Hiroyoshi Iwagami

Satoshi Yokoyama

Shiro Uyama

Yukitaka Yamashita

Springer - Publisher Connector

CASE REPORT Open Access
Successful combination of endoscopic
pyloromyotomy and balloon dilatation for
hypertrophic pyloric stenosis in an older
child: A novel procedure
Satoshi Yokoyama1*, Shiro Uyama1, Hiroyoshi Iwagami2 and Yukitaka Yamashita2
Abstract
Background: Hypertrophic pyloric stenosis (HPS) is a rare cause of gastric outlet obstruction beyond infancy.
Ramstedt pyloromyotomy remains the gold standard treatment for HPS. This type of HPS can be managed
successfully with pyloromyotomy under laparoscopic or open procedures. Endoscopic pyloric balloon dilation
(EPBD) has not been recommended in the treatment of HPS, and there are only a small number of reported
cases who had had successful endoscopic pyloromyotomy (EP) for HPS only in infants.
Case presentation: The patient was suspected of having HPS when the patient was 1 year old after infancy.
However, his parents thought that the vomiting and poor sucking were caused by Down syndrome-associated
muscular hypotonia. Since then, no additional tests have been performed at their request. At 6 years of age, he
was readmitted to our department because of persistent vomiting and failure to thrive, and HPS was diagnosed
again. However, it was unknown whether the HPS had been persisting since infancy or was acquired. The first
EPBD was slightly effective but did not remain effective for a long time. When the second EPBD was performed
in combination with EP, the amount and frequency of vomiting were reduced dramatically.
Conclusions: The combination of EP and EPBD procedure may represent a safe, effective, and minimally invasive
option for selected HPS patients in whom laparotomy would pose a significant risk or who do not respond to
conventional medical treatment. To our knowledge, this is the first report to describe combination treatment
with EP and EPBD in an older child with HPS.
Keywords: Hypertrophic pyloric stenosis (HPS), Endoscopic pyloromyotomy (EP), Endoscopic pyloric balloon
dilation (EPBD)
Background
Hypertrophic pyloric stenosis (HPS) is a disorder that
usually presents with projectile vomiting in infants. The
hypertrophy of the pylorus can progress to near-complete
obstruction of the gastric outlet [1, 2]. The Ramstedt pylor-
omyotomy remains the standard operation for HPS, with a
laparoscopic approach favored because of the shorter
recovery time and lower rate of wound complications
[3–5]. Endoscopic pyloric balloon dilation (EPBD) has
not been recommended in the treatment of HPS because
of reports of its ineffectiveness [6]. However, successful
dilation in selected cases of HPS has also been reported
[7], as have a number of recent case series of successful
endoscopic pyloromyotomy (EP) for HPS in infants have
been reported [8, 9].
Here, we report a case of HPS in an older child success-
fully treated with a combination of EP and EPBD. To our
knowledge, this is the first report to describe combin-
ation treatment with EP and EPBD in an older child
with HPS.
* Correspondence: yokoyama.st@gmail.com
1Department of Pediatric surgery, Japanese Red Cross Society, Wakayama
Medical Center, 4-20 Komatsubara-dori, Wakayama 640-8558, Japan
Full list of author information is available at the end of the article
© The Author(s). 2016 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0
International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and
reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to
the Creative Commons license, and indicate if changes were made.
Yokoyama et al. Surgical Case Reports  (2016) 2:145 
DOI 10.1186/s40792-016-0274-y
Case presentation
The patient was diagnosed with Down syndrome after
birth. At the age of 4 months, he began to experience
frequent non-bilious vomiting along with decreased
weight gain. His parents did not want him to undergo
any diagnostic tests before he became a year old.
At 1 year of age, he was admitted to our department
with long-standing multiple episodes of non-bilious vomit-
ing along with poor weight gain. An upper gastrointestinal
(UGI) series showed no evidence of gastroesophageal reflux
disease (GERD). However, obstruction at the pylorus, which
met the criteria for hypertrophic pyloric stenosis (HPS),
was present, and gastric emptying was significantly delayed.
Abdominal ultrasonography (US) showed the muscle layer
to be 3-mm-thick, and the pylorus to be 15-mm-long.
However, his parents thought that the vomiting and poor
sucking were caused by Down syndrome-associated
muscular hypotonia and did not want the patient to
undergo any tests. Therefore, the patient was observed and
given only conservative treatment with oral H2 blockers
and enterokinetic drugs. Non-bilious vomiting along with
poor weight gain persisted for 5 years.
At 6 years of age, he was readmitted to our department
because of persistent vomiting and failure to thrive. Weight
was 10.3 kg (−3 SD), height was 94.7 cm (−4.1 SD). His
laboratory analyses showed almost normal range. Gastric
outlet obstruction (GOO) was observed on an UGI series
(Fig. 1a). US showed the muscle layer to be 5.6-mm-thick,
and the pylorus to be 15-mm-long (Fig. 1b). An esophago-
gastroduodenoscopy (EGD) demonstrated mild erythema
and marked thickening of the distal esophageal mucosa,
consistent with reflux esophagitis, and a pin-hole open-
ing in the pylorus that did not allow the passage of an
endoscope (Fig. 1c). Finally, the patient was diagnosed
with HPS. However, it was unknown whether the HPS
had been persisting since infancy or was acquired.
Ramstedt pyloromyotomy is generally performed in in-
fants and is seldom performed in six-years-old children.
Therefore, it was unknown whether or not Ramstedt pylor-
omyotomy was definitely effective. At first, we attempted
EPBD because the patient obviously had Down syndrome-
associated muscular hypotonia in addition to chronic re-
spiratory infection. Under general anesthesia, the pediatric
endoscope could not be passed through the pylorus. A
balloon catheter was introduced transorally into the
stomach under fluoroscopic guidance and guided into
the pyloric canal. Catheter balloons (Hercules® 3 Stage
Wire Guided Balloon, Cook Medical, Winston-Salem NC,
USA) of increasing diameters (during the first balloon
dilatation, the balloon was gradually inflated to 8 mm,
12 mm, and 15 mm) were inserted through the biopsy
channel of a 9.9 mm endoscope (GIF Q260J®, Olympus
Fig. 1 a UGI series showed dilated stomach with no contrast seen distally. b US showed the muscle layer to be 5.6 mm thick, and the pylorus to
be 15 mm long. c EGD demonstrated mild erythema of the distal esophagus, markedly enlarged, thickened, and the pin-hole opening pyloric
stenosis that did not allow the passage of an endoscope
Yokoyama et al. Surgical Case Reports  (2016) 2:145 Page 2 of 5
Optical Co.) and inflated with the use of a pressure
gauge system for 60–120 s. The endoscope was able to
be easily passed through the pylorus after the third
session. After the first balloon dilatation, vomiting
gradually decreased in frequency and amount. How-
ever, it recurred after a period of 2 months post-dilation.
Repeat UGI series showed that the pyloric stenosis was not
improved.
Six months after the initial dilation, we treated the
patient with a combination of EP and EPBD. Under gen-
eral anesthesia, the same endoscope (GIF Q260J, Olympus
Optical Co., Ltd., Tokyo, Japan) was used. The pylorus did
not allow passage of the endoscope. Pyloromyotomy was
performed with an electrosurgical needle knife (KD-611L
IT Knife2®, Olympus Optical Co.) and an electrosurgical
generator (VIO® 300 D, Erbe USA, Marietta CA, USA),
with settings of cut 30 and blend 3. Two incisions were
made in the pylorus along the antimesenteric border from
the antrum to the duodenal bulb (Fig. 2a). The incisions
were deepened (2 mm) until longitudinal muscle was
exposed. EPBD was then performed in the same manner
and with the same devices as the procedure for initial
balloon dilation (Fig. 2b). The muscle layer was slowly
loosened and split bluntly along the incisions. Compres-
sion with the balloon dilator provided good hemostasis
(Fig. 2c, d). At the end of the procedure, hemostasis was
confirmed. Introduction of water-soluble radiographic
contrast confirmed free flow across the pylorus. Thereafter,
the endoscope was easily passed through the pylorus into
the duodenum. Feeding with clear liquids was begun 2 h
after the procedure. The patient was discharged the next
day. The second EPBD dramatically reduced the amount
and frequency of vomiting, and repeat UGI series showed
that the pyloric stenosis was improved (Fig. 3). The patient
was doing well and had gained weight on follow-up at
6 months.
Review of the English-language literature (Table 1)
A PubMed search on November 2016 for articles pub-
lished since 1965 with the key words hypertrophic pyloric
stenosis, endoscopic pyloromyotomy, and balloon dilatation
yielded only four articles in the English-language litera-
ture. These publications were all reviewed. Only two
papers describing 19 cases of EP for HPS were found.
Ibarguen-Secchia et al. reported ten infants who underwent
EP without any adverse events [8]. Zhang et al. reported
nine cases in China; one patient underwent a second EP
because of recurrent postoperative vomiting [9]. Two
papers described seven cases of EPBD for HPS. Hayashi
et al. reported six cases; however, EPBD for HPS was
not reliable or safe because of the inadequate split and
Fig. 2 a Two incisions were made in the pylorus along the antimesenteric border from the antrum to the duodenal bulb with an electrosurgical
needle knife. b EPBD was then performed in the same manner and with the same devices as the procedure for initial balloon dilation. c, d The
muscle layer was slowly loosend and split bluntly along the incisions. Compression with the balloon dilator also provided good hemostasis
Yokoyama et al. Surgical Case Reports  (2016) 2:145 Page 3 of 5
possible full-thickness disruption of the pylorus [6].
Ogawa et al. reported a case of successful EPBD for
HPS [7].
Discussion
The combination of EP and EPBD may represent a safe,
effective, and minimally invasive option for HPS. HPS is
the most commonly encountered surgical disease in chil-
dren. Ramstedt pyloromyotomy remains the gold standard
treatment for HPS [1–3]. Laparoscopic pyloromyotomy is
a minimally invasive version of the Ramstedt procedure
that has been associated with a lower incidence of postop-
erative emesis and a shorter hospital stay, but occasionally
results in incomplete pyloromyotomy [4, 5]. Because data
on conservative management is very limited, and because
surgery is both safe and effective, it is suggested that con-
servative management should only be indicated in infants
in whom a surgical approach is either not advisable or
feasible [2].
Endoscopic or image-guided balloon dilation of the
pylorus has been also described for patients with HPS.
However, preliminary reports documented an unacceptably
high rate of both perforation and failure to relieve the ob-
struction [7], limiting its utilization on wide scale. Like the
use of atropine, it may still have a role in children with a
significant contraindication to surgical pyloromyotomy.
Recently, however, a few cases series of successful EP
have been reported for HPS in infants (Table 1). Al-
though the outcome of balloon dilatation for HPS in
infants is not preferable, it has been reported that bal-
loon dilatation is effective for gastric outlet obstruction
(GOO) and pyloric stenosis (PS) in adults or older chil-
dren [10–14]. However, the combination of EP and EPBD
for HPS has not been reported on.
In our case, the patient was not an infant but an older
child. The patient was suspected of having HPS when
the patient was 1 year old after infancy. However, it was
unknown whether the HPS was congenital or acquired.
Therefore, it was unknown whether or not Ramstedt
pyloromyotomy was definitely effective. The first EPBD
was slightly effective but did not remain effective for a
long time. When the second EPBD was performed in
combination with EP, the amount and frequency of
vomiting were reduced dramatically. We suggest that
the combination of EP and EPBD may be also superior
to open and laparoscopic pyloromyotomy for selected
HPS. Therefore, we expected that the combined ther-
apy would be effective. It is difficult to conclusively
prove that the use of the balloon along with the pylor-
ectomy made any difference.
Conclusions
To our knowledge, however, no study has reported
successful treatment of HPS with combination of EP
plus EPBD in an older child. This combination pro-
cedure may represent a safe, effective, and minimally
invasive option for selected HPS patients in whom
laparotomy would pose a significant risk or who do
not respond to conventional medical treatment. Given
the patient’s good condition and absence of any clin-
ical gastric outlet symptoms, confirmation of the reli-
ability of this combination method for selected HPS
cases requires more experience with the procedure
and long-term follow-up.
Fig. 3 UGI series showed smooth passage of contrast through
the pylorus
Table 1 Overview of reports on non-surgical techniques for HPS
Author, reference n Age Type of non-surgical techniques Complication Outcome
[6] 6 4–7 weeks EPBD None No successful dilatation
[7] 1 3.5 months EPBD Redo needed Successful
[8] 10 3–7 weeks EP None Successful
[9] 9 4–10 weeks EP 1 redo needed Successful
Our case 1 6 years EP plus EPBD None Successful
EPBD endoscopic pyloric balloon dilatation
EP endoscopic pyloromyotomy
Yokoyama et al. Surgical Case Reports  (2016) 2:145 Page 4 of 5
Abbreviations
EGD: Esophagogastroduodenoscopy; EP: Endoscopic pyloromyotomy;
EPBD: Endoscopic pyloric balloon dilation; HPS: Hypertrophic pyloric stenosis;
UGI: Upper gastrointestinal
Authors’ contributions
SY made the conception and design of this case report. Authors other than
SA contributed to the collection, analysis, and interpretation of the data. SA
wrote the draft manuscript, and other authors performed the critical revision
of the manuscript. All authors gave final approval of the version to be published.
SA has overall responsibility and guarantees the scientific integrity.
Competing interests
The authors declare that they have no competing interests.
Consent for publication
Written informed consent was obtained from the patient for the publication
of this case report and any accompanying images. A copy of the written
consent is available for review by the Editor-in-Chief of this journal.
Author details
1Department of Pediatric surgery, Japanese Red Cross Society, Wakayama
Medical Center, 4-20 Komatsubara-dori, Wakayama 640-8558, Japan.
2Department of Gastroenterology and Hepatology, Japanese Red Cross
Society, Wakayama Medical Center, 4-20 Komatsubara-dori, Wakayama
640-8558, Japan.
Received: 4 October 2016 Accepted: 1 December 2016
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Successful combination of endoscopic pyloromyotomy and balloon dilatation for hypertrophic pyloric stenosis in an older child: A novel procedure

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Successful combination of endoscopic pyloromyotomy and balloon dilatation for hypertrophic pyloric stenosis in an older child: A novel procedure

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