Detection Rate of Culprit Tumors Causing Osteomalacia Using Somatostatin Receptor PET/CT: Systematic Review and Meta-Analysis.

John O. Prior,Fabio Becce,Nathalie Testart,Giorgio Treglia,Luca Giovanella,Marie Nicod Lalonde,Christian Candrian,Mario Jreige,Christel Kamani,Marie Meyer,Sarah Boughdad,Niklaus Schaefer,Martin Riegger,Barbara Muoio

doi:10.3390/diagnostics10010002

Abstract

Background: Tumor-induced or oncogenic osteomalacia (TIO) is a rare paraneoplastic syndrome in which osteomalacia is a consequence of fibroblast growth factor 23 (FGF23) secretion by a mesenchymal tumor. The localization of the culprit lesion in patients with TIO is often challenging. Several studies have evaluated the detection rate (DR) of these tumors using somatostatin receptor positron emission tomography (SSTR-PET/CT). We aimed to summarize literature findings on this topic providing pooled estimates of DR. Methods: A comprehensive literature search by screening PubMed, Embase and Cochrane library electronic databases through August 2019 was performed. The pooled DR of culprit tumors using SSTR-PET/CT in patients with TIO was calculated using a random-effects statistical model. Results: Fourteen studies on the use of SSTR-PET/CT in detecting the culprit tumor in patients with TIO were included in the qualitative analysis. The pooled DR of SSTR-PET/CT on a per-patient-based analysis calculated using eleven studies (166 patients) was 87.6% (95% confidence interval (95% CI) 80.2–95.1%). Statistical heterogeneity among studies was detected (I-square = 63%), likely due to the use of different radiolabeled somatostatin analogues, as demonstrated by a subgroup analysis. Conclusions: Despite limited literature data due to the rarity of the disease, SSTR-PET/CT demonstrated a very high DR of culprit tumors in patients with TIO and it could be used as first-line imaging method for this indication.

Highlights

Tumor-induced or oncogenic osteomalacia (TIO) is a rare paraneoplastic syndrome [1,2].The majority of TIO cases are caused by phosphaturic mesenchymal tumors [3]
We aimed to perform a systematic review and meta-analysis on the detection rate (DR) of culprit tumors in patients with TIO using SSTR-PET/computed tomography (CT) to provide evidence-based data that could be useful in justifying the use of this technique as first-line imaging method for this indication
The DR of culprit tumors using SSTR-positron emission tomography/CT (PET/CT) in patients with TIO ranged from 53% to 100%, with a pooled outcome measure of 87.6% (Figure 3)

Summary

Introduction

Tumor-induced or oncogenic osteomalacia (TIO) is a rare paraneoplastic syndrome [1,2].The majority of TIO cases are caused by phosphaturic mesenchymal tumors [3]. Tumor-induced or oncogenic osteomalacia (TIO) is a rare paraneoplastic syndrome [1,2]. The culprit tumors of TIO produce fibroblast growth factor 23 (FGF23), a protein which regulates renal phosphate handling and 25-hydroxyvitamin D 1α-hydroxylase activity. Tumor-induced or oncogenic osteomalacia (TIO) is a rare paraneoplastic syndrome in which osteomalacia is a consequence of fibroblast growth factor 23 (FGF23) secretion by a mesenchymal tumor. The pooled DR of culprit tumors using SSTR-PET/CT in patients with TIO was calculated using a random-effects statistical model. Results: Fourteen studies on the use of SSTR-PET/CT in detecting the culprit tumor in patients with TIO were included in the qualitative analysis. The pooled DR of SSTR-PET/CT on a per-patient-based analysis calculated using eleven studies (166 patients) was

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