Case Report

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J Innov Med Technol 2024; 2(2): 69-71

Published online November 30, 2024

https://doi.org/10.61940/jimt.240003

© Korean Innovative Medical Technology Society

Arterio-nodo-gastric fistula as an uncommon cause of upper gastrointestinal bleeding

Han Jo Jeon

Division of Gastroenterology and Hepatology, Department of Internal Medicine, Korea University College of Medicine, Seoul, Korea

Correspondence to : Han Jo Jeon
Division of Gastroenterology and Hepatology, Department of Internal Medicine, Korea University College of Medicine, 73 Goryeodae-ro, Seongbuk-gu, Seoul 02841, Korea
e-mail roadstar82@naver.com
https://orcid.org/0000-0003-2258-1216

Received: May 1, 2024; Revised: September 12, 2024; Accepted: September 12, 2024

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0), which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Gastrointestinal bleeding is an emergency condition requiring immediate intervention. It is essential to localize the bleeding focus in the gastrointestinal tract using endoscopy. Lymph node metastasis in esophageal cancer is common, but it is rare to fistulize the stomach wall. While there are various hemostatic methods available, embolization through the fistula can be highly beneficial. In cases of failed endoscopic hemostasis or hemodynamic instability, embolization could be useful but entails a risk of rebleeding. Therefore, the emergence of new technologies for hemostasis is anticipated.

Keywords Esophageal neoplasms; Gastrointestinal hemorrhage; Gastric fistula

Acute upper gastrointestinal bleeding (AUGIB) is a life-threatening emergent event. These events generally require hospitalization and intervention treatment. The mortality rate of AUGIB varies between 3% and 14%, with a commonly reported all-cause 30-day mortality of 9%–14%1. The predominant causes of AUGIB are nonvariceal, encompassing gastric and duodenal peptic ulcers, mucosal erosive disease of the esophagus, stomach, duodenum, Mallory–Weiss syndrome, Dieulafoy lesion and malignancy. Thus, the milestone of AUGIB is to promptly identify the bleeding source, administer hemostatic treatment, and stabilize the patient’s condition. However, challenging cases in endoscopic hemostasis may necessitate emergency surgery or angiographic embolization.

Esophageal cancer, reported in Globocan 2020, is the sixth leading cause of cancer-related death. Incidence of fistula formation, one of the most fatal complications, is observed in 5.3%–24.1% of patients with esophageal cancer undergoing chemoradiotherapy (CRT)2. Esophageal fistula, which mainly communicates with adjacent organs such as the respiratory or cardiovascular system, is a serious complication. Here, we introduce a case of AUGIB through the fistula formation in an unusual organ between the malignant metastatic regional lymph node and abutting the stomach where endoscopic hemostasis was challenging and failed due to unstable vital.

A 57-year-old male with esophageal squamous cell cancer (ESCC) and metastasis to a number of lymph nodes who had undergone 5-fluorouracil/cisplatin chemotherapy with combined radiotherapy (daily 180 cGy in 34 fractions, dose of 6,120 cGy) was referred to the emergency department for hematemesis, hematochezia, and dizziness. On arrival to the emergency department, the patient’s blood pressure was 71/53 mmHg, his heart rate was 121 beats/min, and his mental status drifted to drowsiness. Emergency angiography detected left gastric arterial bleeding (Fig. 1A). After glue embolization, computed tomography confirmed no residual contrast extravasation, but suspicious gastric perforation presenting as a wall discontinuity between the lesser curvature (LC) of the upper gastric body and the abutting lymph node was observed (Fig. 1B). Diagnostic esophagogastroduodenoscopy to confirm communication between the stomach and peritoneal cavity revealed extensive mucosal necrosis with an abnormal opening at the LC to the posterior wall of the upper body, suggestive of an arterio-nodo-gastric fistula (Fig. 1C).

Figure 1.(A) Ongoing extravasation of contrast (arrow) on the left gastric artery. (B) Abdominal computed tomography showing gastric wall defect (arrows) at the posterior wall of the stomach. (C) Endoscopic appearance of arterio-nodo-gastric fistula.

Up to 10%–29% of patients with unresectable T4 stage ESCC receiving CRT develop esophageal fistula3,4. However, esophageal fistula caused by metastatic lymph nodes is extremely rare5. In this case, esophageal cancer with a metastatic left gastric artery (LGA) lymph node eroded into the stomach, resulting in pathologic communication. A plausible explanation for the bleeding could be repeated CRT-induced inflammation causing tumoral feeding vessel (LGA) erosion at the metastatic lymph node and ongoing tumor necrosis and shrinkage. Akiyama et al.6 reported that the incidence of ESCC originating from the lower esophagus is 27.7%, with 61.5% of lymph nodes metastasizing to the superior gastric lymph nodes to involve LGA. LGA is the most common embolized artery among esophageal cancer patients with bleeding; although LGA embolization is common, bleeding from the arterio-nodo-gastric fistula that originates from an esophageal metastatic lymph node has not yet been documented.

European Society of Gastrointestinal Endoscopy recommends transcatheter angiographic embolization (TAE) for persistent bleeding refractory to endoscopic hemostasis, with surgery indicated if TAE is unavailable or unsuccessful7. Currently, a two-step approach involving endovascular access and subsequent surgical treatment is suggested for aortoesophageal fistula management8. Such an approach was adopted for this case as well. Among hemodynamically unstable esophageal cancer patients with upper GI bleeding, TAE primarily aimed at preventing the leakage of blood flow is recommended. Our endoscopic and radiologic findings of unusual bleeding conditions indicate that embolization should be considered the initial strategy in hemodynamically unstable patients with arterial bleeding from a gastric fistula.

Although previous studies have reported technical success rates as high as 90% to 100%9, TAE also has technical limitations in the cases of vascular complex anatomy, vascular spasm, and stenosis of the bleeding artery. Embolization, which chemically or physically occludes the feeding artery, preventing bleeding, can lead to rare but potential side effects in the gastrointestinal system such as perforation, necrosis of the gastrointestinal wall, fever, and digestive disturbances. The biggest drawback of TAE includes the potential for rebleeding, which is attributed to factors like incomplete embolization leading to rebleeding, rebleeding from adjacent vessels, and ischemia-related or coagulopathy-induced rebleeding after TAE despite immediate GI bleeding treatment. Therefore, it is mandatory to develop a new endoscopic treatment for a more selective hemostasis than TAE.

In recent, there has been a significant interest in the development of smart hemostatic materials tailored to target internal bleeding sites and enhance hemostasis. Several additional studies have investigated the clinical efficacy of topical hemostatic agents, especially Endoclot (EC, Micro-Tech Europe). However, data on the efficacy of EC are still limited and this is due to a lack of adequate randomization in clinical studies or because Forrest 1a bleeding has not yet been researched7. In addition to topical spray, super-thin films, commonly known as nanofilms for hemostasis, have garnered significant attention in the field of nanotechnology. Nanosheet is known to be flexible sufficient to conform to complex substrate topographies and adhere tightly to the substrate even in wet conditions, which appears promising technique compared to conventional hemostatic methods10. Currently, TAE is widely utilized as an alternative therapy to endoscopic treatment. However, introducing new technologies and treatments, such as nanofilms, and nanosheets, would be beneficial in future endoscopic treatments.

No potential conflict of interest relevant to this article was reported.

  1. Hreinsson JP, Kalaitzakis E, Gudmundsson S, Björnsson ES. Upper gastrointestinal bleeding: incidence, etiology and outcomes in a population-based setting. Scand J Gastroenterol 2013;48:439-447. https://doi.org/10.3109/00365521.2012.763174
    Pubmed KoreaMed CrossRef
  2. Pao TH, Chen YY, Chang WL, et al. Esophageal fistula after definitive concurrent chemotherapy and intensity modulated radiotherapy for esophageal squamous cell carcinoma. PLoS One 2021;16:e0251811. https://doi.org/10.1371/journal.pone.0251811
    Pubmed KoreaMed CrossRef
  3. Tsushima T, Mizusawa J, Sudo K, et al. ; Japan Esophageal Oncology Group of Japan Clinical Oncology Group (JCOG). Risk factors for esophageal fistula associated with chemoradiotherapy for locally advanced unresectable esophageal cancer: a supplementary analysis of jcog0303. Medicine (Baltimore) 2016;95:e3699. https://doi.org/10.1097/md.0000000000003699
    Pubmed KoreaMed CrossRef
  4. Kawakami T, Tsushima T, Omae K, et al. Risk factors for esophageal fistula in thoracic esophageal squamous cell carcinoma invading adjacent organs treated with definitive chemoradiotherapy: a monocentric case-control study. BMC Cancer 2018;18:573. https://doi.org/10.1186/s12885-018-4486-3
    Pubmed KoreaMed CrossRef
  5. Reed MF, Mathisen DJ. Tracheoesophageal fistula. Chest Surg Clin N Am 2003;13:271-289. https://doi.org/10.1016/s1052-3359(03)00030-9
    Pubmed CrossRef
  6. Akiyama H, Tsurumaru M, Kawamura T, Ono Y. Principles of surgical treatment for carcinoma of the esophagus: analysis of lymph node involvement. Ann Surg 1981;194:438-446. https://doi.org/10.1097/00000658-198110000-00007
    Pubmed KoreaMed CrossRef
  7. Gralnek IM, Stanley AJ, Morris AJ, et al. Endoscopic diagnosis and management of nonvariceal upper gastrointestinal hemorrhage (NVUGIH): European Society of Gastrointestinal Endoscopy (ESGE) Guideline - Update 2021. Endoscopy 2021;53:300-332. https://doi.org/10.1055/a-1369-5274
    Pubmed CrossRef
  8. Floré B, Heye S, Nafteux P, Maleux G. Single-stage endovascular treatment of an infected subclavian arterio-oesophagal fistula. Eur J Cardiothorac Surg 2014;45:570-572. https://doi.org/10.1093/ejcts/ezt324
    Pubmed CrossRef
  9. Lee S, Kim T, Han SC, Pak H, Jeon HH. Transcatheter arterial embolization for gastrointestinal bleeding: clinical outcomes and prognostic factors predicting mortality. Medicine (Baltimore) 2022;101:e29342. https://doi.org/10.1097/md.0000000000029342
    Pubmed KoreaMed CrossRef
  10. Xuan C, Hao L, Liu X, et al. Wet-adhesive, haemostatic and antimicrobial bilayered composite nanosheets for sealing and healing soft-tissue bleeding wounds. Biomaterials 2020;252:120018. https://doi.org/10.1016/j.biomaterials.2020.120018
    Pubmed CrossRef

Article

Case Report

J Innov Med Technol 2024; 2(2): 69-71

Published online November 30, 2024 https://doi.org/10.61940/jimt.240003

Copyright © Korean Innovative Medical Technology Society.

Arterio-nodo-gastric fistula as an uncommon cause of upper gastrointestinal bleeding

Han Jo Jeon

Division of Gastroenterology and Hepatology, Department of Internal Medicine, Korea University College of Medicine, Seoul, Korea

Correspondence to:Han Jo Jeon
Division of Gastroenterology and Hepatology, Department of Internal Medicine, Korea University College of Medicine, 73 Goryeodae-ro, Seongbuk-gu, Seoul 02841, Korea
e-mail roadstar82@naver.com
https://orcid.org/0000-0003-2258-1216

Received: May 1, 2024; Revised: September 12, 2024; Accepted: September 12, 2024

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0), which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Gastrointestinal bleeding is an emergency condition requiring immediate intervention. It is essential to localize the bleeding focus in the gastrointestinal tract using endoscopy. Lymph node metastasis in esophageal cancer is common, but it is rare to fistulize the stomach wall. While there are various hemostatic methods available, embolization through the fistula can be highly beneficial. In cases of failed endoscopic hemostasis or hemodynamic instability, embolization could be useful but entails a risk of rebleeding. Therefore, the emergence of new technologies for hemostasis is anticipated.

Keywords: Esophageal neoplasms, Gastrointestinal hemorrhage, Gastric fistula

Introduction

Acute upper gastrointestinal bleeding (AUGIB) is a life-threatening emergent event. These events generally require hospitalization and intervention treatment. The mortality rate of AUGIB varies between 3% and 14%, with a commonly reported all-cause 30-day mortality of 9%–14%1. The predominant causes of AUGIB are nonvariceal, encompassing gastric and duodenal peptic ulcers, mucosal erosive disease of the esophagus, stomach, duodenum, Mallory–Weiss syndrome, Dieulafoy lesion and malignancy. Thus, the milestone of AUGIB is to promptly identify the bleeding source, administer hemostatic treatment, and stabilize the patient’s condition. However, challenging cases in endoscopic hemostasis may necessitate emergency surgery or angiographic embolization.

Esophageal cancer, reported in Globocan 2020, is the sixth leading cause of cancer-related death. Incidence of fistula formation, one of the most fatal complications, is observed in 5.3%–24.1% of patients with esophageal cancer undergoing chemoradiotherapy (CRT)2. Esophageal fistula, which mainly communicates with adjacent organs such as the respiratory or cardiovascular system, is a serious complication. Here, we introduce a case of AUGIB through the fistula formation in an unusual organ between the malignant metastatic regional lymph node and abutting the stomach where endoscopic hemostasis was challenging and failed due to unstable vital.

Case Report

A 57-year-old male with esophageal squamous cell cancer (ESCC) and metastasis to a number of lymph nodes who had undergone 5-fluorouracil/cisplatin chemotherapy with combined radiotherapy (daily 180 cGy in 34 fractions, dose of 6,120 cGy) was referred to the emergency department for hematemesis, hematochezia, and dizziness. On arrival to the emergency department, the patient’s blood pressure was 71/53 mmHg, his heart rate was 121 beats/min, and his mental status drifted to drowsiness. Emergency angiography detected left gastric arterial bleeding (Fig. 1A). After glue embolization, computed tomography confirmed no residual contrast extravasation, but suspicious gastric perforation presenting as a wall discontinuity between the lesser curvature (LC) of the upper gastric body and the abutting lymph node was observed (Fig. 1B). Diagnostic esophagogastroduodenoscopy to confirm communication between the stomach and peritoneal cavity revealed extensive mucosal necrosis with an abnormal opening at the LC to the posterior wall of the upper body, suggestive of an arterio-nodo-gastric fistula (Fig. 1C).

Figure 1. (A) Ongoing extravasation of contrast (arrow) on the left gastric artery. (B) Abdominal computed tomography showing gastric wall defect (arrows) at the posterior wall of the stomach. (C) Endoscopic appearance of arterio-nodo-gastric fistula.

Discussion

Up to 10%–29% of patients with unresectable T4 stage ESCC receiving CRT develop esophageal fistula3,4. However, esophageal fistula caused by metastatic lymph nodes is extremely rare5. In this case, esophageal cancer with a metastatic left gastric artery (LGA) lymph node eroded into the stomach, resulting in pathologic communication. A plausible explanation for the bleeding could be repeated CRT-induced inflammation causing tumoral feeding vessel (LGA) erosion at the metastatic lymph node and ongoing tumor necrosis and shrinkage. Akiyama et al.6 reported that the incidence of ESCC originating from the lower esophagus is 27.7%, with 61.5% of lymph nodes metastasizing to the superior gastric lymph nodes to involve LGA. LGA is the most common embolized artery among esophageal cancer patients with bleeding; although LGA embolization is common, bleeding from the arterio-nodo-gastric fistula that originates from an esophageal metastatic lymph node has not yet been documented.

European Society of Gastrointestinal Endoscopy recommends transcatheter angiographic embolization (TAE) for persistent bleeding refractory to endoscopic hemostasis, with surgery indicated if TAE is unavailable or unsuccessful7. Currently, a two-step approach involving endovascular access and subsequent surgical treatment is suggested for aortoesophageal fistula management8. Such an approach was adopted for this case as well. Among hemodynamically unstable esophageal cancer patients with upper GI bleeding, TAE primarily aimed at preventing the leakage of blood flow is recommended. Our endoscopic and radiologic findings of unusual bleeding conditions indicate that embolization should be considered the initial strategy in hemodynamically unstable patients with arterial bleeding from a gastric fistula.

Although previous studies have reported technical success rates as high as 90% to 100%9, TAE also has technical limitations in the cases of vascular complex anatomy, vascular spasm, and stenosis of the bleeding artery. Embolization, which chemically or physically occludes the feeding artery, preventing bleeding, can lead to rare but potential side effects in the gastrointestinal system such as perforation, necrosis of the gastrointestinal wall, fever, and digestive disturbances. The biggest drawback of TAE includes the potential for rebleeding, which is attributed to factors like incomplete embolization leading to rebleeding, rebleeding from adjacent vessels, and ischemia-related or coagulopathy-induced rebleeding after TAE despite immediate GI bleeding treatment. Therefore, it is mandatory to develop a new endoscopic treatment for a more selective hemostasis than TAE.

In recent, there has been a significant interest in the development of smart hemostatic materials tailored to target internal bleeding sites and enhance hemostasis. Several additional studies have investigated the clinical efficacy of topical hemostatic agents, especially Endoclot (EC, Micro-Tech Europe). However, data on the efficacy of EC are still limited and this is due to a lack of adequate randomization in clinical studies or because Forrest 1a bleeding has not yet been researched7. In addition to topical spray, super-thin films, commonly known as nanofilms for hemostasis, have garnered significant attention in the field of nanotechnology. Nanosheet is known to be flexible sufficient to conform to complex substrate topographies and adhere tightly to the substrate even in wet conditions, which appears promising technique compared to conventional hemostatic methods10. Currently, TAE is widely utilized as an alternative therapy to endoscopic treatment. However, introducing new technologies and treatments, such as nanofilms, and nanosheets, would be beneficial in future endoscopic treatments.

Acknowledgments

None.

Conflict of Interest

No potential conflict of interest relevant to this article was reported.

Funding

None.

Fig 1.

Figure 1.(A) Ongoing extravasation of contrast (arrow) on the left gastric artery. (B) Abdominal computed tomography showing gastric wall defect (arrows) at the posterior wall of the stomach. (C) Endoscopic appearance of arterio-nodo-gastric fistula.
Journal of Innovative Medical Technology 2024; 2: 69-71https://doi.org/10.61940/jimt.240003

References

  1. Hreinsson JP, Kalaitzakis E, Gudmundsson S, Björnsson ES. Upper gastrointestinal bleeding: incidence, etiology and outcomes in a population-based setting. Scand J Gastroenterol 2013;48:439-447. https://doi.org/10.3109/00365521.2012.763174
    Pubmed KoreaMed CrossRef
  2. Pao TH, Chen YY, Chang WL, et al. Esophageal fistula after definitive concurrent chemotherapy and intensity modulated radiotherapy for esophageal squamous cell carcinoma. PLoS One 2021;16:e0251811. https://doi.org/10.1371/journal.pone.0251811
    Pubmed KoreaMed CrossRef
  3. Tsushima T, Mizusawa J, Sudo K, et al. ; Japan Esophageal Oncology Group of Japan Clinical Oncology Group (JCOG). Risk factors for esophageal fistula associated with chemoradiotherapy for locally advanced unresectable esophageal cancer: a supplementary analysis of jcog0303. Medicine (Baltimore) 2016;95:e3699. https://doi.org/10.1097/md.0000000000003699
    Pubmed KoreaMed CrossRef
  4. Kawakami T, Tsushima T, Omae K, et al. Risk factors for esophageal fistula in thoracic esophageal squamous cell carcinoma invading adjacent organs treated with definitive chemoradiotherapy: a monocentric case-control study. BMC Cancer 2018;18:573. https://doi.org/10.1186/s12885-018-4486-3
    Pubmed KoreaMed CrossRef
  5. Reed MF, Mathisen DJ. Tracheoesophageal fistula. Chest Surg Clin N Am 2003;13:271-289. https://doi.org/10.1016/s1052-3359(03)00030-9
    Pubmed CrossRef
  6. Akiyama H, Tsurumaru M, Kawamura T, Ono Y. Principles of surgical treatment for carcinoma of the esophagus: analysis of lymph node involvement. Ann Surg 1981;194:438-446. https://doi.org/10.1097/00000658-198110000-00007
    Pubmed KoreaMed CrossRef
  7. Gralnek IM, Stanley AJ, Morris AJ, et al. Endoscopic diagnosis and management of nonvariceal upper gastrointestinal hemorrhage (NVUGIH): European Society of Gastrointestinal Endoscopy (ESGE) Guideline - Update 2021. Endoscopy 2021;53:300-332. https://doi.org/10.1055/a-1369-5274
    Pubmed CrossRef
  8. Floré B, Heye S, Nafteux P, Maleux G. Single-stage endovascular treatment of an infected subclavian arterio-oesophagal fistula. Eur J Cardiothorac Surg 2014;45:570-572. https://doi.org/10.1093/ejcts/ezt324
    Pubmed CrossRef
  9. Lee S, Kim T, Han SC, Pak H, Jeon HH. Transcatheter arterial embolization for gastrointestinal bleeding: clinical outcomes and prognostic factors predicting mortality. Medicine (Baltimore) 2022;101:e29342. https://doi.org/10.1097/md.0000000000029342
    Pubmed KoreaMed CrossRef
  10. Xuan C, Hao L, Liu X, et al. Wet-adhesive, haemostatic and antimicrobial bilayered composite nanosheets for sealing and healing soft-tissue bleeding wounds. Biomaterials 2020;252:120018. https://doi.org/10.1016/j.biomaterials.2020.120018
    Pubmed CrossRef
Journal of Innovative Medical Technology
Nov 30, 2024 Vol.2 No.2, pp. 29~79

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