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Record W4415451142 · doi:10.1210/jendso/bvaf149.706

SUN-756 Diagnosing and Treating Hereditary Hypophosphatemia Rickets with Hypercalciuria

2025· article· en· W4415451142 on OpenAlex

Why this work is in the frame

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

fundA Canadian funder is recorded on the work.
no affNo Canadian affiliation: this work is invisible to an affiliation-only frame.
No Canadian affiliation. An affiliation-only frame, the usual design, would never have seen this work. It is one of the works that make the case for inverting the frame.

Bibliographic record

VenueJournal of the Endocrine Society · 2025
Typearticle
Languageen
FieldMedicine
TopicBiomedical Research and Pathophysiology
Canadian institutionsnot available
FundersSchool of Medicine, Indiana UniversityVanderbilt UniversityMcMaster UniversityYale University
KeywordsHypercalciuriaHypophosphatemiaOsteomalaciaHypophosphatemic RicketsRicketsReabsorptionFanconi syndromeVitamin D and neurologyExcretion

Abstract

fetched live from OpenAlex

Abstract Disclosure: S. Dickerman: None. F.A. Al-Khayer: None. Introduction: Hereditary hypophosphatemia rickets with hypercalciuria (HHRH) is a rare autosomal recessive disorder with an estimated prevalence of 1 in 250,000 caused by mutations in the SLC34A3 gene that clinically manifests as rickets, bone pain, and hypercalciuria. This disorder may present similarly to more common disease states such as osteomalacia or osteoporosis, making its diagnose difficult and often tenuous. Clinical Case: This is the case of a 39-year-old female patient with a complex past medical history significant for renal stones since childhood and pancreatoduodenectomy in 2022 due to a dysplastic pancreatic lesion who initially presented to the office in 2024 with low phosphate, normal alkaline phosphatase, normal B6, high calcitriol, and hypercalciuria. Initial diagnoses considered based on these results included Fanconi syndrome, x-linked hypophosphatemia, nutritional or tumor-induced osteomalacia, and hereditary Vitamin D resistance. However, none of these conditions were consistent with her presenting symptoms and lab values. To work up the etiology of her presentation, the tubular reabsorption of phosphorus (TRP) was calculated using TRP=100(1-[quarinePO4/UCr])(serum Cr/serum PO4). When serum phosphate is low, it is expected that renal phosphate absorption and TRP will increase, yet this patient had a calculated TRP of 15. The tubular maximum reabsorption of phosphate (TmP) was then calculated using TmP=(TRP)(serum PO4), yielding 27. The ratio of TmP/eGFR was calculated to be 0.4, with a normal ratio for females being 3-4.45, 5-4 standard deviations below the age-related mean. Further, the upper limit of normal calcium excretion in 24 hours is 4mg/kg and this patient’s excretion was calculated to be 9.4mg/kg. An excretion rate greater than 8.6mg/kg is consistent with HHRH, as is the low ratio of Tmp/eGFR. The patient was treated with potassium phosphate supplementation. This successfully led to the correction of all the above abnormalities except for the decreased TmP/eGFR ratio and improvement of her symptoms. The diagnosis was later confirmed by detecting the pathogenic SLC34A3 mutation. Conclusion: Making the diagnosis of HHRH is challenging and requires an extensive and often timely workup. This case details the diagnostic process and key findings associated with HHRH: phosphate wasting which causes an appropriately elevated level of calcitriol, increased calcium intestinal absorption, suppression of PTH, hypercalciuria, and phosphate deficiency acting as a potent and direct stimulus to renal 25-hydroxyvitamin D-1-alpha hydroxylase (CYP27B1) activity. Treatment of HHRH and correction of these abnormalities may be accomplished utilizing phosphate supplementation. This case report offers a diagnostic schema for making an obscure diagnosis. Presentation: Sunday, July 13, 2025

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.

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.493
Threshold uncertainty score0.266

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0000.000

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 head0.009
GPT teacher head0.292
Teacher spread0.283 · 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