Introduction
Gastric varices (GVs) are a significant complication of portal hypertension in children.
Endoscopic injection of glue is considered the standard for management of gastric
variceal hemorrhage with a high hemostasis rate [1]. However, rebleeding due to necrosis and ulceration at the sclerosing site or late
bleeding secondary to an incomplete obliteration or extrusion of glue is described
in 10 % to 14 % of patients [2]
[3].
Available endoscopic options for recurrent and rebleeding GVs are scarce. Endoscopic
ultrasound (EUS)-guided placement of coils represents a new technique for management
of GV bleeding in these patients [4]. Nevertheless, there are no reports on use of this technique in pediatric patients.
In this case report, we detail EUS-guided coil placement with cyanoacrylate injection
for management of gastric variceal bleeding after two failed endoscopic cyanoacrylate
injections in a 12-year-old patient.
Case report
A 12-year-old child with the history of cryptogenic liver cirrhosis who was awaiting
a liver transplantation was admitted to our unit due to recurrent upper gastrointestinal
bleeding. The Model End-Stage Liver Disease (MELD) score of the patient was nine points.
Despite two endoscopic sessions of glue injection with N-2 butyl-cyanoacrylate injection
at another center, gastroesophageal varix (GOV) type I (Sarin and Kumar classification)
remained ([Fig. 1]). On admission, after adequate fluid resuscitation, an intravenous infusion of 0.02 mg/kg
terlipressin was maintained. The patient’s hemoglobin level was 8.4 mg/L. Her liver
and renal function tests were within normal limits.
Fig. 1 Endoscopic view of type 1 gastroesophageal varices in accordance to the Sarin classification.
After obtaining informed consent from her parents within 24 hours of hemodynamic stability,
and
due to the patient’s history of nonresponse to cyanoacrylate injection via upper endoscopy,
she
underwent EUS-guided placement of coils with cyanoacrylate injection using a linear
array
echoendoscope (Pentax EG-3870UTK) attached to a Hitachi Avius ultrasound console ([Video 1]). The endoscopic procedure was performed with the patient under general anesthesia
with tracheal intubation and mechanical ventilation.
Video 1 Endoscopic ultrasound-guided placement of coils and cyanoacrylate embolization in
refractory gastric variceal bleeding.
After identification of GV via EUS Doppler flow ([Fig. 2]), 200 mg of intravenous ciprofloxacin was given as antibiotic prophylaxis. Transesophageal
EUS-directed intravascular puncture of the GV was performed using a 19G-FNA needle
(Expect; Boston Scientific, Marlborough, Massachusetts, United States) and two 10-mm
Nester Embolization Coils (Cook Medical, Bloomington, Indiana, United States) were
deployed, followed by injection of 1.2 mL of 2-Octyl-CYA (Dermabond; Ethicon, Piscataway,
New Jersey, United States) ([Video 1]). Absence of flow during EUS Doppler evaluation of the index gastric varices after
the procedure was noted ([Fig. 3]).
Fig. 2 Endoscopic ultrasound evaluation of gastric variceal flow using color Doppler.
Fig. 3 Absence of flow during EUS Doppler evaluation of the index gastric varices after
EUS-guided coiling and cyanoacrylate injection.
Primary endoscopic hemostasis was achieved with normalization of vital signs and no
hemoglobin drop, need for blood transfusion, or incidence of hematemesis or melena,
or rebleeding within 48 hours of gastric varix obliteration. On follow-up via upper
endoscopy and EUS evaluation 1 month after the index procedure, complete eradication
of the GV was observed. Neither rebleeding nor need for reintervention occurred during
the 12-month follow-up period ([Fig. 4]).
Fig. 4 Endoscopic view of the gastric varices 12 months after EUS-guided therapy.
Discussion
Acute variceal hemorrhage secondary to rupture of gastric varices is a life-threatening
event in children due to the volume and severity of bleeding, with high rebleeding,
mortality, and morbidity rates. Management of GV hemorrhage is challenging, requiring
a high level of expertise in endoscopy and interventional radiology techniques. Studies
regarding management of GV bleeding in the pediatric population are limited.
Endoscopic gastric varix obliteration with direct injection of N-butyl-2-cyanoacrylate
into the varix, transjugular intrahepatic portosystemic shunt (TIPS), and balloon-occluded
retrograde transvenous obliteration (B-RTO) are alternatives for treatment of GV hemorrhage;
however, these techniques require skillful endoscopic and radiological expertise.
Moreover, cyanoacrylate injection through standard gastroscopes is associated with
a 25 % to 50 % rebleeding rate [5], and even death secondary to glue embolism has been reported [6]
[7].
The Baveno VI consensus suggests use of N-butyl-2-cyanoacrylate for treatment of IGV
and GOV-2, but no definitive recommendations exist, and data regarding the safety
and efficacy of N-butyl-2-cyanoacrylate in management of bleeding gastric varices
in children are scarce [8]
[9]. After failed obliteration of GV with endoscopic injection of cyanoacrylate, alternatives
are limited in the pediatric population. EUS-guided transesophageal-transcrural variceal
targeting with combined coil and 2-octyl-cyanoacrylate embolization represents a novel
approach in adults with excellent results [10]. This approach was safe and feasible in this pediatric case, without any reported
adverse events, and complete obliteration and varix disappearance was achieved [5].
Emergent shunt surgery, TIPS, and B-RTO are not available options in our center, and
hemostasis in our patient was imperative. We preferred 2-octyl-cyanoacrylate over
N-butyl-cyanoacrylate because the former has a similar efficacy for hemostasis but
also has longer polymerization time, allowing a longer injection time and reducing
risk of endoscope damage. It also does not require dilution with Lipiodol (which makes
injection more difficult due to viscosity) [8].
EUS-guided embolization with coils and cyanoacrylate might be more beneficial because
less glue is required, reducing risk of embolization and increasing the procedure
success and obliteration rate in adults. No AEs were reported after the procedure
or during the 12-month follow-up period in this patient. EUS-guided coil and cyanoacrylate
embolization was a safe and effective technique for treating GV hemorrhage in this
pediatric patient and might be considered as rescue therapy for GV hemorrhage in pediatric
cases.
