Evidence for Metaphyseal Fractures Typical of Abuse in Osteogenesis Imperfecta: A Systematic Review.
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All Authors
Riley, E.
Paddock M.
Fairhurst J.
Halliday K.
Jeanes A.
Johnson K.
Offiah A.
LTHT Author
Jeanes, Annmarie
LTHT Department
Radiology
Non Medic
Publication Date
2023
Item Type
Conference Abstract
Language
Subject
Subject Headings
Abstract
OBJECTIVE: To search for evidence of metaphyseal fractures (also known as classic metaphyseal lesions, CMLs) in children aged 0 to 2 years with osteogenesis imperfecta (OI) to differentiate from inflicted injury in infants with unexplained fractures. METHOD(S): A systematic review of Medline, Cinahl and Web of Science was conducted. Inclusion criteria: Studies of children < 2 years of age with OI which report the number of CMLs. Studies were excluded in which the full article was not retrievable, not in English or the authors neither define nor illustrate what they mean by 'metaphyseal fracture'. All radiographs included of 'metaphyseal' fractures were independently reviewed by 6 paediatric radiologists as to the quality of the illustrated images and whether a CML was present. The quality of the included papers will be assessed using the QUADAS-2 tool. RESULT(S): A total of 256 articles were retrieved with 81 duplicates. There were 175 articles screened based on abstract and title and 151 excluded. A total of 26 articles were reviewed with a hand search of the references adding 2 articles. Only 4 articles were included. Article 1 reviewed 41 children with OI and found 7 with 'metaphyseal' fractures, aged between 1 day and 3 years. Article 2 reported 3 'metaphyseal' fractures in a 16-month-old male. Despite a diagnosis of type IV OI (not genetically confirmed), there remained concerns about physical abuse. Article 3 reported a 7-month-old female who presented with multiple fractures including 2 'corner' fractures. The child was suspected both to have OI and to have been abused. Article 4 reported a pair of siblings with 'metaphyseal' fractures who both had a FKBP10 genetic mutation causing type XI OI. The 4 studies included 8 images which the authors purport to show 9 'metaphyseal' fractures. Of these, 3 images were considered of diagnostic quality, and none were classed as CMLs by all 6 radiologists. CONCLUSION(S): There is limited scientific evidence that OI predisposes infants and young children to CMLs. Physical abuse should always be considered as a differential diagnosis in infants and young children with CMLs, even in the context of confirmed OI.
Journal
JBMR Plus