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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 6  |  Issue : 2  |  Page : 136-139

Cystic pancreatic lymphangioma diagnosed with endoscopic ultrasound-guided fine needle aspiration


Department of Gastroenterology and Hepatology, Changi General Hospital, Simei, Singapore

Date of Submission15-Oct-2015
Date of Acceptance22-Apr-2016
Date of Web Publication20-Apr-2017

Correspondence Address:
Ikram Hussain
Department of Gastroenterology and Hepatology, Changi General Hospital, 2 Simei Street 3
Singapore
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2303-9027.204807

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  Abstract 

Pancreatic lymphangiomas are rare, but benign neoplasms. Historically, the diagnoses in various case reports were mostly made after surgical resection. There are emerging data concerning the utility of endoscopic ultrasound-guided fine needle aspiration (EUS-FNA) to differentiate it from more sinister pancreatic cystic neoplasms. A confident preoperative diagnosis with EUS-FNA is crucial to avoid unnecessary workup or surgery, especially for small and asymptomatic lesions. Here, we present a rare case of asymptomatic pancreatic lymphangioma which was diagnosed with EUS-FNA. The case highlights that a preoperative diagnosis of pancreatic lymphangioma can be made with certainty.

Keywords: Endoscopic fine needle aspiration, endoscopic ultrasound, lymphangioma, pancreatic cys


How to cite this article:
Hussain I, Ang TL. Cystic pancreatic lymphangioma diagnosed with endoscopic ultrasound-guided fine needle aspiration. Endosc Ultrasound 2017;6:136-9

How to cite this URL:
Hussain I, Ang TL. Cystic pancreatic lymphangioma diagnosed with endoscopic ultrasound-guided fine needle aspiration. Endosc Ultrasound [serial online] 2017 [cited 2020 Jan 24];6:136-9. Available from: http://www.eusjournal.com/text.asp?2017/6/2/136/204807


  Introduction Top


With the widespread use of high-resolution abdominal cross-sectional imaging, more asymptomatic pancreatic cystic lesions are being detected. The key diagnostic concern is the malignant potential of these lesions and the need for surgical intervention. Endoscopic ultrasound (EUS) can provide characteristic high-resolution morphologic images for diagnosis and management. EUS-guided fine needle aspiration (EUS-FNA) has an important diagnostic role, based on the acquisition of cytology and biochemical markers of cyst fluid.[1]

We report a rare case of pancreatic cystic lesion due to pancreatic lymphangioma that was diagnosed by EUS-FNA. Pancreatic lymphangiomas cause symptoms when they become large or when they compress upon crucial surrounding structures. Historically, these lesions have been diagnosed after surgical resection of symptomatic lesions. In last decade, only a handful of cases has been preoperatively diagnosed with EUS-FNA, thus avoiding surgery for asymptomatic lesions.[2],[3],[4],[5]


  Case Report Top


A 49-year-old female was referred by the surgeon to our clinic due to incidental detection of a cystic lesion of pancreas. She underwent computed tomography (CT) of the abdomen 2 months earlier during a hospitalization for severe enteritis. A lobulated cyst of 3.4 cm × 3.0 cm was identified in the body of the pancreas [Figure 1]. The rest of the pancreas was unremarkable, and the main pancreatic duct was not dilated. This had been detected 15 months earlier by CT, which was performed whereas evaluating for suspected appendicitis. She denied any chronic abdominal pain except a painful episode of 1 week during the period of enteritis 2 months ago. She had no nausea, vomiting, weight loss, anorexia, or postprandial distress. She was not a smoker and did not consume alcohol. She had past medical history of thalassemia, appendectomy, and hemorrhoidectomy. She denied history of any abdominal trauma. She was not on any long-term medications. The laboratory analysis showed mild anemia. The liver and renal biochemical tests were within normal range.
Figure 1: Computed tomography image of cystic pancreatic lesion

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EUS [Figure 2] was performed for further characterization of the cyst. A discrete, multiloculated cystic structure, with anechoic contents separated by thin septae, was visualized in the body of the pancreas. The maximum diameter of the cyst was 38 mm. There was no mural nodule or calcification in the cyst. A connection with the main pancreatic duct was absent. The remaining pancreas did not show features of chronic pancreatitis, and the diameter of the main pancreatic duct was normal. After puncturing with a 22G needle (Echotip ® Ultra; Cook Medical, Winston-Salem, USA), 10 mL of lipemic, white, opaque, and thick fluid was aspirated in a single pass [Figure 3]. The complete collapse of cyst was seen postaspiration. The biochemical analysis of the fluid showed normal carcinoembryonic antigen level (35 mcg/L), high amylase level (325 U/L), and very high triglycerides level (1451.3 mg/dL). The cytology of fluid demonstrated a moderate yield of mixed lymphoid cells in a proteinaceous background. There were no malignant cells. Based on the findings on EUS-FNA combined with high triglycerides level, a diagnosis of pancreatic lymphangioma was made. Since the patient was asymptomatic, a nonsurgical approach with monitoring of the lesion was adopted.
Figure 2: Endoscopic ultrasound image of cystic pancreatic lesion

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Figure 3: White fluid was aspirated by endoscopic ultrasound-guided fine needle aspiration

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  Discussion Top


Lymphangiomas are benign and rare neoplasms. Considered congenital in nature, these are most commonly diagnosed in children. Females are more commonly affected. The disruption of lymphatic vessels during embryogenesis is postulated to result in progressive local accumulation of lymph surrounded by thin wall septae of endothelial cells, giving rise to cysts which are mostly multiloculated. Such lymphatic obstruction may also rarely occur after trauma, surgery and radiation therapy, and potentially result in lymphangioma. The head and neck region of the body harbors around 90% of all lymphangiomas, and a minority are found elsewhere (e.g., axillae, spleen, small intestines). The pancreas, harboring <1% of all lymphangiomas, is relatively an uncommon location. On the other hand, only about 0.2% of the pancreatic lesions are suspected to be lymphangiomas.[6]

Pancreatic lymphangiomas most commonly come to attention when they start to cause symptoms (e.g., abdominal pain, nausea, weight loss, etc.). Due to the rarity of such lesions, the natural history is uncertain at present although no case of malignant transformation has ever been reported. There are no specific features on CT, EUS, and magnetic resonance imaging which may confidently differentiate a lymphangioma from other pancreatic cysts. In the majority of reported cases, the pancreatic lymphangiomas were surgically resected either to alleviate symptoms or to characterize the true nature of the cyst histologically. Although surgical resection is the most definitive treatment, the issue of morbidity and mortality must be considered, and small and asymptomatic lesions may be monitored safely. The key lies in a confident preoperative diagnosis.

EUS is an excellent modality for imaging of pancreatic cystic lesions and EUS-FNA provides a sample for fluid analysis to increase diagnostic certainty [Table 1]. Usually in such imaging, a pancreatic lymphangioma appears as a well-defined, multi-cystic cavity with thin septae. Sometimes, a concomitant solid component due to collapsed cavities may be confused with a more sinister diagnosis (e.g., malignancy). The characteristic appearance of a chylous, milky-white fluid with a high triglyceride level allows the diagnosis of lymphangioma to be made confidently. With a confident preoperative diagnosis by EUS-FNA, surgery can be avoided.[2],[3],[4],[5] In other rare instances of preoperative diagnosis, surgical resection was still performed due to the presence of symptoms.[7],[8] However as a cautionary note, EUS-FNA of a pancreatic cyst may result in complications such a bleeding and infection. The decision for EUS-FNA to achieve a confident diagnosis, although occasionally risky, should be individualized, and taken in conjunction with the patient.
Table 1: Pancreatic lymphangiomas diagnosed only with endoscopic ultrasound-guided fine needle aspiration

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To conclude, lymphangioma should be considered in the differential diagnosis of pancreatic cystic lesions. EUS-FNA has an important diagnostic role with the potential to guide therapy.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.[11]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
But DY, Poley JW. To fine needle aspiration or not? An endosonographer's approach to pancreatic cystic lesions. Endosc Ultrasound 2014;3:82-90.  Back to cited text no. 1
    
2.
Jathal A, Arsenescu R, Crowe G, et al. Diagnosis of pancreatic cystic lymphangioma with EUS-guided FNA: Report of a case. Gastrointest Endosc 2005;61:920-2.  Back to cited text no. 2
    
3.
Dries AM, McDermott J. Diagnosis of cystic lymphangioma of the pancreas with endoscopic ultrasound-guided fine needle aspiration. Am J Gastroenterol 2008;103:1049-50.  Back to cited text no. 3
    
4.
Applebaum B, Cunningham JT. Two cases of cystic lymphangioma of the pancreas: A rare finding in endoscopic ultrasonography. Endoscopy 2006;38 Suppl 2:E24-5.  Back to cited text no. 4
    
5.
Sanaka MR, Kowalski TE. Cystic lymphangioma of the pancreas. Clin Gastroenterol Hepatol 2007;5:e10-1.  Back to cited text no. 5
    
6.
Paal E, Thompson LD, Heffess CS. A clinicopathologic and immunohistochemical study of ten pancreatic lymphangiomas and a review of the literature. Cancer 1998;82:2150-8.  Back to cited text no. 6
    
7.
Barnes EL, Lee LS. Got milk? An unusual cause of abdominal pain. Gastroenterology 2015;148:e1-2.  Back to cited text no. 7
    
8.
Bhatia V, Rastogi A, Saluja SS, et al. Cystic pancreatic lymphangioma. The first report of a preoperative pathological diagnosis by endoscopic ultrasound-guided cyst aspiration. JOP 2011;12:473-6.  Back to cited text no. 8
    
9.
Barresi L, Tarantino I, Curcio G, et al. Pancreatic cystic lymphangioma in a 6-year-old girl, diagnosed by endoscopic ultrasound (EUS) fine needle aspiration. Endoscopy 2011;43 Suppl 2:E61-2.  Back to cited text no. 9
    
10.
Coe AW, Evans J, Conway J. Pancreas cystic lymphangioma diagnosed with EUS-FNA. JOP 2012;13:282-4.  Back to cited text no. 10
    
11.
Mansour NM, Salyers WJ Jr. Recurrence of a pancreatic cystic lymphangioma after diagnosis and complete drainage by endoscopic ultrasound with fine-needle aspiration. JOP 2013;14:280-2.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1]


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