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A rare case of intra-pancreatic cystic lesion (with video)

1 Digestive Endoscopy Center, Shanghai Tenth People's Hospital, Tongji University School of Medicine, Shanghai, China
2 Department of Gastroenterology, Jiangyin Hospital of Traditional Chinese Medicine, Jiangyin Hospital Affiliated to Nanjing University of Chinese Medicine, Jiangyin, Jiangsu Province, China

Date of Submission26-Dec-2021
Date of Acceptance24-Apr-2022
Date of Web Publication05-Oct-2022

Correspondence Address:
Feng Liu,
Digestive Endoscopy Center, Shanghai Tenth People's Hospital, Tongji University School of Medicine, 301 Mid. Yanchang Road, Shanghai 200072
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/EUS-D-21-00277

How to cite this URL:
Li J, Shen M, Liu F. A rare case of intra-pancreatic cystic lesion (with video). Endosc Ultrasound [Epub ahead of print] [cited 2022 Nov 28]. Available from: http://www.eusjournal.com/preprintarticle.asp?id=357882

A 60-year-old woman presented with mild epigastric pain after aspirin medication for prophylactic purposes. A contrast-enhanced computed tomography (CT) revealed an intra-pancreatic enhancing lesion suspicious for neuroendocrine tumor or vascular malformation [Figure 1]a. Blood tests showed no abnormalities. The patient was referred to our center for EUS evaluation of the intra-pancreatic lesion with a plan to perform fine-needle aspiration. Under EUS, an unilocular anechoic lesion measuring about 13.6 mm × 6.4 mm was seen in the pancreatic tail [Figure 1]b. The lesion was positive of Doppler flow with communication to the splenic vein [Figure 1]c and [Video 1 [Additional file 1]]. The patient denied any history of trauma, surgery, liver cirrhosis, or pancreatitis. A CT angiography was scheduled and confirmed the lesion to be connected with the splenic vein but early enhanced during the arterial phase, indicating an arteriovenous malformation [Figure 1]d. The patient subsequently provided a history of epistaxis, oral, and finger bleeding concomitant with epigastric pain after aspirin medication. These symptoms were relieved after aspirin suspension. Physical examination revealed multiple dilated dark purple vessels on her tongue [Figure 2]a and right thumb [Figure 2]b. She also gave a family history of similar lesions on the face, lips, or fingers of her grandmother, father, sister, and son. A diagnosis of hereditary hemorrhagic telangiectasia (HHT) is made according to the Curaçao criteria.[1]
Figure 1: Radiology and EUS images of the pancreatic lesion. (a) Contrast-enhanced CT showed an enhancing lesion in the pancreatic tail (yellow arrow); (b and c), EUS showed an anechoic lesion (yellow arrow) with positive Doppler flow and communication to the Sv; (d) CT angiography showed the lesion to be early enhanced during the arterial phase with connection to the splenic vein (yellow arrow). CT: Computed tomography; Sv: splenic vein

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Figure 2: Clinical images. Multiple dilated dark purple vessels on the tongue (a) and right thumb (b)

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Also known as Rendu-Osler-Weber disease, HHT is an autosomal dominant condition characterized by the development of telangiectasias, arteriovenous malformations, and aneurysms.[2] It predominantly involves the brain, lung, liver, gastrointestinal tract, nasal mucosa, and skin. Pancreatic involvement is rare in HHT. In the present case, the patient was examined later by a series of brain and thoracic CT and gastrointestinal endoscopy. Involvement of these organs was excluded and the pancreatic lesion was managed by conservative surveillance. To the best of our knowledge, this is the first reported case of HHT with solitary pancreatic lesion but no other visceral involvement. EUS could display anatomic features of such lesions, provide direct vision of the internal bloodstream, and trace the vessels to reveal their connections. Accurate diagnosis of such a case is crucial to avoid fine-needle aspiration with potentially catastrophic consequences.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Faughnan ME, Mager JJ, Hetts SW, et al. Second International Guidelines for the Diagnosis and Management of Hereditary Hemorrhagic Telangiectasia. Ann Intern Med 2020;173:989-1001.  Back to cited text no. 1
Tortora A, Riccioni ME, Gaetani E, et al. Rendu-Osler-Weber disease: A gastroenterologist's perspective. Orphanet J Rare Dis 2019;14:130.  Back to cited text no. 2


  [Figure 1], [Figure 2]


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