Utility of Gallium‐68 <scp>DOTANOC PET</scp>/<scp>CT</scp> in the localization of Tumour‐induced osteomalacia

Bhavani, Nisha; Asirvatham, Adlyne Reena; Kallur, Kumar; Arun, Chankramath S; Pavithran, Praveen V; Nair, Vasantha; Vasukutty, Jayakumar R.; Menon, Usha; Kumar, Harish

doi:10.1111/cen.12822

Cited by 29 publications

(31 citation statements)

References 19 publications

(22 reference statements)

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“…But he failed to include 68Ga DOTATATE PET/CT in localizing these tumors. However, an increasing number of researches have demonstrated that 68Ga DOTATATE PET/CT is an accurate imaging modality (overall accuracy 97.7%) and can be used as the first imaging modality in the detection of obscure TIO tumors [28][29][30] . Once the neoplasm is located, complete resection of the neoplasm with wide margins will be the most appropriate treatment.…”

Section: Discussionmentioning

confidence: 99%

The diagnostic dilemma of tumor induced osteomalacia: a retrospective analysis of 144 cases

et al. 2017

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Abstract. Diagnostic delay of tumor induced osteomalacia (TIO) is common in clinic practice. To investigate the diagnostic condition of TIO in China and raise clinicians' awareness of TIO, we retrospectively analyzed clinical manifestations, biochemical features, and specially evaluated missed diagnoses and misdiagnoses among 144 TIO patients from Peking Union Medical College Hospital during December 1982 to December 2014. Clinical presentations of TIO mainly included bone pain, difficulty in walking, pathological fractures, muscle weakness, and height loss. TIO patients demonstrated hypophosphatemia (0.48±0.13 mmol/L), elevated serum alkaline phosphatase (277.9±152.6 U/L), reduced tubular maximum for phosphorus/glomerular filtration rate (0.39±0.14) and markedly elevated serum fibroblast growth factor 23 (FGF23) (median level 302.9 pg/mL). The average time from onset to a correct diagnosis was 2.9±2.3 years while the mean duration from onset to tumor resection was 5.4±4.2 years. The initial misdiagnosis rate was 95.1% (137/144) and 240 case-times of misdiagnoses occurred among the 144 cases. The most frequent misdiagnoses were intervertebral disc herniation, spondyloarthritis (including ankylosing spondylitis) and osteoporosis. A total of 43.1% (62/144) cases with hypophosphatemia presented on their laboratory sheets were neglected and missed diagnosed. Our study showed that TIO was frequently misdiagnosed and missed diagnosed due to its rarity, insidious onset, nonspecific clinical manifestations and clinicians' poor recognition. It is necessary to test serum phosphorus in patients with musculoskeletal symptoms and difficulty in walking. The measurement of serum FGF23 is rather valuable. Once hypophosphatemia is discovered, TIO should be suspected and it is highly recommended to search for tumors and perform curative surgery.

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Section: Discussionmentioning

confidence: 99%

The diagnostic dilemma of tumor induced osteomalacia: a retrospective analysis of 144 cases

et al. 2017

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“…No additional records were found screening the references of these articles, whereas six articles were excluded after the analysis of the full text. Therefore, 14 articles were included in the qualitative analysis (systematic review) [16][17][18][19][20][21][22][23][24][25][26][27][28][29]. Three articles were excluded from the meta-analysis for possible patient data overlap [18,25,26].…”

Section: Literature Searchmentioning

confidence: 99%

“…Three articles were excluded from the meta-analysis for possible patient data overlap [18,25,26]. Overall, 11 articles (166 patients with TIO) were included in the quantitative analysis (meta-analysis) [16,17,[19][20][21][22][23][24][27][28][29]. The characteristics of the 14 studies included in the systematic review are presented in Tables 1 and 2.…”

Section: Literature Searchmentioning

confidence: 99%

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

et al. 2019

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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.

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“…have been utilized for localizing these small tumors. Ga 68 DOTANOC PET/CT scan is said to have the greatest sensitivity and specificity for detecting small mesenchymal lesions, giving rise to TIO . The standard treatment of TIO is surgical excision of the tumor.…”

Section: Introductionmentioning

confidence: 99%

“…Ga 68 DOTANOC PET/CT scan is said to have the greatest sensitivity and specificity for detecting small mesenchymal lesions, giving rise to TIO. (6,7) The standard treatment of TIO is surgical excision of the tumor. The patients with TIO can be cured completely if the tumor is localized correctly and excised adequately.…”

Section: Introductionmentioning

confidence: 99%

Successful Management Of Tumor‐Induced Osteomalacia with Radiofrequency Ablation: A Case Series

et al. 2019

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Tumor‐induced osteomalacia (TIO) is a curable condition when the tumor is correctly located and completely removed. These tumors are, however, small and located in regions that make surgical removal difficult and sometimes risky in some patients. Experience of radiofrequency ablation (RFA) in the management of TIO is limited. We describe 3 patients with TIO who were treated in our hospital with RFA. They had suspected lesions in surgically difficult locations and were subjected to single sessions of RFA. The response was documented in terms of improvement in symptoms, normalization of hypophosphatemia and hyperphosphaturia, and disappearance of uptake on follow‐up Ga 68 DOTANOC PET/CT imaging. All 3 patients had a clinical and biochemical profile consistent with TIO. The first patient (patient 1) had an intensely Ga 68 DOTANOC avid lesion involving the roof of right acetabulum. The second patient (patient 2) had a Ga 68 DOTANOC avid intramuscular lesion in left pectineus muscle and the third patient (patient 3) had a Ga 68 DOTANOC avid expansile osteolytic lesion involving the angle and ramus of right mandible. All 3 patients achieved complete biochemical as well as clinical remission with single sessions of RFA. Six months after the procedure, Ga 68 DOTANOC imaging revealed the absence of uptake at the previous sites, corroborating with the clinical improvement and normalization of hypophosphatemia and hyperphosphaturia. In conclusion, although surgical resection is the standard of care, RFA can be used successfully for treating patients with TIO. It can be an effective, less invasive, and safe modality of treatment in those patients where resection of the lesion is not possible because of inaccessible anatomical location or comorbidity that prohibits surgery. © 2019 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.

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Utility of Gallium‐68 DOTANOC PET/CT in the localization of Tumour‐induced osteomalacia

Abstract: We conclude that Ga-68 DOTANOC PET/CT can be used as the first imaging modality in patients diagnosed with TIO. The extremely good outcome following the resection of these small otherwise undiagnosed tumours far outweighs its cost even in resource limited settings.

Cited by 29 publications

References 19 publications

The diagnostic dilemma of tumor induced osteomalacia: a retrospective analysis of 144 cases

The diagnostic dilemma of tumor induced osteomalacia: a retrospective analysis of 144 cases

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

Successful Management Of Tumor‐Induced Osteomalacia with Radiofrequency Ablation: A Case Series

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