Massive gastrointestinal hemorrhage and iodine-125 seed migration due to hepatogastric fistula: a case report and literature review

Introduction

Hepatocellular carcinoma (HCC) is one of the most deadly cancers worldwide, ranking third among cancer-related deaths worldwide (1,2). Hepatogastric fistula (HGF), typically associated with hepatic abscess, is rarely reported as a complication in patients with HCC. For patients with HCC, the utilization of permanent low-dose iodine-125 (¹²⁵I) seed implantation is considered an effective modality. This approach delivers a high radiation dose to the tumor, minimizing hepatic damage and gastroduodenal complications, which are commonly observed in external radiotherapy (3). This method has demonstrated favorable therapeutic outcomes (4).

While the reported incidence of seed migration in HCC is 20.3% with the chest, including the lung and heart, being the most frequent site, there have been no reported cases of ¹²⁵I seed migration to the stomach through HGF.

Herein, we detail a case wherein the caudate lobe communicated with the lesser curvature of the stomach, forming an HGF. Subsequently, intermittent massive gastrointestinal hemorrhage occurred, and seed implantation in the caudate lobe migrated to the stomach through the HGF. Additionally, we provide a literature review of gastrointestinal hemorrhage due to HGF. We present this article in accordance with the CARE reporting checklist (available at https://tcr.amegroups.com/article/view/10.21037/tcr-24-1052/rc).

Case presentation

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompany images. A copy of the written consent is available for review by the editorial office of this journal.

A 32-year-old man with chronic B viral hepatitis underwent emergency surgery at another hospital abroad to control bleeding due to HCC rupture. One month postoperatively, an active residual tumor (44 mm × 33 mm) was found in the caudate lobe of the liver. Given the tumor’s proximity to the first hepatic portis and the main blood vessels, the patient was admitted for percutaneous microwave coagulation therapy (PMCT) (SurBlate, Vison Medical USA, Santa Clara, CA, USA) (number of antennas: 2, power: 50 watts, duration: 3 minutes) and ¹²⁵I seed implantation sequentially. No postoperative discomfort was observed. Enhanced magnetic resonance imaging (MRI) performed at the 2-month follow-up revealed complete tumor necrosis without abnormal thickening of the gastric wall. Subsequent intrahepatic HCC distant recurrences were successfully managed using PMCT and systemic treatments (molecular targeted drug and checkpoint inhibitor).

Twenty months after the initial seed implantation in the caudate lobe, the patient was referred again owing to intrahepatic tumor recurrence in the right lobe and underwent repeat PMCT (SurBlate, Vison Medical USA) (number of antennas: 1, power: 60 watts, duration: 4 minutes and 20 seconds) and ¹²⁵I seed implantation. Two days after the second ¹²⁵I seed implantation, the patient presented with severe upper gastrointestinal bleeding and epigastric pain. On endoscopic examination, the esophageal varices were excluded, and an oozing fistulous opening was observed in the lesser curvature of the stomach (Figure 1). Bleeding temporarily ceased with conservative treatment, but recurred after. Arteriography revealed no abnormalities. Enhanced MRI showed that the size of the lesion in the caudate lobe had obviously decreased compared to previous findings (Figure 2). The caudate lobe adhered to the stomach, and there was no clear boundary. Abdominal radiography revealed a decreased number of seeds in the caudate lobe (Figure 3). The above analysis indicated that emergency surgery and multiple interventional procedures resulted in adhesion of the caudate lobe of the liver to the lesser curvature of the stomach, causing necrosis in the caudate lobe to form an abscess. The liver abscess ruptured into the gastric lumen and formed the HGF. Finally, intermittent massive gastrointestinal hemorrhage occurred, and the seeds in the caudate lobe migrated to the stomach through the HGF.

Figure 1 Endoscopy discovered a fistulous opening [black arrow in (A)], which was oozing white pus [green arrow in (B)], in lesser curvature of the stomach.

Figure 2 Magnetic resonance imaging of the liver showed the size of lesion [red arrows in (A,B)] in caudate lobe decreased significantly.

Figure 3 Plain film of the abdomen revealed the number of seeds in the caudate lobe [red arrow in (A)] decreased; seeds in right lobe [green arrow in (B)] were implanted two days before the first bleeding.

After multidisciplinary consultation, endoscopy, surgery, or conservative treatment was suggested to repair the internal fistula. Seventeen days after the second hemorrhage, massive bleeding recurred, and an emergency operation was performed because of hemorrhagic shock.

During surgery, fistulization of the caudate lobe into the adjacent lesser curvature of the gastric antrum was observed. In addition, a ¹²⁵I seed was detected in the fistula. Gastric perforation repair was performed, and the upper gastrointestinal hemorrhage was permanently resolved.

On the second postoperative day, the patient was comatose and was diagnosed with cerebral infarction. The patient’s condition improved after neurosurgery, and he was safely discharged.

Discussion

¹²⁵I irradiation has a short penetration radius of 1.7 cm, reducing hepatic and gastroduodenal damage, which commonly occurs in external radiotherapy. PMCT is a thermal ablation modality that has clear advantages in terms of minimal trauma, excellent curative effects, and rapid recovery in recent years. The review identified 29 studies investigating microwave ablation (MWA) and reported technical success (range, 91.6–100%) and ablation success (range, 73.1–100%), as well as major complications (range, 0–9.1%) (5). Besides, the temperature had not been monitored during PMCT as the previous studies reported. In this case, PMCT was used to eradicate cancer cells inside the tumor, which reduced ¹²⁵I seeds consumption. ¹²⁵I seeds were implanted at sites around the tumor and near large vessels where PMCT could not reach.

In most HCC cases, the seeds migrate through the venous system to the lungs and heart. In this case, the seeds migrated through the HGF to the stomach and were discharged through bleeding.

HGF, a very rare internal fistula, develops as a complication of rupture of a liver abscess or tumor into the stomach (6). In this case, the carcinoma in the caudate lobe was eradicated by PMCT and seed implantation, which was confirmed by no residual tumor on 1-month follow-up via MRI; however, several months later, the necrotic tissue in the caudate lobe was infected and formed an abscess, which subsequently induced HGF.

Massive upper gastrointestinal bleeding and seed migration caused by HGF in HCC are yet to be described. To date, few case reports on gastrointestinal hemorrhage following HGF have been published. The literature review is presented in Table 1.

Conclusions

In conclusion, clinicians should be aware of rare cases of upper gastrointestinal bleeding and seed migration in patients with HCC. According to previous reports, medical, endoscopic, interventional, and surgical methods for HGF have favorable outcomes. Upper gastrointestinal hemorrhage and an unaccountable deep fistula at an unusual site should urge doctors to investigate further. A detailed clinical medical history and high vigilance are key to diagnosing and treating this rare complication.

Acknowledgments

Funding: This work was supported by Take-off Program (No. TF2024TJYQ02).

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://tcr.amegroups.com/article/view/10.21037/tcr-24-1052/rc

Peer Review File: Available at https://tcr.amegroups.com/article/view/10.21037/tcr-24-1052/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://tcr.amegroups.com/article/view/10.21037/tcr-24-1052/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompany images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

References

1. Bray F, Laversanne M, Sung H, et al. Global cancer statistics 2022: GLOBOCAN estimates of incidence and mortality worldwide for 36 cancers in 185 countries. CA Cancer J Clin 2024;74:229-63. [Crossref] [PubMed]
2. Yu X, Feng B, Wu J, et al. A novel anoikis-related gene signature can predict the prognosis of hepatocarcinoma patients. Transl Cancer Res 2024;13:1834-47. [Crossref] [PubMed]
3. Zhao XH, Li HL, Guo CY, et al. Downstaging and Conversation Strategy for Advanced Hepatocellular Carcinoma with Portal Vein Branch Tumor Thrombus: TACE, (125)I Seed Implantation, and RFA for Hepatocellular Carcinoma with Portal Vein Branch Tumor Thrombus. J Hepatocell Carcinoma 2023;10:231-40. [Crossref] [PubMed]
4. Zhang L, Wang J, Li Y, et al. Implanting Iodine-125 Seed Strand Inside the Portal Vein Stent: An Improved Approach to Endovascular Brachytherapy for Treatment of Patients with Hepatocellular Carcinoma and Main Portal Vein Tumor Thrombus. J Hepatocell Carcinoma 2023;10:2187-96. [Crossref] [PubMed]
5. Barrow B, Martin Ii RCG. Microwave ablation for hepatic malignancies: a systematic review of the technology and differences in devices. Surg Endosc 2023;37:817-34. [Crossref] [PubMed]
6. Malakar S, Mathur A, Elhence A, et al. Hepatogastric fistula: A rare complication of liver abscess. Indian J Gastroenterol 2024;43:854-6. [Crossref] [PubMed]
7. Püspök A, Kiener HP, Susani M, et al. Gastric perforation of a left lobe amoebic liver abscess. Eur J Gastroenterol Hepatol 2000;12:961-2. [Crossref] [PubMed]
8. Kim ES, Lee SY, Yeom JO, et al. Education and Imaging. Hepatobiliary and pancreatic: Liver abscess complicated by a hepatogastric fistula. J Gastroenterol Hepatol 2013;28:206. [Crossref] [PubMed]
9. Sayana H, Yousef O, Clarkston WK. Massive upper gastrointestinal hemorrhage due to invasive hepatocellular carcinoma and hepato-gastric fistula. World J Gastroenterol 2013;19:7472-5. [Crossref] [PubMed]
10. Agarwal J, Hasan A, Mehrotra M. Hepatobiliary and Pancreatic: Hepatogastric fistula. J Gastroenterol Hepatol 2017;32:1284. [Crossref] [PubMed]
11. Bale A, Shetty S, Shetty A, et al. Upper Gastrointestinal Bleed Due to Invasive Hepatocellular Carcinoma and Hepato-Gastric Fistula. J Clin Exp Hepatol 2018;8:104-5. [Crossref] [PubMed]
12. Liu CA, Chiu NC, Chiou YY. Massive hematemesis after radiofrequency ablation of metastatic liver tumor with successful hemostasis achieved through transarterial embolization. Clin Imaging 2018;51:192-5. [Crossref] [PubMed]
13. Dhali A, Sarkar A, Ray S, et al. Hepatic artery pseudo-aneurysm rupturing into hepato-gastric fistula, a rare cause of massive upper gastrointestinal hemorrhage: Case report. Radiol Case Rep 2021;17:133-6. [Crossref] [PubMed]