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Korean J Gastroenterol  <  Volume 83(3); 2024 <  Articles

Korean J Gastroenterol 2024; 83(3): 119-122  https://doi.org/10.4166/kjg.2024.014
Life-threatening Gastrointestinal Bleeding from a Dieulafoy’s Lesion in the Duodenum: A Case Report
Jun Hyuk Son
Department of Internal Medicine, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
Correspondence to: Jun Hyuk Son, Department of Internal Medicine, Inje University College of Medicine, Division of Gastroenterology and Hepatology, Inje University Ilsan Paik Hospital, 170 Juhwa-ro, Ilsanseo-gu, Goyang 10380, Korea. Tel: +82-31-910-7200, Fax: +82-31-910-7219, E-mail: jun1981@paik.ac.kr, ORCID: https://orcid.org/0000-0003-3477-6985
Received: January 29, 2024; Revised: February 8, 2024; Accepted: February 8, 2024; Published online: March 25, 2024.
© The Korean Journal of Gastroenterology. All rights reserved.

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
Dieulafoy’s lesion is a rare cause of gastrointestinal bleeding, accounting for approximately 1–2% of all cases of gastrointestinal bleeding. Dieulafoy’s lesion usually occurs in the lesser curvature of the stomach within six centimeters of the gastroesophageal junction. On the other hand, extragastric Dieulafoy’s lesions are uncommon. Diagnosing an extragastric Dieulafoy’s lesion by endoscopy can be challenging because of its small size and obscure location. The key elements for an accurate diagnosis include heightened awareness and a careful early endoscopic evaluation following a bleeding episode. Various endoscopic hemostatic techniques can be used for treatment. This paper presents a case of successful hemostasis using argon plasma coagulation for a life-threatening duodenal Dieulafoy’s lesion.
Keywords: Dieulafoy’s lesion; Duodenum; Bleeding; Hemostasis
INTRODUCTION

A Dieulafoy’s lesion (DL) is a rare cause of gastrointestinal (GI) bleeding and is responsible for approximately 1–2% of cases of GI bleeding.1,2 DL is characterized by an abnormally enlarged arteriole protruding from the submucosa. Without any abnormality, such as ulcers or erosions, this tortuous artery can be defective and cause massive active pulsatile bleeding. DLs are located most often in the stomach, while DLs of other parts of the GI tract are rare. This paper reports a case of life-threatening bleeding from a DL in the duodenum.

CASE REPORT

A 72-year-old male attended the emergency room with melena for one day. The patient had hypertension. He was not taking any nonsteroidal anti-inflammatory drugs (NSAIDs), antiplatelet medications, or anticoagulation agents. His initial vital signs were as follows: blood pressure 103/56 mmHg, heart rate 112 beats per minute, respiration rate 16 breaths per minute, and body temperature 36.4°C. A physical examination revealed a soft abdomen with epigastric tenderness. The initial laboratory tests were as follows: white blood cell count 20,530/mm3, hemoglobin 7.5 g/dL, platelet count 382,000/mm3, blood urea nitrogen 41.0 mg/dL, creatinine 0.93 mg/dL, total bilirubin 0.32 mg/dL, aspartate aminotransferase 14 U/L, alanine aminotransferase 11 U/L, and international normalized ratio 1.26. Abdominal computed tomography revealed possible active GI bleeding in the duodenal second portion (Fig. 1). While awaiting admission in the emergency room, the patient exhibited hematochezia several times. His blood pressure decreased to 70/40 mmHg, and he was admitted to intensive care unit. The blood pressure stabilized with the administration of vasopressors and packed red blood cells in the intensive care unit. On the other hand, the hemoglobin level decreased to 4.5 g/dL. An emergency esophagogastroduodenoscopy was performed, revealing no evidence of an active bleeding lesion in the stomach. However, fresh blood emanating from the duodenum was observed in the antrum of the stomach. While observing the duodenum, an exposed vessel with active pulsatile bleeding was observed at the superior duodenal angle. The surrounding mucosa appeared normal without evidence of erosion or ulceration (Fig. 2). Argon plasma coagulation (APC) was applied to the exposed vessel, and successful hemostasis was achieved. His hemoglobin level remained stable at 9.5 g/dL after the procedure. He was discharged home after 10 days of hospitalization.

Figure 1. Abdominal CT scan showed active gastrointestinal bleeding in the duodenum (arrow). (A) Axial view. (B) Coronal view.

Figure 2. Endoscopy findings. (A) Active bleeding in the superior duodenal angle was noted. (B) Successful hemostasis was achieved with APC. APC, argon plasma coagulation.
DISCUSSION

Dr. Gallard first reported DL in 1884. The condition was properly distinguished and described by Dr. Dieulafoy, a French surgeon, in 1898. Since then, this rare condition has commonly been referred to as DL or Dieulafoy’s disease. DL is a condition where abnormal tortuous submucosal arteries erode through small overlying gastrointestinal mucosal defects, leading to bleeding without surrounding ulcers or erosions.3 Alternatively, it can manifest as fresh, densely adherent clots with a small attachment point to the mucosal defect or normal-appearing mucosa.4 The pathogenesis of DL remains unclear. It has been proposed that pulsations in a large submucosal vessel may disrupt the overlying epithelium, leading to localized ischemia and exposure of the vessel to bowel contents, resulting in erosion and rupture.5 Another hypothesis suggests that prolonged weakening of the wall by gastric wear-and-tear, coupled with the development of intravascular thrombi, leads to necrosis and bleeding.6 Predisposing factors for bleeding from DL include comorbid conditions, such as cardiovascular diseases, chronic kidney disease, diabetes mellitus, and the use of NSAIDs, aspirin, or anticoagulants.3,7

DL is a rare condition, accounting for approximately 1–2% of upper gastrointestinal bleeding cases, but it can lead to severe hemorrhage. DL is encountered most commonly in the stomach, with 80–95% reported to occur within six centimeters of the gastroesophageal junction on the lesser curve of the stomach.8,9 Although rare outside the stomach, DL can occur in the esophagus, duodenum, jejunum, ileum, colon, and rectum, albeit infrequently. Approximately 15% of DLs are reported to originate in the duodenum.10-12

Clinically, DL often presents with sudden onset hematemesis, melena, or hematochezia without preceding prodromal symptoms. The bleeding is characterized by a temporary cessation after a massive hemorrhage, with a high frequency of rebleeding. Substantial blood loss can lead to hypovolemic shock and even death.

A diagnosis of DL is challenging because the lesions are very small, causing obscured, intermittent gastrointestinal bleeding with normal surrounding mucosa.13 The most effective modality for diagnosing DL is endoscopy, with a reported diagnostic yield of approximately 70%.14 Nevertheless, a diagnosis by endoscopy can be challenging in cases where the lesion is subtle, actively bleeding profusely, or if bleeding has temporarily ceased.15 In such cases, repeated endoscopic examinations may be necessary, and angiography can be helpful in the diagnostic process.7,16

The treatment of DL is generally performed through endoscopic hemostasis, with a reported success rate of approximately 90%.3 Commonly used endoscopic hemostatic techniques include electrocoagulation, thermocoagulation, band ligation, sclerotherapy, and hemoclips. In cases where DL occurs in the small intestine, including the duodenum, as in the present case, hemostasis can be challenging, and the options for hemostatic procedures may be limited because of the narrow lumen and the relatively thin intestinal wall. In this case, the DL was located at the superior duodenal angle, making it challenging to perform mechanical techniques such as hemoclipping. Therefore, APC was attempted, and successful hemostasis was achieved.

The literature on hemostasis using APC in DL is limited but generally considered an effective hemostatic procedure. The reported success rates of hemostasis with APC are comparable to those of other hemostatic methods.17 The coagulation depth of APC is self-limited, but it extends to the submucosa, appearing adequate for complete obliteration of the submucosal arteries in DL.18 Moreover, APC can be applied to lesions using frontal and lateral probes, regardless of location. APC is considered a relatively safe procedure because of its limited penetration depth. Complications related to APC arise primarily from tissue contact with the probe tip and forceful insufflation of gas into the submucosa, potentially leading to emphysema and perforation in rare cases.19 Angiography can be considered as a rescue therapy if endoscopic hemostasis fails. Finally, surgical resection may be necessary if all other methods are unsuccessful.

In conclusion, duodenal DL is extremely rare but can result in recurrent and life-threatening massive bleeding. Increased awareness and early endoscopy are crucial for prompt diagnosis and treatment. APC can be used effectively for bleeding control of duodenal DL.

Financial support

None.

Conflict of interest

None.

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