scholarly journals TIO Associated with Hyperparathyroidism: A Rarity, a Rule, or a Novel HPT-PMT Syndrome—A Case Study with Literature Review

2021 ◽  
Vol 2021 ◽  
pp. 1-4
Author(s):  
Michael Salim ◽  
Mohannad Samy Behairy ◽  
Elena Barengolts

Objective. Association of primary hyperparathyroidism (pHPT) with phosphaturic mesenchymal tumors (PMT) is rarely reported. This report entertains the hypothesis of the causal association of HPT with tumor-induced osteomalacia (TIO) and of the existence of HPT-PMT syndrome. Case Presentation. A 49-year-old man presented with fragility rib fractures, generalized bone pain, and muscle weakness worsening over the past 3 years. Initial tests demonstrated hypophosphatemia and high PTH. The diagnosis of pHPT was entertained, but parathyroid scan was negative. During a 2-year follow-up, the patient reported minimal improvement of symptoms after intermittent treatment with calcitriol and phosphate. Biochemical evaluation showed persistent hypophosphatemia with renal phosphate wasting, elevated FGF23, and osteopenia on DXA scan. TIO was suspected. Multiple MRIs and whole-body FDG-PET scans were inconclusive. The patient subsequently underwent 68Ga-DOTATATE PET-CT, which revealed a somatostatin receptor-positive lesion in the lung. The resected mass was confirmed as PMT. The patient had dramatically improved symptoms, normal phosphate, calcium, and FGF23. During follow-up over 3 years postsurgery, the patient had slowly rising calcium and persistently elevated PTH. Conclusion. The debate whether the patient had pHPT or tertiary HPT prompted literature review showing that aberrant genes including FGFR1, FGF1, fibronectin 1, and Klotho were mechanistically involved in the HPT-PMT association. This case highlights the pitfalls contributing to delayed diagnosis and treatment of TIO and hypothesizes the association between pHPT and PMT.

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A174-A175
Author(s):  
Michael Salim ◽  
Elena Barengolts

Abstract Background: Localization of tumor-induced osteomalacia (TIO) is often challenging. Primary hyperparathyroidism (HPT) following curative surgery for TIO is rarely reported. Clinical Case: A 49-year-old man presented with fragility rib fractures, generalized bone pain, and muscle weakness worsening over the past 3 years. Rheumatologic workup was negative. Initial tests showed elevated levels of parathyroid hormone (PTH) 114.1 pg/mL (14–72 pg/mL) and alkaline phosphatase (ALP) 283 IU/L (44–174 IU/L), reduced levels of 25(OH)D 16 ng/mL (30–100 ng/mL), 1,25(OH)2D 9 pg/mL (18–72 pg/mL), and phosphorus 1.6 mg/dL (2.5–4.9 mg/dL), calcium levels of 9.2 mg/dL (8.5–10.1 mg/dL), and eGFR 58 mL/min/1.73 m2. A sestamibi scan showed normal parathyroid uptake. The diagnosis was secondary HPT due to chronic kidney disease and vitamin D deficiency. The patient was treated with D3 and phosphate. During a 2-year follow-up, the patient reported improvement of pain and weakness with no additional fractures. Further investigations showed persistent hypophosphatemia with elevated urinary fractional phosphate excretion (44%, ref. <20%), indicating renal phosphate wasting. Fibroblast Growth Factor 23 (FGF23) was high, 291 RU/mL (0–180 RU/mL). DXA results were consistent with osteopenia. TIO was suspected. At a 3-year follow-up, investigations included three whole-body 18F-FDG PET-CT scans revealing several areas suspicious for tumor presence. However, multiple MRIs were inconclusive. Laboratory tests showed persistent hypophosphatemia (despite D3 and phosphate treatment), elevated FGF23 (1330 RU/mL) and PTH (274.4 pg/mL), and normal calcium, 25(OH)D, and 1,25(OH)2D. The patient subsequently underwent 68Ga DOTATATE PET-CT, which revealed a somatostatin receptor-positive lesion involving the left upper lobe of the lung. The mass was resected without complications. Histopathology was compatible with a phosphaturic mesenchymal tumor. At a 6-month postoperative follow-up, the patient reported dramatically improved symptoms with decreased weakness and pain, normal phosphate, calcium, ALP, and FGF23 (160 RU/mL) levels, while DXA results were significantly improved. Phosphorus supplementation was discontinued. At follow-up 3 years post-surgery, the patient had slowly rising PTH (126.3 pg/mL) and calcium (10.1–10.6 mg/dL) levels with normal phosphate, 25(OH)D, and FGF23 (174 RU/mL) levels. A diagnosis of primary HPT was made. Further evaluation was deemed unnecessary since the patient did not meet the criteria for surgical treatment. The development of primary HPT was considered mechanistically related to long-standing hypophosphatemia and hypovitaminosis D stimulating PTH production. Conclusion: This case report highlights the pitfalls contributing to delayed diagnosis of TIO and alerts clinicians to the potential development of primary HPT after curative surgery for TIO.


ANALES RANM ◽  
2021 ◽  
Vol 138 (138(02)) ◽  
pp. 176-179
Author(s):  
M.N. Cabrera-Martín ◽  
P. Romero Fernández ◽  
M.K. Meneses Navas ◽  
P. Pérez-Segura ◽  
M. Yus Fuertes ◽  
...  

Sometimes, the clinical presentation of a brain tumour mimics that of stroke or viceversa. In these cases it is useful to evaluate follow-up with MR. The coregistration of PET and MR images improves the diagnostic performance of both techniques. In the area of interest, FDG uptake of any degree, with reference to the adjacent brain tissue uptake, should be considered suggestive of tumor if it corresponds to brain injury in MR, even when the FDG uptake is equal to or lower than that of the normal cerebral cortex. We present a case in which coregistration of images from both techniques contributed to the differential diagnosis of stroke and high-grade glioma, whole-body PET/CT ruled out metastatic etiology, and the results led to surgery.


2018 ◽  
Vol 69 (1) ◽  
pp. 63-70 ◽  
Author(s):  
Scott J. Adams ◽  
Rajan Rakheja ◽  
Rhonda Bryce ◽  
Paul S. Babyn

Purpose The study sought to determine the incidence of incidental findings on whole-body positron emission tomography with computed tomography (PET/CT) imaging and the average costs of investigations to follow-up or further characterize incidental findings. Methods Imaging reports of 215 patients who underwent whole-body PET/CT imaging were retrospectively reviewed. Our provincial picture archiving and communication system was queried and patient charts were reviewed to identify all investigations performed to follow-up incidental findings within 1 year of the initial PET/CT study. Costs of follow-up imaging studies (professional and technical components) and other diagnostic tests and procedures were determined in Canadian dollars (CAD) and U.S. dollars (USD) using the 2015 Ontario Health Insurance Plan Schedule of Benefits and Fees and 2016 U.S. Medicare Physician Fee Schedule, respectively. Results At least 1 incidental finding was reported in 161 reports (74.9%). The mean number of incidental findings ranged from 0.64 in patients <45 years of age to 2.2 in patients 75 years of age and older. Seventy-five recommendations for additional investigations were made for 64 (30%) patients undergoing PET/CT imaging, and 14 of those were carried out specifically to follow-up incidental findings. Averaged across all 215 patients, the total cost of investigations recommended to follow-up incidental findings was CAD$105.51 (USD$127.56) per PET/CT study if all recommendations were acted on, and CAD$22.77 (USD$29.14) based on investigations actually performed. Conclusions As the incidence of incidental findings increases with age and a larger proportion of elderly patients is expected as population demographics change, it will be increasingly important to consider incidental findings on PET/CT imaging with standardized approaches to follow-up.


2020 ◽  
Vol 38 (5) ◽  
pp. 472-479 ◽  
Author(s):  
Susan Adil Ali ◽  
Darine Helmy Amin ◽  
Yasser Ibrahim Abdelkhalek
Keyword(s):  
Fdg Pet ◽  
Pet Ct ◽  
18F Fdg ◽  

2019 ◽  
Vol 12 (4) ◽  
pp. e227910
Author(s):  
Kanhaiyalal Agrawal ◽  
P Sai Sradha Patro ◽  
C Preetam

There is literature evidence showing utility of somatostatin receptor (SSTR) positron emission tomography-CT (PET-CT) imaging in differentiated thyroid cancer with Thyroglobulin Elevated and Negative Iodine Scan (TENIS). These patients are less benefited with I-131 therapy and surgery remains only curable option if disease could be localised. If surgery is not feasible, other therapeutic options are not promising. However, if these patients show strongly positive SSTR imaging, then possibility of peptide receptor radionuclide therapy may be explored. As SSTR PET-CT imaging is expensive and not widely available, Technetium-99m (Tc-99m) hydrazinonicotinyl-Tyr3-octreotide (HYNIC-TOC), which is a Single photon emission computed tomography (SPECT) tracer, can be used. We are documenting a case of raised serum thyroglobulin antibody and negative I-131 whole body scan with disease recurrence localised on Tc-99m HYNIC-TOC scan.


2015 ◽  
Vol 173 (3) ◽  
pp. R115-R130 ◽  
Author(s):  
Massimo Salvatori ◽  
Bernadette Biondi ◽  
Vittoria Rufini

In recent years, 2-[18F]-fluoro-2-deoxy-d-glucose positron emission tomography/computed tomography (FDG-PET/CT) has emerged as an important tool for the postoperative management of patients with differentiated thyroid cancer (DTC) and it is widely used in selected clinical situations. The most valuable role that FDG-PET/CT plays in clinical practice is that it can be used to obtain prognostic information in patients with increasing thyroglobulin (Tg) levels and negative 131I whole-body scan post-thyroidectomy and radioiodine (RAI) ablation. FDG-PET/CT may also have a potential role in the initial staging and follow-up of high-risk patients with aggressive histological subtypes, in the identification of patients who are at the highest risk of disease-specific mortality, in the management of patients with RAI-refractory disease, in clinical trials of novel targeted therapies in patients with advanced metastatic disease, and in the evaluation of thyroid nodules with indeterminate fine-needle aspiration for cytology. However, several controversies remain to be resolved, namely: the cutoff value of Tg in the selection of DTC patients for FDG-PET/CT, whether FDG-PET/CT scanning should be performed under thyrotropin stimulation or suppression, and the clinical significance of thyroid FDG-PET/CT incidentalomas. The aim of the present article is to provide an overview of the data about the molecular basis for, clinical indications of, and controversies related to the use of FDG-PET/CT in patients with DTC.


2016 ◽  
Vol 55 (03) ◽  
pp. 99-103 ◽  
Author(s):  
Ina Binse ◽  
Andreas Bockisch ◽  
Sandra Rosenbaum-Krumme ◽  
Marcus Ruhlmann

SummaryIn a previous paper, we published the impact of initial [18F]FDG PET/CT (FDG-PET/CT) in high-risk patients with differentiated thyroid cancer (DTC) and described the changes in therapy management. The aim of the present study was to evaluate the prognostic impact of the initial FDG-PET/CT on a patient’s follow-up over three years and the rate of complete remission. Patients, methods: This study included 109 DTC patients who underwent radioiodine treatment (RIT), including posttherapeutic whole-body scintigraphy with FDG-PET/CT and a follow-up over three years. The follow-up included high-resolution sonography of the neck and determination of serum Tg as well as Tg antibodies every six months. The results of initial FDG-PET/CT and whole-body scintigraphy were compared with the status after three years of follow-up. Results: 24/109 patients (22%) presented FDG-positive lesions, 22/109 patients (20%) only iodine-positive lesions, and 63/109 patients (58%) neither FDG-positive nor iodine-positive lesions. After three years, 83/109 patients (76%) revealed full remission, 15/109 patients (14%) tumour persistence and 11/109 patients (10%) a progressive disease. The negative predictive value (NPV) was calculated for patients without FDG-positive lesions (NPV 85%) and patients without any lesions (NPV 91%) regarding full remission in the follow-up. Conclusion: FDG-PET/CT has a high NPV (85% to 91%) in DTC patients regarding recurrence-free follow-up after three years. The change in patient management in patients with iodine-negative lesions can lead to a higher rate of full remissions in the follow-up after additional surgery. Therefore, FDG-PET/ CT should be performed in all high-risk DTC patients in the context of the first RIT to improve patient management and risk stratification.


Blood ◽  
2014 ◽  
Vol 124 (21) ◽  
pp. 5707-5707
Author(s):  
Adrian Alegre ◽  
Beatriz Aguado ◽  
Miriam González-Pardo ◽  
Evelyn Acuña ◽  
Álvaro Arriero ◽  
...  

Abstract Introduction: Conventional radiography remains the “gold standard” technique for bone involvement assessment in patients with multiple myeloma (MM). Newer imaging modalities such as whole-body Magnetic Resonance (MR) and 18FDG-PET/CT have emerged as more sensitive techniques than routine skeletal survey in the detection of bone involvement in the diagnostic and follow up of patients with MM. The advantages and disadvantages of MR and 18FDG-PET/CT are discussed. Patients and methods: We have retrospectively analyzed 12 patients since 2012 to 2014 with multiple myeloma in our institution whose bone involvement was evaluated with MR and 18FDG-PET/CT. Age range: 36-70. Seven patients were female and five were male. Eight cases were treated with an induction regimen containing bortezomib, three cases with chemotherapy with alternating VBCMP/VBAD and one of them with VAD. After induction, ten of them received autologous stem cell transplantation (ASCT), one patient allogeneic stem cell transplantation and one patient no transplantation. Results: All patients presented bone lesions on MR and all of them were also positive at PET/CT. One showed leptomeningeal involvement on RM and PET. Regarding extramedullar disease two patients presented soft tissue masses and in other two cases there was ganglionar involvement, all of them positives by both techniques. Of nine evaluable patients after complete treatment, six of them have a negative PET and three have a low positive SUV value, however eight of them still had persistent residual lesions on MR, what could indicate not stringent complete response. The patient with leptomeningeal involvement had both MR and PET negative result after treatment. Comments and conclusions: Our data suggest that whole-body MR and 18FDG-PET/CT provide valuable complementary information, MR could be superior to assess extent of lesions and PET to monitor disease activity and to detect asymptomatic relapse. The optimal imaging technique for the management of patients with MM is not well defined and our proposal is a multimodality imaging approach according to individualized criteria. References: Caers J et al. The role of positron emission tomography-computed and magnetic resonance imaging in diagnosis and follow-up of multiple myeloma. Haematologica 2014;99(4):629-637. doi:10.3324/haematol.2013.091918. Agarwal A et al. Evolving Role of FDG PET/CT in Multiple Myeloma Imaging and Management. AJR 2013;200:884-890. Dimopoulos D et al. International myeloma working group consensus statement and guidelines regarding the current role of imaging techniques in the diagnosis and monitoring of multiple Myeloma. Leukemia 2009, 1–12. doi:10.1038/leu.2009.89 Disclosures Alegre: Celgene: Membership on an entity's Board of Directors or advisory committees, Research Funding; Jansen: Membership on an entity's Board of Directors or advisory committees, Research Funding.


2020 ◽  
Vol 0 (0) ◽  
Author(s):  
Jelena Saponjski ◽  
Djuro Macut ◽  
Dragana Sobic Saranovic ◽  
Branislava Radovic ◽  
Vera Artiko

AbstractBackgroundThe aim of the study was evaluation of 18F-fluorodeoxyglucose (18F-FDG) positron emission tomography with computed tomography (PET/CT) in the detection of active disease in the patients with suspected recurrence of the medullary thyroid carcinoma (MTC).Patients and methods18F-FDG PET/CT investigation was performed in 67 patients, investigated from 2010 to 2019. _ Follow up was performed from 6 to 116 months after surgery (median 16.5 months, x± SD = 29±28.9 months). Twenty five of 67 patients underwent 99mTc-dimercaptosuccinic acid (99mTc-DMSA) scintigraphy, 11 underwent somatostatin receptor scintigraphy (SRS) with 99mTc-HYNIC TOC while 11 123I-metaiodobenzylguanidine (MIBG) scintigraphy.ResultsFrom 67 patients, 35 (52.2%) had true positive 18F-FDG PET/CT findings (TP). Average maximal standardized uptake value (SUVmax) for all TP lesions was 5.01+3.6. In 25 (37.3%) patients findings were true negative (TN). Four (6%) patients had false positive (FP) findings while three (4.5%) were false negative (FN). Thus, sensitivity of the 18F-FDG PET/ CT was 92.11%, specificity 86.21%, positive predictive value 89.74%, negative predictive value 89.29% and accuracy 89.55%. In 27 patients (40%) 18F-FDG PET/CT finding influenced further management of the patient.Conclusions18F-FDG PET/CT has high accuracy in the detection of metastases/recurrences of MTC in patients after thyroidectomy as well as in evaluation and the appropriate choice of the therapy.


2013 ◽  
Vol 31 (6_suppl) ◽  
pp. 188-188
Author(s):  
Anthonius J. Breeuwsma ◽  
Maxim Rybalov ◽  
Anna Maria Leliveld ◽  
Rudi A. Dierckx ◽  
Jan Pruim ◽  
...  

188 Background: 11C-choline PET/CT has proven to be a sensitive technique for re-staging after radiation therapy (RT). The aim of this study was to analyze the clinical impact of 11C-choline-PET/CT in the selection of patients with biochemical recurrence (BCR) after RT for salvage cryoablation of the prostate. Methods: This prospective study was conducted between November 2006 and February 2012 on patients considered as candidates for salvage cryoablation. 74 patients, mean age 69.2 years, median – 70.3 years (range 49-79), who were being followed up after RT for histological proven prostate cancer (according to ASTRO-Phoenix) were included. Until 2009 we used PET/CT fusion, but from 2009 all patients were examined with an integrated PET/CT system. After receiving 400 MBq 11C-choline intravenously, a whole body scan was made. As reference we used biopsy-proven histology from site of suspicion, confirmative imaging modalities (bonescan, CT) or clinical follow-up. PSA doubling time and velocity was calculated. Results: According to the PET/CT results, 40 (54%) patients had a local recurrence, 20 (27%) had regional/distant metastases and 14 (19%) had a negative scan. The positive PET findings were proved by histology from prostate biopsies and/or pelvic lymph node dissections in 63% of cases. Considering PET/CT results: 50/74 (68%) patients received cryoablation, for 24/74 (32%) treatment was changed (active surveillance or androgen deprivation therapy). Conclusions: 11C-choline-PET/CT could be useful for the selection of patients with BCR after RT for salvage cryoablation of the prostate. 11C-choline-PET/CT was decisive and led to therapy change in 32% of cases. [Table: see text]


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