‘Peptide receptor radionuclide therapy in the management of advanced pheochromocytoma and paraganglioma: A systematic review and meta‐analysis’

Satapathy, Sujata; Mittal, Bhagwant Rai; Bhansali, Anil

doi:10.1111/cen.14106

Cited by 86 publications

(64 citation statements)

References 30 publications

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“…Three patients had a concomitant pancreatic neuroendocrine tumor (PNET) of whom two were genetically proven to have von Hippel Lindau syndrome. The indication for PRRT was metastasis ( 8 ) and inoperability ( 7 ). A median of three cycles (1–6) was administered with median cumulative radioactivity of 28 GBq (19–40 GBq).…”

Section: Resultsmentioning

confidence: 99%

“…Controlled disease (CD) was defined as a combination of all the responses except PD (SD + PR + MR). CD was calculated to make the data more comparable to previously published literature ( 5 , 7 ).…”

Section: Assessment Of Efficacymentioning

confidence: 99%

“…177 Lu-tetra-aza-cyclo-dodecanetetraacetic acid–DPhe1-Tyr3-octreotate (DOTATATE) has shown favorable efficacy in controlling symptoms and tumor progression in most of the previous studies ( 5 , 6 ). A recently published meta-analysis has reported good efficacy (disease control rate: 80% (95% CI: 77–89%)) with an encouraging safety profile ( 7 ). But availability, cost, and potential adverse effects limit the use of PRRT.…”

Section: Introductionmentioning

confidence: 99%

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177Lu-DOTATATE therapy in metastatic/inoperable pheochromocytoma-paraganglioma

Jaiswal¹,

Sarathi

Memon³

et al. 2020

Endocrine Connections

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Introduction: 177Lu-DOTATATE-based peptide receptor radionuclide therapy (PRRT) is a promising therapy for metastatic and/ inoperable pheochromocytoma and paraganglioma (PPGL). We aim to evaluate the efficacy and safety of and identify predictors of response to 177Lu-DOTATATE therapy in metastatic and/ inoperable PPGL. Methods: This retrospective study involved 15 patients of metastatic or unresectable PPGL, who received 177Lu-DOTATATE PRRT therapy. Clinical, biochemical (Plasma free normetanephrine), and radiological (anatomical and functional) responses were compared before and after the last therapy. Results: A total of 15 patients PCC (4), sPGL (4), HNPGL (5), PCC+sPGL (1), HNPGL+sPGL (1) were included. The median duration of follow up was 27 (range: 11-62) months from the start of PRRT. Based on the RECIST (1.1) criteria, progressive disease was seen in three (20%), stable disease in eight (53 %), partial response in one (7%), and minor response in three (20%) and controlled disease in 12 (80%). On linear regression analysis presence of PGL (p= 0.044) and baseline SUVmax >21 (p < 0.0001) were significant positive predictors of early response to PRRT. Encouraging safety profiles were noted with no long term nephrotoxicity and haematotoxicity. Conclusion: 177Lu-DOTATATE therapy is an effective and safe modality of treatment for patients with metastatic/inoperable PPGL. Although it is not prudent to withhold PRRT in metastatic PPGL with baseline SUVmax < 21, baseline SUVmax >21 can be used to predict early response to PRRT.

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Section: Resultsmentioning

confidence: 99%

Section: Assessment Of Efficacymentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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177Lu-DOTATATE therapy in metastatic/inoperable pheochromocytoma-paraganglioma

Jaiswal¹,

Sarathi

Memon³

et al. 2020

Endocrine Connections

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“…Targeted radiotherapy using 131 I MIBG (Azedra®) is an option in systemic treatment. Radiolabeled somatostatin analogues are being investigated [41,42]. Less than 40% of patients with metastatic PHEO respond (usually partial rather than complete response) to currently used therapeutic modalities such as 131 I MIBG or chemotherapy [43] (Fig.…”

Section: Management Of Advanced Diseasementioning

confidence: 99%

Emerging Treatments for Advanced/Metastatic Pheochromocytoma and Paraganglioma

Ilanchezhian

Jha

Pacák

et al. 2020

Curr. Treat. Options in Oncol.

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Opinion statement The incidence of metastatic pheochromocytoma (PHEO) and paraganglioma (PGL) may occur in as many as 35% of patients particularly with PGL and even more frequently in those with specific mutations. Biochemical, morphological, and molecular markers have been investigated for use in the distinction of benign from malignant PHEO/PGL. PHEO/PGL metastasizes via hematogenous or lymphatic routes and shows differences based on mutational status. The most common sites of involvement in patients that have an SDHB mutation are the bone (78%), lungs (45%), lymph nodes (36%), and liver (35%). In patients with sporadic PHEO/PGL, the most common sites of metastasis are the bones (64%), lungs (47%), lymph nodes (36%), and liver (32%). Metastases may be present at presentation or may occur later. Metastases to the liver and lungs are associated with a shorter survival. Overall, the estimated 5-year survival rates are between 34 and 74%. Currently, treatments for metastatic PHEO/PGL are essentially palliative. Surgery is potentially curative; however, tumor dissemination limits the chance for a curative resection. When surgical intervention is not amenable, the therapeutic options include radiolabeled MIBG (Azedra®—iobenguane 131 was recently FDA-approved for patients > 12 years and older with iobenguane scan positive) or systemic chemotherapy with cyclophosphamide, vincristine, and dacarbazine (CVD) with an overall objective response rate (ORR) of less than 40%; however, it is not clear if the administration of CVD impacts overall survival, as nearly all patients develop progressive and ultimately fatal disease. Other treatment modalities under investigation include cytoreductive techniques, novel radiopharmaceuticals, chemotherapy, radiotherapy, immunotherapy, and experimental therapies. Here we are discussing emerging treatment for advanced/metastatic PHEO/PGL.

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“…Additionally, this agent has demonstrated substantial safety and efficacy based on data from 1200 patients treated for gastroenteropancreatic and bronchial NETs ( 5 ). 177 Lu-DOTATATE has also been effectively utilized in the treatment of inoperable, metastatic pheochromocytomas and paragangliomas (PPGLs) ( 6 , 7 ). 177 Lu radionuclide exerts its anti-tumor effects by emitting medium energy beta particles and has a maximal tissue penetration of 2 mm ( 8 ).…”

Section: Introductionmentioning

confidence: 99%

Case Report: Primary Hypothyroidism Associated With Lutetium 177-DOTATATE Therapy for Metastatic Paraganglioma

et al. 2021

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BackgroundLutetium 177 (177Lu) - DOTATATE is a form of peptide receptor radionuclide therapy (PRRT) utilized in the treatment of neuroendocrine tumors. Data on 177Lu-DOTATATE-induced thyroid dysfunction is limited.Case DescriptionA 29-year-old male with SDHB positive metastatic paraganglioma enrolled under the 177Lu-DOTATATE trial (NCT03206060) underwent thyroid function test (TFT) evaluation comprised of thyroid stimulating hormone (TSH) and free thyroxine (FT4) immunoassay measurements per protocol prior to 177Lu-DOTATATE therapy. The TSH was suppressed [<0.01 µIU/ml (0.27–4.2 µIU/ml)], and FT4 was normal [1.3 ng/dl (0.9–1.7 ng/dl)]. The TSH receptor antibody and thyroid stimulating immunoglobulin index were undetectable [<1 IU/L (≤1.75 IU/L), and <1 (≤1.3) respectively], while the anti-thyroid peroxidase (anti-TPO) and anti-thyroglobulin (anti-Tg) antibodies were elevated [605 IU/ml (0.0–34.9 IU/ml), and 178 IU/ml (0.0-40.0 IU/ml) respectively]. Mass spectrometry on a stored (-80°C) plasma sample obtained one-month pre-PRRT revealed elevated total triiodothyronine (TT3) [235 ng/dl (65–193 ng/dl)] and FT4 [3.9 ng/dl (1.2–2.9 ng/dl)] levels. The patient was diagnosed with Hashimoto’s thyrotoxicosis. However, the patient was asymptomatic. One month after the first dose of 200mCi 177Lu-DOTATATE, the patient noted fatigue and a 2.6 Kg weight gain. The TSH (73.04 µIU/ml), anti-TPO antibodies (>1,000 IU/ml), and anti-Tg antibodies (668 IU/ml) had substantially increased, with reductions in FT4 (0.3 ng/dl) and TT3 [54 ng/dl (87–169 ng/dl)]. Diagnostic gallium 68 - DOTATATE positron emission tomography-computed tomography performed prior to 177Lu-DOTATATE treatment revealed diffuse thyroid uptake. Post-therapy single-photon emission computed tomography also revealed diffuse uptake of 177Lu-DOTATATE in the thyroid gland. Levothyroxine therapy was initiated, and the patient’s symptoms resolved.SummaryWe report, for the first time, a patient with asymptomatic primary hyperthyroidism who rapidly developed symptomatic primary hypothyroidism 1 month after 177Lu-DOTATATE therapy, accompanied by marked changes in TFTs and thyroid auto-antibody titers, with functional imaging evidence of diffuse uptake of 177Lu-DOTATATE in the thyroid gland.ConclusionsThyroid dysfunction can be associated with PRRT. Thyroid uptake patterns on pre-treatment diagnostic somatostatin analog scans might predict individual susceptibility to PRRT-associated TFT disruption. Therefore, periodic evaluation of TFTs should be considered in patients receiving PRRT.

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‘Peptide receptor radionuclide therapy in the management of advanced pheochromocytoma and paraganglioma: A systematic review and meta‐analysis’

Cited by 86 publications

References 30 publications

177Lu-DOTATATE therapy in metastatic/inoperable pheochromocytoma-paraganglioma

177Lu-DOTATATE therapy in metastatic/inoperable pheochromocytoma-paraganglioma

Emerging Treatments for Advanced/Metastatic Pheochromocytoma and Paraganglioma

Case Report: Primary Hypothyroidism Associated With Lutetium 177-DOTATATE Therapy for Metastatic Paraganglioma

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