Cavernous hemangioma of an intrapancreatic accessory spleen mimicking a pancreat

lNTRODUCTlON

An intrapancreatic accessory spleen(IPAS)is an uncommon condition,with a prevalence ranging from 1.1%-3.4% in individuals[1,2].An IPAS is typically asymptomatic and has an innocuous nature.However,overlapping imaging manifestations of an IPAS and primary pancreatic tumors may lead to unnecessary surgery[3].A typical IPAS demonstrates a solid lesion with a round,oval or triangular shape,which is similar to the spleen on both precontrast and contrast-enhanced images.Therefore,this disorder is frequently confused with adenocarcinomas,neuroendocrine tumors or other solid pancreatic entities.When compared with a solid IPAS,cystic lesions arising from an IPAS are rare but necessitate a differential diagnosis with pancreatic cystic neoplasms,especially those possessing the potential to be malignant.Moreover,when considering the high likelihood of false-negative results,biopsy of cystic pancreatic lesions is seldom performed,and surgery is ultimately performed in most patients.

Herein,we report such a case involving a patient who underwent laparoscopic pancreatic body and tail resections because of an indeterminate pancreatic cystic lesion.Postoperative pathology confirmed this lesion as being a cavernous hemangioma arising from an IPAS.Furthermore,the clinical and imaging characteristics of IPAS and pancreatic cystic neoplasms(according to the previous literature)were also reviewed(Table 1).

CASE PRESENTATlON

Chief complaints

A 32-year-old male was referred to our hospital because of a suspicious lesion neighboring the hilum of the spleen,which was detectedconventional grayscale ultrasound in a local community hospital.The patient did not complain of obvious discomfort.

History of past illness

The patient had a history of chronic hepatitis B.

Physical examination

The patient did not complain of abdominal pain or any remarkable discomfort during the physical examination.

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Laboratory examinations

In addition to a slightly increased albumin-globulin ratio(2.96)and glutamine transpeptidase level(63 IU/L),no abnormal laboratory test results,including those of related tumor markers,were found.

Imaging examinations

The patient underwent contrast-enhanced ultrasound(CEUS)in our department.Before the CEUS,a baseline ultrasound illustrated a complicated cystic nodule measuring 2 cm,with a well-defined border in the tail of the pancreas without salient blood supply on color Doppler ultrasound(Figure 1).For the CEUS,a bolus injection of the US contrast agent SonoVue(Bracco,Milan,Italy)was administered through the antecubital vein,followed by a flush of 5 mL of 0.9% normal saline.The lesion demonstrated peripheral nodular and internal septal isoenhancement in the arterial phase,followed by slight hyperenhancement of the enhanced area in the venous phase.The predominant cystic area of the lesion did not show any enhancement in either phase.According to the aforementioned enhancing pattern in the CEUS,the lesion was suspected to be a pancreatic cystadenomaCEUS(Figure 1).

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Contrast-enhanced computed tomography(CECT)was performed to further examine the lesion.On the unenhanced CT,a nodule with a diameter of 2.2 cm and slightly low density was identified in the tail of the pancreas.Septa were observed,whereas no significant enhancement was presented within the lesion(Figure 2).The nodule was diagnosed as being a pancreatic cystic lesionthe CECT.Moreover,no salient abnormalities were found in the liver,kidney,spleen or biliary systemimaging evaluations.

FlNAL DlAGNOSlS

Informed consent was obtained from the patient for the publication of any potentially identifiable images or data included in this article.

TREATMENT

After multidisciplinary discussion and communication with the patient,as well as with his family,laparoscopic pancreatic body and tail resections were performed.

Typical IPAS presents as a solid lesion and demonstrates similar manifestations to the spleen on both precontrast and contrast-enhanced ultrasound[7,8].However,cystic neoplasm development in IPASs is rare.Sporadic cases of epidermoid cysts in IPASs(known as ECIPASs)have been reported[6,9-11].The walls of ECIPASs are irregularly thickened and thicker than those of mucinous cystic neoplasms(MCNs)and intraductal papillary mucinous neoplasms(IPMNs)[9].Moreover,the evident contrast enhancement of the partially thickened wall of ECIPAS(which is similar to that of the spleen)makes it possible to distinguish ECIPASs from MCNs or IPMNs.

The authors would like to express their gratitude to the patient and his family.

OUTCOME AND FOLLOW-UP

Postoperative pathology demonstrated that the lesion was a splenic cavernous hemangioma in the pancreas(Figure 3).After an uneventful postoperative course,the patient was discharged on postoperative day 5.No obvious abnormality was found in a follow-up abdominal US one month later(Timeline of diagnosis and treatment of the pancreatic lesion is presented in Supplementary Figure 1).

DlSCUSSlON

Cavernous hemangioma in the intrapancreatic accessory spleen may mimic pancreatic cystadenoma,which is a condition with the potential for malignancy.Imaging follow-ups or surgical interventions may be helpful for the exclusion of malignant risks in complicated cystic lesions,especially those with parietal and septal enhancements.

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The differential diagnosis was even more considerable in our case.The cystic cavernous hemangioma in the IPAS(known as CHIPAS)presented peripheral nodular and internal septal enhancements,which are frequently observed in pancreatic mucinous cystadenomas(MCAs).Furthermore,the majority of MCAs are located in the tail of the pancreas,where IPASs are also frequently discovered[12].Therefore,this increases the difficulty of an accurate diagnosis.However,the ancillary features of a fibrous pseudocapsule or calcified contents inside of the MCNs have also been reported[13].Another pancreatic cystic lesion that warrants vigilant discrimination from the CHIPAS is an IPMN.An IPMN in the main duct possesses a high risk of malignancy,with 38%-68% being confirmed as high-grade dysplasia or pancreatic cancer in postoperative specimens[14].Fortunately,CEUS is sensitive in being demonstrated in the dilated main pancreatic duct and the polycystic lesion connecting to the pancreatic duct or in developing within the duct in cases of IPMNs[15].

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To our knowledge,there is only one case report of solid cavernous hemangioma detected in both the spleen and the IPAS[16].In this case,the CHIPAS was accurately identified by the investigators because of a similar enhancement pattern of the pancreatic lesion and the splenic lesions on CECT and contrastenhanced magnetic resonance imaging.An accurate diagnosis was more difficult,as in our patient,because there was no lesion in the spleen for comparison.Moreover,a splenic hemangioma typically shows a hyperechoic and solid appearance.The atypical cystic appearance in our patient increased the difficulty of making an accurate diagnosis.

Herein,we presented on an extremely rare case of a cystic cavernous hemangioma arising from an IPAS.Contrast-enhanced ultrasound is sensitive in demonstrating the enhancements of the septa and the parietal nodule.However,an accurate diagnosis of cystic cavernous hemangioma arising from an IPASimaging tools is challenging.Imaging follow-ups or surgical interventions may be needed,due to the possible malignancy risk of a complicated cystic lesion with parietal and septal enhancements.

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CONCLUSlON

Intrapancreatic accessory spleen is a rare congenital condition,compared with an accessory spleenlocated at the hilum of the spleen[2,4].Due to its innocuous nature and infrequent induction of symptoms,IPAS seldom requires therapy unless they cause symptoms as a result of the compression,torsion or spontaneous rupture of a hemorrhage[5,6].

ACKNOWLEDGEMENTS

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FOOTNOTES

Luo Y performed the contrast-enhanced ultrasound examination for the patient and proposed writing it up as a case report;Huang JY collected the clinical information of the patient,reviewed the literature and contributed to manuscript drafting;Yang R provided the pathological data and helped with creating the figures;Li JW contributed to revising the grammar of the manuscript;Luo Y and Lu Q were responsible for the revision of the manuscript for important intellectual content;all of the authors issued final approval for this version of the manuscript to be submitted.

the National Natural Science Foundation of China,No.81571697.

The lesion was misdiagnosed as pancreatic cystadenoma by CEUS and CECT.

The authors declare that they have no conflicts of interest.

The authors have read the CARE Checklist(2016),and the manuscript was prepared and revised according to the CARE Checklist(2016).

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This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers.It is distributed in accordance with the Creative Commons Attribution NonCommercial(CC BYNC 4.0)license,which permits others to distribute,remix,adapt,build upon this work non-commercially,and license their derivative works on different terms,provided the original work is properly cited and the use is noncommercial.See:http://creativecommons.org/Licenses/by-nc/4.0/

China

Jia-Yan Huang 0000-0002-1918-2874;Rui Yang 0000-0002-7733-0636;Jia-Wu Li 0000-0003-0844-5883;Qiang Lu 0000-0002-4057-1997;Yan Luo 0000-0003-2985-1768.

Zhang H

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