Introduction
Dieulafoy’s lesion (DL), also called “caliber persistent artery”, is a rare cause
of gastrointestinal bleeding. It accounts for about 6 % of gastrointestinal nonvariceal
bleeding, and 1 % to 2 % of all acute gastrointestinal hemorrhages [1]
[2]. The incidence of DL is potentially higher than reports suggest because of the difficulty
of making the correct diagnosis. It was initially described as “miliary aneurysms
in stomach” by Gallard in 1884, and given a more exact definition by French surgeon
Georges Dieulafoy in 1898, on the basis of his study of alimentary tract hemorrhage
in three young men. Since then, with the development of gastrointestinal endoscopy,
more and more cases of DL have been reported worldwide. According to most of the reports,
DL can occur in the stomach (71 %), duodenum (15 %), esophagus (8 %), colon (2 %),
rectum (2 %), jejunum-ileum (1 %), and gastric anastomosis (1 %) [3]
[4].
We present a case of DL in a young man who had a history of anal receptive sexual
intercourse.
Case report
A 21-year-old man was admitted to our digestive department because of intermittent
bloody stool passage for a week. The patient did not suffer from abdominal pain, his
vital signs were stable (heart rate 71 bpm, blood pressure 115/75 mmHg), and he had
no other clinical manifestations apart from hematochezia. He had no history of gastrointestinal
disease or liver disease, no history of alcohol, smoking or drug abuse, and was negative
for Helicobacter pylori infection. The only point worth mentioning was a history of anal receptive intercourse.
Physical examination did not reveal any significant alterations. Laboratory examination
showed a normal hemoglobin level (13.5 g/dL), and platelet count and coagulation parameters
were also normal. The patient underwent emergency colonoscopy after a cleansing enema,
and a nipple-like protuberance (about 5 mm diameter) was discovered. The lesion was
located in the rectum 5 cm away from the anal verge; it was concave at the top and
was accompanied by hyperemia ( [Fig. 1] and [Fig. 2]). Endoscopic ultrasound (EUS) did not find any occupancy lesions but a local vascular
structure was present in the intestinal wall ( [Fig. 3]). The lesion was consistent with Dieulafoy’s lesion (DL), and was treated with two
hemostatic clips simultaneously ([Fig. 4]). No further bleeding was recorded after the procedure, and the patient was discharged.
A colonoscopy performed 1 month later did not demonstrate any abnormality but a residual
hemostatic clip was present on the lesion. Furthermore, bloody stools have not recurred
during a 6-month follow-up period.
Fig. 1 Colonoscopic examination revealed a nipple-like protuberance (about 5 mm diameter),
which was concave at the top and accompanied by hyperemia.
Fig. 2 Higher magnification view of Dieulafoy’s lesion seen in Fig. 1.
Fig. 3 Endoscopic ultrasound (EUS) showing the vascular structure passing through the intestinal
wall from the subserosa to the mucosa, with a diameter of 1.5 mm. The Doppler signal
was very clear, and the pulsed waveform indicated that it was an artery.
Fig. 4 Two hemostatic clips were deployed, and the lesion showed no signs of bleeding.
Discussion
DL is an uncommon but well-recognized cause of gastrointestinal bleeding. This disease
often occurs in males, and there is a wide range of age at time of occurrence, with
reports of the lesion in infants as well as in a 93-year-old patient [5]. The majority of DLs are located in the proximal stomach within 6 cm of the gastroesophageal
junction; however, they can occur anywhere in the gastrointestinal tract, including
the rectum [6]. Excessive alcohol intake and nonsteroidal anti-inflammatory drug use may increase
the chances of bleeding from a gastric lesion by causing mucosal erosions [3]; however, some researchers report that there is no relationship between alcohol
intake and DL [7]. In the colon, solid bowel contents may lead to ulceration with resultant exposure
of an artery and hemorrhage [8], while in the rectum, anal receptive intercourse should be considered to be a direct
cause as a result of the repeated mechanical stimulation which occurs in men during
anal sexual intercourse. The sudden onset of extensive bleeding, which is usually
intermittent and recurrent, is the same presentation in all ages. Since the bleeding
is arterial, the amount of blood lost is usually massive. Depending upon the location
of bleeding and the amount, the manifestations can range from iron deficiency anemia
to life-threatening hemorrhagic shock [9], which has been reported to occur in up to 87 % of patients with DL [10].
The pathogenesis of DL is not yet clear. The normal gastroenteric vascular network
narrows progressively as it reaches the mucosa, and forms a capillary network mostly
in the submucosa. A DL is described as an arteriole, of which the diameter of the
vascular network remains unchanged (1 – 3 mm) through the serosa and reaches the mucosa.
The DL is generally 10 – 20 times thicker than a normal capillary, and is covered
by a thin mucosal membrane without symptoms until the insidious onset of acute gastrointestinal
bleeding begins [2]
[3].
Patients with gastrointestinal bleeding as a result of DL usually have no history
of chronic liver disease or gastrointestinal disease, while DL can actually be exacerbated
by portal hypertension or liver transplantation [11]. Patients may or may not have taken non-steroidal anti-inflammatory drugs before.
The typical location of DL is the proximal stomach, usually within 6 cm of the cardio-esophageal
junction, and a variety of other sites including the esophagus, small bowel, and large
bowel have been reported in the literature, including 2 % in the colon, and 2 % in
the rectum. Hematochezia complicated by hypovolemic shock is the most important sign
of a colorectal DL. Endoscopic examination is primarily preferred for diagnosis, and
provides the following valuable diagnostic criteria: (1) micropulsatile bleeding from
small (< 3 mm) mucosal defects surrounded by normal mucosa; (2) the presence of protruding
vessels; (3) fresh clots attached to a small mucosal defect or to normal mucosa [12]. The choice of gastroscopy, colonoscopy or enteroscopy mainly depends on the clinical
manifestations and the doctor’s experience. EUS may be valuable in identifying the
unaltered vessel.
Endoscopic diagnosis can sometimes be delayed owing to the difficulty of localizing
the bleeding site in situations with a small lesion, intermittent bleeding or poor
visualization. Under these circumstances, emergent mesenteric angiography or CT angiography
is an alternative. A typical angiographic image shows a caliber-persistent, convoluted
artery in a position tangential to the lumen [13]. Multidetector row computed tomography, a noninvasive technique, could also help
pinpoint potential bleeding therefore allowing the endoscopist to locate the lesion
more accurately.
Hemostasis can be successfully achieved by endoscopic therapy in about 90 % of patients
with DL, and this has dramatically decreased the mortality rate [14]. Endoscopic treatments cover several major techniques including epinephrine and
sclerotherapy injection, bipolar electro-coagulation (BICAP), and mechanical methods;
however, in practice, neither the injection therapy nor BICAP are feasible because
the hemostatic effect and rebleeding rate are unsatisfactory when compared to using
mechanical methods. Thus, the mechanical methods currently used such as hemoclipping
and band ligation are reliable and effective for the treatment of colonic DLs [15]. Repeated endoscopic treatment is strongly recommended when rebleeding occurs.
Arterial embolization is an alternative therapy in patients resistant to endoscopic
treatment and patients who cannot endure surgery. Increasing evidence has suggested
that EUS-guided treatment with vascular therapy and hemoclipping offers a less invasive
and more practical option than surgery, on account of the precise delivery of thrombotic
agent into the target vessel or endoscopic hemoclipping of DL [16]
[17].
Emergent resection surgery, which plays a role in traditional therapy for the treatment
of colorectal DL, is indicated on the occasions when patients are suffering from hemorrhagic
shock, and bleeding cannot be successfully controlled by endoscopic or angiographic
methods. It consists of resection of the bleeding bowel segment or subtotal colectomy
if bleeding cannot be accurately localized. Laparoscopic resection with the assistance
of endoscopy has been successfully applied in some patients.
In conclusion, in our case, Dieulafoy’s lesion was diagnosed with the help of both
colonoscopy and ultrasonography before successful treatment with hemostatic clips.
A history of anal receptive intercourse may play a role in mechanical damage. Ultrasonography
and fine flow Doppler should be used in the diagnosis of this disease and endoscopic
hemostatic clips can be effective in controlling bleeding from a rectal Dieulafoy’s
lesion.
