scholarly journals Effective localization in tumor-induced osteomalacia using 68Ga-DOTATOC-PET/CT, venous sampling and 3T-MRI

Author(s):  
Shintaro Kawai ◽  
Hiroyuki Ariyasu ◽  
Yasushi Furukawa ◽  
Reika Yamamoto ◽  
Shinsuke Uraki ◽  
...  

Summary Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by renal phosphate wasting leading to hypophosphatemia due to excessive actions of fibroblast growth factor 23 (FGF23) produced by the tumors. Although the best way of curing TIO is complete resection, it is usually difficult to detect the culprit tumors by general radiological modalities owing to the size and location of the tumors. We report a case of TIO in which the identification of the tumor by conventional imaging studies was difficult. Nonetheless, a diagnosis was made possible by effective use of multiple modalities. We initially suspected that the tumor existed in the right dorsal aspect of the scapula by 68Ga-DOTATOC positron emission tomography/computed tomography (68Ga-DOTATOC-PET/CT) and supported the result by systemic venous sampling (SVS). The tumor could also be visualized by 3T-magnetic resonance imaging (MRI), although it was not detected by 1.5T-MRI, and eventually be resected completely. In cases of TIO, a stepwise approach of 68Ga-DOTATOC-PET/CT, SVS and 3T-MRI can be effective for confirmation of diagnosis. Learning points: TIO shows impaired bone metabolism due to excessive actions of FGF23 produced by the tumor. The causative tumors are seldom detected by physical examinations and conventional radiological modalities. In TIO cases, in which the localization of the culprit tumors is difficult, 68Ga-DOTATOC-PET/CT should be performed as a screening of localization and thereafter SVS should be conducted to support the result of the somatostatin receptor (SSTR) imaging leading to increased diagnosability. When the culprit tumors cannot be visualized by conventional imaging studies, using high-field MRI at 3T and comparing it to the opposite side are useful after the tumor site was determined.

Diagnostics ◽  
2019 ◽  
Vol 10 (1) ◽  
pp. 2
Author(s):  
Marie Meyer ◽  
Marie Nicod Lalonde ◽  
Nathalie Testart ◽  
Mario Jreige ◽  
Christel Kamani ◽  
...  

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.


2021 ◽  
Vol 8 ◽  
Author(s):  
Jiale Hou ◽  
Yi Yang ◽  
Na Chen ◽  
Dengming Chen ◽  
Shuo Hu

Purpose: A meta-analysis was conducted to investigate the value of the volume parameters based on somatostatin receptor (SSTR)-positron emission tomography (PET) in predicting the prognosis in patients with neuroendocrine tumors (NETs).Material: PUBMED, EMBASE, Cochrane library, and Web of Knowledge were searched from January 1990 to May 2021 for studies evaluating prognostic value of volume-based parameters of SSTR PET/CT in NETs. The terms used were “volume,” “positron emission tomography,” “neuroendocrine tumors,” and “somatostatin receptor.” Pooled hazard ratio (HR) values were calculated to assess the correlations between volumetric parameters, including total tumor volume (TTV) and total-lesion SSTR expression (TL-SSTR), with progression-free survival (PFS) and overall survival (OS). Heterogeneity and subgroup analysis were performed. Funnel plots, Begg's and Egger's test were used to assess possible underlying publication bias.Results: Eight eligible studies involving 593 patients were included in the meta-analysis. In TTV, the pooled HRs of its prognostic value of PFS and OS were 2.24 (95% CI: 1.73–2.89; P < 0.00001) and 3.54 (95% CI, 1.77–7.09; P = 0.0004), respectively. In TL-SSTR, the pooled HR of the predictive value was 1.61 (95% CI, 0.48–5.44, P = 0.44) for PFS.Conclusion: High TTV was associated with a worse prognosis for PFS and OS in with patients NETs. The TTV of SSTR PET is a potential objective prognosis predictor.


2016 ◽  
Vol 36 (suppl_1) ◽  
Author(s):  
Chase W Kessinger ◽  
Ahmed Tawakol ◽  
Gregory R Wojtkiewicz ◽  
Peter K Henke ◽  
Ralph Weissleder ◽  
...  

Objective: While venous thrombosis (VT)-induced inflammation facilitates thrombus resolution, inflammation causes vein wall scarring (VWS). Recently, statins have shown to improve VT resolution and reduce VT inflammatory components. In this study, we hypothesized that early VT inflammation detected by 18F-FDG positron emission tomography/computed tomography (PET/CT) could predict subsequent late stage VWS, and would be attenuated by statin therapy. Methods: Stasis VT was induced in 8-12 week old male C57BL/6 mice (n=31) in either the right jugular vein (n=13) or inferior vena cava (IVC,n=18). Animals in the IVC VT cohort were randomized to statin (n=8) or control (n=10) treatment. Statin, rosuvastatin (5mg/kg), was administered by oral gavage, daily starting 24 hours prior to VT induction; control mice received saline. All mice underwent survival FDG-PET/CT venography imaging on day 2. FDG inflammation signals (standard uptake value=SUV) were measured in the thrombosed vein and compared to the sham-operated venous segments or treatment control. On day 14, mice were sacrificed and VT tissue was resected. Picrosirius red staining allowed measurement of collagen and vein wall thickness in VT sections. Results: FDG-PET/CT at day 2 revealed increased inflammation signal activity in jugular VT (SUV 1.43 ± 0.3 VT vs. 0.81 ± 0.3 contralateral vein, p<0.0001). Statin-treated mice showed a trend of decreased inflammation signal at day 2 in the IVC VT models (SUV 1.02 ± 0.1 statin VT vs. 1.42 ± 0.2 control VT, p=0.07). Day 14 histological analysis revealed significantly reduced vein wall injury in statin-treated animals (thickness, 32±9.4 μm statin; vs. 56.2±14.7 μm control, p=0.02). Day 2 FDG-PET inflammation in VT correlated positively with the magnitude of day 14 VWS (jugular VT, Spearman r=0.62, p=0.02; IVC VT r=0.74, p<0.001, respectively). Conclusions: Quantitative FDG-PET/CT imaging demonstrates that early in vivo VT inflammation predicts subsequent VWS, a driver of post-thrombotic syndrome (PTS). The overall findings strengthen: (i) the link between inflammation and PTS; (ii) the translational potential of FDG-PET inflammation to predict VWS and PTS; and (iii) the concept that statins and other anti-inflammatory therapies could reduce VWS and PTS.


2019 ◽  
Vol 104 (12) ◽  
pp. 6129-6138 ◽  
Author(s):  
Mikkel Andreassen ◽  
Emma Ilett ◽  
Dominik Wiese ◽  
Emily P Slater ◽  
Marianne Klose ◽  
...  

Abstract Introduction Diagnosis and pathological classification of insulinomas are challenging. Aim To characterize localization of tumors, surgery outcomes, and histopathology in patients with insulinoma. Methods Patients with surgically resected sporadic insulinoma were included. Results Eighty patients were included. Seven had a malignant tumor. A total of 312 diagnostic examinations were performed: endoscopic ultrasonography (EUS; n = 59; sensitivity, 70%), MRI (n = 33; sensitivity, 58%), CT (n = 55; sensitivity, 47%), transabdominal ultrasonography (US; n = 45; sensitivity, 40%), somatostatin receptor imaging (n = 17; sensitivity, 29%), 18F-fluorodeoxyglucose positron emission tomography/CT (n = 1; negative), percutaneous transhepatic venous sampling (n = 10; sensitivity, 90%), arterial stimulation venous sampling (n = 20; sensitivity, 65%), and intraoperative US (n = 72; sensitivity, 89%). Fourteen tumors could not be visualized. Invasive methods were used in 7 of these 14 patients and localized the tumor in all cases. Median tumor size was 15 mm (range, 7 to 80 mm). Tumors with malignant vs benign behavior showed less staining for insulin (3 of 7 vs 66 of 73; P = 0.015) and for proinsulin (3 of 6 vs 58 of 59; P < 0.001). Staining for glucagon was seen in 2 of 6 malignant tumors and in no benign tumors (P < 0.001). Forty-three insulinomas stained negative for somatostatin receptor subtype 2a. Conclusion Localization of insulinomas requires many different diagnostic procedures. Most tumors can be localized by conventional imaging, including EUS. For nonvisible tumors, invasive methods may be a useful diagnostic tool. Malignant tumors showed reduced staining for insulin and proinsulin and increased staining for glucagon.


2020 ◽  
Vol 10 (9) ◽  
pp. 2062-2066
Author(s):  
Lang Yang ◽  
Xiaoxia Hu.

Objective: Investigating the value of 18Fluorine-Fluorodesoxyglucose (18F-FDG) positron emission computed tomography/computed tomography (PET/CT) before and after the comprehensive treatment of gynecological malignant tumors is the study purpose. Method: In this study, the patients with gynecological malignant tumor who are examined by 18F-FDG PET/CT test are taken as the study objects, supplemented by image fusion algorithm to achieve the best fusion effect. At the same time, routine imaging examination is performed and confirmed by histopathology. Before and after comprehensive treatment, in terms of PET/CT, its predictive rate of positive as well as negative, sensitivity, specificity as well as accuracy are compared with conventional imaging. Results: Before comprehensive treatment, the positive predictive rate of PET/CT diagnosis is 100%, the sensitivity is 95.00%, and the accuracy is 95.00%. The positive predictive rate of conventional imaging diagnosis is 100%, the sensitivity is 73.75%, and the accuracy is 73.75%. In terms of the PET/CT sensitivity as well as accuracy, conventional imaging is lower than that of it. After comprehensive treatment, the positive predictive rate of PET/CT diagnosis is 96.30%, the negative predictive rate is 100.00%, the sensitivity is 100.00%, the specificity is 92.86%, and the accuracy is 95.50%. The positive predictive rate of conventional imaging diagnosis is 94.87%, the negative predictive rate is 68.29%, the sensitivity is 74.00%, the specificity is 93.33%, and the accuracy is 81.25%. In terms of the above indexes, conventional imaging is lower than PET/CT diagnosis. Conclusion: When it is compared with conventional imaging, 18F-FDG PET/CT has higher accuracy. Besides, it is more comprehensive when diagnosing gynecological cancer before and after comprehensive treatment, which is worthy of clinical promotion.


2016 ◽  
Vol 102 (1) ◽  
pp. 195-199 ◽  
Author(s):  
Pavel Nockel ◽  
Bruna Babic ◽  
Corina Millo ◽  
Peter Herscovitch ◽  
Dhaval Patel ◽  
...  

Abstract Context: Reliable localization of insulinoma is critical for successful treatment. Objective: This study compared the accuracy of 68Gallium DOTA-(Tyr3)-octreotate (Ga-DOTATATE) positron emission tomography (PET)/computed tomography (CT) to anatomic imaging modalities, selective arterial secretagogue injection (SASI), and intraoperative ultrasound (IO ultrasound) and palpation for localizing insulinoma in patients who were biochemically cured. Design, Setting, and Patients: We conducted a retrospective analysis of 31 patients who had an insulinoma. The results of CT, magnetic resonance imaging (MRI), ultrasound, IO ultrasound, 68Ga-DOTATATE PET/CT, SASI, and operative findings were analyzed. Intervention, Main Outcome Measures, and Results: The insulinomas were correctly localized in 17 out of 31 (55%) patients by CT, in 17 out of 28 (61%) by MRI, in 6 out of 28 (21%) by ultrasound, and in 9 out of 10 (90%) by 68Ga-DOTATATE. In 29 of 31 patients (93.5%) who had IO ultrasound, an insulinoma was successfully localized. Thirty patients underwent SASI, and the insulinoma was regionalized in 28 out of 30 patients (93%). In 19 out of 23 patients (83%), manual palpation identified insulinoma. In patients who had all 4 noninvasive imaging studies, CT was concordant with 68Ga-DOTATATE in 6 out of 9 patients (67%), MRI in 8 out of 9 (78%), ultrasound in 0 out of 9; the lesion was only seen by 68Ga-DOTATATE in 1 out of 9 (11%). Conclusions 68Ga-DOTATATE PET/CT identifies most insulinomas and may be considered as an adjunct imaging study when all imaging studies are negative and when a minimally invasive surgical approach is planned.


2018 ◽  
Vol 59 (12) ◽  
pp. 1466-1474 ◽  
Author(s):  
Ying Kan ◽  
Shuxin Zhang ◽  
Wei Wang ◽  
Jie Liu ◽  
Jigang Yang ◽  
...  

Background Metastatic pheochromocytoma and paraganglioma (PCs/PGLs) show high germline mutation, and 18F-FDG and 68Ga-DOTA peptide positron emission tomography/computed tomography (PET/CT) imaging are recommended for the diagnosis of metastatic of PCs. However, there has been lack of direct comparison of the two modalities in the diagnosis of metastatic of PCs up to now. Purpose To evaluate and compare the value of localization of 68Ga-somatostatin receptor analogs and 18F-FDG in the diagnosis of metastatic PCs/PGLs. Material and Methods A comprehensive literature search of PubMed/MEDLINE, ScienceDirect, and Web of Science was performed in accordance with the Preferred Reporting Items for Systematic Review and Meta-analysis (PRISMA) guidelines published in August 2016. We critically reviewed all studies based on the PICOS criteria. QUADAS-2 was used to evaluate the quality of the methodology of the included studies. Results This meta-analysis included 17 studies (629 patients, average age [mean ± SD] = 42.7 ± 6.3 years). The pooled sensitivity and specificity of 18F-FDG and 68Ga peptides were 0.85 (95% confidence interval [CI] = 0.78–0.91) and 0.55 (95% CI = 0.37–0.73), and 0.95 (95% CI = 0.92–0.97) and 0.87 (95% CI = 0.63–0.96), respectively. The area under the sROC curves of the 18F-FDG and 68Ga peptides were 0.88 (95% CI = 0.85–0.91) and 0.78 (95% CI = 0.74–0.81), respectively. A subgroup analysis demonstrated that the difference at the per-lesion level and gene mutation level was significant. Conclusion Compared to 18F-FDG PET/CT, the 68Ga-somatostatin receptor demonstrated good performance in the localization of metastatic PCs/PGLs, especially those with germline mutations. The use of the 68Ga-somatostatin receptor can be a new tool in the diagnosis of metastatic PCs/PGLs.


2021 ◽  
Author(s):  
Ke Zhao ◽  
Ke Rao ◽  
Xin Chen ◽  
Si Chen ◽  
Haifeng Xu

Abstract BackgroundChoriocarcinoma is a rare malignant tumor and rarely occurs outside the gonads. Primary hepatic choriocarcinoma is more infrequent, with hidden clinical manifestations, rapid progress, and extremely poor prognosis. Only more than 10 cases were publicly reported in the world. Therefore, there is still a lack of deep understanding of the diagnosis and treatment of the disease.Case presentationWe report a case of primary hepatic choriocarcinoma in a man diagnosed by pathology. A 65-year-old male patient presented with fever and anorexia, nothing but mild jaundice of the skin and sclera was found on physical examination. Abdominal enhanced magnetic resonance imaging (MRI) showed a huge mass in the right hepatic lobe. Fludeoxyglucose-positron emission tomography-computed tomography (FDG-PET/CT) scan showed increased uptake in the liver and sigmoid colon and no uptake in the testes. The patient underwent the right hepatectomy, and postoperative pathology showed that the tumor was primary hepatic choriocarcinoma. Then he received one course of adjuvant chemotherapy. Then he developed severe myelosuppression and was transferred to the intensive care unit for further treatment. He eventually died of severe liver failure about 100 days after surgery. Primary hepatic choriocarcinoma is extremely rare, and its diagnosis is challenging.ConclusionsPrimary hepatic choriocarcinoma is a rare and highly malignant tumor with a poor prognosis. We believe that this differential diagnosis should be considered in liver tumor patients. The effective treatment for this disease is still to be explored.


2021 ◽  
Vol 55 (3) ◽  
pp. 163-168
Author(s):  
Huajing (Jing) Ni ◽  
Roderick Clifton-Bligh ◽  
Malgorzata Monika Brzozowska

Abstract Objective. Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by chronic hypophosphatemia and osteomalacia. We present case of a patient with a protracted clinical course of TIO. TIO profoundly affected every aspect of his life with subsequent profound physical and psychosocial disabilities. Method. The review of a complex clinical presentation, serial laboratory investigations, and imaging modalities of a patient with TIO caused by a mesenchymal tumor. Results. The patient presented with chronic lower back pain, severe bilateral leg weakness, and multiple pathological fractures due to severe osteoporosis. His investigations revealed hypophosphatemia, low 1,25 dihydroxyvitamin D, phosphaturia and normal serum calcium, and parathyroid hormone. Elevated fibroblast growth factor 23 (FGF23) confirmed the diagnosis of TIO and 68Ga-DOTATATE-positron emission tomography/computed tomography (PET/CT) imaging correctly identified a tumor in the left femoral head. His clinical features and biochemical abnormalities promptly recovered after successful surgical resection of the mesenchymal tumor. Conclusion. The present case demonstrated the need to extensively investigate causes of generalized bone pain in patients with hypophosphatemia, as TIO is highly curable. Importantly, 68Ga-DOTATATE PET/CT imaging successfully identified the FGF23 producing tumor, which was undetectable by conventional imaging, favoring its early use in suspected TIO presentation. The present report highlights the importance of timely diagnosis of this complex medical condition, aiming to improve general awareness and enable better clinical outcomes for this rare disorder.


Sign in / Sign up

Export Citation Format

Share Document