Fibroblast growth factor 23 in oncogenic osteomalacia and X-linked hypophosphatemia - PubMed (original) (raw)
. 2003 Apr 24;348(17):1656-63.
doi: 10.1056/NEJMoa020881.
Richard Zahradnik, Tobias Larsson, Kenneth E White, Toshitsugu Sugimoto, Yasuo Imanishi, Takehisa Yamamoto, Geeta Hampson, Hiroyuki Koshiyama, Osten Ljunggren, Koichi Oba, In Myung Yang, Akimitsu Miyauchi, Michael J Econs, Jeffrey Lavigne, Harald Jüppner
Affiliations
- PMID: 12711740
- DOI: 10.1056/NEJMoa020881
Free article
Fibroblast growth factor 23 in oncogenic osteomalacia and X-linked hypophosphatemia
Kenneth B Jonsson et al. N Engl J Med. 2003.
Free article
Abstract
Background: Mutations in fibroblast growth factor 23 (FGF-23) cause autosomal dominant hypophosphatemic rickets. Clinical and laboratory findings in this disorder are similar to those in oncogenic osteomalacia, in which tumors abundantly express FGF-23 messenger RNA, and to those in X-linked hypophosphatemia, which is caused by inactivating mutations in a phosphate-regulating endopeptidase called PHEX. Recombinant FGF-23 induces phosphaturia and hypophosphatemia in vivo, suggesting that it has a role in phosphate regulation. To determine whether FGF-23 circulates in healthy persons and whether it is elevated in those with oncogenic osteomalacia or X-linked hypophosphatemia, an immunometric assay was developed to measure it.
Methods: Using affinity-purified, polyclonal antibodies against [Tyr223]FGF-23(206-222)amide and [Tyr224]FGF-23(225-244)amide, we developed a two-site enzyme-linked immunosorbent assay that detects equivalently recombinant human FGF-23, the mutant form in which glutamine is substituted for arginine at position 179 (R179Q), and synthetic human FGF-23(207-244)amide. Plasma or serum samples from 147 healthy adults (mean [+/-SD] age, 48.4+/-19.6 years) and 26 healthy children (mean age, 10.9+/-5.5 years) and from 17 patients with oncogenic osteomalacia (mean age, 43.0+/-13.3 years) and 21 patients with X-linked hypophosphatemia (mean age, 34.9+/-17.2 years) were studied.
Results: Mean FGF-23 concentrations in the healthy adults and children were 55+/-50 and 69+/-36 reference units (RU) per milliliter, respectively. Four patients with oncogenic osteomalacia had concentrations ranging from 426 to 7970 RU per milliliter, which normalized after tumor resection. FGF-23 concentrations were 481+/-528 RU per milliliter in those with suspected oncogenic osteomalacia and 353+/-510 RU per milliliter (range, 31 to 2335) in those with X-linked hypophosphatemia.
Conclusions: FGF-23 is readily detectable in the plasma or serum of healthy persons and can be markedly elevated in those with oncogenic osteomalacia or X-linked hypophosphatemia, suggesting that this growth factor has a role in phosphate homeostasis. FGF-23 measurements might improve the management of phosphate-wasting disorders.
Copyright 2003 Massachusetts Medical Society
Comment in
- Oncogenic osteomalacia--a complex dance of factors.
Carpenter TO. Carpenter TO. N Engl J Med. 2003 Apr 24;348(17):1705-8. doi: 10.1056/NEJMe030037. N Engl J Med. 2003. PMID: 12711747 No abstract available. - Fibroblast growth factor 23 in oncogenic osteomalacia and X-linked hypophosphatemia.
Kida Y. Kida Y. N Engl J Med. 2003 Jul 31;349(5):505-6; author reply 505-6. doi: 10.1056/NEJM200307313490517. N Engl J Med. 2003. PMID: 12890852 No abstract available. - Fibroblast growth factor 23 in oncogenic osteomalacia and X-linked hypophosphatemia.
Fukumoto S, Yamashita T. Fukumoto S, et al. N Engl J Med. 2003 Jul 31;349(5):505-6; author reply 505-6. N Engl J Med. 2003. PMID: 12892105 No abstract available.
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