing whole HNF1B gene was detected. Cascade screening showed that the patient's mother was unaffected. Discussion: HNF1B nephropathy displays substantial phenotypic heterogeneity. We present a case of HNF1B nephropathy clinically mimicking ADPKD with extreme kidney enlargement and loss of kidney function, without hypomagnesemia, hyperuricemia, or MODY. Phenocopies are not uncommon, and this case further exemplifies the role of genetic testing to obtain a concrete diagnosis. This case report contributes to the understanding of phenotypic variability in HNF1B nephropathy and emphasizes the importance of considering this diagnosis in patients with large kidney cysts.

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)0.001
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 score0.007
Threshold uncertainty score0.015

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.003
Meta-epidemiology (narrow)0.0030.002
Meta-epidemiology (broad)0.0020.002
Bibliometrics0.0050.003
Science and technology studies0.0040.002
Scholarly communication0.0030.003
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 head0.010
GPT teacher head0.263
Teacher spread0.253 · 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

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