Evidence map›Paper›PMID 35604455›Full record

ArticleEuropean spine journal : official publication of the European Spine Society, the European Spinal Deformity Society, and the European Section of the Cervical Spine Research Society2022

Prevalence of scoliosis and impaired pulmonary function in patients with type III osteogenesis imperfecta.

M C Keuning, S J G Leeuwerke, P R van Dijk, A G J Harsevoort, H P Grotjohan, A A M Franken, G J M Janus

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Article in European spine journal : official publication of the European Spine Society, the European Spinal Deformity Society, and the European Section of the Cervical Spine Research Society, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.

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7citing papers in PubMed
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1 · What the graph read from it

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2 · The registry

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3 · Its place in the literature

Who cites it

7 citing papers in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

7 authors.

M C KeuningDepartment of Orthopaedic Surgery, University Medical Center, University of Groningen, Groningen, The Netherlands. m.c.keuning@umcg.nl.ORCID 0000-0002-3203-7874
S J G LeeuwerkeDepartment of Surgery, University Medical Center, University of Groningen, Groningen, The Netherlands.
P R van DijkDepartment of Endocrinology, University Medical Center, University of Groningen, Groningen, The Netherlands.
A G J HarsevoortDepartment of Orthopaedic Surgery, Isala, Zwolle, The Netherlands.
H P GrotjohanDepartment of Pulmonary Medicine, Isala, Zwolle, The Netherlands.
A A M FrankenDepartment of Internal Medicine, Isala, Zwolle, The Netherlands.
G J M JanusDepartment of Orthopaedic Surgery, Isala, Zwolle, The Netherlands.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

purposeOsteogenesis Imperfecta (OI) is a rare group of congenital genetic disorders that consists of a collagen synthesis defect. The most severe phenotype is type III OI. Characterized by progressive bone deformity, fragility and pulmonary impairment, causing significant morbidity and mortality. Also, multilevel spine deformities are observed, such as scoliosis. The literature on the pathophysiology of pulmonary impairment in relation to scoliosis in these patients is scarce and conflicting. This study aims to determine the prevalence of scoliosis and its relation to pulmonary function in type III OI patients.

methodsThis retrospective cohort study took place between April 2020 and November 2021. Forty-two patients with type III OI were included. Anterior-posterior spine radiographs were evaluated for scoliosis. Pulmonary function was assessed using spirometry and partial pressure of carbon dioxide.

resultsAll 42 patients had scoliosis, with a mean curve of 66° (95% CI of range). Vital lung capacity was decreased, compared to a non-OI population (mean 1.57 L). This was correlated to the degree of scoliosis (st. β - 0.40, P = 0.03), especially in increasing thoracic curves. Restrictive lung pathophysiology was shown in our study population with a mean FEV1/FVC ratio of 0.85.

conclusionsIncreasing thoracic scoliosis was correlated with decreased vital lung capacity in our study population of type III OI patients. High FEV1/FVC ratios found in this study population show restrictive lung pathophysiology. Therefore, it is plausible that the pulmonary impairment found in type III OI patients is a combined issue, partly associated to scoliosis and partly intrinsic to OI.

Indexed as

Lung DiseasesOsteogenesis ImperfectaScoliosisHumansPrevalenceRetrospective StudiesOsteogenesis imperfectaPulmonary functionScoliosis

Identifiers

PMID35604455

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