|
 
 |
| CASE REPORT |
|
| Year : 2021 | Volume
: 54
| Issue : 1 | Page : 25-27 |
|
Dieulafoy's lesion in small intestine
Premananda Patnaik1, Rashmi Patnayak2, Mohita Ray2, Prateek Das2, Debahuti Mahapatra2
1 Department of Surgical Gastroenterology, Institute of Medical Sciences and SUM Hospital, Bhubaneswar, Odisha, India
2 Department of Pathology, Institute of Medical Sciences and SUM Hospital, Bhubaneswar, Odisha, India
| Date of Submission | 13-Dec-2019 |
| Date of Decision | 21-Apr-2020 |
| Date of Acceptance | 24-Jul-2020 |
| Date of Web Publication | 22-Jan-2021 |
Correspondence Address:
Rashmi Patnayak
Department of Pathology, Institute of Medical Sciences and SUM Hospital, Bhubaneswar, Odisha
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/fjs.fjs_108_19
Dieulafoy's lesion is a rare cause of gastrointestinal (GI) bleeding. It accounts for 0.3%–1.5% of major GI bleed. It is typically found in the stomach. Small intestinal Dieulafoy's lesion is very rare. In the small intestine, Dieulafoy's lesion is most commonly seen in the jejunum. Hereby, we present one such case. A 36-year-old female attended the department of gastrosurgery with complaints of pain abdomen and generalized weakness for 15 days. She also had on and off melena and hematochezia. She was admitted with a diagnosis of obscure GI bleeding and was taken up for laparotomy. On table, enterotomy and retrograde enteroscopy was performed. There was a lesion over the distal jejunum. That segment of the intestine was resected. The specimen was sent for histopathological examination. The luminal surface of the intestine showed a small, whitish nodule measuring 0.3 cm in diameter. This nodule was noted beneath the mucosa, in the subepithelium. The overlying mucosa was ulcerated. Histopathological examination established the lesion to be of Dieulafoy's. Awareness, careful endoscopy, and histopathological examination are essential to diagnose Dieulafoy's lesion.
Keywords: Bleeding, Dieulafoy's lesion, gastrointestinal bleeding, small intestine
How to cite this article:
Patnaik P, Patnayak R, Ray M, Das P, Mahapatra D. Dieulafoy's lesion in small intestine. Formos J Surg 2021;54:25-7 |
| Introduction | |  |
Dieulafoy's lesions are described as a rare cause of gastrointestinal (GI) hemorrhage. Gallard first identified this lesion in 1884 and described it as a miliary aneurysm of the stomach.[1],[2] It was also designated as “exulceratio simplex.”[3] Formally, Dieulafoy described it in 1897, after whom the terminology Dieulafoy's lesion has been coined. The most common site of Dieulafoy's lesion is stomach. Small-bowel Dieulafoy's lesion is very rare.[1],[2] It is commonly seen in elderly males.
Dieulafoy's lesion is also called caliber-persistent artery. It is an arteriovenous malformation. The pathogenesis of Dieulafoy's lesion is not clear. It is believed to be congenital in origin. The common presentations are hematemesis or melena.[1],[2],[3] This bleeding ulcer is characterized by presence of a single, unusually large-diameter mural arteriole penetrating the submucosa. This large vessel erodes through the mucosa, resulting in massive bleeding.
Macroscopically, it appears like a small pea-sized lesion with protrusion of the artery from its base. Microscopically, a thick-walled artery is seen in the submucosa. This vessel is larger than the surrounding submucosal vessels.
Dieulafoy's lesion accounts for 0.3%–1.5% of major GI bleeds. They are often the cause of obscure, massive GI hemorrhage. Although Dieulafoy's lesion is relatively rare, it does carry a significantly high risk for mortality.[4]
The diagnosis of Dieulafoy's lesion is difficult with conventional investigations such as endoscopy, arteriography, and even laparotomy.[5] They are increasingly diagnosed nowadays due to recent advances in endoscopy.[6]
| Case Report | | |
A 36-year-old female had pain abdomen, generalized weakness, on and off melena, and hematochezia for 15 days. She had a previous history of hospitalization. At that time, upper GI endoscopy and colonoscopy (up to terminal ileum) was performed, which did not show any abnormality. On further evaluation, her hemoglobin was 3.8 g/dL and total red blood cell was (1.82 × 106/μL). Her coagulation profile was within normal limits. She was not a known diabetic or hypertensive. She was admitted with the diagnosis of obscure GI bleeding and underwent laparotomy. Computed tomography (CT) angiography was done, but it could not detect the site of bleed or any causative lesion. Enterotomy and retrograde enteroscopy was performed through the mid-ileum up to D-J flexure to locate the source of bleed. A submucosal lesion measuring 15 mm ×15 mm, with central umbilication and visible vessel over the center, was noted. Active bleed was seen in the distal jejunum [Figure 1]a and [Figure 1]a, [Figure 1]b. Anterograde enteroscopy showed altered blood in the distal ileum. The small bowel was extracted outside with a small 5-cm incision to do intraoperative enteroscopy. A clinical diagnosis of submucosal GI stromal tumor was made. That segment of the intestine with the lesion was resected and sent for histopathological examination [Figure 1]c. | Figure 1: (a and b) Retrograde enteroscopy showing a submucosal lesion with central umbilication and visible vessel over the center and active bleed (c) Small intestine with the lesion (d and e) Histopathology showing eroded intestinal lining epithelium by a large blood vessel within the submucosa (hematoxylin and eosin, ×40) (f) Congested and thrombosed blood vessel within the submucosa (hematoxylin and eosin, ×100)
Click here to view |
In the luminal aspect, there was a small, whitish nodule measuring 0.3 cm in diameter, present beneath the mucosa with ulceration of the overlying epithelium. The serosa appeared unremarkable.
The histopathological examination showed erosion of the intestinal mucosa by a large, congested, thrombosed blood vessel. This vessel was present within the submucosa. The muscularis propria and serosa were unremarkable [Figure 1]d, [Figure 1]e, [Figure 1]f. Based on these findings, a histopathological diagnosis of Dieulafoy's lesion was made.
She had an uneventful postoperative course and is currently doing well.
| Discussion | | |
Dieulafoy's lesion is a relatively rare but potentially life-threatening cause of GI tract hemorrhage. The true incidence of Dieulafoy's lesion is difficult to determine as it remains silent until presentation. It remains usually underdiagnosed. Even with conventional investigations, Dieulafoy's lesion is not easily identified. It is believed to account for only 1%–2% of acute GI bleeding. Lack of awareness attributes to increase in morbidity in Dieulafoy's lesion.[7]
The exact etiology is not known. It is more common in males than in females. It can affect any age group but is more commonly seen in the elderly. Various literatures have noted that Dieulafoy's lesion is associated with comorbidities such as cardiopulmonary dysfunction and chronic renal failure in majority of patients.[7] Holleran et al. have reported a possible link between chronic liver disease and small-intestinal Dieulafoy's lesions. Few reports describe the association of Dieulafoy's lesion with intake of nonsteroidal anti-inflammatory drug and anticoagulant drugs.[8] However, our patient did not have any such association.
The predominant location of Dieulafoy's lesion is the upper GI tract.[9] Stomach is the most common site. Approximately 80%–95% of the Dieulafoy's lesion is located within 6 cm of the gastroesophageal junction. At this location, it is present in direct correlation with the left gastric artery. Other sites include the esophagus, duodenum, jejunum, ileum, large bowel, and rectum.[10]
Approximately one-third of the lesions are extragastric. Small-intestinal Dieulafoy's lesion is uncommon.
In Dieulafoy's lesion, aberrant submucosal arteries are present which may cause severe GI bleeding. The cause of mucosal injury is yet to be discovered. One theory suggested that the pulsation within the large submucosal artery may apply pressure to the epithelium, resulting in erosion and thereby causing rupture of the vessel toward the lumen. However, the point against this theory is that the lesion is often intermittent in nature. Another hypothesis suggested that a thrombus within the artery may result in continuous necrosis of the arterial walls, which can, in turn, lead to arterial rupture.[10]
There is no available consensus regarding the treatment of Dieulafoy's lesion. Therapeutic endoscopy can control bleeding in majority of patients. In the case of an inaccessible lesion, angiography can act as a valuable alternative to endoscopy. Surgical intervention is used in those cases, where therapeutic endoscopic or angiographic interventions have failed.[7] In our case, CT angiography was done, but it could not detect the site of bleed or any causative lesion. Technetium-99m-labeled red blood cell scan (RBC scan) has the advantage of detecting small amounts of extravasation into the gut.[7] RBC scan could not be done in the present case due to logistic issues. It is not available in our setup.
With increased detection of Dieulafoy's lesion by advanced endoscopy, a significant decrease in the mortality has been noted. Recent reports suggest that laparoscopic surgery may play a role in managing symptomatic Dieulafoy's lesion.[7] As there is a need to perform intraoperative enteroscopy, laparoscopic method is not ideal because handling the endoscope with laparoscopic instruments is not easy.
Dieulafoy's lesion should be included in the differential diagnosis of any acute or obscure GI hemorrhage.[6] It can cause life-threatening bleeding in 10% of cases. Awareness about Dieulafoy's lesion and careful endoscopy are essential for diagnosis. Many a time, repeat endoscopy may be necessary to establish the diagnosis.[7]
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal the identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Dieulafoy P. Lecons clinique de l'Hotel de Paris. Paris: Mason; 1897. p. 2:1.  |
| 2. | Fox A, Ravi K, Leeder PC, Britton BJ, Warren BF. Adult small bowel Dieulafoy lesion. Postgrad Med J 2001;77:783-4. |
| 3. | Senger JL, Kanthan R. The evolution of Dieulafoy's lesion since 1897: Then and now-A journey through the lens of a pediatric lesion with literature review. Gastroenterol Res Pract 2012;2012:432517. |
| 4. | Blecker D, Bansal M, Zimmerman RL, Fogt F, Lewis J, Stein R, et al. Dieulafoy's lesion of the small bowel causing massive gastrointestinal bleeding: Two case reports and literature review. Am J Gastroenterol 2001;96:902-5. |
| 5. | Schmulewitz N, Baillie J. Dieulafoy lesions: A review of 6 years of experience at a tertiary referral center. Am J Gastroenterol 2001;96:1688-94. |
| 6. | Joarder AI, Faruque MS, Nur-E-Elahi M, Jahan I, Siddiqui O, Imdad S, et al. Dieulafoy's lesion: An overview. Mymensingh Med J 2014;23:186-94. |
| 7. | Baxter M, Aly EH. Dieulafoy's lesion: Current trends in diagnosis and management. Ann R Coll Surg Engl 2010;92:548-54. |
| 8. | Holleran G, Hussey M, McNamara D. Small bowel Dieulafoy lesions: An uncommon cause of obscure bleeding in cirrhosis. World J Gastrointest Endosc 2016;8:568-71. |
| 9. | Dulic-Lakovic E, Dulic M, Hubner D, Fuchssteiner H, Pachofszky T, Stadler B, et al. Bleeding Dieulafoy lesions of the small bowel: A systematic study on the epidemiology and efficacy of enteroscopic treatment. Gastrointest Endosc 2011;74:573-80. |
| 10. | Khan R, Mahmad A, Gobrial M, Onwochei F, Shah K. The Diagnostic Dilemma of Dieulafoy's Lesion. Gastroenterology Res 2015;8:201-6. |
[Figure 1]
|
Search Pubmed for