Evidence map›Paper›PMID 29548779›Full record

ArticleAmerican journal of kidney diseases : the official journal of the National Kidney Foundation2018

Dialysis as a Treatment Option for a Patient With Normal Kidney Function and Familial Tumoral Calcinosis Due to a Compound Heterozygous FGF23 Mutation.

Patrícia T Goldenstein, Precil D Neves, Bruno E Balbo, Rosilene M Elias, Alexandre C Pereira, Luiz F Onuchic, Harald Jüppner, Vanda Jorgetti, Hugo Abensur, Rosa Maria Moysés

Open access · greenAbstract readCase Reports
In one paragraph

Article in American journal of kidney diseases : the official journal of the National Kidney Foundation, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed, 1 pooled it
0.8field-weighted citation impact, top 31% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

3 citing papers in PubMed, 1 synthesis or guideline pooled it, 7 citations in OpenAlex.

  1. Pooled it
  2. Review
  3. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

10 authors at 2 institutions in 2 countries.

Patrícia T GoldensteinNephrology Division, University of São Paulo School of Medicine, Sao Paulo-SP, Brazil.
Precil D NevesNephrology Division, University of São Paulo School of Medicine, Sao Paulo-SP, Brazil.
Bruno E BalboNephrology Division, University of São Paulo School of Medicine, Sao Paulo-SP, Brazil.
Rosilene M EliasNephrology Division, University of São Paulo School of Medicine, Sao Paulo-SP, Brazil.
Alexandre C PereiraLaboratory of Genetics and Molecular Cardiology, Heart Institute, University of Sao Paulo, Sao Paulo-SP, Brazil; Genetics Department, Harvard Medical School, Boston, MA.
Luiz F OnuchicNephrology Division, University of São Paulo School of Medicine, Sao Paulo-SP, Brazil.
Harald JüppnerEndocrine, Massachusetts General Hospital and Harvard Medical School, Boston, MA; Pediatric Nephrology Units, Massachusetts General Hospital and Harvard Medical School, Boston, MA.
Vanda JorgettiNephrology Division, University of São Paulo School of Medicine, Sao Paulo-SP, Brazil.
Hugo AbensurNephrology Division, University of São Paulo School of Medicine, Sao Paulo-SP, Brazil.
Rosa Maria MoysésNephrology Division, University of São Paulo School of Medicine, Sao Paulo-SP, Brazil; Universidade Nove de Julho, São Paulo-SP, Brazil. Electronic address: rosa.moyses@uol.com.br.
Universidade de São Paulo · BRMassachusetts General Hospital · US

Funding

PTH/PTHRP RECEPTOR DEFECTS IN PSEUDOHYPOPARATHYROIDISMR01DK046718 · NIDDK · MASSACHUSETTS GENERAL HOSPITAL · PI JUEPPNER, HARALD W. · 1993 to 2024
$6.0M
NIDDK NIH HHS R01 DK046718
6 · The paper itself

Abstract

Primary tumoral calcinosis is a rare autosomal recessive disorder characterized by ectopic calcified tumoral masses. Mutations in 3 genes (GALNT3, FGF23, and KL) have been linked to this human disorder. We describe a case of a 28-year-old man with a history of painful firm masses over his right and left gluteal region, right clavicle region, knees, and left elbow. Biochemical analysis disclosed hyperphosphatemia (phosphate, 9.0 mg/dL) and normocalcemia (calcium, 4.8 mg/dL), with normal kidney function and fractional excretion of phosphate of 3%. Parathyroid hormone was suppressed (15 pg/mL), associated with a low-normal 25-hydroxyvitamin D (26 ng/mL) concentration but high 1,25-dihydroxyvitamin D concentration (92 pg/mL). Serum intact FGF-23 (fibroblast growth factor 23) was undetectable. Genetic analysis revealed tumoral calcinosis due to a compound heterozygous mutation in FGF23, c.201G>C (p.Gln67His) and c.466C>T (p.Gln156*). Due to lack of other treatment options and because the patient was facing severe vascular complications, we initiated a daily hemodialysis program even in the setting of normal kidney function. This unusual therapeutic option successful controlled hyperphosphatemia and reduced metastatic tumoral lesions. This is a report of a new mutation in FGF23 in which dialysis was an effective treatment option for tumoral calcinosis with normal kidney function.

Indexed as

Renal DialysisAdultCalcinosisFibroblast Growth Factor-23Fibroblast Growth FactorsHumansHyperostosis, Cortical, CongenitalHyperphosphatemiaKidneyMaleMutationTreatment OutcomeFGF23 protein, humanFibroblast Growth Factor-23Fibroblast Growth FactorsCase reportextra-osseous calcificationsFGF23 mutationfibroblast growth factor 23 (FGF-23)genetic diseasehemodialysishyperphosphatemiametabolic disordermutationtumoral calcinosis

Identifiers

PMID29548779
PMCPMC6109594
OpenAlexW2793220359

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.