Dieulafoy lesions: One patient, two different localizations

ABSTRACT

Dieulafoy lesions (DLs) are dilated submucosal arterial structures visualized on endoscopy as bleeding foci on the superficial mucosa without erosion or ulceration. DLs account for 1–5.8% of acute non-variceal upper gastrointestinal bleeding cases. A 72-year-old male patient with known Alzheimer’s disease and coronary artery disease, being followed up at a nursing home, presented to our emergency department with foul-smelling, loose, and tarry stool. Esophagogastroduodenoscopy revealed a 3 mm DL immediately adjacent to the Z line in the distal esophagus, demonstrating a fresh blood clot without the appearance of a surrounding ulcer. Two endoscopic hemoclips were applied to this lesion. The patient was monitored at the intensive care unit for the following 2 days and later transferred to internal medicine inpatient unit. He developed hematochezia on the 8^th day of hospitalization. Emergent rectosigmoidoscopy was performed showing two separate 3 and 4 mm sized DLs, located immediately proximal to the dentate line. These lesions were successfully treated using two endoscopic band ligations. DLs can occur synchronously, albeit very rarely, and a careful search for multiple lesions is necessary to avoid further bleeding.

INTRODUCTION

Dieulafoy lesions (DLs) are dilated submucosal arterial structures visualifzed on endoscopy as bleeding foci on the superficial mucosa without erosion or ulceration. They are believed to be caused by localized mucosal ischemic damage and acquired vascular ectasia eroding through atrophic mucosa. DLs account for 1–5.8% of acute non-variceal upper gastrointestinal (GI) bleeding cases. Although DLs are generally asymptomatic, melena is seen in 44%, hematemesis in 30%, both melena and hematemesis in 18%, hematochezia in 6%, and iron deficiency anemia in 1% among symptomatic cases.[1]

DLs are more commonly seen in men and older age group.[2] Comorbidities are present in 90% of patients, with the most common being cardiovascular disease, hypertension, diabetes mellitus, liver disease, and kidney failure.[3] Disease etiology includes nonsteroidal anti-inflammatory medication and anticoagulant use, alcoholism, and fecalomas.[4] Approximately 70% of DLs are detected in the stomach and of those gastric lesions, 75% are localized in the proximal stomach, especially within 6 cm of the gastroesophageal junction. DLs can also occur as a source of lower GI bleeding, developing along the GI tract, including the duodenum, jejunum, ileum, cecum, appendix, colon, and anal canal.[1,2] Here, we describe a peculiar case, presented with recurrent upper and lower GI bleeding at the same inpatient setting due to separate synchronous DLs in the esophagus and rectum, which were successfully treated using endoscopy.

CASE REPORT

A 72-year-old male patient with known Alzheimer’s disease and coronary artery disease, being followed up at a nursing home, presented to our emergency department with foul-smelling, loose, and tarry stool. At the time of presentation, his pulse was 126 beats/min, blood pressure was 126/86 mmHg, and melena was detected on rectal examination. The patient was taking 20 mg/day memantine and 100 mg/day acetylsalicylic acid. His hemoglobin (Hb) level (11.9 g/dl), platelet count (531,000/mm³), and coagulation parameters were within normal limits. Esophagogastroduodenoscopy (Olympus, GIFHD 190, Tokyo, Japan) revealed a 3 mm DL immediately adjacent to the Z line in the distal esophagus, demonstrating a fresh blood clot without the appearance of a surrounding ulcer (Fig. 1a). Two endoscopic hemoclips (QuickClip 2™ Olympus) were applied to this lesion (Fig. 1b).

Figure 1.

(a) Dieulafoy lesion (white arrow) in the distal of the esophagus and (b) endoscopic hemoclips applied to the lesion.

The patient was monitored at the intensive care unit for the following 2 days and later transferred to internal medicine inpatient unit. He developed hematochezia on the 8^th day of hospitalization. His Hb level was 7.9 g/dl, and subsequent computed tomography angiography (CTA) demonstrated active extravasation from the distal rectum. Subsequently, an emergent rectosigmoidoscopy was performed (Olympus GIFHD 190, Tokyo, Japan) showing two separate 3 and 4 mm sized DLs, located immediately proximal to the dentate line (Fig. 2a). These lesions were successfully treated using two endoscopic band ligations (Speedband Superview Super 7™ Boston multiple band ligator), as shown in Figure 2b.

Figure 2.

(a) Dieulafoy lesion (white arrow) in the rectum and (b) endoscopic band ligation to the lesion (white arrow).

The patient was transfused with two units of erythrocyte suspension during the hospital stay. Since his hemogram parameters remained stable during the follow-up without any evidence of bleeding, he was discharged on the 10^th day of hospitalization. The patient, who has been followed up by phone since then, has been taking low-dose acetylsalicylic acid for 5 months and no recurrent bleeding has occurred.

DISCUSSION

Although not common, DLs are severe and life-threatening causes of GI bleeding. They are most frequently localized in the stomach, they have also been described in the duodenum, colon, esophagus, and rectum.[1,2,4] DLs are generally seen in a single localization, and to the best of our knowledge, no case report presenting with synchronous DLs in the esophagus and rectum, was reported in the English literature before; thus, our paper is the first case report in this respect. According to Reilly and Al-Kawas, only 49% of DLs are identified during the first endoscopic examination, 33% of patients require a second examination, and 18% require explorative laparotomy for an accurate diagnosis. DLs can easily be overlooked on endoscopy due to mucosal inflammation and lack of ulceration. Therefore, DLs must be suspected in all patients without a clear GI bleeding focus.[5,6] CTA must be considered when investigating less affected areas, such as the esophagus and rectum, as possible localizations of DLs. In the current case, CTA was performed because of hematochezia. The presence of active extravasation from the distal rectum, as demonstrated in CTA, indicates the need for a more careful examination.

Hemostasis can be endoscopically achieved by mechanical methods, such as thermal or argon plasma coagulation, regional epinephrine or cyanoacrylate injection, endoscopic band ligation, and hemoclip application in patients with DLs. Mechanical methods are generally more effective than sclerotherapy and coagulation techniques. The recurrent bleeding rate in mechanical therapy combined with epinephrine injection is lower than that in monotherapy.[7–9] In the current case, hemostasis was achieved by endoscopic hemoclip application and band ligation to the lesions in the esophagus and rectum, respectively, without any recurrent bleeding.

Our case is highly peculiar because the patient presented with synchronous DLs in two different rare foci, as well as different presenting findings, such as melena and hematochezia that developed within a week. In the available English literature, we found a report of a 3-year-old child with four different DLs detected in the stomach that was treated endoscopically.[10] Another case report described a 15-year-old child with a DL in the stomach that was first treated with injection sclerotherapy, but surgical treatment was required after recurrent bleeding and detection of a synchronous separate focus in the jejunum on CTA.[11]

Conclusion

To the best of our knowledge, our patient is the first adult case in the literature in which synchronous DLs caused bleeding both in the esophagus and the rectum, treated successfully with endoscopic approach. DLs can occur synchronously, albeit very rarely, and a careful search for multiple lesions is necessary to avoid further bleeding.