Management of symptoms
Fluid and electrolyte replacement
Initial treatment of VIPoma involves the correction of dehydration with saline-based intravenous fluids and the repletion of electrolytes with standard electrolytes (e.g., potassium chloride, magnesium sulfate, phosphorus).[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
[17]Hofland J, Falconi M, Christ E, et al. European Neuroendocrine Tumor Society 2023 guidance paper for functioning pancreatic neuroendocrine tumour syndromes. J Neuroendocrinol. 2023 Aug;35(8):e13318.
https://onlinelibrary.wiley.com/doi/10.1111/jne.13318
http://www.ncbi.nlm.nih.gov/pubmed/37578384?tool=bestpractice.com
[36]Modlin IM, Pavel M, Kidd M, et al. Review article: somatostatin analogues in the treatment of gastroenteropancreatic neuroendocrine (carcinoid) tumours. Aliment Pharmacol Ther. 2010 Jan 15;31(2):169-88.
http://onlinelibrary.wiley.com/doi/10.1111/j.1365-2036.2009.04174.x/full
http://www.ncbi.nlm.nih.gov/pubmed/19845567?tool=bestpractice.com
Somatostatin analogs
Somatostatin analogs (e.g., octreotide, lanreotide) are the standard of care for controlling VIPoma related symptoms, including diarrhea, and decreasing VIP secretion.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
[35]Del Rivero J, Perez K, Kennedy EB, et al. Systemic therapy for tumor control in metastatic well-differentiated gastroenteropancreatic neuroendocrine tumors: ASCO guideline. J Clin Oncol. 2023 Nov 10;41(32):5049-67.
https://ascopubs.org/doi/10.1200/JCO.23.01529
http://www.ncbi.nlm.nih.gov/pubmed/37774329?tool=bestpractice.com
[36]Modlin IM, Pavel M, Kidd M, et al. Review article: somatostatin analogues in the treatment of gastroenteropancreatic neuroendocrine (carcinoid) tumours. Aliment Pharmacol Ther. 2010 Jan 15;31(2):169-88.
http://onlinelibrary.wiley.com/doi/10.1111/j.1365-2036.2009.04174.x/full
http://www.ncbi.nlm.nih.gov/pubmed/19845567?tool=bestpractice.com
Up to 80% of patients with VIPoma achieve a clinical response (e.g., improvement in diarrhea) with somatostatin analogs, and a small percentage also have tumor shrinkage.[37]Oberg K. Chemotherapy and biotherapy in the treatment of neuroendocrine tumours. Ann Oncol. 2001;12 Suppl 2:S111-4.
http://www.ncbi.nlm.nih.gov/pubmed/11762335?tool=bestpractice.com
[38]Maton PN, Gardner JD, Jensen RT. Use of long-acting somatostatin analog SMS 201-995 in patients with pancreatic islet cell tumors. Dig Dis Sci. 1989;34(3 suppl):28-39S.
http://www.ncbi.nlm.nih.gov/pubmed/2537716?tool=bestpractice.com
Patients are initiated on a short-acting somatostatin analog (e.g., octreotide) for rapid symptom control. Patients are transitioned to a long-acting somatostatin analog (either octreotide or lanreotide), with dose titration for optimal symptom control. Short-acting analogs may be used to manage breakthrough symptoms.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
Patients with VIPoma often require lifelong somatostatin analog therapy to maintain symptom control.[35]Del Rivero J, Perez K, Kennedy EB, et al. Systemic therapy for tumor control in metastatic well-differentiated gastroenteropancreatic neuroendocrine tumors: ASCO guideline. J Clin Oncol. 2023 Nov 10;41(32):5049-67.
https://ascopubs.org/doi/10.1200/JCO.23.01529
http://www.ncbi.nlm.nih.gov/pubmed/37774329?tool=bestpractice.com
Patients receiving somatostatin analogs in the long term may develop gallstones due to biliary stasis.[39]Brighi N, Panzuto F, Modica R, et al. Biliary stone disease in patients with neuroendocrine tumors treated with somatostatin analogs: a multicenter study. Oncologist. 2020 Mar;25(3):259-65.
https://academic.oup.com/oncolo/article/25/3/259/6443343?login=false
http://www.ncbi.nlm.nih.gov/pubmed/32162819?tool=bestpractice.com
Cholecystectomy may be performed if long-term use of somatostatin analogs is expected, based on the patient’s clinical course.
Drug resistance can occur with long-term use of somatostatin analogs; dose escalation may be required.
Treatment for metastatic disease
More than half of patients with VIPoma will have distant metastasis (most commonly to the liver) at the time of diagnosis.[42]Perry RR, Vinik AI. Clinical review 72: diagnosis and management of functioning islet cell tumors. J Clin Endocrinol Metab. 1995 Aug;80(8):2273-8.
http://www.ncbi.nlm.nih.gov/pubmed/7629220?tool=bestpractice.com
Surgery, embolization, or ablation
Surgical treatment in the metastatic setting is palliative and should be individualized based on patient factors (e.g., age, comorbidities) and disease factors (e.g., location, distribution, and grade of metastases).[34]Pavel M, O'Toole D, Costa F, et al; Vienna Consensus Conference participants. ENETS consensus guidelines update for the management of distant metastatic disease of intestinal, pancreatic, bronchial neuroendocrine neoplasms (NEN) and NEN of unknown primary site. Neuroendocrinology. 2016;103(2):172-85.
http://www.ncbi.nlm.nih.gov/pubmed/26731013?tool=bestpractice.com
Patients with metastatic disease may undergo surgical resection to remove the primary tumor and regional lymph nodes, and debulking of metastatic lesions (e.g., surgical resection, radiofrequency ablation, cryoablation) to reduce tumor burden and alleviate symptoms.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
[24]Pavel M, Öberg K, Falconi M, et al. Gastroenteropancreatic neuroendocrine neoplasms: ESMO clinical practice guidelines for diagnosis, treatment and follow-up. Ann Oncol. 2020 Jul;31(7):844-60.
https://www.annalsofoncology.org/article/S0923-7534(20)36394-8/fulltext
[41]Howe JR, Merchant NB, Conrad C, et al. The North American Neuroendocrine Tumor Society consensus paper on the surgical management of pancreatic neuroendocrine tumors. Pancreas. 2020 Jan;49(1):1-33.
http://www.ncbi.nlm.nih.gov/pubmed/31856076?tool=bestpractice.com
Patients who are not candidates for surgical resection of hepatic metastasis may undergo liver-directed therapies such as transarterial embolization, transarterial chemoembolization, radiofrequency ablation, cryoablation, or selective internal radiation therapy to ablate functional tumors.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
[43]Ho AS, Picus J, Darcy MD, et al. Long-term outcome after chemoembolization and embolization of hepatic metastatic lesions from neuroendocrine tumors. AJR Am J Roentgenol. 2007 May;188(5):1201-7.
http://www.ajronline.org/doi/pdf/10.2214/AJR.06.0933
http://www.ncbi.nlm.nih.gov/pubmed/17449759?tool=bestpractice.com
[44]King J, Quinn R, Glenn DM, et al. Radioembolization with selective internal radiation microspheres for neuroendocrine liver metastases. Cancer. 2008 Sep 1;113(5):921-9.
http://www.ncbi.nlm.nih.gov/pubmed/18618495?tool=bestpractice.com
Generally, ablation is only considered if hepatic metastases are small (<3 cm) and there are no more than 4 lesions.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
Liver transplantation has been successfully performed in highly selected patients with resected primary tumor who have long-term stable and unresectable metastatic disease limited to the liver.[45]Johnston PC, Ardill JE, Johnston BT, et al. Vasoactive intestinal polypeptide secreting pancreatic tumour with hepatic metastases: long term survival after orthotopic liver transplantation. Ir J Med Sci. 2010 Sep;179(3):439-41.
http://www.ncbi.nlm.nih.gov/pubmed/18825477?tool=bestpractice.com
[46]Máthé Z, Tagkalos E, Paul A, et al. Liver transplantation for hepatic metastases of neuroendocrine pancreatic tumors: a survival-based analysis. Transplantation. 2011 Mar 15;91(5):575-82.
http://www.ncbi.nlm.nih.gov/pubmed/21200365?tool=bestpractice.com
However, it is rarely used.
Medical treatment
Medical treatment can help control tumor growth and treat tumor-related symptoms.[24]Pavel M, Öberg K, Falconi M, et al. Gastroenteropancreatic neuroendocrine neoplasms: ESMO clinical practice guidelines for diagnosis, treatment and follow-up. Ann Oncol. 2020 Jul;31(7):844-60.
https://www.annalsofoncology.org/article/S0923-7534(20)36394-8/fulltext
Optimal sequencing of medical treatment is unclear.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
Somatostatin analogs (e.g., octreotide, lanreotide) are recommended for initial medical treatment if not already used.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
[35]Del Rivero J, Perez K, Kennedy EB, et al. Systemic therapy for tumor control in metastatic well-differentiated gastroenteropancreatic neuroendocrine tumors: ASCO guideline. J Clin Oncol. 2023 Nov 10;41(32):5049-67.
https://ascopubs.org/doi/10.1200/JCO.23.01529
http://www.ncbi.nlm.nih.gov/pubmed/37774329?tool=bestpractice.com
As well as controlling diarrhea, these agents may have an antiproliferative effect.[34]Pavel M, O'Toole D, Costa F, et al; Vienna Consensus Conference participants. ENETS consensus guidelines update for the management of distant metastatic disease of intestinal, pancreatic, bronchial neuroendocrine neoplasms (NEN) and NEN of unknown primary site. Neuroendocrinology. 2016;103(2):172-85.
http://www.ncbi.nlm.nih.gov/pubmed/26731013?tool=bestpractice.com
[47]Merola E, Panzuto F, Delle Fave G. Antiproliferative effect of somatostatin analogs in advanced gastro-entero-pancreatic neuroendocrine tumors: a systematic review and meta-analysis. Oncotarget. 2017 Jul 11;8(28):46624-34.
https://www.doi.org/10.18632/oncotarget.16686
http://www.ncbi.nlm.nih.gov/pubmed/28402955?tool=bestpractice.com
In phase 3 studies, octreotide and lanreotide both significantly prolonged progression-free survival compared with placebo.[48]Rinke A, Müller HH, Schade-Brittinger C, et al. Placebo-controlled, double-blind, prospective, randomized study on the effect of octreotide LAR in the control of tumor growth in patients with metastatic neuroendocrine midgut tumors: a report from the PROMID Study Group. J Clin Oncol. 2009 Oct 1;27(28):4656-63.
https://www.doi.org/10.1200/JCO.2009.22.8510
http://www.ncbi.nlm.nih.gov/pubmed/19704057?tool=bestpractice.com
[49]Caplin ME, Pavel M, Ćwikła JB, et al. Lanreotide in metastatic enteropancreatic neuroendocrine tumors. N Engl J Med. 2014 Jul 17;371(3):224-33.
https://www.nejm.org/doi/10.1056/NEJMoa1316158
http://www.ncbi.nlm.nih.gov/pubmed/25014687?tool=bestpractice.com
Everolimus (a mammalian target of rapamycin [mTOR] inhibitor) and sunitinib (a multi-targeted receptor tyrosine kinase inhibitor) prolonged progression-free survival in randomized placebo-controlled trials of patients with pancreatic neuroendocrine tumors.[50]Raymond E, Dahan L, Raoul JL, et al. Sunitinib malate for the treatment of pancreatic neuroendocrine tumors. N Engl J Med. 2011 Feb 10;364(6):501-13.
http://www.ncbi.nlm.nih.gov/pubmed/21306237?tool=bestpractice.com
[51]National Institute for Health and Care Excellence. Everolimus and sunitinib for treating unresectable or metastatic neuroendocrine tumours in people with progressive disease. June 2017 [internet publication].
https://www.nice.org.uk/guidance/ta449
[52]Yao JC, Shah MH, Ito T, et al. Everolimus for advanced pancreatic neuroendocrine tumors. N Engl J Med. 2011 Feb 10;364(6):514-23.
https://www.doi.org/10.1056/NEJMoa1009290
http://www.ncbi.nlm.nih.gov/pubmed/21306238?tool=bestpractice.com
These agents may be combined with a somatostatin analog or used as an alternative therapy; however, their specific use in VIPoma has not been evaluated in clinical trials.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
[53]Capdevila J, Teulé A, Barriuso J, et al. Phase II study of everolimus and octreotide LAR in patients with nonfunctioning gastrointestinal neuroendocrine tumors: the GETNE1003_EVERLAR study. Oncologist. 2019 Jan;24(1):38-46.
https://www.doi.org/10.1634/theoncologist.2017-0622
http://www.ncbi.nlm.nih.gov/pubmed/29794066?tool=bestpractice.com
[54]Bajetta E, Catena L, Pusceddu S, et al. Everolimus in combination with octreotide long-acting repeatable in a first-line setting for patients with neuroendocrine tumors: a 5-year update. Neuroendocrinology. 2018;106(4):307-11.
http://www.ncbi.nlm.nih.gov/pubmed/28743120?tool=bestpractice.com
Chemotherapy can be considered for patients who have clinically significant tumor burden or symptomatic progressive disease.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
[35]Del Rivero J, Perez K, Kennedy EB, et al. Systemic therapy for tumor control in metastatic well-differentiated gastroenteropancreatic neuroendocrine tumors: ASCO guideline. J Clin Oncol. 2023 Nov 10;41(32):5049-67.
https://ascopubs.org/doi/10.1200/JCO.23.01529
http://www.ncbi.nlm.nih.gov/pubmed/37774329?tool=bestpractice.com
Combination chemotherapy regimens containing streptozocin or temozolomide are often used (e.g., streptozocin plus fluorouracil; streptozocin plus fluorouracil plus doxorubicin; temozolomide plus capecitabine).[55]Moertel CG, Lefkopoulo M, Lipsitz S, et al. Streptozocin-doxorubicin, streptozocin-fluorouracil or chlorozotocin in the treatment of advanced islet-cell carcinoma. N Engl J Med. 1992 Feb 20;326(8):519-23.
https://www.doi.org/10.1056/NEJM199202203260804
http://www.ncbi.nlm.nih.gov/pubmed/1310159?tool=bestpractice.com
[56]Dilz LM, Denecke T, Steffen IG, et al. Streptozocin/5-fluorouracil chemotherapy is associated with durable response in patients with advanced pancreatic neuroendocrine tumours. Eur J Cancer. 2015 Jul;51(10):1253-62.
http://www.ncbi.nlm.nih.gov/pubmed/25935542?tool=bestpractice.com
[57]Kouvaraki MA, Ajani JA, Hoff P, et al. Fluorouracil, doxorubicin, and streptozocin in the treatment of patients with locally advanced and metastatic pancreatic endocrine carcinomas. J Clin Oncol. 2004 Dec 1;22(23):4762-71.
https://www.doi.org/10.1200/JCO.2004.04.024
http://www.ncbi.nlm.nih.gov/pubmed/15570077?tool=bestpractice.com
[58]Strosberg JR, Fine RL, Choi J, et al. First-line chemotherapy with capecitabine and temozolomide in patients with metastatic pancreatic endocrine carcinomas. Cancer. 2011 Jan 15;117(2):268-75.
https://www.doi.org/10.1002/cncr.25425
http://www.ncbi.nlm.nih.gov/pubmed/20824724?tool=bestpractice.com
A somatostatin analog can be given in combination with chemotherapy to improve the control of syndromic symptoms.[17]Hofland J, Falconi M, Christ E, et al. European Neuroendocrine Tumor Society 2023 guidance paper for functioning pancreatic neuroendocrine tumour syndromes. J Neuroendocrinol. 2023 Aug;35(8):e13318.
https://onlinelibrary.wiley.com/doi/10.1111/jne.13318
http://www.ncbi.nlm.nih.gov/pubmed/37578384?tool=bestpractice.com
Peptide receptor radionuclide therapy with lutetium Lu 177 dotatate (a radiolabeled somatostatin analog) is an option for patients with somatostatin receptor-positive tumors (confirmed by somatostatin receptor-based imaging) who have progressed following treatment with conventional and/or long-acting somatostatin analogs.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
[35]Del Rivero J, Perez K, Kennedy EB, et al. Systemic therapy for tumor control in metastatic well-differentiated gastroenteropancreatic neuroendocrine tumors: ASCO guideline. J Clin Oncol. 2023 Nov 10;41(32):5049-67.
https://ascopubs.org/doi/10.1200/JCO.23.01529
http://www.ncbi.nlm.nih.gov/pubmed/37774329?tool=bestpractice.com
[59]Hope TA, Bodei L, Chan JA, et al. NANETS/SNMMI consensus statement on patient selection and appropriate use of (177)Lu-DOTATATE peptide receptor radionuclide therapy. J Nucl Med. 2020 Feb;61(2):222-7.
https://www.doi.org/10.2967/jnumed.119.240911
http://www.ncbi.nlm.nih.gov/pubmed/32015164?tool=bestpractice.com
In a phase 3 trial of patients with advanced or progressive somatostatin receptor-positive midgut neuroendocrine tumors, lutetium Lu 177 dotatate significantly improved progression-free survival and response rate compared with high-dose, long-acting octreotide.[60]Strosberg J, El-Haddad G, Wolin E, et al. Phase 3 Trial of (177)Lu-Dotatate for midgut neuroendocrine tumors. N Engl J Med. 2017 Jan 12;376(2):125-35.
https://www.doi.org/10.1056/NEJMoa1607427
http://www.ncbi.nlm.nih.gov/pubmed/28076709?tool=bestpractice.com
Final analysis of overall survival favored lutetium Lu 177 dotatate, but was not statistically significant.[61]Strosberg JR, Caplin ME, Kunz PL, et al. (177)Lu-Dotatate plus long-acting octreotide versus high‑dose long-acting octreotide in patients with midgut neuroendocrine tumours (NETTER-1): final overall survival and long-term safety results from an open-label, randomised, controlled, phase 3 trial. Lancet Oncol. 2021 Dec;22(12):1752-63.
http://www.ncbi.nlm.nih.gov/pubmed/34793718?tool=bestpractice.com