Evidence map›Paper›PMID 35546999›Full record

ArticleFrontiers in endocrinology2022

Prevalence and Hospital Admissions in Patients With Osteogenesis Imperfecta in The Netherlands: A Nationwide Registry Study.

Silvia Storoni, Sanne Treurniet, Alessandra Maugeri, Gerard Pals, Joost G van den Aardweg, Stéphanie L van der Pas, Mariet W Elting, Peter Kloen, Dimitra Micha, Elisabeth Marelise W Eekhoff

Open access · goldAbstract read
In one paragraph

Article in Frontiers in endocrinology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed, 1 pooled it
3.9field-weighted citation impact, top 6% 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

14 citing papers in PubMed, 1 synthesis or guideline pooled it, 23 citations in OpenAlex.

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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 1 institution in 1 country.

Silvia StoroniDepartment of Internal Medicine, Section Endocrinology, Amsterdam Rare Bone Disease/Amsterdam Bone Center, Amsterdam University Medical Center, location VUmc, Amsterdam, Netherlands.
Sanne TreurnietDepartment of Internal Medicine, Section Endocrinology, Amsterdam Rare Bone Disease/Amsterdam Bone Center, Amsterdam University Medical Center, location VUmc, Amsterdam, Netherlands.
Alessandra MaugeriDepartment of Human Genetics, Amsterdam Movement Sciences, Amsterdam Rare Bone Disease/Amsterdam Bone Center, Amsterdam University Medical Center, location VUmc, Amsterdam, Netherlands.
Gerard PalsDepartment of Human Genetics, Amsterdam Movement Sciences, Amsterdam Rare Bone Disease/Amsterdam Bone Center, Amsterdam University Medical Center, location VUmc, Amsterdam, Netherlands.
Joost G van den AardwegDepartment of Respiratory Medicine, Amsterdam University Medical Center, location AMC, Amsterdam, Netherlands.
Stéphanie L van der PasDepartment of Epidemiology and Data Science (EDS), Amsterdam University Medical Center, location VUmc, Amsterdam, Netherlands.
Mariet W EltingDepartment of Human Genetics, Amsterdam University Medical Center, location VUmc, Amsterdam, Netherlands.
Peter KloenDepartment of Orthopedic Surgery, Amsterdam Movement Sciences, Amsterdam Rare Bone Disease/Amsterdam Bone Center, Amsterdam University Medical Center, location AMC, Amsterdam, Netherlands.
Dimitra MichaDepartment of Human Genetics, Amsterdam Movement Sciences, Amsterdam Rare Bone Disease/Amsterdam Bone Center, Amsterdam University Medical Center, location VUmc, Amsterdam, Netherlands.
Elisabeth Marelise W EekhoffDepartment of Internal Medicine, Section Endocrinology, Amsterdam Rare Bone Disease/Amsterdam Bone Center, Amsterdam University Medical Center, location VUmc, Amsterdam, Netherlands.
Amsterdam University Medical Centers · NL

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Osteogenesis Imperfecta (OI) is a complex disease caused by genetic alterations in production of collagen type I, and collagen-related proteins. Bone fragility is the most common patient issue, but extraskeletal complications also present an adverse factor in the quality of life and prognosis of patients with OI. However, still little is known about the morbidity and mortality of these patients. The objective of this paper is to determine and describe to what extent OI impacts patients' life in terms of hospitalization and complications describing the incidence and prevalence of the Dutch cohort of OI patients and the characteristics of their hospital admissions. Information regarding OI patients and their hospital admission was extracted from the Statistics Netherlands Database and matched to the OI Genetics Database of Amsterdam UMC. Hospital admission data was available for 674 OI patients. This OI nationwide registry study shows that the life expectancy of OI patients is adversely affected by the disease. The median annual incidence risk of OI between 1992 and 2019 was 6.5 per 100,000 live births. Furthermore, patients with OI had a 2.9 times higher hospitalization rate compared to the general Dutch population. The highest hospitalization rate ratio of 8.4 was reported in the patient group between 0 and 19 years old. OI type and severity had impact on extraskeletal manifestations, which play a key role in the numerous hospital admissions. More awareness about the impact of OI on patients' life is needed to improve and implement prevention and follow-up guidelines.

Indexed as

Osteogenesis ImperfectaAdolescentAdultChildChild, PreschoolHospitalizationHospitalsHumansInfantInfant, NewbornNetherlandsPrevalenceQuality of LifeRegistriesYoung AdultCBS registryhospital admissionmorbidityosteogenesis imperfectapathogenic variant

Identifiers

PMID35546999
PMCPMC9082351
OpenAlexW4224862073

What OpenQuestion holds

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LicenceCC BY
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Registered trials

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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.