| No. . | Study . | Title and description . | Reason for exclusion . |
|---|---|---|---|
| SR on burosumab vs conventional therapy or no treatment | |||
| 1 | Gadion, 2022 (20) | Burosumab and Dental Abscesses in Children With X-Linked Hypophosphatemia. | With only 1 observational study and 1 RCT (17) meeting our inclusion criteria, the RCT was the study that we included. It is not possible to pool the dental data from the observational study with that of the RCT and in this case meta-analysis was not possible. |
| 2 | Imel, 2023 (21) | Burosumab Versus Phosphate/Active Vitamin D in Pediatric X-Linked Hypophosphatemia: A Sub-group Analysis by Dose Level. | Post hoc analysis of included RCT (17), no additional data on outcomes of interest. |
| 3 | Akta, 2023 (22) | The ankle in XLH: Reduced Motion, Power and Quality of Life. | Cross-sectional study, same as reason (No.1). |
| 4 | Ward, 2022 (23) | Effect of Burosumab Compared with Conventional Therapy on Younger vs Older Children With X-linked Hypophosphatemia. Post hoc analysis of included RCT (17); compared surrogate outcomes between older (≥5 years) and younger children (<5 years). | Post hoc analysis of included RCT (17), no additional data on outcomes of interest. |
| 5 | Ariceta G, 2023 (24) | The International X-Linked Hypophosphatemia (XLH) Registry: First Interim Analysis of Baseline Demographic, Genetic and Clinical Data. Baseline characteristics of children treated with conventional therapy and burosumab; ongoing international, multicenter, non-interventional clinical study for children < 18 years; n = 165 (burosumab), n = 114 (conventional therapy), and 2 untreated. | Observational, offered baseline data only. |
| 6 | Baronio, 2023 (25) | X-Linked Hypophosphatemic Rickets: Cases Series and Literature Review With a Focus on Neurosurgical Management. | Case series. |
| 7 | Barros, 2023 (26) | X-Linked Hypophosphatemia in 4 Generations Due to an Exon 13-15 Duplication in PHEX, in the Absence of the c.*231A>G Variant. | Case series. |
| 8 | Demirbaş, 2023 (27) | A Novel PHEX Mutation in A Case Followed Up With A Diagnosis of X-Linked Hypophosphatemic Rickets. | Case study. |
| 9 | Ewert, 2023 (28) | Effects of Burosumab Treatment on Mineral Metabolism in Children and Adolescents With X-linked Hypophosphatemia. A prospective national registry from Germany, involved 65 children, 28 adolescents with XLH. | Prospective, single-arm study. |
| SR on conventional therapy vs no treatment | |||
| 1 | Verge, 1991 (29) | Effects of Therapy In X-Linked Hypophosphatemic Rickets. Retrospective study from Australia involved children 1 to 16 years; n = 19 (conventional therapy) and 16 (no treatment). | No outcome data on controls other than baseline. |
| 2 | Cheung, 2013 (30) | Cortical and Trabecular Bone Density in X-Linked Hypophosphatemic Rickets. Cross-sectional study from Canada in children < 18 years; n = 21 (conventional therapy) and 6 (no treatment). | Mixed patient population; unable to separate adults data vs children, especially when assessing the impact of intervention vs no intervention on outcomes of interest. |
| 3 | Grote, 2023 (31) | Predicting Rates of Angular Correction After Hemiepiphysiodesis in Patients With X-Linked Hypophosphatemic Rickets. | Retrospective, control group does not have XLH. |
| 4 | Taylor, 1995 (32) | Nephrocalcinosis in X-Linked Hypophosphatemia: Effect of Treatment Versus Disease. Interventional study from the United States; involved adults and children; n = 8 (conventional therapy) and 4 (no treatment). | Provided baseline data only. |
| No. . | Study . | Title and description . | Reason for exclusion . |
|---|---|---|---|
| SR on burosumab vs conventional therapy or no treatment | |||
| 1 | Gadion, 2022 (20) | Burosumab and Dental Abscesses in Children With X-Linked Hypophosphatemia. | With only 1 observational study and 1 RCT (17) meeting our inclusion criteria, the RCT was the study that we included. It is not possible to pool the dental data from the observational study with that of the RCT and in this case meta-analysis was not possible. |
| 2 | Imel, 2023 (21) | Burosumab Versus Phosphate/Active Vitamin D in Pediatric X-Linked Hypophosphatemia: A Sub-group Analysis by Dose Level. | Post hoc analysis of included RCT (17), no additional data on outcomes of interest. |
| 3 | Akta, 2023 (22) | The ankle in XLH: Reduced Motion, Power and Quality of Life. | Cross-sectional study, same as reason (No.1). |
| 4 | Ward, 2022 (23) | Effect of Burosumab Compared with Conventional Therapy on Younger vs Older Children With X-linked Hypophosphatemia. Post hoc analysis of included RCT (17); compared surrogate outcomes between older (≥5 years) and younger children (<5 years). | Post hoc analysis of included RCT (17), no additional data on outcomes of interest. |
| 5 | Ariceta G, 2023 (24) | The International X-Linked Hypophosphatemia (XLH) Registry: First Interim Analysis of Baseline Demographic, Genetic and Clinical Data. Baseline characteristics of children treated with conventional therapy and burosumab; ongoing international, multicenter, non-interventional clinical study for children < 18 years; n = 165 (burosumab), n = 114 (conventional therapy), and 2 untreated. | Observational, offered baseline data only. |
| 6 | Baronio, 2023 (25) | X-Linked Hypophosphatemic Rickets: Cases Series and Literature Review With a Focus on Neurosurgical Management. | Case series. |
| 7 | Barros, 2023 (26) | X-Linked Hypophosphatemia in 4 Generations Due to an Exon 13-15 Duplication in PHEX, in the Absence of the c.*231A>G Variant. | Case series. |
| 8 | Demirbaş, 2023 (27) | A Novel PHEX Mutation in A Case Followed Up With A Diagnosis of X-Linked Hypophosphatemic Rickets. | Case study. |
| 9 | Ewert, 2023 (28) | Effects of Burosumab Treatment on Mineral Metabolism in Children and Adolescents With X-linked Hypophosphatemia. A prospective national registry from Germany, involved 65 children, 28 adolescents with XLH. | Prospective, single-arm study. |
| SR on conventional therapy vs no treatment | |||
| 1 | Verge, 1991 (29) | Effects of Therapy In X-Linked Hypophosphatemic Rickets. Retrospective study from Australia involved children 1 to 16 years; n = 19 (conventional therapy) and 16 (no treatment). | No outcome data on controls other than baseline. |
| 2 | Cheung, 2013 (30) | Cortical and Trabecular Bone Density in X-Linked Hypophosphatemic Rickets. Cross-sectional study from Canada in children < 18 years; n = 21 (conventional therapy) and 6 (no treatment). | Mixed patient population; unable to separate adults data vs children, especially when assessing the impact of intervention vs no intervention on outcomes of interest. |
| 3 | Grote, 2023 (31) | Predicting Rates of Angular Correction After Hemiepiphysiodesis in Patients With X-Linked Hypophosphatemic Rickets. | Retrospective, control group does not have XLH. |
| 4 | Taylor, 1995 (32) | Nephrocalcinosis in X-Linked Hypophosphatemia: Effect of Treatment Versus Disease. Interventional study from the United States; involved adults and children; n = 8 (conventional therapy) and 4 (no treatment). | Provided baseline data only. |
Abbreviations: RCT, randomized controlled trial; XLH, X-linked hypophosphatemia.
| No. . | Study . | Title and description . | Reason for exclusion . |
|---|---|---|---|
| SR on burosumab vs conventional therapy or no treatment | |||
| 1 | Gadion, 2022 (20) | Burosumab and Dental Abscesses in Children With X-Linked Hypophosphatemia. | With only 1 observational study and 1 RCT (17) meeting our inclusion criteria, the RCT was the study that we included. It is not possible to pool the dental data from the observational study with that of the RCT and in this case meta-analysis was not possible. |
| 2 | Imel, 2023 (21) | Burosumab Versus Phosphate/Active Vitamin D in Pediatric X-Linked Hypophosphatemia: A Sub-group Analysis by Dose Level. | Post hoc analysis of included RCT (17), no additional data on outcomes of interest. |
| 3 | Akta, 2023 (22) | The ankle in XLH: Reduced Motion, Power and Quality of Life. | Cross-sectional study, same as reason (No.1). |
| 4 | Ward, 2022 (23) | Effect of Burosumab Compared with Conventional Therapy on Younger vs Older Children With X-linked Hypophosphatemia. Post hoc analysis of included RCT (17); compared surrogate outcomes between older (≥5 years) and younger children (<5 years). | Post hoc analysis of included RCT (17), no additional data on outcomes of interest. |
| 5 | Ariceta G, 2023 (24) | The International X-Linked Hypophosphatemia (XLH) Registry: First Interim Analysis of Baseline Demographic, Genetic and Clinical Data. Baseline characteristics of children treated with conventional therapy and burosumab; ongoing international, multicenter, non-interventional clinical study for children < 18 years; n = 165 (burosumab), n = 114 (conventional therapy), and 2 untreated. | Observational, offered baseline data only. |
| 6 | Baronio, 2023 (25) | X-Linked Hypophosphatemic Rickets: Cases Series and Literature Review With a Focus on Neurosurgical Management. | Case series. |
| 7 | Barros, 2023 (26) | X-Linked Hypophosphatemia in 4 Generations Due to an Exon 13-15 Duplication in PHEX, in the Absence of the c.*231A>G Variant. | Case series. |
| 8 | Demirbaş, 2023 (27) | A Novel PHEX Mutation in A Case Followed Up With A Diagnosis of X-Linked Hypophosphatemic Rickets. | Case study. |
| 9 | Ewert, 2023 (28) | Effects of Burosumab Treatment on Mineral Metabolism in Children and Adolescents With X-linked Hypophosphatemia. A prospective national registry from Germany, involved 65 children, 28 adolescents with XLH. | Prospective, single-arm study. |
| SR on conventional therapy vs no treatment | |||
| 1 | Verge, 1991 (29) | Effects of Therapy In X-Linked Hypophosphatemic Rickets. Retrospective study from Australia involved children 1 to 16 years; n = 19 (conventional therapy) and 16 (no treatment). | No outcome data on controls other than baseline. |
| 2 | Cheung, 2013 (30) | Cortical and Trabecular Bone Density in X-Linked Hypophosphatemic Rickets. Cross-sectional study from Canada in children < 18 years; n = 21 (conventional therapy) and 6 (no treatment). | Mixed patient population; unable to separate adults data vs children, especially when assessing the impact of intervention vs no intervention on outcomes of interest. |
| 3 | Grote, 2023 (31) | Predicting Rates of Angular Correction After Hemiepiphysiodesis in Patients With X-Linked Hypophosphatemic Rickets. | Retrospective, control group does not have XLH. |
| 4 | Taylor, 1995 (32) | Nephrocalcinosis in X-Linked Hypophosphatemia: Effect of Treatment Versus Disease. Interventional study from the United States; involved adults and children; n = 8 (conventional therapy) and 4 (no treatment). | Provided baseline data only. |
| No. . | Study . | Title and description . | Reason for exclusion . |
|---|---|---|---|
| SR on burosumab vs conventional therapy or no treatment | |||
| 1 | Gadion, 2022 (20) | Burosumab and Dental Abscesses in Children With X-Linked Hypophosphatemia. | With only 1 observational study and 1 RCT (17) meeting our inclusion criteria, the RCT was the study that we included. It is not possible to pool the dental data from the observational study with that of the RCT and in this case meta-analysis was not possible. |
| 2 | Imel, 2023 (21) | Burosumab Versus Phosphate/Active Vitamin D in Pediatric X-Linked Hypophosphatemia: A Sub-group Analysis by Dose Level. | Post hoc analysis of included RCT (17), no additional data on outcomes of interest. |
| 3 | Akta, 2023 (22) | The ankle in XLH: Reduced Motion, Power and Quality of Life. | Cross-sectional study, same as reason (No.1). |
| 4 | Ward, 2022 (23) | Effect of Burosumab Compared with Conventional Therapy on Younger vs Older Children With X-linked Hypophosphatemia. Post hoc analysis of included RCT (17); compared surrogate outcomes between older (≥5 years) and younger children (<5 years). | Post hoc analysis of included RCT (17), no additional data on outcomes of interest. |
| 5 | Ariceta G, 2023 (24) | The International X-Linked Hypophosphatemia (XLH) Registry: First Interim Analysis of Baseline Demographic, Genetic and Clinical Data. Baseline characteristics of children treated with conventional therapy and burosumab; ongoing international, multicenter, non-interventional clinical study for children < 18 years; n = 165 (burosumab), n = 114 (conventional therapy), and 2 untreated. | Observational, offered baseline data only. |
| 6 | Baronio, 2023 (25) | X-Linked Hypophosphatemic Rickets: Cases Series and Literature Review With a Focus on Neurosurgical Management. | Case series. |
| 7 | Barros, 2023 (26) | X-Linked Hypophosphatemia in 4 Generations Due to an Exon 13-15 Duplication in PHEX, in the Absence of the c.*231A>G Variant. | Case series. |
| 8 | Demirbaş, 2023 (27) | A Novel PHEX Mutation in A Case Followed Up With A Diagnosis of X-Linked Hypophosphatemic Rickets. | Case study. |
| 9 | Ewert, 2023 (28) | Effects of Burosumab Treatment on Mineral Metabolism in Children and Adolescents With X-linked Hypophosphatemia. A prospective national registry from Germany, involved 65 children, 28 adolescents with XLH. | Prospective, single-arm study. |
| SR on conventional therapy vs no treatment | |||
| 1 | Verge, 1991 (29) | Effects of Therapy In X-Linked Hypophosphatemic Rickets. Retrospective study from Australia involved children 1 to 16 years; n = 19 (conventional therapy) and 16 (no treatment). | No outcome data on controls other than baseline. |
| 2 | Cheung, 2013 (30) | Cortical and Trabecular Bone Density in X-Linked Hypophosphatemic Rickets. Cross-sectional study from Canada in children < 18 years; n = 21 (conventional therapy) and 6 (no treatment). | Mixed patient population; unable to separate adults data vs children, especially when assessing the impact of intervention vs no intervention on outcomes of interest. |
| 3 | Grote, 2023 (31) | Predicting Rates of Angular Correction After Hemiepiphysiodesis in Patients With X-Linked Hypophosphatemic Rickets. | Retrospective, control group does not have XLH. |
| 4 | Taylor, 1995 (32) | Nephrocalcinosis in X-Linked Hypophosphatemia: Effect of Treatment Versus Disease. Interventional study from the United States; involved adults and children; n = 8 (conventional therapy) and 4 (no treatment). | Provided baseline data only. |
Abbreviations: RCT, randomized controlled trial; XLH, X-linked hypophosphatemia.
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