Evidence map›Paper›PMID 38678283›Full record

ArticleOrphanet journal of rare diseases2024

'BLUES' procedure for assessing the blue level of the sclera in Osteogenesis Imperfecta.

Valerio Di Martino, Fabiana Mallone, Alessandro Lambiase, Mauro Celli, Alice Mannocci, Luca Celli, Pietro Mangiantini, Pasquale Fino, Antonietta Moramarco

Open access · goldAbstract read
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In one paragraph

Article in Orphanet journal of rare diseases, 2024. 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
0.6field-weighted citation impact, top 29% 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

0 citing papers in PubMed, 1 citations in OpenAlex.

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

9 authors at 2 institutions in 1 country.

Valerio Di MartinoDepartment of Sense Organs, Sapienza University of Rome, Viale del Policlinico 155, Rome, 00161, Italy.
Fabiana MalloneDepartment of Sense Organs, Sapienza University of Rome, Viale del Policlinico 155, Rome, 00161, Italy.
Alessandro LambiaseDepartment of Sense Organs, Sapienza University of Rome, Viale del Policlinico 155, Rome, 00161, Italy. Alessandro.lambiase@uniroma1.it.ORCID 0000-0002-8974-991X
Mauro CelliDepartment of Pediatrics and Child Neuropsychiatry, Sapienza University of Rome, Rome, Italy.
Alice MannocciDepartment of Public Health and Infectious Diseases, Sapienza University of Rome, Rome, Italy.
Luca CelliDepartment of Pediatrics and Child Neuropsychiatry, Sapienza University of Rome, Rome, Italy.
Pietro MangiantiniDepartment of Sense Organs, Sapienza University of Rome, Viale del Policlinico 155, Rome, 00161, Italy.
Pasquale FinoDepartment of Clinical Internal, Anesthesiologic and Cardiovascular Sciences, Sapienza University of Rome, Rome, Italy.
Antonietta MoramarcoDepartment of Sense Organs, Sapienza University of Rome, Viale del Policlinico 155, Rome, 00161, Italy.
Policlinico Umberto I · ITSapienza University of Rome · IT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

purposeBlue sclera is a characteristic and common clinical sign of Osteogenesis Imperfecta (OI). However, there is currently no widely accepted, objective method for assessing and grading blue sclera in individuals with OI. To address this medical need, this study is aimed to design and validate a new method called 'BLUES' (BLUe Eye Sclera) to objectively identify and quantify the blue color in the sclera of patients affected by OI.

methodsSixty-two patients affected by OI and 35 healthy controls were enrolled in the present prospective study, for a total of 194 eyes analyzed. In the 'BLUES' procedure, eye images from patients with OI and control subjects were analyzed to assess and grade the blue level of the sclera using Adobe Photoshop Software. The validation process then involved comparing the results obtained with the 'BLUES' procedure to the judgement of experienced ophthalmologists (JEO). A receiver-operating characteristic (ROC) curve analysis was used to examine the overall discriminatory power. The sensitivity and specificity levels and the Cohen's Kappa (K) indexes of 'BLUES' and 'JEO' were estimated versus the standard OI diagnosis. The K indexes of 'BLUES' versus 'JEO' were also evaluated.

resultsThe optimal cut-off point of the scleral blue peak was calculated at 17%. Our findings demonstrated a sensitivity of 89% (CI95%: 0.835-0.945) and specificity of 87% (CI95%: 0.791-0.949) for the 'BLUES' procedure with an agreement versus the diagnosis of OI of 0.747. In comparison, the sensitivity and specificity of 'JEO' ranged from 89 to 94% and 77% to 100%, respectively, with an agreement ranging from 0.663 to 0.871 with the diagnosis of OI. The agreement between 'BLUES 'and 'JEO' evaluations ranged from 0.613 to 0.734.

conclusionsOur findings demonstrated an 89% sensitivity and an impressive 87% specificity of our method to analyze the blue sclera in OI. The results indicated high agreement with disease diagnosis and were consistent with evaluations by experienced ophthalmologists. The 'BLUES' procedure appears to be a simple, reliable and objective method for effectively identify and quantify the blue color of the sclera in OI.

Indexed as

Osteogenesis ImperfectaScleraAdolescentAdultChildChild, PreschoolFemaleHumansMaleProspective StudiesROC CurveYoung AdultBlue sclera‘BLUES’ procedureComputed analysisDiagnosisOsteogenesis Imperfecta (OI)Rare diseases

Identifiers

PMID38678283
PMCPMC11056038
OpenAlexW4395690409

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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.