Recurrent massive bleeding due to dissecting intramural hematoma of the esophagus: Treatment with therapeutic angiography

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World J Gastroenterol 2009 November 7; 15(41): 5232-5235
wjg@wjgnet.com World Journal of Gastroenterology ISSN 1007-9327
doi:10.3748/wjg.15.5232 © 2009 The WJG Press and Baishideng. All rights reserved.
ORIGINAL ARTICLES
CASE REPORT
Recurrent massive bleeding due to dissecting intramural
hematoma of the esophagus: Treatment with therapeutic
angiography
Jaejun Shim, Jae Young Jang, Young Hwangbo, Seok Ho Dong, Joo Hyeong Oh, Hyo Jong Kim, Byung-Ho Kim,
Young Woon Chang, Rin Chang
Jaejun Shim, Jae Young Jang, Young Hwangbo, Seok Ho
dissecting intramural hematoma of the esophagus: Treatment
Dong, Hyo Jong Kim, Byung-Ho Kim, Young Woon Chang,
with therapeutic angiography. World J Gastroenterol 2009;
Rin Chang, Department of Internal Medicine, Kyung Hee
15(41): 5232-5235 Available from: URL: http://www.wjgnet.
University School of Medicine, Hoegi-dong 1, Dongdaemoon-
com/1007-9327/15/5232.asp DOI: http://dx.doi.org/10.3748/
gu, Seoul 130-702, South Korea
wjg.15.5232
Joo Hyeong Oh, Department of Diagnostic Radiology,
Kyung Hee University School of Medicine, Hoegi-dong 1,
Dongdaemoon-gu, Seoul 130-702, South Korea
Author contributions: Jang JY, Oh JH and Hwangbo Y
designed the procedure and took part in patient management;
INTRODUCTION
Shim J and Hwangbo Y analyzed the data; Shim J wrote the
Dissecting intramural hematoma of the esophagus (DIHE)
paper; all the authors discussed the results and commented on
the manuscript.
is a rare but wel -known esophageal injury[1,2]. It is char-
Correspondence to: Jae Young Jang, MD, Department of
acterized by a concentric or eccentric intramural hema-
Internal Medicine, Kyung Hee University School of Medicine,
toma associated with dissection of the esophageal wal [2].
Hoegi-dong 1, Dongdaemoon-gu, Seoul 130-702,
Forceful vomiting, mechanical insult, and underlying co-
South Korea. jyjang@khu.ac.kr
agulopathy are common causes. It can also occur sponta-
Telephone: +82-2-9588248 Fax: +82-2-9681848
neously without any evident cause[3]. DIHE is considered
Received: August 5, 2009 Revised: September 8, 2009
Accepted: September 15, 2009
a benign disease. The hemorrhage from DIHE does not
Published online: November 7, 2009
have clinical y significant consequences, although in rare
cases massive bleeding with hypovolemic shock can oc-
cur. We report a case of DIHE presenting as recurrent
massive intraluminal bleeding that was treated by transar-
Abstract
terial embolization.
Spontaneous or traumatic intramural bleeding of the
esophagus, which is often associated with overlying
CASE REPORT
mucosal dissection, constitutes a rare spectrum of
esophageal injury cal ed dissecting intramural hema-
A 57-year-old man visited the emergency room com-
toma of the esophagus (DIHE). Chest pain, swal owing
plaining of a sore throat and difficulty swal owing for
difficulty, and minor hematemesis are common, which
5 d. He had a history of alcoholic liver cirrhosis (Child-
resolve spontaneously in most cases. This case report
Pugh class B), but he continued drinking. Laryngoscopy
describes a patient with spontaneous DIHE with recur-
and cervical computed tomography (CT) revealed severe
rent massive bleeding which required critical manage-
laryngopharyngitis. Upper gastrointestinal endoscopy
ment and highlights a potential role for therapeutic
showed marked laryngeal edema and a smal ulceration
angiography as an alternative to surgery.
in the upper esophageal sphincter. There was no intralu-
minal mass, except for small varices in the distal esopha-
© 2009 The WJG Press and Baishideng. Al rights reserved.
gus. The gastric mucosa was moderately congested. The
patient denied any history of cervical trauma or instru-
Key words: Esophagus; Intramural hematoma; Therapeutic
mentation. He was treated medical y with antibiotics, and
angiography
his initial symptoms subsided within 5 d.
One week later, the patient complained that he
Peer reviewer: Dr. Andreas G Schreyer, Department of Radiology,
University Hospital Regensburg, Franz-Josef-Strauss-Allee 11,
regurgitated blood from the throat. He continued to
Regensburg 93053, Germany
spit up smal volumes of fresh blood repeatedly. His
hemoglobin dropped from 13.9 to 6.5 g/dL, and 4 U of
Shim J, Jang JY, Hwangbo Y, Dong SH, Oh JH, Kim HJ, Kim
packed red cel s were transfused. Emergency endoscopy
BH, Chang YW, Chang R. Recurrent massive bleeding due to
revealed a smal opening in the cervical esophagus and
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Shim J et al . Dissecting intramural hematoma of the esophagus 5233
A
Figure 3 Celiac angiography
showing a hyperstaining pseu-
doaneurysmal lesion (arrow)
in an esophageal branch of the
left gastric artery. The branch
was embolized using glue and
lipiodol.
B
A
Figure 1 Endoscopic view. A: A small opening in the cervical esophagus; B:
Mucosal bridging with a large mucosal defect in the esophagogastric junction.
B
A
B
Figure 4 Fol ow-up chest CT. A: The transverse view of the proximal esophagus
showing a resolved intramural hematoma and improved dissection (ar ow); B: The
Figure 2 Chest CT images. A: The transverse view of the proximal esophagus
embolized lesion is seen as focal lipiodol uptake in the distal esophagus (ar ow).
shows a concentric intramural hematoma and mucosal dissection with an air-
fluid level in the false lumen (arrow). Bilateral pleural effusions are seen; B: The
showed no bleeding at the previously treated site. How-
sagittal view shows an extensive intramural hematoma of the esophagus.
ever, continuous oozing of blood from the distal esoph-
agus was observed. His vital signs were unstable.
mucosal bridging with a large mucosal defect around the
Since surgery was very risky due to underlying liver
esophagogastric junction (Figure 1). Active bleeding was
cirrhosis, less invasive celiac angiography was performed
detected from a vessel exposed on the ulcer base in the
for hemostasis. A smal hyperstaining pseudoaneurysmal
cardia. After hemostasis with endoscopic clipping, his vital
lesion was observed at an esophageal branch of the
signs and hemoglobin stabilized. Chest CT performed
left gastric artery (Figure 3). The branch was embolized
because of the esophageal lesions revealed dissection of
using glue and lipiodol. After the procedure, the patient’s
the wal and a large circumferential intramural hematoma
vital signs and hemoglobin stabilized. However, he
in the esophagus (Figure 2). Based on the endoscopy and
experienced a few bouts of minor hematemesis 10 d
chest CT findings, DIHE with cardiac ulcer bleeding was
later. Transarterial embolization using glue and coils was
diagnosed. The patient was treated conservatively without
reattempted, and no further bleeding occurred. Fol ow-
oral intake.
up chest CT 4 wk later showed a resolving hematoma
Eight days later, the hematemesis recurred. Conser-
and double lumens separated by the dissected mucosa.
vative treatment with massive transfusion of packed red
The embolization site was seen as focal lipiodol uptake in
cel s and fresh frozen plasma was ineffective. Endoscopy
the distal esophagus (Figure 4). Endoscopic examination
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5234 ISSN 1007-9327 CN 14-1219/R World J Gastroenterol November 7, 2009 Volume 15 Number 41
Table 1 Summary of 11 patients with dissecting intramural hematoma of the esophagus presenting major hemorrhage
Ref.
Sex/age (yr)
Underlying diseases
Precipitating factor
Primary symptoms
Treatment
Outcome
Atefi et al[9]
M/29
Diabetes
Retching
Hematemesis
Conservative
Died
Pneumonia
Hemodialysis
Kerr[15]
F/72
Hypertension
None
Epigastric pain
Conservative
Resolved
Ortiz[16]
F/80
None
Food (rice)
Chest pain, dysphagia
Surgical
Died
Freeman et al[14]
F/59
None
None
Chest pain
Conservative
Resolved
de Vries et al[12]
M/88
None
Minor head trauma
Retrosternal pain
Conservative
Resolved
Folan et al[13]
M/58
Alcoholism
Vegetable (broccoli)
Dysphagia
Surgical
Resolved
Takaoka et al[17]
F/87
Suppurative cholangitis
Nasobiliary catheter
Hematemesis
Conservative
Resolved
Cullen et al[11]
F/74
Hypertension
Heparin IV
Chest pain, dysphagia
Conservative
Resolved
Yamashita et al[18]
F/67
Cerebral aneurysm
Heparin IV
Hematemesis
Conservative
Resolved
Retching
Bandyopadhyay et al[10]
M/86
Hypertension
Warfarin
Hematemesis
Conservative
Died
Ischemic heart disease
Present case
M/57
Liver cirrhosis
None
Hematemesis
Therapeutic
Resolved
angiography
was declined. Supportive care with parenteral nutrition
the hematoma, however, the exact mechanism remains
was maintained. The patient was discharged without
unclear. Although half of patients are reported to
complications after 6 wk.
experience hematemesis, the volume of bleeding is usual y
smal , and major bleeding with hypovolemic shock is
DISCUSSION
uncommon.
We reviewed 119 patients who were presented in 87
The esophagus is susceptible to various extrinsic injuries
English-language reports since 1968. Major bleeding
(from ingested foods, instruments, and bougienage) or
was defined as more than 500 mL hematemesis with
intrinsic sheering forces induced by retching, vomiting,
hypovolemic shock or bleeding that required transfusion
or coughing. DIHE lies in the spectrum of esophageal
of at least 4 U. Eleven cases (9.2%), including the present
injuries between a mucosal tear (Mallory-Weiss syn-
case, were col ected and summarized in Table 1[9-18]. Seven
drome) and a transmural laceration (Boerhaave’s syn-
cases were elderly patients (five females and two males).
drome)[2,4]. Although these syndromes are usually associ-
The bleeding was attributed to anticoagulation therapy in
ated with severe vomiting, DIHE is not always caused by
three cases[10,11,18]. The majority were treated conservatively.
emesis[4]. One out of five patients reports no history of
However, this type of treatment was not always effective.
trauma. However, an underlying coagulopathy is found
An elderly patient who was treated conservatively died
in many patients with so-cal ed spontaneous DIHE[1].
after 5 d[10]. Two patients were treated with surgery[13,16];
Portal hypertension and endoscopic variceal sclerothera-
one of them had a 5-cm longitudinal tear with a pulsatile
py are also associated with DIHE in cirrhotic patients[5].
bleeding vessel between 25 and 30 cm from the alveolar
Although the direct cause was not clear in this present
case, the cervical esophageal ulcer and underlying portal
ridge on endoscopy, and the active arterial bleeder was
hypertension may have been precipitating factors.
identified and treated with an emergency thoracotomy[13].
Acute chest pain is a common presenting symptom
In our case, angiography located and treated the bleeding
and should be differentiated from acute myocardial
site at an esophageal branch of the left gastric artery.
infarction and aortic dissection[6]. Hematemesis and
Extensive DIHE and major bleeding might be caused
difficulty swal owing may ensue, and these are helpful for
by a smal bleeding artery. In this situation, therapeutic
differentiating from other critical diseases. The typical
angiography may be effective.
triad of DIHE (chest pain, dysphagia, and hematemesis)
Therapeutic angiography has been used to control
is evident only in one third of cases[1]. Therefore, the
major non-variceal gastrointestinal bleeding and has been
rarity of DIHE and atypical symptoms can delay correct
reported to be safe, effective, and durable[19]. It is usually
diagnosis. In our case, repeated hematemesis with
considered when endoscopic therapy has failed or when
hypovolemia was the only clinical feature.
emergency surgery carries a high risk of mortality. It has
DIHE is general y benign. Most patients recover ful y
also been effective in the hemostasis of uncontrol ed
with conservative management[1]. Esophageal obstruction
Mal ory-Weiss syndrome[20] and in the treatment of a
and major bleeding constitute two major complications
giant gastric intramural hematoma[21].
of DIHE. Esophageal obstruction by the hematoma
In conclusion, massive intraluminal bleeding can be
may cause or aggravate difficulty swal owing. Successful
a complication of DIHE. Although most bleeding in
endoscopic decompression or surgical treatment have
DIHE can be managed medical y, prompt, appropriate
been reported in such cases[7,8].
treatment should be attempted in hemodynamically
A major intraluminal hemorrhage seems to be
unstable patients. In such cases, therapeutic angiography
caused by overlying mucosal rupture and evacuation of
may be a useful treatment alternative to surgery.
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Shim J et al . Dissecting intramural hematoma of the esophagus 5235
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S- Editor Tian L L- Editor Logan S E- Editor Zheng XM
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