Tumor-Induced Osteomalacia: Treatment Progress Using Burosumab, an Anti-FGF23 Monoclonal Antibody

Whyte, Michael P.

doi:10.1002/jbmr.4280

Cited by 4 publications

(5 citation statements)

References 6 publications

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“…Treatment with Burosumab improves biochemical abnormalities (serum phosphate, alkaline phosphatase, and 1,25(OH)2D levels), radiographic signs of rickets (delayed growth, bow legs, weakness, and bone pain due to hypophosphatemia), growth, fracture healing, and impaired mineralization in patients with XLH (Dahir et al, 2020; Fukumoto, 2014, 2021; Gladding et al, 2021; Imel, 2021; Michigami, 2019; Schindeler et al, 2020; Verbueken & Moe, 2021). In adults with TIO, Burosumab was associated with improvements in phosphate metabolism and osteomalacia (Imel et al, 2015; Jan de Beur et al, 2021; Whyte, 2021).…”

Section: Development Of Fgf Pathway Inhibitors As Pharmaceuticalsmentioning

confidence: 99%

New developments in the biology of fibroblast growth factors

Ornitz

Itoh

2022

WIREs Mechanisms of Disease

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The fibroblast growth factor (FGF) family is composed of 18 secreted signaling proteins consisting of canonical FGFs and endocrine FGFs that activate four receptor tyrosine kinases (FGFRs 1-4) and four intracellular proteins (intracellular FGFs or iFGFs) that primarily function to regulate the activity of voltagegated sodium channels and other molecules. The canonical FGFs, endocrine FGFs, and iFGFs have been reviewed extensively by us and others. In this review, we briefly summarize past reviews and then focus on new developments in the FGF field since our last review in 2015. Some of the highlights in the past 6 years include the use of optogenetic tools, viral vectors, and inducible transgenes to experimentally modulate FGF signaling, the clinical use of small molecule FGFR inhibitors, an expanded understanding of endocrine FGF signaling, functions for FGF signaling in stem cell pluripotency and differentiation, roles for FGF signaling in tissue homeostasis and regeneration, a continuing elaboration of mechanisms of FGF signaling in development, and an expanding appreciation of roles for FGF signaling in neuropsychiatric diseases.

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Section: Development Of Fgf Pathway Inhibitors As Pharmaceuticalsmentioning

confidence: 99%

New developments in the biology of fibroblast growth factors

Ornitz

Itoh

2022

WIREs Mechanisms of Disease

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“…In addition to convenient dosing, it has better efficacy and requires minimal monitoring compared with traditional medical management 2. While long-term studies are still pending to determine safety and optimal treatment duration, burosumab is currently the best option for most patients with unresectable tumours since, at adequate doses, can result in a rapid resolution of symptoms and relatively rapid resolution of bone fractures 3 9. Our case demonstrates these points nicely.…”

Section: Discussionmentioning

confidence: 63%

Tumour-induced osteomalacia due to residual benign glomangioma

2022

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Tumour-induced osteomalacia (TIO) is a rare paraneoplastic syndrome. The constellation of findings of unprovoked fractures, hypophosphataemia, urinary phosphate wasting and a negative genetic evaluation suggest a TIO diagnosis. Tumours leading to TIO are often small and difficult to localise using standard imaging studies. The68Ga-DOTATATE CT/positron emission tomography, a somatostatin receptor imaging modality, is the radiographical study of choice for localisation. It is highly sensitive and specific since tumours that cause oncogenic osteomalacia have been shown to express somatostatin receptors. Complete surgical resection is the treatment of choice; however, it may not always be feasible. Burosumab, a human anti-fibroblast growth factor-23 monoclonal antibody, is a therapeutic option in cases of unresectable TIO to normalise phosphorus levels and improve fracture healing. Our patient was initiated on burosumab, which led to healing of his fractures and profound symptomatic improvement of his pain. TIO is often undiagnosed for many years, leading to significant patient morbidity.

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“…Nine cases (9/13, 62.9 %) had the average peak blood phosphorus levels at the 24th week, while six cases (6/13, 46.2 %) had the average low blood phosphorus level at the 48th week [133] . For TIO patients whose tumors cannot be removed, burosumab is a potential effective treatment method [133] , [134] , [135] , [136] , [137] . The recommended initial dose of burosumab for TIO treatment is 0.5 mg/kg, once every 4 weeks; the maximum dose is 2 mg/kg (not exceeding 180 mg) every 2 weeks.…”

Section: Other Management Optionsmentioning

confidence: 99%

“…The anti FGF-23 monoclonal antibodies require more evidence to support their effectiveness and safety in TIO treatment. Surgery is the frontline method for TIO treatment [28] , [137] , [140] . Summarizing the experience of orthopedic surgical treatment for TIO with causative tumors located in different parts is of great clinical value, and is of crucial significance for improving the effectiveness of orthopedic surgical treatment.…”

Section: Future Prospectsmentioning

confidence: 99%

Preoperative evaluation and orthopedic surgical strategies for tumor-induced osteomalacia

Liu,

Zhou,

Liu

et al. 2024

Journal of Bone Oncology

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Tumor-Induced Osteomalacia: Treatment Progress Using Burosumab, an Anti-FGF23 Monoclonal Antibody

Cited by 4 publications

References 6 publications

New developments in the biology of fibroblast growth factors

New developments in the biology of fibroblast growth factors

Tumour-induced osteomalacia due to residual benign glomangioma

Preoperative evaluation and orthopedic surgical strategies for tumor-induced osteomalacia

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