Elevated FGF23 in a patient with hypophosphatemic osteomalacia associated with neurofibromatosis type 1.


Journal

Bone
ISSN: 1873-2763
Titre abrégé: Bone
Pays: United States
ID NLM: 8504048

Informations de publication

Date de publication:
12 2019
Historique:
received: 22 03 2019
revised: 10 08 2019
accepted: 28 08 2019
pubmed: 3 9 2019
medline: 29 8 2020
entrez: 3 9 2019
Statut: ppublish

Résumé

The mechanism behind hypophosphatemia in the setting of neurofibromatosis type 1 (NF1) is not known. We describe a possible role of fibroblast growth factor-23 (FGF23) in the pathophysiology of hypophosphatemia in a patient with NF1. A 34-year woman with NF1 presented with severe hypophosphatemia, osteomalacia, and elevated plasma FGF23. The patient had considerable improvement on replacement of oral phosphate. Two Ga68 DOTANOC PET-CT scans over a period of 2 years failed to detect any localized uptake. Immuno-staining for FGF23 was absent in the neural-derived tumour cells of the neurofibromas in the proband. The patient with NF1 had elevated circulating FGF23. Tumour cells in the neurofibroma tissues did not stain for FGF23 on IHC. It is unlikely for neurofibromas to contribute to high circulating FGF23 levels in the proband.

Identifiants

pubmed: 31476437
pii: S8756-3282(19)30345-X
doi: 10.1016/j.bone.2019.115055
pii:
doi:

Substances chimiques

FGF23 protein, human 0
Fibroblast Growth Factors 62031-54-3
Fibroblast Growth Factor-23 7Q7P4S7RRE

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

115055

Informations de copyright

Copyright © 2019 Elsevier Inc. All rights reserved.

Auteurs

Saroj Kumar Sahoo (SK)

Department of Endocrinology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India.

Priyanka Kushwaha (P)

Division of Endocrinology, Central Drug Research Institute, Lucknow, India.

Niharika Bharti (N)

Department of Pathology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India.

Vikram Khedgikar (V)

Division of Endocrinology, Central Drug Research Institute, Lucknow, India.

Ritu Trivedi (R)

Division of Endocrinology, Central Drug Research Institute, Lucknow, India.

Vinita Agrawal (V)

Department of Pathology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India.

Naseer Ahmad (N)

Division of Endocrinology, Central Drug Research Institute, Lucknow, India.

Ghazala Zaidi (G)

Department of Endocrinology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India.

Lily Pal (L)

Department of Pathology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India.

Nobuaki Ito (N)

Division of Nephrology and Endocrinology, The University of Tokyo Hospital, Tokyo, Japan.

Eesh Bhatia (E)

Department of Endocrinology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India. Electronic address: ebhatia@sgpgi.ac.in.

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