Evidence map›Paper›PMID 41079531›Full record

ReviewFrontiers in nephrology2025

The assessment and treatment of the musculoskeletal manifestations of cystinosis.

Priya Singh, D'Arcy Marsh, Melinda Sharkey

Abstract readReview
In one paragraph

Review in Frontiers in nephrology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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

3 authors.

Priya SinghDepartment of Orthopaedic Surgery, Montefiore Health System, Bronx, NY, United States.
D'Arcy MarshDepartment of Orthopaedic Surgery, Montefiore Health System, Bronx, NY, United States.
Melinda SharkeyDepartment of Orthopaedic Surgery, Montefiore Health System, Bronx, NY, United States.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Cystinosis is a rare autosomal recessive lysosomal storage disease caused by a defective lysosomal cystine carrier protein, cystinosin, resulting in formation and deposition of cystine crystals throughout the body. The renal manifestations of the disease have long been studied, but the musculoskeletal consequences of the disease are generally less well understood. Limb deformities, scoliosis, myopathy and low bone mineral density are associated with cystinosis and can lead to pain, fragility fractures, bone deformity, and difficulty ambulating. Although potentially exacerbated by renal disease and post-transplant medications, it has been found that the musculoskeletal manifestations of cystinosis are also due to inherent dysfunction caused by the mutation of cystinosin. Surgical intervention can provide solutions to the bony symptoms of cystinosis. Early referral to an orthopedic surgeon and evaluation for corrective scoliosis surgery, guided growth for growing children with lower extremity deformity and formal osteotomies for deformity correction in skeletally mature individuals may improve physical function and decrease pain. Standard principles of operative treatment of scoliosis and of bone deformity correction utilized for the treatment of bone deformity in other metabolic bone disease may be applied to patients with cystinosis in the absence of cystinosis-specific studies of the efficacy and outcomes of orthopedic surgery.

Indexed as

bone deformityfragility fractureslong bone fractureslow bone mineral densitymyopathyrenal osteodystrophyscoliosisvertebral insufficiency fractures

Identifiers

PMID41079531
PMCPMC12507550

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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.