Željko Jeleč, Dragan Primorac, Vilim Molnar, Darko Antičević
{"title":"解决严重成骨不全和长期双膦酸盐治疗患者的手术挑战:髓内硬化和技术解决方案。","authors":"Željko Jeleč, Dragan Primorac, Vilim Molnar, Darko Antičević","doi":"10.1007/s00264-025-06645-7","DOIUrl":null,"url":null,"abstract":"<p><strong>Purpose: </strong>Osteogenesis imperfecta (OI) is commonly managed with bisphosphonates (BPs) which are associated with significant side effects. This study aimed to investigate intramedullary sclerosis as a potential side effect of prolonged BP use in paediatric patients.</p><p><strong>Methods: </strong>Thirteen children with OI underwent surgery at our hospital. We assessed 21 non-operated bone segments from 13 paediatric patients using a tailored protocol to measure sclerosis severity and its impact on surgical outcomes.</p><p><strong>Results: </strong>Among 21 non-operated segments, 17 exhibited intramedullary sclerosis. Pamidronate cycles inversely correlated with the percentage of the sclerosing area and varus deformity, plateauing after 15 cycles. Angulation in the sagittal plane decreased progressively.</p><p><strong>Conclusions: </strong>Our study introduces intramedullary sclerosis as a possible novel BP-related side-effect in pediatric OI patients. Our findings suggest a critical review of prolonged BP therapy in OI patients, advocating for a surgical intervention threshold after fifteen BP cycles to mitigate complications and optimize treatment outcomes.</p>","PeriodicalId":14450,"journal":{"name":"International Orthopaedics","volume":" ","pages":"2415-2421"},"PeriodicalIF":2.6000,"publicationDate":"2025-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Addressing surgical challenges in patients with severe form of osteogenesis imperfecta and with prolonged bisphosphonate treatment: intramedullary sclerosis and technical solutions.\",\"authors\":\"Željko Jeleč, Dragan Primorac, Vilim Molnar, Darko Antičević\",\"doi\":\"10.1007/s00264-025-06645-7\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Purpose: </strong>Osteogenesis imperfecta (OI) is commonly managed with bisphosphonates (BPs) which are associated with significant side effects. This study aimed to investigate intramedullary sclerosis as a potential side effect of prolonged BP use in paediatric patients.</p><p><strong>Methods: </strong>Thirteen children with OI underwent surgery at our hospital. We assessed 21 non-operated bone segments from 13 paediatric patients using a tailored protocol to measure sclerosis severity and its impact on surgical outcomes.</p><p><strong>Results: </strong>Among 21 non-operated segments, 17 exhibited intramedullary sclerosis. Pamidronate cycles inversely correlated with the percentage of the sclerosing area and varus deformity, plateauing after 15 cycles. Angulation in the sagittal plane decreased progressively.</p><p><strong>Conclusions: </strong>Our study introduces intramedullary sclerosis as a possible novel BP-related side-effect in pediatric OI patients. Our findings suggest a critical review of prolonged BP therapy in OI patients, advocating for a surgical intervention threshold after fifteen BP cycles to mitigate complications and optimize treatment outcomes.</p>\",\"PeriodicalId\":14450,\"journal\":{\"name\":\"International Orthopaedics\",\"volume\":\" \",\"pages\":\"2415-2421\"},\"PeriodicalIF\":2.6000,\"publicationDate\":\"2025-10-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"International Orthopaedics\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1007/s00264-025-06645-7\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2025/9/3 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q2\",\"JCRName\":\"ORTHOPEDICS\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"International Orthopaedics","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s00264-025-06645-7","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/9/3 0:00:00","PubModel":"Epub","JCR":"Q2","JCRName":"ORTHOPEDICS","Score":null,"Total":0}
Addressing surgical challenges in patients with severe form of osteogenesis imperfecta and with prolonged bisphosphonate treatment: intramedullary sclerosis and technical solutions.
Purpose: Osteogenesis imperfecta (OI) is commonly managed with bisphosphonates (BPs) which are associated with significant side effects. This study aimed to investigate intramedullary sclerosis as a potential side effect of prolonged BP use in paediatric patients.
Methods: Thirteen children with OI underwent surgery at our hospital. We assessed 21 non-operated bone segments from 13 paediatric patients using a tailored protocol to measure sclerosis severity and its impact on surgical outcomes.
Results: Among 21 non-operated segments, 17 exhibited intramedullary sclerosis. Pamidronate cycles inversely correlated with the percentage of the sclerosing area and varus deformity, plateauing after 15 cycles. Angulation in the sagittal plane decreased progressively.
Conclusions: Our study introduces intramedullary sclerosis as a possible novel BP-related side-effect in pediatric OI patients. Our findings suggest a critical review of prolonged BP therapy in OI patients, advocating for a surgical intervention threshold after fifteen BP cycles to mitigate complications and optimize treatment outcomes.
期刊介绍:
International Orthopaedics, the Official Journal of the Société Internationale de Chirurgie Orthopédique et de Traumatologie (SICOT) , publishes original papers from all over the world. The articles deal with clinical orthopaedic surgery or basic research directly connected with orthopaedic surgery. International Orthopaedics will also link all the members of SICOT by means of an insert that will be concerned with SICOT matters.
Finally, it is expected that news and information regarding all aspects of orthopaedic surgery, including meetings, panels, instructional courses, etc. will be brought to the attention of the readers.
Manuscripts submitted for publication must contain a statement to the effect that all human studies have been approved by the appropriate ethics committee and have therefore been performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki. It should also be stated clearly in the text that all persons gave their informed consent prior to their inclusion in the study. Details that might disclose the identity of the subjects under study should be omitted.
Reports of animal experiments must state that the "Principles of laboratory animal care" (NIH publication No. 85-23, revised 1985) were followed, as well as specific national laws (e.g. the current version of the German Law on the Protection of Animals) where applicable.
The editors reserve the right to reject manuscripts that do not comply with the above-mentioned requirements. The author will be held responsible for false statements or for failure to fulfil the above-mentioned requirements.