Case Report Open Access
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Aug 14, 2022; 28(30): 4211-4220
Published online Aug 14, 2022. doi: 10.3748/wjg.v28.i30.4211
Contrast-enhanced ultrasound of a traumatic neuroma of the extrahepatic bile duct: A case report and review of literature
Zhi-Qiang Yuan, Hua-Lin Yan, Jia-Wu Li, Yan Luo, Department of Medical Ultrasound, West China Hospital of Sichuan University, Chengdu 610041, Sichuan Province, China
ORCID number: Zhi-Qiang Yuan (0000-0002-3037-7576); Hua-Lin Yan (0000-0003-1338-1124); Jia-Wu Li (0000-0003-0844-5883); Yan Luo (0000-0003-2985-1768).
Author contributions: Yuan ZQ performed the literature review and wrote the manuscript; Yan HL and Li JW supported the data collection and manuscript revision; Luo Y supervised the writing and revision of the manuscript; all authors read and approved the final manuscript.
Supported by National Natural Science Foundation of China, No. 82071940.
Informed consent statement: Written informed consent for publication was obtained from the patient.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest to report.
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: Yan Luo, Doctor, Professor, Department of Medical Ultrasound, West China Hospital of Sichuan University, No. 37 Guo Xue Xiang, Chengdu 610041, Sichuan Province, China. yanluo@scu.edu.cn
Received: February 10, 2022
Peer-review started: February 10, 2022
First decision: April 5, 2022
Revised: April 17, 2022
Accepted: July 16, 2022
Article in press: July 16, 2022
Published online: August 14, 2022
Processing time: 180 Days and 23.4 Hours

Abstract
BACKGROUND

Traumatic neuromas result from nerve injury after trauma or surgery but rarely occur in the bile duct. However, it is challenging to diagnose traumatic neuromas correctly preoperatively. Although some previous reports have described the imaging features of traumatic neuroma in the bile duct, no features of traumatic neuromas in the bile duct have been identified by using contrast-enhanced ultrasound (CEUS) imaging before.

CASE SUMMARY

A 55-year-old male patient presented to our hospital with a 3-mo history of abdominal distension and anorexia and history of cholecystectomy 4 years ago. Grayscale ultrasound demonstrated mild to moderate intrahepatic bile duct dilatation. Meanwhile, a hyperechoic nodule was found in the upper extrahepatic bile duct. The lesion approximately 0.8 cm × 0.6 cm with a regular shape and clear margins. The nodule of the bile duct showed slight hyperenhancement in the arterial phase and isoenhancement in the venous phase on CEUS. Laboratory tests showed that alanine aminotransferase and aspartate aminotransferase were increased significantly, while the tumor marker carbohydrate antigen 19-9 was increased slightly. Then, hilar bile duct resection and end-to-end bile ductal anastomosis were performed. The histological examination revealed traumatic neuroma of the extrahepatic bile duct. The patient had an uneventful recovery after surgery.

CONCLUSION

The current report will help enhance the current knowledge regarding identifying traumatic neuromas by CEUS imaging and review the related literature.

Key Words: Traumatic neuroma; Bile duct; Contrast-enhanced ultrasound; Enhancement; Cholangiocarcinoma; Case report

Core Tip: A traumatic neuroma results from nerve injury after trauma or surgery but rarely occurs in the bile duct. Herein, we present some of the sonographic features of ultrasound and contrast-enhanced ultrasound in a case of a traumatic neuroma. We report this unusual case and review the related literature to improve the diagnosis and differential diagnosis of a traumatic neuroma of the bile duct and related imaging findings.



INTRODUCTION

A traumatic neuroma is a chronic reparative proliferative response of the nerve after trauma or surgery. It is composed of disorganized nerve fiber bundles with fibrous stroma, Schwann cells, perineural cells, axons, and endoneural fibroblasts[1]. The common sites of traumatic neuromas are the necks and extremities[2,3]. Although some studies have described traumatic neuromas in the bile duct, cases of sonographic features of contrast-enhanced ultrasound (CEUS) have not been published before. The clinical manifestation and imaging examination of a traumatic neuroma of the bile duct are not specific, which makes it challenging be accurately diagnosed preoperatively. Herein, we report a traumatic neuroma of the extrahepatic bile duct with detailed ultrasonographic imaging features. We also reviewed the literature on the imaging findings for traumatic neuromas.

CASE PRESENTATION
Chief complaints

A 55-year-old man was admitted to our hospital with unexplained abdominal distension and anorexia 3 mo ago.

History of present illness

The patient suffered from unexplained abdominal distension and anorexia for 3 mo. The patient developed darkened urine 2 mo ago. He experienced a weight loss of 5 kg over the course of the disease. He underwent contrast-enhanced computed tomography (CECT) examination at a local hospital, and a lesion was found in the extrahepatic bile duct, which was believed to be a tumor.

History of past illness

The patient underwent cholecystectomy for gallbladder stones with an uneventful postoperative recovery 4 years ago. He had a 10-year history of hypertension.

Personal and family history

There was no other personal or family history of acute or chronic disease.

Physical examination

The patient showed no tenderness, rebound tenderness or muscle tension on abdominal palpation.

Laboratory examinations

The liver function tests demonstrated increased levels of alanine aminotransferase (185 IU/L, normal range: < 50 IU/L), aspartate aminotransferase (148 IU/L, normal range: < 40 IU/L) and total bilirubin (37.0 μmol/L, normal range: 5 µmol/L to 28 µmol/L). Tumor markers included carbohydrate antigen 19-9 (98.6 U/mL, normal range: < 22 U/mL), carcinoembryonic antigen (0.97 ng/mL, normal range: < 5 ng/mL), and alpha-fetoprotein (4.67 ng/mL, normal range: < 7 ng/mL).

Imaging examinations

The patient underwent an abdominal ultrasound (US) examination by a Resona7 US system (Mindray Medical International, Shenzhen, Guangdong Province, China) equipped with an SC6-1U (1-6 MHz) transducer. The US revealed mild to moderate dilatation of the intrahepatic bile duct, and the diameter of the upper extrahepatic bile duct was 1.2 cm (Figure 1A). A hyperechoic nodule sized 0.8 cm × 0.6 cm was found in the upper extrahepatic bile duct with an almost regular shape and slightly clear margins (Figure 1B). The patient underwent CEUS with the patient’s consent for further diagnosis. A 2.4-mL US contrast agent SonoVue (Bracco, Milan, Italy) suspension was injected through the left cubital vein followed by a flush with 5 mL saline. In the arterial phase, the nodule showed slight heterogeneous hyperenhancement without rim-like enhancement (Figure 1C). The nodule appeared heterogeneous isoenhancement in the venous phase (Figure 1D). Additional CECT in our hospital showed a hypoenhancement nodule approximately 1.3 cm × 1.0 cm in size in the upper extrahepatic bile duct (Figure 2).

Figure 1
Figure 1 Ultrasound images of the patient. A and B: The ultrasound (US) showed mild to moderate intrahepatic bile duct dilatation (orange arrow) and a hyperechoic nodule sized 0.8 cm × 0.6 cm (orange arrow) in the extrahepatic bile duct; C and D: In the arterial phase, contrast-enhanced US (CEUS) showed slight hyperenhancement (orange arrow); in the venous phase, CEUS showed isoenhancement (orange arrow).
Figure 2
Figure 2 Contrast-enhanced computed tomography images of the patient. Contrast-enhanced computed tomography showed a hypoenhancement nodule in the upper extrahepatic bile duct (orange arrow).
FINAL DIAGNOSIS

Based on the incidence of bile duct diseases, imaging findings and laboratory tests, the patient's clinical diagnosis was hilar cholangiocarcinoma. However, postoperative pathology of the common bile duct lesion showed a neoplastic proliferation of submucosal nerve tissue and fibrous tissue (Figure 3A), and an immunohistochemistry marker was positive for S-100 (Figure 3B). The above pathological findings indicated that the lesion in the bile duct was a traumatic neuroma.

Figure 3
Figure 3 Postoperative histopathological images of the patient. A: Hematoxylin and eosin staining showed proliferation of submucosal nerve tissue (magnification, × 100); B: Immunohistochemical staining displayed S100(+) (magnification, × 100).
TREATMENT

During the surgery, intraoperative frozen pathology showed no tumor cells within the bile duct lesion. Therefore, hilar bile duct resection and end-to-end bile ductal anastomosis (EE) were performed. The patient recovered uneventfully after surgery.

OUTCOME AND FOLLOW-UP

There was no obvious abnormality on CECT for half a year after the operation.

DISCUSSION

Extrahepatic bile duct masses are commonly malignant tumors, while benign tumors account for only 6%[4-7]. Consequently, the possibility that extrahepatic bile duct lesions are traumatic neuromas is easily overlooked. It has been reported that most traumatic neuromas of the biliary tract arise in the cystic duct stump after cholecystectomy[8]. If a nerve is transected and its continuity cannot be reestablished, a traumatic neuroma may develop[9].

We reviewed the literature from 2000 to 2021 and found 18 publications regarding the imaging features of traumatic neuromas in the bile ducts[2,10-26]. The clinical findings and imaging features of these 18 reported cases are summarized in Table 1. Finally, 22 patients were included in the literature review for further analysis. The age of patients ranged from 17 to 81 years of age, and there was a significant male predominance, with 15 males (68.2%), 2 females, and 5 patients of unreported sex. Most cases were secondary to cholecystectomy, but a few were secondary to liver transplantation, hepatectomy and hilar cholangiocarcinoma. The major symptoms found in these patients were jaundice, abdominal pain, and weight loss, while some patients had no apparent symptoms.

Table 1 Traumatic neuroma of the bile duct reported in the literature between 2000 and 2020.
Ref.
Age
Sex
Symptoms
Location
Imaging findings
Preoperative diagnosis
Treatment
Shimura et al[10]70FAbdominal discomfortExtrahepatic bile ductUS: Hypoechoic tumor, bile duct slightly dilatedDid not indicate bile duct carcinomaBile duct excision and a Roux-en-Y hepaticojejunostomy
CT: Round, hyperdense, distinct margin tumor
Angiography: No encasement of the surrounding major vessels
Endoscopic retrograde cholangiography: A protuberant nodule
Intraductal ultrasonography: A smooth hypoechoic tumor
Watanabe et al[11]48MJaundiceExtrahepatic bile ductCholangiogram via the percutaneous transhepatic biliary drainage tube: The extrahepatic bile duct severely stenoticNDBile duct excision and a Roux-en-Y hepaticojejunostomy
Iannelli et al[12]81MJaundiceCommon bile ductUS: Dilatation of the intrahepatic bile ductsNDBile duct excision and a Roux-en-Y hepaticojejunostomy
MRCP: A focal stricture
Ueno et al[2]60MJaundiceMid-common bile ductUS: Dilatation of the bile ducts, a mildly echogenic massCould not confirm benign or malignant natureBile duct excision and a hepato-jejunal anastomosis
CT: Dilatation of the bile ducts, a markedly enhanced nodule
MRI: Dilatation of the bile ducts. Homogeneous enhanced nodule with an iso-intense to the aorta, both in the arterial and portal phase
Percutaneous transhepatic cholangiography: Dilatation of the bile ducts and a smooth stricture
Choi et al[13]46MIncreased liver enzymesRight hepatic ductCT: A mass approximately 2 cmA bile duct cancer could not be excludedRight hemihepatectomy
MRI: A mass approximately 2 cm
Kim et al[14]76MNDMid-bile ductCT: A small enhancing noduleNDSegmental resection with a Roux-en-Y hepaticojejunostomy
MRC: Eccentric wall thickening of the bile duct consistent with a neoplasm
Cheng et al[15]33FJaundice and weight lossRemnant choledochal cystMRI: A massCholangiocarcinomaExcision of the remnant choledochal cyst and a new hepaticojejunostomy
Cheng et al[16]56MJaundice, abdominal pain and weight lossDistal extrahepatic bile ductUS: Dilatation of bile ductAmpullary or periampullary carcinomaPancreaticoduodenectomy
MRI: Dilatation of bile duct, a filling-defect in the distal bile duct and a thickened biliary wall around the ampulla of Vater
Cheng et al[17]68MProgressive jaundice and abdominal painBifurcation of the left and right hepatic ductMRI: A mass with enhancement, a stricture of the hilar bile duct, dilatation of bile ductsCholangiocarcinomaExcision of the mass and a new Roux-en-Y hepaticojejunostomy
Navez et al[18]NDNDJaundice (3 patients) or liver function test alteration (1 patient), a retro-obstructive choleperitoneum on the downstream biliary stenosis (1 patient)Anastomotic biliary strictureCT: Anastomotic biliary stricture (4 patients)NDTraumatic biliary neuromas resection combined with hepaticojejunostomy (1 patient); traumatic biliary neuromas resection and duct-to-duct biliary reconstruction protected by a T-tube (4 patients)
MRI: A markedly homogeneous high intensity nodule enhanced on portal-phase (1 patient), anastomotic biliary stricture (4 patients)
Terzi et al[19]17FPersistent elevated transaminase and bilirubin levelsAnastomotic biliaryPercutaneous transhepatic cholangiography: A biliary stricture at the anastomosisNDResection of the bile duct stricture and a Roux-en-Y hepaticojejunostomy
Toyonaga et al[20]76FA bile duct noduleProximal common bile ductCT: An 8 mm, smooth, and uniformly enhanced noduleSubmucosal tumorBiopsy, observation for 1 year, no changes to the nodule
Contrast enhanced endoscopic ultrasonography: A clear boundary and a low echoic nodule, uniformly enhanced at early
Cholangioscopy: A smooth elevated lesion, covered with normal mucosa
Yang et al[21]65MJaundiceRight bile ductMRI: A 1.0 cm × 1.5 cm massCholangiocarcinomaResection of the mass and Roux-en-Y hepaticojejunostomy.
Hirohata et al[22]60FNo chief complaintJunction of the cystic ductUS: A 6 mm round tumor, surrounding lymph nodes were not swollenCholangiocarcinomaSurgery
MRI: A slightly high signal on T2 and the periphery remnant cystic duct of the tumor presented as a high-intensity lesion on T2
EUS: A residual cystic duct tumor with enhancement
ERCP: Not invade the common bile duct
Yasuda et al[23]76MNDStump of the dilatated cystic ductEUS: A hypoechoic oval mass with a hyperechoic rim on the surface, 14 mm in diameter, hypervascularityAmputation neuromaBiopsy, observation
Cholangiogram: A hemispherical defect
Cholangioscopy: A hemispherical mass covered with thin normal cystic duct epithelium
Lalchandani et al[24]41MEpigastric pain, weight loss, tea-colored urineCommon hepatic ductUS: Dilation of the bile ductsAcute cholangitisFirst: Biliary stent Finally: Bile duct resection and hepaticojejunostomy
ERCP: A 3-4 cm stricture
Kim et al[25]72MA duodenal subepithelial tumor during a medical checkupNear the duodenal wall and the cystic duct stumpCT: A 1.4 cm massDuodenal subepithelial tumorResection of the mass and duodenal wall, en-block resection of the mass and cystic duct origin
EUS: An 18 mm hypoechoic mass
Nechi et al[26]76MJaundiceThe transition zone between the common hepatic duct and the main bile ductUS: Dilation of the bile ducts, a 5 mm hypoechoic noduleCould not confirm benign or malignant natureResection of the main bile duct with a choledocho-duodenal anastomosis
MRI: Dilation of the common hepatic duct

Unfortunately, no specific imaging features for traumatic neuromas of the bile duct have been found at present. Although some imaging modalities, such as US, computed tomography (CT), and nuclear magnetic resonance imaging (MRI), are valuable to some extent, it remains a challenge to diagnose traumatic bile duct neuromas preoperatively[17]. Imaging findings in these 22 patients varied from nodules or masses to localized bile duct stenosis with dilatation of the upper bile duct. It has been reported in the literature that the US imaging findings of extraabdominal nerve tumors and traumatic neuromas are generally hypoechoic masses, larger than the nerve trunk and continuous with the nerve[27]. However, the nerve injury related to cholecystectomy may be too small, so we could not find that the nerve is connected to traumatic neuroma of the bile duct. US was performed in 5 of the 22 patients, 2 of whom showed hypoechoic nodules, and the remaining 3 patients showed stenosis and dilatation of the bile ducts. However, our patient's US sonogram showed a hyperechoic nodule, indicating that the echogenicity of the nodule of traumatic neuroma was variable.

CECT was performed in 2 of the 18 cases, and an enhancing nodule was seen, which was consistent with the CECT findings of our patient. Traumatic neuromas also show enhancement on MRI when a contrast agent is used[28], which may be related to a damaged peripheral nerve blood barrier that occurred during a prior insult to the nerve[29-32]. One of these 18 cases described the enhancement pattern of traumatic neuroma on MRI in detail, which showed a marked homogeneously enhanced nodule that was iso-intense to the aorta in the atrial phase and a homogeneously enhanced nodule that was iso-intense to the aorta in the portal phase. There have been a few reports of other imaging techniques for diagnosing traumatic neuromas, such as magnetic resonance cholangiopancreatography, endoscopic US, contrast-enhanced harmonic endoscopic ultrasonography, intraductal ultrasonography and percutaneous transhepatic cholangiography. None of these imaging methods revealed specific features for traumatic neuromas.

It is challenging to distinguish bile duct traumatic neuroma from other lesions before surgery, so it is often misdiagnosed. The diagnosis of bile duct traumatic neuroma was correctly diagnosed in 1 of the 18 cases examined and confirmed by biopsy. The remaining cases were not correctly diagnosed, and it was difficult to distinguish between benign and malignant lesions in most cases. Therefore, surgery would be performed on a large proportion of patients. Once the patient underwent surgery, an intraoperative frozen section examination helped to confirm that the lesion was benign and extensive surgical resection of the traumatic neuroma was avoided[2,3]. The primary treatment reported in the literature consists of bile duct excision and hepaticojejunostomy (HJ). Although HJ is frequently recommended for reconstruction, the indications, surgical options and suture selection are also controversial. Some investigators also recommend EE because it is more physiological and can maintain physiological balance[33]. It is possible to achieve excellent long-term results and high quality of life using both HJ and EE when it is feasible for the proximal and distal ductal ends to permit EE[34]. Therefore, the choice of the optimum method is strictly correlated with the morphological nature of the lesion, which is different from one stage to the other, depending upon the moment of detection, and therefore have different surgical implications[35]. The surgeon found that the anastomosed edges blood supply was good and that there was no tension of the anastomosed edges in this patient. Therefore, according to the actual conditions of patients, as well as to maintain physiological balance, our hospital professor implemented EE for this patient.

In this patient, the symptoms of anorexia, weight loss and jaundice mimicked those often caused by malignant tumors of extrahepatic bile ducts. CEUS and CECT showed enhancement of the nodule. Based on the incidence of bile duct diseases and imaging findings, the surgeons misdiagnosed it as cholangiocarcinoma. Periductal infiltrative cholangiocarcinomas account for the majority of extrahepatic cholangiocarcinomas[36]. Extrahepatic cholangiocarcinomas may show hyperenhancement, isoenhancement, or hypoenhancement in the early phase of CEUS, and most of them show hypoenhancement in the late phase[37]. If we find a nodule in the bile duct, we should rule out the diagnosis of cholangiocarcinoma when the nodule does not show hypoenhancement in the late phase of CEUS. However, when traumatic neuroma presents as localized bile duct stenosis, it is relatively difficult to distinguish it from malignant lesions. When a patient has a history of biliary system surgery and the tumor markers are not significantly elevated, suspicion of traumatic neuroma increases. If conditions permit, patients can be protected from unnecessary surgeries by confirming the diagnosis with a biopsy. CEUS is beneficial for differentiating cholangiocarcinoma from traumatic neuromas, but more cases are needed to summarize the sonographic features of this disease. Recognizing of traumatic neuromas may aid in preoperative work up, planning, and patient counseling[24].

CONCLUSION

It is difficult to correctly diagnose traumatic neuroma of the bile duct before surgery. We should rule out malignant differential diagnoses, such as cholangiocarcinoma preoperatively, to avoid unnecessary surgery. The enhancement mode of CEUS may provide information to distinguish traumatic neuromas from malignant lesions. We need to combine the history of biliary tract surgery, clinical findings, imaging findings and laboratory tests to diagnose this disease.

Footnotes

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

Peer-review model: Single blind

Corresponding Author's Membership in Professional Societies: Society of Ultrasound, Abdomen Ultrasound Subcommittee, Chinese Medical Doctor Association, No. 199174.

Specialty type: Gastroenterology and hepatology

Country/Territory of origin: China

Peer-review report’s scientific quality classification

Grade A (Excellent): 0

Grade B (Very good): B, B

Grade C (Good): 0

Grade D (Fair): 0

Grade E (Poor): 0

P-Reviewer: Cochior D, Romania; Koganti S, United States S-Editor: Yan JP L-Editor: A P-Editor: Yan JP

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