Abstract
Objectives
XLHR in children with a Rickets Severity Score ≥2 can now be treated with the new antibody drug, Burosumab, which prevents bone deformities and increases gait endurance. This study illustrates the extent of complications in the traditional orthopedic treatment of XLHR. The impact of surgery and severity of complications in this patient population has not been systematically assessed before.
Methods
The search strategy resulted in 215 studies and data were collected from 19 eligible studies and complications were categorized. Four medical charts of patients with XLHR at Aalborg University Hospital were assessed.
Results
One complication occurred on average per surgical procedure for XLHR in the published literature. The 168 reported complications were categorized as follows: Type I (n=79): Complications with minimal intervention required and treatment goal still achieved, Type II (n=41): Complications with substantial change in treatment plan and treatment goal still achieved, Type IIIA (n=23): Complications with failure to achieve treatment goal and no new pathology or permanent sequelae, Type IIIB (n=25): Complications with failure to achieve treatment goal and/or new pathology or permanent sequelae.
Conclusions
In average, one complication occurred per surgery and the severity of complications were substantial. The treatment goal was not achieved in 28% of surgeries whereof half of them resulted in permanent sequalae or new pathology. Our findings support the use of Burosumab for treatment of the skeletal changes in XLHR as the reported side-effects in Burosumab treatment appear negligible compared to the impact of surgeries and related complications (Imel EA, Glorieux FH, Whyte MP, Munns CF, Ward LM, Nilsson O, et al. Burosumab versus conventional therapy in children with X-linked hypophosphataemia: a randomised, active-controlled, open-label, phase 3 trial. Lancet 2019;393:2416–27). However, orthopedic surgery might still be needed for correcting deformities restricting activities of daily living in XLHR patients.
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Research funding: None declared.
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Author contributions: All authors have accepted responsibility for the entire content of this manuscript and approved its submission.
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Competing interests: Authors state no conflict of interest.
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Informed consent: Informed consent was obtained from all individuals included in this study.
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Ethical approval: The local Institutional Review Board deemed the study exempt from review.
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Supplementary Material
The online version of this article offers supplementary material (https://doi.org/10.1515/jpem-2021-0775).
© 2022 Walter de Gruyter GmbH, Berlin/Boston
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- Frontmatter
- Review Article
- Effects of vitamin D supplementation on glycemic control of children and adolescents with type 1 diabetes mellitus: a systematic review
- Original Articles
- A preliminary study on the relationship between environmental endocrine disruptors and precocious puberty in girls
- Tissue concentration of aldosterone in fetal adrenals of intrauterine death cases
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- Comparison of insulin sensitivity indices for detection of double diabetes in Indian adolescents with type 1 diabetes
- Evaluation of the growth and nutritional status of preschool children: a pilot study in the cold area of China
- Arterial hypertension is associated with an increased risk of metabolic complications in pediatric patient with obesity
- From pregnancy to breastfeeding: adequate maternal body mass index is essential to prevent a high body mass index in your children
- Screening of non-syndromic early-onset child and adolescent obese patients in terms of LEP, LEPR, MC4R and POMC gene variants by next-generation sequencing
- The association between vitamin B12, folate, homocysteine levels, and carotid intima-media thickness in children with obesity: a cross-sectional study
- Comprehensive assessment of cardiovascular disease risk in children with short stature due to isolated growth hormone deficiency: a case-control study
- Visceral adiposity is related to insulin sensitivity and inflammation in adolescents with obesity and mild sleep disordered breathing
- Recurrent hypoglycemic seizure as a presenting symptom of post-TBI hypopituitarism in children: a case report, review and proposed protocol
- Evaluation of the etiological and clinical characteristics of pediatric central diabetes insipidus
- Case Reports
- Clinical and radiologic evaluation of a Turkish family with hypochondroplasia and a rare FGFR3 variant
- Rituximab therapy in ROHHAD(NET) syndrome
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