VIPoma is characterised by profuse watery diarrhoea, hypokalaemia, metabolic acidosis, and hypochlorhydria or achlorhydria, in the presence of raised serum vasoactive intestinal peptide (VIP). The clinical syndrome associated with VIPomas is sometimes referred to as WDHA syndrome (watery diarrhoea, hypokalaemia, and achlorhydria), Verner-Morrison syndrome, or pancreatic cholera.[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
Multi-phase contrast-enhanced computed tomography (CT) or magnetic resonance imaging (MRI) scan is recommended to identify the primary tumour.[3]National Comprehensive Cancer Network. NCCN clinical practice guidelines in oncology: neuroendocrine and adrenal tumors [internet publication].
https://www.nccn.org/guidelines/category_1
History
Patients usually present with profuse watery diarrhoea (100% >700 mL/day); stool volumes typically exceed 3 L/day (70% to 80% of patients).[18]Vinik AI, Woltering EA, Warner RR, et al. NANETS consensus guidelines for the diagnosis of neuroendocrine tumor. Pancreas. 2010 Aug;39(6):713-34.
https://journals.lww.com/pancreasjournal/Fulltext/2010/08000/NANETS_Consensus_Guidelines_for_the_Diagnosis_of.3.aspx
http://www.ncbi.nlm.nih.gov/pubmed/20664471?tool=bestpractice.com
[19]Kulke MH, Anthony LB, Bushnell DL, et al. NANETS treatment guidelines: well-differentiated neuroendocrine tumors of the stomach and pancreas. Pancreas. 2010 Aug;39(6):735-52.
https://journals.lww.com/pancreasjournal/Fulltext/2010/08000/NANETS_Treatment_Guidelines__Well_Differentiated.4.aspx
http://www.ncbi.nlm.nih.gov/pubmed/20664472?tool=bestpractice.com
[20]Una Cidon E. Vasoactive intestinal peptide secreting tumour: an overview. World J Gastrointest Oncol. 2022 Apr 15;14(4):808-19.
http://www.ncbi.nlm.nih.gov/pubmed/35582098?tool=bestpractice.com
Large tumours can result in 6-8 L stool volumes per day.[21]de Herder WW, Hofland J. Vasoactive intestinal peptide-secreting tumor (VIPoma). 2023 Apr 5. In: Feingold KR, Anawalt B, Blackman MR, et al. eds. Endotext [Internet]. South Dartmouth (MA): MDText.com, Inc.; 2000.
https://www.ncbi.nlm.nih.gov/books/NBK278960
http://www.ncbi.nlm.nih.gov/pubmed/25905195?tool=bestpractice.com
Characteristically, the diarrhoea is odourless and tea-coloured, and persists even after 48-72 hours of fasting.
Patients may complain of headache and other dehydration-related symptoms. Flushing, secondary to vasodilation, may occur in up to 33% of patients.[9]Ghaferi AA, Chojnacki KA, Long WD, et al. Pancreatic VIPomas: subject review and one institutional experience. J Gastointest Surg. 2008 Feb;12(2):382-93.
http://www.ncbi.nlm.nih.gov/pubmed/17510774?tool=bestpractice.com
Medical history may reveal a history or family history of multiple endocrine neoplasia type 1 (MEN1). However, this is an uncommon finding; VIPoma is reported in <1% of patients with MEN1.[10]Thakker RV, Newey PJ, Walls GV, et al. Clinical practice guidelines for multiple endocrine neoplasia type 1 (MEN1). J Clin Endocrinol Metab. 2012 Sep;97(9):2990-3011.
https://academic.oup.com/jcem/article/97/9/2990/2536740
http://www.ncbi.nlm.nih.gov/pubmed/22723327?tool=bestpractice.com
[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
Physical examination
Physical examination findings will reflect hypokalaemia (e.g., muscle weakness, muscle cramps), and volume depletion (e.g., weight loss, poor skin turgor, dry mucus membranes). There may be evidence to suggest metastasis (e.g., hepatomegaly, weight loss).[9]Ghaferi AA, Chojnacki KA, Long WD, et al. Pancreatic VIPomas: subject review and one institutional experience. J Gastointest Surg. 2008 Feb;12(2):382-93.
http://www.ncbi.nlm.nih.gov/pubmed/17510774?tool=bestpractice.com
Laboratory investigations
A chemistry panel is the initial laboratory investigation in patients presenting with severe and persistent watery diarrhoea.
Chemistry panel will typically reveal hypokalaemia and low bicarbonate levels; hypercalcaemia and hyperglycaemia may also be present. Low bicarbonate levels may result in non-anion gap metabolic acidosis, which can be confirmed by arterial blood gas analysis.
Liver function tests are recommended; abnormal results may suggest hepatic metastasis.[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
Gastric pH is increased in patients with VIPoma due to inhibition of gastric acid secretion by VIP.[21]de Herder WW, Hofland J. Vasoactive intestinal peptide-secreting tumor (VIPoma). 2023 Apr 5. In: Feingold KR, Anawalt B, Blackman MR, et al. eds. Endotext [Internet]. South Dartmouth (MA): MDText.com, Inc.; 2000.
https://www.ncbi.nlm.nih.gov/books/NBK278960
http://www.ncbi.nlm.nih.gov/pubmed/25905195?tool=bestpractice.com
Gastric pH (via nasogastric tube aspiration) is rarely measured, but useful to assess for hypochlorhydria or achlorhydria if there is diagnostic uncertainty.[20]Una Cidon E. Vasoactive intestinal peptide secreting tumour: an overview. World J Gastrointest Oncol. 2022 Apr 15;14(4):808-19.
http://www.ncbi.nlm.nih.gov/pubmed/35582098?tool=bestpractice.com
Vasoactive intestinal peptide (VIP) levels
VIPoma patients usually have markedly raised levels of VIP (reference value <75 picograms/mL).[9]Ghaferi AA, Chojnacki KA, Long WD, et al. Pancreatic VIPomas: subject review and one institutional experience. J Gastointest Surg. 2008 Feb;12(2):382-93.
http://www.ncbi.nlm.nih.gov/pubmed/17510774?tool=bestpractice.com
One review of case reports and case series reported serum VIP levels ranging from 293 to 1500 picograms/mL.[9]Ghaferi AA, Chojnacki KA, Long WD, et al. Pancreatic VIPomas: subject review and one institutional experience. J Gastointest Surg. 2008 Feb;12(2):382-93.
http://www.ncbi.nlm.nih.gov/pubmed/17510774?tool=bestpractice.com
Diagnosis of VIPoma can be made based on raised VIP levels and the presence of clinical syndrome. The only pancreatic neuroendocrine tumour to secrete VIP is VIPoma.[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
VIP levels are measured by radioimmunoassay in symptomatic fasting patients (ideally during a bout of diarrhoea). If VIP levels are normal, the test should be repeated because VIP secretion from the tumour may be episodic.[22]Scottish Neuroendocrine Tumour Group. Consensus guidelines for the management of patients with neuroendocrine tumours. July 2015 [internet publication].
https://www.woscan.scot.nhs.uk/wp-content/uploads/FINAL-PUBLISHED-SCONET-Guideline-v1.1-July-2015.pdf
Neuroendocrine tumour markers
Neuroendocrine tumour markers (chromogranin A and pancreatic polypeptide) have limited utility as they are non-specific and secreted by a broad variety of neuroendocrine tumours.[23]O'Toole D, Kianmanesh R, Caplin M. ENETS 2016 consensus guidelines for the management of patients with digestive neuroendocrine tumors: an update. Neuroendocrinology. 2016;103(2):117-8.
https://karger.com/nen/article/103/2/117/220108/ENETS-2016-Consensus-Guidelines-for-the-Management
If VIP levels are normal but concerns regarding neuroendocrine tumour persist, chromogranin A and pancreatic polypeptide can be measured to aid diagnosis.
Imaging
Accurately locating the tumour and metastases is important for staging and guiding management.[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
Anatomical and functional imaging techniques should be used where available and as clinically indicated.
Anatomical imaging
Due to the hypervascular nature of pancreatic neuroendocrine tumours, multi-phasic CT and MRI scans are recommended to identify primary tumours and metastatic disease.[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
[25]Kunz PL, Reidy-Lagunes D, Anthony LB, et al. Consensus guidelines for the management and treatment of neuroendocrine tumors. Pancreas. 2013 May;42(4):557-77.
https://journals.lww.com/pancreasjournal/fulltext/2013/05000/consensus_guidelines_for_the_management_and.2.aspx
http://www.ncbi.nlm.nih.gov/pubmed/23591432?tool=bestpractice.com
CT
VIPomas are usually solitary tumours, >3 cm in diameter at diagnosis. Therefore, most can be located by multi-phase abdominal contrast-enhanced CT scan.[26]Sundin A, Arnold R, Baudin E, et al. ENETS consensus guidelines for the standards of care in neuroendocrine tumors: radiological, nuclear medicine & hybrid imaging. Neuroendocrinology. 2017;105(3):212-44.
http://www.ncbi.nlm.nih.gov/pubmed/28355596?tool=bestpractice.com
MRI
Multi-phase MRI of the abdomen effectively locates VIPomas, and is warranted if radiation exposure is to be avoided (e.g., pregnancy).[26]Sundin A, Arnold R, Baudin E, et al. ENETS consensus guidelines for the standards of care in neuroendocrine tumors: radiological, nuclear medicine & hybrid imaging. Neuroendocrinology. 2017;105(3):212-44.
http://www.ncbi.nlm.nih.gov/pubmed/28355596?tool=bestpractice.com
MRI is superior to CT scan in identifying hepatic metastasis.[26]Sundin A, Arnold R, Baudin E, et al. ENETS consensus guidelines for the standards of care in neuroendocrine tumors: radiological, nuclear medicine & hybrid imaging. Neuroendocrinology. 2017;105(3):212-44.
http://www.ncbi.nlm.nih.gov/pubmed/28355596?tool=bestpractice.com
Endoscopic ultrasound
For smaller tumours that prove elusive (rare), endoscopic ultrasound can allow for a definitive diagnosis via ultrasound-guided fine needle aspiration of the mass.[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
[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
Functional imaging
If imaging from CT and MRI is inconclusive, then functional somatostatin receptor-based imaging techniques should be considered if available. Somatostatin receptors are expressed in 80% to 90% of VIPomas.[9]Ghaferi AA, Chojnacki KA, Long WD, et al. Pancreatic VIPomas: subject review and one institutional experience. J Gastointest Surg. 2008 Feb;12(2):382-93.
http://www.ncbi.nlm.nih.gov/pubmed/17510774?tool=bestpractice.com
Somatostatin receptor-based imaging techniques have increased sensitivity for detecting neuroendocrine tumours (including metastases) compared with CT and MRI, making them particularly useful for staging and guiding treatment.[26]Sundin A, Arnold R, Baudin E, et al. ENETS consensus guidelines for the standards of care in neuroendocrine tumors: radiological, nuclear medicine & hybrid imaging. Neuroendocrinology. 2017;105(3):212-44.
http://www.ncbi.nlm.nih.gov/pubmed/28355596?tool=bestpractice.com
[27]Hope TA, Bergsland EK, Bozkurt MF, et al. Appropriate use criteria for somatostatin receptor PET imaging in neuroendocrine tumors. J Nucl Med. 2018 Jan;59(1):66-74.
https://www.doi.org/10.2967/jnumed.117.202275
http://www.ncbi.nlm.nih.gov/pubmed/29025982?tool=bestpractice.com
These modalities can identify patients with sufficient tumour somatostatin receptor expression who may benefit from treatment with somatostatin analogues.[26]Sundin A, Arnold R, Baudin E, et al. ENETS consensus guidelines for the standards of care in neuroendocrine tumors: radiological, nuclear medicine & hybrid imaging. Neuroendocrinology. 2017;105(3):212-44.
http://www.ncbi.nlm.nih.gov/pubmed/28355596?tool=bestpractice.com
[27]Hope TA, Bergsland EK, Bozkurt MF, et al. Appropriate use criteria for somatostatin receptor PET imaging in neuroendocrine tumors. J Nucl Med. 2018 Jan;59(1):66-74.
https://www.doi.org/10.2967/jnumed.117.202275
http://www.ncbi.nlm.nih.gov/pubmed/29025982?tool=bestpractice.com
Somatostatin receptor scintigraphy (using a radiolabelled somatostatin analogue, e.g., octreotide) can confirm the location of the tumour and detect occult hepatic metastasis. However, it has been mostly replaced by positron emission tomography (PET)-CT/MRI.
PET-CT/MRI employing novel somatostatin receptor PET tracers (e.g., 68Ga-DOTATATE; 64Cu-DOTATATE; 68Ga-DOTATOC) has increased sensitivity for detecting neuroendocrine tumours and metastases compared with conventional CT, MRI, and scintigraphy.[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
[26]Sundin A, Arnold R, Baudin E, et al. ENETS consensus guidelines for the standards of care in neuroendocrine tumors: radiological, nuclear medicine & hybrid imaging. Neuroendocrinology. 2017;105(3):212-44.
http://www.ncbi.nlm.nih.gov/pubmed/28355596?tool=bestpractice.com
[27]Hope TA, Bergsland EK, Bozkurt MF, et al. Appropriate use criteria for somatostatin receptor PET imaging in neuroendocrine tumors. J Nucl Med. 2018 Jan;59(1):66-74.
https://www.doi.org/10.2967/jnumed.117.202275
http://www.ncbi.nlm.nih.gov/pubmed/29025982?tool=bestpractice.com
[28]Gabriel M, Decristoforo C, Kendler D, et al. 68Ga-DOTA-Tyr3-octreotide PET in neuroendocrine tumors: comparison with somatostatin receptor scintigraphy and CT. J Nucl Med. 2007 Apr;48(4):508-18.
https://www.doi.org/10.2967/jnumed.106.035667
http://www.ncbi.nlm.nih.gov/pubmed/17401086?tool=bestpractice.com
[29]Barrio M, Czernin J, Fanti S, et al. The impact of somatostatin receptor-directed PET/CT on the management of patients with neuroendocrine tumor: a systematic review and meta-analysis. J Nucl Med. 2017 May;58(5):756-61.
https://www.doi.org/10.2967/jnumed.116.185587
http://www.ncbi.nlm.nih.gov/pubmed/28082438?tool=bestpractice.com
Biopsy
In patients with surgically resectable disease, preoperative biopsy is not indicated. For unresectable disease tissue diagnosis is recommended with endoscopic ultrasound-guided fine needle aspiration. Pathological evaluation focuses on Ki-67 and mitosis per high power field to determine tumour grade and prognosis.[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
Operative exploration
Intraoperative ultrasound can be used during operative exploration to localise smaller tumours. However, most tumours are easily visualised and palpated at pancreatic exploration.