Case Report Open Access
Copyright ©The Author(s) 2024. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Feb 16, 2024; 12(5): 995-1003
Published online Feb 16, 2024. doi: 10.12998/wjcc.v12.i5.995
Imaging, pathology, and diagnosis of solitary fibrous tumor of the pancreas: A case report and review of literature
Wen-Wen Wang, Xiang Wu, Luo-Luo Wang, Yi Ruan, Li-Li Qiu, Xin-Hua Zhou, Department of Hepatobiliary and Pancreatic Surgery, The Affiliated Li Huili Hospital, Ningbo University School of Medicine, Ningbo 315000, Zhejiang Province, China
Shu-Ping Zhou, Ningbo College of Health Sciences, Ningbo College of Health Sciences, Ningbo 315000, Zhejiang Province, China
Xiang Wu, Health Science Center, Ningbo University, Ningbo 315000, Zhejiang Province, China
Jun Lu, Department of Hepatobiliary and Pancreatic Surgery, The Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou, 310006, Zhejiang Province, China
Hai-Li Li, Xu-Ling Ni, Department of Diagnosis, Ningbo Diagnostic Pathology Center, Ningbo 315000, Zhejiang Province, China
ORCID number: Xiang Wu (0000-0002-2134-1184); Jun Lu (0000-0001-6225-1225); Xin-Hua Zhou (0000-0001-6707-9655).
Co-first authors: Wen-Wen Wang and Xiang Wu.
Co-corresponding authors: Xin-Hua Zhou and Li-Li Qiu.
Author contributions: Wang WW, Zhou SP and Wu X, investigation, data curation, writing-original draft; Wang LL investigation, provide image pictures; Ruan Y investigation, funding acquisition, medical history collection; Lu J investigation, supervision; Li HL pathology to provide; Ni XL pathology to provide; Qiu LL and Zhou XH resources, writing-review and editing, supervision, project administration funding acquisition; all authors read and approved the final manuscript. The author contributions of Wang WW and Wu X as co-first authors, Qiu LL and Zhou XH as corresponding authors are as follows: Wang WW and Wu X are co-first authors because they made equal contributions to the research and were involved in all stages of the study, from design to data collection and analysis. They both contributed significantly to the writing of the manuscript and are responsible for the accuracy and validity of the results reported. Qiu LL and Zhou XH are corresponding authors because they supervised the research project, provided guidance and expertise throughout the study, and ensured the quality and reliability of the data presented. As senior researchers, they are responsible for the overall content and implications of the study and act as points of contact for further information or queries.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: 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 BY-NC 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 non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Xin-Hua Zhou, Doctor, PhD, Chief Doctor, Doctor, Department of Hepatobiliary and Pancreatic Surgery, The Affiliated Li Huili Hospital, Ningbo University School of Medicine, No. 1111 Jiangnan Road, Yinzhou District, Ningbo 315000, Zhejiang Province, China. zhouxinhua1002@163.com
Received: October 13, 2023
Peer-review started: October 13, 2023
First decision: November 28, 2023
Revised: December 1, 2023
Accepted: January 18, 2024
Article in press: January 18, 2024
Published online: February 16, 2024

Abstract
BACKGROUND

A solitary fibrous tumor (SFT) is often located in the pleura, while SFT of the pancreas is extremely rare. Here, we report a case of SFT of the pancreas and discuss imaging, histopathology, and immunohistochemistry for accurate diagnosis and treatment.

CASE SUMMARY

A 54-year-old man presented to our hospital with pancreatic occupancy for over a month. There were no previous complaints of discomfort. His blood pressure was normal. Blood glucose, tumor markers, and enhanced computed tomography (CT) suggested a malignant tumor. Because the CT appearance of pancreatic cancer varies, we could not confirm the diagnosis; therefore, we performed endoscopic ultrasound-guided fine-needle biopsy (EUS-FNB). Pathology and immunohistochemistry were consistent with SFT of the pancreas. The postoperative pathology and immunohistochemistry were consistent with the puncture results. The patient presented for a follow-up examination one month after discharge with no adverse effects.

CONCLUSION

Other diseases must be excluded in patients with a pancreatic mass that cannot be diagnosed. CT and pathological histology have diagnostic value for pancreatic tumors. Endoscopic puncture biopsy under ultrasound can help diagnose pancreatic masses that cannot be diagnosed preoperatively. Surgery is an effective treatment for SFT of the pancreas; however, long-term follow-up is strongly recommended because of the possibility of malignant transformation of the tumor.

Key Words: Pancreas, Neoplasm fibrous tumor, Endoscopic ultrasound-guided fine-needle biopsy, Treatment, Case report

Core Tip: We need to be more vigilant for indeterminate pancreatic masses, and then computed tomography and histopathology can play a very important role in clinical diagnosis. Surgery is an effective treatment for solitary fibrous tumor of the pancreas; however, long-term follow-up is strongly recommended because of the possibility of malignant transformation of the tumor.



INTRODUCTION

A solitary fibrous tumor (SFT) is histologically characterized as a mesenchymal tumor, probably fibroblastic in origin, located primarily in the pleura; however, it can be found in any other extrapleural region[1-3]. Extrapleural areas include the liver, peritoneum, kidney, and salivary glands[4-7]. SFT of the pancreas is rare, with only about 30 cases reported to date[1-3,6-35]. SFT of the pancreas is usually asymptomatic, and most are detected by physical examination, computed tomography (CT), or ultrasound as pancreatic masses[6,30,32]. The final diagnosis depends on histopathology and immunohistochemistry[7,31].

Here, we report a case of SFT of the pancreas and present the radiological and pathological differential diagnosis.

CASE PRESENTATION
Chief complaints

A 54-year-old man was admitted to our hospital with a pancreatic space-occupying mass of one month’s duration, identified on a physical exam.

History of present illness

A 54-year-old man had been one month before a medical CT finding of pancreas space-occupying lesions, with no adverse reactions, patients for further treatment at our hospital.

History of past illness

The patient had no other significant medical history. History of hypertension, diabetes, coronary heart disease, and other chronic disease was denied.

Personal and family history

The patient had no significant personal or family history.

Physical examination

The patient had no discomfort after the physical examination.

Laboratory examinations

There was no abnormal carcinoembryonic antigen [< 0.5 ng/mL (normal 0-5 ng/mL)], carbohydrate antigen 199 3.9 U/mL (average 0-7 U/mL), alpha-fetoprotein 2.4 ng/mL (normal 0-8.8 ng/mL), carbohydrate antigen 125 12.5 (average 0-30.2 U/mL). Fasting glucose was 5. 19 mmol/L (normal 3.89-6. 11 mmol/L).

Imaging examinations

A review of an abdominal enhanced CT showed a tumor of about 3 cm × 2 cm in the tail of the pancreatic body, showing uneven enhancement after enhancement, consistent with a malignant tumor (Figure 1).

Figure 1
Figure 1 Abdominal computed tomography scan showing a 5.52 cm × 2.82 cm × 2 cm mass in the pancreas (orange arrows). A: No enhancement in the arterial region. B: Heterogeneous enhancement in the venous area.
FINAL DIAGNOSIS

A SFT of the pancreas.

TREATMENT

CT revealed a mass with mixed density and inadequate blood supply; these findings were inconsistent with a pancreatic tumor; therefore, we considered a pseudopapillary tumor and a non-functional pancreatic neuroendocrine tumor. We performed an ultrasound endoscopic tissue biopsy. The pathology and immunohistochemistry suggested SFT of the pancreas. After excluding contraindications to surgery and obtaining informed written consent, we performed a laparoscopic distal pancreatectomy with splenectomy. No significant adhesions were seen in the peripancreatic tissue. The pancreatic body was approximately 3 cm × 2 cm (Figure 2). Intraoperative frozen sections showed negative margins. Intraoperative blood loss was 100 mL and no blood transfusion was required.

Figure 2
Figure 2 Postoperative surgical specimen: Pancreatic tail and spleen (tumor cut open chart) (orange arrows).

The patient had no postoperative pancreatic fistula, abdominal infection, or bleeding. Ten days after surgery, he was discharged from the hospital after removing the drainage tube. One month after surgery, the patient returned to the hospital for examination. He did not complain of discomfort. The complete blood count, liver enzymes and renal function were normal.

Histopathological and immunohistochemical results of the postoperative specimen suggested an SFT of the pancreas of 3.0 cm × 2.5 cm × 1.0 cm, negative margins, no tumor involvement in the surrounding lymph nodes, and no tumor involvement in the spleen. Markers were as follows: Signal transducer and activator of transcription 6 (STAT6) (+), cluster of differentiation (CD) 34 (+), B cell CLL/lymphoma-2 (Bc1-2) (+), vimentin (+), CD99 (+), CD117 (-), Ki-67 (+40%), discovered on GIST-1 (+), transducin-like enhancer protein 1 (+), S- 100 (-), cytokeratin pan (pan) (-), somatostatin receptor 2 (-) (Figure 3).

Figure 3
Figure 3 Representative results of hematoxylin and eosin and immunohistochemical staining of surgical specimens of solitary fibrous tumor of the pancreas. A: Hematoxylin and Eosin staining (hematoxylin and Shuhong); B: Immunohistochemistry (original magnifcation of × 400) signal transducer and activator of transcription 6; C: CD34; D: CD99; E: Vimentin; F: Vimentin; G: Ki-67.
OUTCOME AND FOLLOW-UP

No specific treatment was given after the patient was discharged from the hospital, and he had no complaints for three months after the procedure. He returned for regular follow-up. No abnormalities were found on complete blood counts, blood glucose, tumor markers, or CT.

DISCUSSION

SFT is a mesenchymal tumor comprising less than 2% of soft tissue tumors[36]. About 65% of SFTs originate from the pleura[3]; however, they can also be found in extrapleural areas[6], with only 34 cases reported to date, including the present case (Tables 1 and 2). SFT of the pancreas is extremely rare. We searched PubMed and Google Scholar for pancreatic tumors and SFT and found 34 cases. Of these, 14 (41. 1%) were male, and 20 (58.9%) were female. The mean age was 54. 17 ± 15.4, and the median age was 54; 17 patients had lesions in the pancreatic tumor head [three (17.6%) male and 13 (76.4%) female]. Seventeen had tumors in the tail of the pancreatic body [ten (58.8%) male and seven (41.2%) female]. The mean tumor diameter was 5.2 cm ± 3.8 cm. Of the 34 patients, 12 presented with pain (12/34), 12 were discovered on physical examination (12/34), four presented with jaundice (4/34), one presented with an abdominal mass (1/34), and five were detected by other means (5/34) (Table 1).

Table 1 Characteristics of pancreatic solitary fibrous tumors.
No
Ref.
Age
Sex
Pancreatic site
Symptoms
Size (cm)
Pancreatic surgery
1Lüttges et al[1]50FBodyIncidental55DP
2Chatti et al[8]41MBodyAbdominal pain13DP
3Gardini et al[9]62FHeadAbdominal pain3PD
4Miyamoto et al[10]41FHeadAbdominal pain18 × 15Enucleation
5Kwon et al[11]54MBodyIncidental76 × 6MS
6Srinivasan et al[12]78FBodyBack pain, weight loss5DP
7Chetty et al[13]67FHeadIncidental26PD
8Ishiwatari et al[14]58FHeadIncidental3PD
9Sugawara et al[15]55FHeadIncidental6 × 4PD
10Santos et al[16]40MBodyIncidental3Enucleation
11Tasdemir et al[17]24FBodyEpigastric pain11Enucleation
12van der et al[18]67FHeadAbdominal pain28 × 16Enucleation
13Chen et al[19]49FHeadAbdominal pain13PD
14Hwang et al[20]53FHeadIncidental52 × 45 × 40PHR
15Baxter et al[21]58FHeadAbdominal pain35 × 3LPD
16Estrella et al[22]52FHeadJaundice15 × 10 × 10LPD
17Han et al[23]77FHeadJaundice15 × 14Biopsy
18Murakami et al[24]82MBodyHypokalemia hypertension, edema6DP
19Spasevska et al[3]47MHeadjaundice35 × 2 × 18LPD
20Paramythiotis et al[7]55MBodyAbdominal pain31 × 28DP
21D'Amico FE et al[25]52MBodyIncidental12DP
22Oana et al[26]73MHeadAbdominal discomfort65 × 55Enucleation
23Sheng et al[27]1MHeadJaundice20DP
24Geng et al[28]48MBodyHypoglycemia65 × 5DP
25Qian et al[29]46MBodyHypoglycemia70 × 61DP
26Rogers et al[30]37FHeadAbdominal pain23PD
27Taguchi et al[31]60MHeadPalpable mass9 × 7 × 7PD
28Jariwalla et al[32]64FBodyAbdominal pain19DP
29Marotti et al[33]75FBodyIncidental13Enucleation
30Addeo et al[6]59MBodyIncidental4DP
31Rodriguez et al[2]48FBodyAbdominal pain13 × 10 × 95TP
32Jones et al[34]61FBodyNA27DP
33Liu et al[35]54FHeadIncidental31 × 23LDPPHRt
34Present case54MBodyIncidental3 × 2DP
Table 2 Histological features and outcomes of pancreatic solitary fibrous tumors.
No
Ref.
Immunohistochemistry
Outcome
Follow-up
1Lüttges et al[1]CD34, CD99, Bcl-2, vimentinAlive20 months
2Chatti et al[8]CD34, CD99, Bcl-2, vimentinDeath3 d
3Gardini et al[9]CD34, CD99, Bcl-2, vimentin, SMAAlive16 months
4Miyamoto et al[10]CD34, Bcl-2Alive7 months
5Kwon et al[11]CD34, CD99, vimentinNANA
6Srinivasan et al[12]CD34, Bcl-2Alive7 months
7Chetty et al[13]CD34, CD99, Bcl-242 mo6 v
8Ishiwatari et al[14]CD34, Bcl-2Alive42 months
9Sugawara et al[15]CD34NANA
10Santos et al[16]CD34, betacateninNANA
11Tasdemir et al[17]CD34, Bcl-2, beta-catenin, vimentin, Ki67 < 2%Alive3 months
12van der et al[18]CD34, CD99, Bcl-2NANA
13Chen et al[19]CD34, Bcl-2, vimentin, CD68, muscle-specific actinAlive30 months
14Hwang et al[20]CD34, Bcl-2, muscle-specific actin, CD10, ER, PRAlive30 months
15Baxter et al[21]CD34, Bcl-2NANA
16Estrella et al[22]CD34, Bcl-2, keratin (rare), p16, p53Alive40 months
17Han et al[23]CD34, CD99No progression10 months
18Murakami et al[24]STAT6, CD34, Bcl-2, ACTH, POMC, NSEDeath4 months
19Spasevska et al[3]CD34, vimentin, CD99, Bcl-2, nuclear betacateninDeath1 wk
20Paramythiotis et al[7]CD34, CD99, Bcl-2 vimentin, S-100Alive40 months
21D'Amico FE et al[25]STAT6, CD34Alive24 months
22Oana et al[26]CD34, Bcl-2Alive36 months
23Sheng et al[27]CD34, vimentin, SMA, Ki67 < 3%Alive12 months
24Geng et al[28]STAT6, CD34, Bcl-2, CD31, PHH-3, D2-40, Ki67 > 10%Alive6 months
25Qian et al[29]STAT6, CD34, Bcl-2, Ki67 10%Alive10 months
26Rogers et al[30]STAT6, CD34, Bcl-2, CD99Alive4 months
27Taguchi et al[31]STAT6, CD34, Bcl-2, vimentin, cytokeratin AE1/AE3Alive12 months
28Jariwalla et al[32]STAT6, CD34NANA
29Marotti et al[33]STAT6, CD34Alive6 months
30Addeo et al[6]STAT6, CD34, Bcl-2, Ki67 7%NANA
31Rodriguez et al[2]STAT6Alive12 months
32Jones et al[34]STAT6, CD34Alive1 months
33Liu et al[35]CD34, STAT6, CD99Alive6 months
34Present case
TAT6, CD34, Bc1-2, Vimentin, CD99, Ki67 40%Alive3 months

Most SFTs of the pancreas are detected by physical examination; clinical signs and symptoms include abdominal pain and jaundice. Because these are not typical symptoms, it is challenging to differentiate SFT from other pancreatic diseases. Histopathology and immunohistochemistry are the gold standards for diagnosis. We recommend ultrasound endoscopic aspiration biopsy for space-occupying pancreatic lesions that cannot be diagnosed on imaging.

Our preoperative diagnosis relied on ultrasound endoscopic puncture biopsy in the present case. The preoperative and postoperative pathological histological examination and immunohistochemistry were consistent with SFT of the pancreas with no tumor involvement in the peripheral lymph nodes, no tumor involvement in the incised margin of the pancreas, and no tumor involvement in the spleen.

The immunohistochemical differential diagnosis of SFT of the pancreas should include spindle cell tumors such as gastrointestinal stromal tumor (GIST), smooth muscle sarcoma, nerve sheath tumor, fibrous mucinous sarcoma, perivascular epithelioid cell tumor, and vascular tumors[3,16,20,37]. The immunomarkers of SFT of the pancreas include STAT6, CD34, bc1-2, vimentin, and CD99[34]. These features help to distinguish SFT from other mesenchymal tumors[34,37]. SFT expresses CD34 and vimentin in 80%-90% of cases and CD99 and bcl-2 in 70%. SFTs are usually negative for c-kit (CD117), smooth muscle actin, junctional protein, S-100 protein, and cytokeratin (markers for GIST, smooth muscle sarcoma, nerve sheath tumor, and fibrous mucinous sarcoma, respectively) are negative[3]. NAB2-STAT6 fusion is a driver mutation in SFT, where transcriptional repressors of the cytokinesis pathway are converted into transcriptional activators[31,38,39]. STAT6 has a sensitivity of 98% and a specificity of 85% for SFT and is therefore considered the most characteristic SFT marker[40,41]. In our case, the tumor was positive for STAT6, while CD34, bc1-2, vimentin, and CD99 were positive.

In this case, CT showed no enhancement in the arterial phase and heterogeneous enhancement in the venous area. We believe that it should be distinguished from neuroendocrine tumors, which show enhanced CT from the arterial phase to the portal venous phase[13,37], which makes it difficult for us to distinguish the disease, so many scholars before us also misdiagnosed it before surgery[1,10,11,13,26]. At the same time, we believe that it should also be differentiated from pancreatic cancer and solid pseudopapillary tumors of the pancreas. The imaging features of this tumor have been described in detail in our previous work on pancreatic tumors[42].

Most SFTs are benign[43], and malignant SFTs account for 10%-15%[30,39,44,45]. The histopathological features of malignant SFT: (1) Hypercellularity; (2) more than four mitotic figures per ten high-power fields; (3) nuclear pleomorphism; (4) hemorrhage and necrosis; (5) tumor diameter ≥10 cm; and (6) positive margins[15,21,46]. Ki-67 can also differentiate benign from malignant tumors, with a cutoff value of 0%-5% (indeterminate in 5%-10%) for benign tumors and > 10% for malignant SFTs[40,47]. In our case, our patient had a Ki-67 proliferation index of 40%; therefore, the tumor was possibly malignant. Because SFT of the pancreas is rare, there are no uniform treatment criteria; nevertheless, complete resection is the treatment of choice for intra-abdominal SFTs[1,7,10-12,15], and post-surgical follow-up is critical because SFTs have a high recurrence rate. Due to the increasing number of reported cases of SFT, we believe there will be a complete system of treatment.

CONCLUSION

Because of the non-specific clinical symptoms and radiological features of SFT of the pancreas, the diagnosis is challenging with preoperative radiological and laboratory examinations alone. A definitive diagnosis relies on histopathology and immunohistochemistry. In cases where the tumor is found in the pancreas, and the diagnosis cannot be confirmed, it is recommended to obtain histopathology with ultrasound aspiration. As this presentation is rarely reported, there is a lack of uniform treatment criteria, and surgery is effective. However, the tumor may lead to potential recurrence or metastasis; therefore, long-term follow-up is recommended.

ACKNOWLEDGEMENTS

We thank the patient's family members for providing detailed treatment information and Dr. Kevin Li for revising the language.

Footnotes

Provenance and peer review: Unsolicited article; Externally peer reviewed.

Peer-review model: Single blind

Specialty type: Medicine, research and experimental

Country/Territory of origin: China

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P-Reviewer: Triantopoulou C, Greece S-Editor: Qu XL L-Editor: A P-Editor: Yu HG

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