Literature DB >> 30937490

[Tumor-induced osteomalacia caused by an FGF23-secreting myopericytoma : Case report and literature review].

N Muro Bushart1, L Tharun2, R Oheim3, A Paech3, J Kiene3.   

Abstract

BACKGROUND: The symptoms of muscle weakness, bone pain and fragility fractures can be an indication of osteomalacia. Phosphate is often not considered within osteologic parameters, decreased levels are therefore easily overseen. The additional test for fibroblast growth factor 23 (FGF23) as indicator for tumor-induced osteomalacia (TIO) is still largely unfamiliar.
OBJECTIVE: By emphasizing the role of phosphate and furthermore FGF23 in bone metabolism illustrated by the long-term disease process of our clinical case we would like to introduce these parameters to a broader public.
METHODS: We performed a literature search via PubMed and Google Scholar with the relevant key words and summarized the diagnostic and therapeutic information. The studies evaluated were mainly case reports. We present a case report of a 70-year-old patient with TIO and a myopericytoma and retrospectively analyzed the clinical case. The follow-up was 6 months.
RESULTS: Our literature search found one case of TIO and evidence of FGF23 among 124 cases of myopericytomas in total. Over 300 cases of TIO are reported. In our case, we retrospectively found an FGF23-secreting myopericytoma in the phosphaturic mesenchymal tumors (PMT) group to be the cause of pseudarthrosis on the right humerus shaft and increasing disablement in a patient with osteomalacia. After surgical resection the patient was mobile again, and the osteologic parameters, especially phosphate, normalized from 0.21 to 1.52 mmol/l.
CONCLUSION: Low phosphate levels are the decisive indication of TIO in our case. Therefore, we should always think of phosphate level control when dealing with osteomalacia. A hypophosphatemia and hyperphosphaturia should be recognized in time and be diagnostically verified. The additional FGF23 test (c-terminal and intact FGF23) should be considered.

Entities:  

Keywords:  Adult rickets; Fibroblast growth factor 23, human; Hypophosphatemia; Neoplasia; Phosphate

Year:  2020        PMID: 30937490     DOI: 10.1007/s00132-019-03719-4

Source DB:  PubMed          Journal:  Orthopade        ISSN: 0085-4530            Impact factor:   1.087


  108 in total

1.  A myopericytoma of the nose.

Authors:  Ikuko Numata; Satoshi Nakagawa; Satoshi Hasegawa; Setsuya Aiba
Journal:  Acta Derm Venereol       Date:  2010-03       Impact factor: 4.437

Review 2.  Myopericytoma at the Craniocervical Junction: Clinicopathological Report and Review of a Rare Perivascular Neoplasm.

Authors:  Jimmy C Yang; Andrew S Venteicher; Matthew J Koch; Christopher J Stapleton; Gabriel N Friedman; Emma M Venteicher; John H Shin
Journal:  Neurosurgery       Date:  2019-08-01       Impact factor: 4.654

Review 3.  Tumour-induced osteomalacia.

Authors:  Salvatore Minisola; Munro Peacock; Seijii Fukumoto; Cristiana Cipriani; Jessica Pepe; Sri Harsha Tella; Michael T Collins
Journal:  Nat Rev Dis Primers       Date:  2017-07-13       Impact factor: 52.329

Review 4.  Primary hepatic myopericytoma with rhabdoid-like cells: Report of the first case and review of Literature.

Authors:  Jun Li; Qihan You; Lijun Wang; Liming Xu; Han Zhang; Xinru Yu
Journal:  Pathol Int       Date:  2015-01-14       Impact factor: 2.534

5.  Arterial myopericytoma resulting in Guyon's canal syndrome.

Authors:  A Michot; B Chaput; J-M Alet; P Pelissier
Journal:  Hand Surg Rehabil       Date:  2016-05-05       Impact factor: 0.969

6.  An unusual complex karyotype in myopericytoma.

Authors:  Aaron W James; Le Chang; Swati Shrestha; Carlos A Tirado; Sarah M Dry
Journal:  J Orthop       Date:  2015-01-29

7.  Myopericytoma: a pleural-based spindle cell neoplasm off the beaten path.

Authors:  Allison Edgecombe; Rebecca A Peterson; Farid M Shamji; Susan Commons; Harman Sekhon; Marcio M Gomes
Journal:  Int J Surg Pathol       Date:  2011-04       Impact factor: 1.271

8.  Oncogenous osteomalacia. Review of the world literature of 42 cases and report of two new cases.

Authors:  E A Ryan; E Reiss
Journal:  Am J Med       Date:  1984-09       Impact factor: 4.965

9.  Fibroblast growth factor 23 impairs phosphorus and vitamin D metabolism in vivo and suppresses 25-hydroxyvitamin D-1alpha-hydroxylase expression in vitro.

Authors:  Farzana Perwad; Martin Y H Zhang; Harriet S Tenenhouse; Anthony A Portale
Journal:  Am J Physiol Renal Physiol       Date:  2007-08-15

Review 10.  Intravascular myopericytoma in the heel: case report and literature review.

Authors:  José Valero; José L Salcini; Luis Gordillo; José Gallart; David González; Javier Deus; Manuel Lahoz
Journal:  Medicine (Baltimore)       Date:  2015-03       Impact factor: 1.889

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