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

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.

PET with Different Radiopharmaceuticals in Neuroendocrine Neoplasms: An Umbrella Review of Published Meta-Analyses

“…In this umbrella review, we did not consider the use of PET with different radiopharmaceuticals in patients with congenital hyperinsulinism (CHI) or oncogenic osteomalacia (OO) because these conditions are not caused by neuroendocrine neoplasms sensu stricto. However, we should underline that several meta-analyses have demonstrated that 18 F-FDOPA PET/CT has high sensitivity and specificity in differentiating between the focal and diffuse form of CHI and in localizing the focal form of CHI [54][55][56], whereas in detecting culprit tumors causing OO 68 Ga-SSA PET/CT has better sensitivity than other PET and SPECT methods and should be used as first-line imaging modality [57][58][59].…”

Delayed diagnosis of occult phosphaturic mesenchymal tumor in the foot

“…All the 3 Gallium 68 labeled DOTA-peptides currently available for use, DOTATATE, DOTATOC and DOTANOC, have a higher and and wider affinity for SSTR, resulting in better visualization of somatostatin avid lesions compared to Indium 111-labeled octreotide single-photon emission and computed tomography (SPECT) [12] . Data from a recent systematic review and meta-analysis support the use of Gallium 68 DOTA-peptides PET-CT as first-line imaging study in tumor-induced osteomalacia [13] . Lesions identified on functional imaging require a focused anatomic assessment with CT and/or MRI for preoperative planning.…”

Comparison of 18F‐FDG PET/CT and 68Ga‐DOTATATE PET/CT in the Targeted Imaging of Culprit Tumors Causing Osteomalacia

“…The systematic review and meta‐analysis showed that since SSTRs is highly expressed in most of the culprit tumors that cause osteomalcic, the culprit tumors can be well diagnosed in the exploration of TIO patients using SSTRS‐PET/CT. Even if false negative findings are possible, in about 10% of cases, it should be underlined that SSTR‐PET/CT has allowed the detection of culprit tumors which remained occult with conventional imaging methods in most of the cases 19,20 . The main reason for the false negative results of conventional imaging methods is that the primary culprit of osteomalacia is the small size and variable location of the tumor 21 .…”