Atypical Femoral Fracture in Hypophosphatasia: A Systematic Review
Objective. To summarize the characteristics of all reported patients with hypophosphatasia (HPP) who sustained atypical femoral fracture (AFF) and identify all available evidence to quantify the rate of coexistence between HPP and AFF. Methods. Potentially eligible articles were identified from the...
| Những tác giả chính: | , , , , , , |
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| Định dạng: | Bài viết |
| Ngôn ngữ: | English |
| Được phát hành: |
Wiley
2023-01-01
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| Loạt: | International Journal of Endocrinology |
| Truy cập trực tuyến: | http://dx.doi.org/10.1155/2023/5544148 |
| _version_ | 1826820638535319552 |
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| author | Nipith Charoenngam Jerapas Thongpiya Pitchaporn Yingchoncharoen Ben Ponvilawan Mehmet S. Marangoz Jirat Chenbhanich Patompong Ungprasert |
| author_facet | Nipith Charoenngam Jerapas Thongpiya Pitchaporn Yingchoncharoen Ben Ponvilawan Mehmet S. Marangoz Jirat Chenbhanich Patompong Ungprasert |
| author_sort | Nipith Charoenngam |
| collection | DOAJ |
| description | Objective. To summarize the characteristics of all reported patients with hypophosphatasia (HPP) who sustained atypical femoral fracture (AFF) and identify all available evidence to quantify the rate of coexistence between HPP and AFF. Methods. Potentially eligible articles were identified from the MEDLINE and EMBASE databases from its inception to September 2022, using a search strategy consisting of terms related to “Hypophosphatasia” and “Atypical femoral fracture.” Eligible articles must report one of the following information: (1) individual data of patients diagnosed with HPP and AFF, (2) prevalence of HPP among patients with AFF, or (3) prevalence of AFF among patients of HPP. Characteristics of patients reported in each study were extracted. Results. A total of 148 articles were identified. After the systematic review, 24 articles met the eligibility criteria. A total of 28 patients with AFF and HPP were identified. The mean ± SD age of the reported patients was 53.8 ± 12.5 years, and 22 patients (78.6%) were female. Nine patients (32.1%) received antiresorptive medication (bisphosphonate and/or denosumab), and two patients (7.1%) received teriparatide prior to the development of AFF. Seven (25.0%) and eighteen (64.3%) patients sustained unilateral and bilateral AFF, respectively (laterality not reported in three cases). Thirteen patients (46.4%) had a history of fractures at other sites. Four (14.3%) and seven (25.0%) patients received asfotase alfa and teriparatide after sustaining AFF. Two studies reported the prevalence of AFF among patients with HPP of approximately 10%. One study reported one HPP patient in a cohort of 72 patients with AFF. Conclusions. Based on the limited evidence, AFF occurred in up to 10% of patients with HPP. Based on the 28 case reports, about two-thirds did not receive antiresorptive treatment, suggesting that the HPP itself could potentially be a risk factor for AFF. |
| first_indexed | 2024-03-11T23:36:03Z |
| format | Article |
| id | doaj.art-842ff92352cd475286adb24bd7596037 |
| institution | Directory Open Access Journal |
| issn | 1687-8345 |
| language | English |
| last_indexed | 2025-02-16T06:32:56Z |
| publishDate | 2023-01-01 |
| publisher | Wiley |
| record_format | Article |
| series | International Journal of Endocrinology |
| spelling | doaj.art-842ff92352cd475286adb24bd75960372025-02-03T06:45:35ZengWileyInternational Journal of Endocrinology1687-83452023-01-01202310.1155/2023/5544148Atypical Femoral Fracture in Hypophosphatasia: A Systematic ReviewNipith Charoenngam0Jerapas Thongpiya1Pitchaporn Yingchoncharoen2Ben Ponvilawan3Mehmet S. Marangoz4Jirat Chenbhanich5Patompong Ungprasert6Department of MedicineDepartment of MedicineDepartment of MedicineDepartment of MedicineDepartment of Endocrinology and MetabolismDepartment of Genetics and Genomic SciencesDepartment of Rheumatic and Immunologic DiseasesObjective. To summarize the characteristics of all reported patients with hypophosphatasia (HPP) who sustained atypical femoral fracture (AFF) and identify all available evidence to quantify the rate of coexistence between HPP and AFF. Methods. Potentially eligible articles were identified from the MEDLINE and EMBASE databases from its inception to September 2022, using a search strategy consisting of terms related to “Hypophosphatasia” and “Atypical femoral fracture.” Eligible articles must report one of the following information: (1) individual data of patients diagnosed with HPP and AFF, (2) prevalence of HPP among patients with AFF, or (3) prevalence of AFF among patients of HPP. Characteristics of patients reported in each study were extracted. Results. A total of 148 articles were identified. After the systematic review, 24 articles met the eligibility criteria. A total of 28 patients with AFF and HPP were identified. The mean ± SD age of the reported patients was 53.8 ± 12.5 years, and 22 patients (78.6%) were female. Nine patients (32.1%) received antiresorptive medication (bisphosphonate and/or denosumab), and two patients (7.1%) received teriparatide prior to the development of AFF. Seven (25.0%) and eighteen (64.3%) patients sustained unilateral and bilateral AFF, respectively (laterality not reported in three cases). Thirteen patients (46.4%) had a history of fractures at other sites. Four (14.3%) and seven (25.0%) patients received asfotase alfa and teriparatide after sustaining AFF. Two studies reported the prevalence of AFF among patients with HPP of approximately 10%. One study reported one HPP patient in a cohort of 72 patients with AFF. Conclusions. Based on the limited evidence, AFF occurred in up to 10% of patients with HPP. Based on the 28 case reports, about two-thirds did not receive antiresorptive treatment, suggesting that the HPP itself could potentially be a risk factor for AFF.http://dx.doi.org/10.1155/2023/5544148 |
| spellingShingle | Nipith Charoenngam Jerapas Thongpiya Pitchaporn Yingchoncharoen Ben Ponvilawan Mehmet S. Marangoz Jirat Chenbhanich Patompong Ungprasert Atypical Femoral Fracture in Hypophosphatasia: A Systematic Review International Journal of Endocrinology |
| title | Atypical Femoral Fracture in Hypophosphatasia: A Systematic Review |
| title_full | Atypical Femoral Fracture in Hypophosphatasia: A Systematic Review |
| title_fullStr | Atypical Femoral Fracture in Hypophosphatasia: A Systematic Review |
| title_full_unstemmed | Atypical Femoral Fracture in Hypophosphatasia: A Systematic Review |
| title_short | Atypical Femoral Fracture in Hypophosphatasia: A Systematic Review |
| title_sort | atypical femoral fracture in hypophosphatasia a systematic review |
| url | http://dx.doi.org/10.1155/2023/5544148 |
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