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Revista argentina de cirugía

versión impresa ISSN 2250-639Xversión On-line ISSN 2250-639X

Rev. argent. cir. vol.112 no.3 Cap. Fed. jun. 2020

http://dx.doi.org/10.25132/raac.v112.n3.1464.es 

Articles

Neuroendocrine cell hyperplasia mimicking a solid pancreatic tumor

José G. Yaryura Montero1  * 

Mario A. Cafaro1 

Ramiro X. Casa1 

José S. García1 

Lucas Granero1 

1 Servicio de Cirugía Ge neral, Sanatorio Allende. Córdoba, Argentina.

Pancreatic endocrine cell hyperplasia (PECH), a condition observed in 10% of adults, is defined as an increase in the number of cells of Langerhans islets1.

We report the case of a male patient with imaging tests suggestive of tail of pancreas tumor who underwent laparoscopic distal pancreatectomy and splenectomy. The pathological examination of the surgical specimen demonstrated PECH.

The aim of this paper is to describe an atypical presentation of this disease with review of the literature.

We report the case of a 72-year-old male patient who sought medical care due to a single episode of pain in the epigastric and left hypochondriac regions. The laboratory tests performed in other institution were normal and the abdominal ultrasound revealed the presence of a 30 x 30 mm hyperechogenic mass in the tail of the pancreas.

In our institution, the patient underwent triple-phase computed tomography (CT) scan which showed pancreas tail thickness of 29 mm with arterial phase enhancement of 65 Hounsfield units with higher density (112 Hounsfield units) in the portal-venous phase (Fig. 1-A). A gadolinium-enhanced magnetic resonance imaging (MRI) described a 35 x 25 mm irregular lesion in the distal pancreas with heterogeneous enhancement, hypointense on T1-weighted images, hyperintense on T2-weighted fat suppressed images and hyperintense on T1-weighted fat suppressed images and intravenous contrast (Fig. 1-B).

Figure 1 A. CT scan, portal-venous phase: distal pancreas tumor (black arrow). B. T1-weighted MRI images: distal pancreas tumor (black arrowhead) 

The images were evaluated by two specialists in diagnostic imaging, who concluded that the images corresponded to a solid malignancy.

The possible diagnoses were neuroendocrine tumor (NET) due to the characteristics described on MRI or distal pancreatic ductal adenocarcinoma due to the CT scan images. The tumor markers (CEA, CA 19-9 and CA 125) were within normal ranges. Chromogranin A and endoscopic ultrasound were not performed.

After discussing the case in the pancreatic surgery unit, laparoscopic distal pancreatectomy and splenectomy was decided. The spleen was not preserved because of the proximity with the suspected pancreatic malignancy.

The macroscopic section of the specimen showed a hard consistency area of 2.5 x 2 cm in size, which was whitish and poorly defined. A brownish nodular lesion with net edges, 0.6 centimeters in diameter, was identified near this area (Fig. 2). The microscopic examination described the presence of increased numbers of eosinophils in the pancreatic islets without anaplasia, consistent with PECH and cells suggestive of intrapancreatic splenic tissue adjacent to the PECH. The immunohistochemical analysis was positive for neuron-specific enolase, chromogranin, CD56, synaptophysin and a Ki-67 proliferation index of less than 2% (Fig. 3). These histologic findings confirmed the diagnosis of PECH.

The patient evolved without complications and was discharged three days after surgery.

Figure 2 Surgical specimen. Thickened distal pancreas (white arrow); intrapan creatic accessory spleen (white arrowhead). 

Figure 3 Histological examination. A. Neuroendocrine cell hyperplasia, hematoxylin and eosin stain (HE 40×). B. Immunostaining for chromogranin, 20x (long black arrow). C. Immunostaining for Ki-67 antigen (hollow black arrowhead), 20x. D. Immunostai ning for synaptophysin (hollow white arrowhead), 20x. 

There is no agreement to define the diagnostic criteria of PCHE1. While some authors define this condition as an expansion of the endocrine cell compartment of the pancreas > 2% of the total pancreatic mass in adults1,2, others define it as islets diameter > 250 μm with expansion in the number of islets1,2. All types of endocrine cells can be hyperplastic, but hyperplasia is more common in β and α cells1.

Most of the patients do not develop symptoms, but some cases can be associated with multiple endocrine neoplasia type 1 (MEN1), von Hippel-Lindau disease1, alpha1-antitrypsin deficiency hyperinsulinism and Von Recklinghausen disease2.

In patients with MEN1, the pancreas presents hyperplastic islets and NETs1,3. These lesions could be considered as precursor lesions of NETs in patients with MEN-1; yet, this theory is not completely clear1,3. As there have been no reports of similar precursor lesions for sporadic NETs, it is possible that PNETs develop from such pre-malignant lesions1,3.

There are exceptional cases in the medical literature of patients undergoing surgery for a suspected neuroendocrine tumor in the distal pancreas, in whom the pathological examination reported PECH4, as occurred in our case. However, typical PECH does not appear as a tumor and is usually asymptomatic4.

Nowadays, the diagnosis of solid and cystic pancreatic lesions has increased with the advent of CT scan and MRI4. The sensitivity and specificity of endoscopic ultrasound-guided fine needle aspiration to characterize pancreatic lesions is not of 100%5. This test should be considered for the diagnosis although it is difficult to differentiate between neuroendocrine tumor and PECH4.

Bearing this in mind, our case was presented in the pancreatic surgery unit of our institution and surgery was decided based on the reports of CT scan and the MRI without performing endoscopic ultrasound.

When malignancy cannot be ruled out by routine complementary tests and biopsy is not feasible, surgical resection should be considered5 with subsequent histological and immunohistochemical analysis3. Curative resection via laparoscopy is the treatment of choice for tumors of the distal pancreas6.

In conclusion, we reported a case of a pancreatic tumor with a pathological diagnosis of PECH which poses the differential diagnosis with other more common pancreatic tumors. Laparoscopic surgery is feasible when the preoperative diagnosis by imaging tests or biopsy is not possible.

Referencias bibliográficas /References

1. Ouyang D, Dhall D, Yu R. Pathologic pancreatic endocrine cell hy perplasia. World J Gastroenterol. 2011;17(2):137. doi:10.3748/wjg.v17.i2.137. [ Links ]

2. Rindi G, Solcia E. Endocrine Hyperplasia and Dysplasia in the Pathogenesis of Gastrointestinal and Pancreatic Endocri ne Tumors. Gastroenterol Clin North Am. 2007;36(4):851-65. doi:10.1016/j.gtc.2007.08.006. [ Links ]

3. Ro C, Chai W, Yu VE, Yu R. Pancreatic neuroendocrine tumors: bio logy, diagnosis, and treatment. Chin J Cancer. 2013;32(6):312-24. [ Links ]

4. Mori R, Takeda Y, Sakamoto T, et al. A resected case of tumorige nic pancreatic endocrine cell hiperplasia. Gan To Kagaku Ryoho. 2019;46(3):549-51. [ Links ]

5. Okun SD, Lewin DN. Non-neoplastic pancreatic lesions that may mimic malignancy. Semin Diagn Pathol. 2016;33(1):31-42. doi:10.1053/j.semdp.2015.09.005. [ Links ]

6. Wakasugi M, Tori M, Akamatsu H, Ueshima S, Omori T, Tei M, et al. Laparoscopic distal pancreatectomy for multiple epithelial cysts in an intrapancreatic accessory spleen. A case report and review of literature. JOP. 2013;14(6):636-41. doi:10.6092/1590-8577/1784. [ Links ]

Received: November 11, 2019; Accepted: March 02, 2020

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