Case Report
Submucosal Tunneling Endoscopic Septum Division for Congenital Pseudo-Pylorus: A New Technology in a Rare Disease (With Video)
Xinyang Liu1, Ying Fang2, Hong-Bin Yang2, Xiao-Xia Ren2, Ya-Nan Han2, Quan-Lin Li1* and Ping-Hong Zhou1*
1Department of Endoscopy, Fudan University, China
2Department of Gastroenterology, The Children’s Hospital of Xi’an City, China
*Corresponding author: Ping-Hong Zhou, Department of
Endoscopy, Fudan University, China
sh.cn
Quan-Lin Li, Department of Endoscopy
Published: 22 Oct, 2018
Cite this article as: Liu X, Fang Y, Yang H-B, Ren X-X,
Han Y-N, Li Q-L, et al. Submucosal
Tunneling Endoscopic Septum Division
for Congenital Pseudo-Pylorus: A New
Technology in a Rare Disease (With
Video). Clin Surg. 2018; 3: 2169.
Abstract
Background: Pseudo-pylorus is a congenital or an acquired septum (secondary to ulcer scaring)
in the antral area that might simulate the pylorus. It could easily cause obstruction thus requires
intervention. We report the use of Submucosal Tunneling Endoscopic Septum Division (STESD)
on a pediatric case of congenital pseudo-pylorus.
Methods: This video illustrates the STESD procedure in an 8-year-old Chinese boy. With a
congenital pseudo-pyloric stricture that occurred proximal to the original pylorus, he was suffering
from recurrent vomiting, epigastric pain and growth retardation. A gastroscopy demonstrated
pseudo-pyloric stricture with severe reflux esophagitis. STESD was performed according to the
standard protocol (step 1: mucosal incision; step 2: submucosal tunneling; step 3: septum division;
step 4: mucosal closure.) with appropriate preoperative and postoperative care.
Results: The operation time was 35minutes.Intraoperative pneumoperitoneum was relieved by
paracentesis. The pseudo pylorus disappeared and symptoms were completely relieved after the
procedure. The patient was discharged uneventfully at postoperative day 10. In four-month followup,
we observed complete symptom resolution, significant improvement of nutritional status and
no complications.
Conclusion: We suggest STESD as a safe and efficient technology in treating congenital pseudopylorus.
Validation in other benign strictures of the gastrointestinal tract is warranted.
Keywords: STESD; Pseudo-pylorus; Congenital; Endoscopic treatment
Introduction
Pseudo-pylorus is a congenital or an acquired septum (secondary to ulcer scaring) in the
antral area that might simulate the pylorus [1]. Different from the normal intermittent, ring-like
contraction of the pyloric loop, it could easily cause obstruction.
Although in some literature, the term pseudo-pylorus was sometimes confused with “double
pylorus” (also known as pyloric diaphragm, antral mucosal band and pyloric septum) [2-3], another
rare abnormality usually arising as an acquired complication of chronic peptic ulcer disease and
rarely congenital [4-8], the two terms are essentially different.
In double pylorus, the two pylori occur “in parallel”, with a gastro duodenal fistula consisting of
a short accessory channel between the distal stomach and the duodenal bulb, such that the gastric
antrum and the duodenal bulb are connected by two separate openings [4-8]. In this case, it is
asymptomatic. However, as the pseudo-pylorus occurs “in series” with the anatomic-pylorus, It is a
benign stricture that causes obstructive symptoms and thus requires intervention [1].
Herein, we report the use of a new method recently developed by our institution for the treatment
of Zenker’s diverticulum----Submucosal Tunneling Endoscopic Septum Division (STESD) [9], for
treating a pediatric case of congenital pseudo-pylorus.
Case Presentation
The procedure was performed on an 8-year-old Chinese boy who had been suffering from
recurrent vomiting, epigastric pain and growth retardation. The
past medical history revealed cryptorchism and was otherwise
unremarkable. A gastroscopy was performed and demonstrated
pyloric stricture with severe reflux esophagitis. A single-channel
gastroscope with a diameter of 9.6 mm (GIFQ 260; Olympus Medical
Systems Co. Tokyo, Japan) could not pass through the stricture.
Interestingly, after passing through the above-mentioned “pyloric”
stricture with a 5.8 mm gastroscope (GIF-XP290N; Olympus Medical
Systems Co, Tokyo, Japan), another pylorus was observed, implicating
that the previous “pylorus” was a congenital pseudo-pylorus that
occurred in series with and proximal to the original pylorus. Rapid
urease test showed that H. pylori was negative. His hemoglobin level
was low at 5.82 g/dL, and then corrected by transfusion to 9.0 g/dL
preoperatively.
A single-channel gastroscope (GIF-Q260J; Olympus Medical
Systems Co, Tokyo, Japan) was used with a hybrid knife (ERBE;
Erbe Elektromedizin GmbH, Tubingen, Germany). Carbon dioxide
(CO2) gas was used for insufflation during the procedure with a
CO2 insufflator (UCR; Olympus). A transparent cap (D-201-11802,
Olympus) was attached to gastroscope tip. Prophylactic intravenous
antibiotics were administered. STESD was performed as follows
(Video 1 and Figure 1): Step 1: Mucosal incision. Mucosal entry was
made 3 cm proximal to the stricture of the pseudo-pylorus. A 2.0 cm
longitudinal mucosal incision was performed at the tunnel entry after
injection of saline mixed with indigo carmine and epinephrine.
Step 2: Submucosal tunneling. A submucosal tunnel was created
between the mucosal and muscular layers. The tunnel was created
from the incision point till 2 cm distal to the stricture to ensure
enough working space for myotomy.
Step 3: Septum division. The thickened septal muscle fibers at the
stricture were dissected by longitudinal incisions till 2 cm beyond the
stricture.
Step 4: Mucosal closure. Mucosal closure of the tunnel entry was
made with hemostatic clips after careful hemostasis.
The procedure took 35 mins. Abdominal distention was observed
during the procedure, suggesting pneumoperitoneum, and was
relieved by paracentesis. No mucosal injury occurred. Intravenous
antibiotics were continued for one week postoperatively. Nasal
feeding started at postoperative day 7 and the patient was discharged
at postoperative day 10. The follow-up gastroscopy 4 months after
the operation confirmed the relaxation of the pseudo-pylorus and the
patient’s symptoms were relieved completely.
Figure 1
Figure 1
Submucosal Tunneling Endoscopic Septum Division (STESD) for a pediatric case of congenital pseudo-pylorus. (A) Endoscopic view of the pseudopylorus.
(B) Submucosal injection and mucosal incision 3 cm proximal to the pseudo-pylorus. (C) Creation of the submucosal tunnel and clear exposure of the
muscle fibers of the septum inside the tunnel. (D) Septum division under direct endoscopic view. (E) Closure of the mucosal tunnel entry. (F) Endoscopic view of the
relaxed pseudo-pylorus (red arrow) and the anatomic pylorus (blue arrow). (G) Follow-up endoscopic view of the relaxed pseudo-pylorus at month 4. (H) Follow-up
endoscopic view of the relaxed pseudo-pylorus (red arrow) and the anatomic pylorus (blue arrow) at month 4.
Video 1
Video 1
Submucosal tunneling endoscopic septum division for congenital pseudo-pylorus: mucosal incision; submucosal tunneling; septum division; mucosal closure
Discussion
Previous publications have reported cases with congenital or
acquired two pylori “in parallel” [4-8], known as “double pylorus”,
while pseudo-pylorus with two pylori “in series” has been barely
known [1]. In our case, the pseudo-pylorus appeared “in series” with
the anatomic pylorus, causing recurrent vomiting and subsequent
severe malnutrition due to the stricture. Therefore, surgical
intervention was necessary. Pseudo-pylorus usually forms secondary
to a peptic ulcer scar. However, this boy had no history of peptic
ulcer, and gastroscopy also detected no H. pylori infection or peptic
ulcer scar, suggesting the congenital origination of this condition.
The pathogenesis of this condition is unknown and we propose
the imbalance between contraction and relaxation of the circular
muscular fibers might be one explanation.
STESD is a novel technique recently developed by our institution
for the treatment of Zenker’s diverticulum [9]. It was inspired by the
peroral endoscopic myotomy (POEM) technique [10]. Theoretically,
it could release the stricture of the gastrointestinal tract with
maintenance of mucosal integrity when selectively dividing the
muscular septum, which in this case equals the thickened circular
muscular fibers at the pseudo-pylorus. Therefore, it advantages in
possibly reducing the risk of postoperative perforation and secondary
infection. Complete division of thickened muscles and protection of
the gastric wall is also facilitated by adequate exposure and enough
working space by the submucosal tunnel. In this pediatric case, we
believe it is better than balloon dilation, which is not long-lasting
and usually requires repeated procedures. In the meantime, it is also
less invasive than an open surgery. To our knowledge, this is the first
application of STESD in this rare disease. Besides pseudo-pylorus,
STESD is also potentially applicable in other conditions of benign
stricture of the gastrointestinal tract.
Acknowledgement
This study was supported by the grants from the National Natural Science Foundation of China (81470811, 81570595, and 81670483), Major Project of Shanghai Municipal Science and Technology Committee (16411950400), Outstanding Young Doctor Training Project of Shanghai Municipal Commission of Health and Family Planning (2017YQ026) and Shanghai Sailing Program of Shanghai Municipal Science and Technology Committee (18YF1403700).
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