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Received: 12 April 2021    Accepted: 5 July 2021

DOI: 10.1111/petr.14105

CASE REPORT

A novel technique for large lateral enterocutaneous fistula

management after intestinal transplantation

Agustina R Oliva1  | Paula Violo Gonzalez1 | Luciana Lerendegui1 |

Rodrigo Sanchez Clariá2 | Juan Moldes1 | Pablo Lobos1,2

1
Department Pediatric Surgery, Hospital
Italiano de Buenos Aires, Buenos Aires, Abstract
Argentina
Background: Enterocutaneous fistula (ECF) is a serious and complex problem when
2
Center for Rehabilitation and Transplant,
Hospital Italiano de Buenos Aires, Buenos
affecting children, being responsible for a high morbidity burden, with an estimated
Aires, Argentina mortality rate of 10 to 20%. There are many therapeutic options, including surgery

Correspondence
Oliva Agustina René, Peron 4190, Ciudad
Autonoma de Buenos Aires, Buenos Aires
C1199ABB, Argentina.
Email: rene.oliva@hospitalitaliano.org.ar
and a wide variety of nonoperative strategies. Prognosis of ECF closure depends on
the output and also on the patency of distal bowel. Spontaneous closure without
operative intervention occurs in approximately 50% of patients with lateral ECF and
distal bowel transit, but this drastically decreases in high output fistulas. High-­volume
fistula output and consequent skin damage are a great challenge for the health-­care
team.
Methods: We describe a postoperative complication that required a new nonopera-
tive technique for the transient management of a lateral high-­output ECF, involving
the insertion of an occlusive device in order to redirect intestinal content to the distal
bowel, reducing the fistula output.
Results and conclusions: The main benefit of this nonoperative technique is the abil-
ity to occlude a high-­output fistula, allowing the distal flow to be restored and reduc-
ing abdominal wall damage, as a bridge to definitive surgical closure.

KEYWORDS
enterocutaneous lateral fistula, fistula occlusion, high-­output fistula

1  |  I NTRO D U C TI O N Treatment of ECF represents a great challenge for the surgeon,

ECF is a serious and complex problem for affected children. They are
responsible for a high morbidity burden, with an estimated mortality
rate of 10 to 20% 1.
An ECF is an aberrant connection between the intra-­abdominal
gastrointestinal (GI) tract and the skin or wound 2. Most of them
are acquired, secondary to a surgical trauma. 3. Complications such
as anastomotic dehiscence, unnoticed perforations, multiple reop-
erations, and the resultant hostile abdomen are responsible for the
genesis of ECF.
and affected patients should be approached by an experienced mul-
tidisciplinary team 4 5. A common acronym used to describe ECF care
protocol is “SNAP,” which stands for management of skin and sepsis,
nutrition, definition of fistula anatomy, and proposing a procedure to
manage the fistula 2.
Prognosis of ECF closure depends on the output and also on the
patency of distal bowel. Spontaneous closure without operative in-
tervention occurs in approximately 50% of patients with lateral ECF
and distal bowel transit, but this drastically decreases in high-­output
fistulas 6. There are many treatment alternatives proposed for ECF

Abbreviations: BN, bottle nipple; ECF, enterocutaneous fistula; VAC, vacuum-­assisted closure.

Pediatric Transplantation. 2021;00:e14105. wileyonlinelibrary.com/journal/petr |

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management, including surgery and a wide variety of nonoperative

strategies. Among them, vacuum-­assisted closure (VAC) has been
widely used 3, 4, 7. On the other hand, stenting the fistulous tract
with different devices (Foley tubes, endotracheal tubes, Penrose
drainage, condoms) in order to divert the content to an external
reservoir without damaging the surrounding skin has also been de-
scribed as useful methods 4.
Once the fusion between the intestinal mucosa and the ab-
dominal wall occurs, expectation of spontaneous fistula closure is
markedly reduced, being surgery the only treatment option in most
of the cases. However, unfavorable patient clinical status or local
F I G U R E 1  Fistula occlusion device consisting of a 6-­French
abdominal conditions (such as a hostile abdomen secondary to pre-
Foley tube and a silicone bottle nipple. A balloon is inflated to fit
vious operations or complications) may cause a significant delay in exactly on top of the bottle nipple for traction (red arrow)
scheduling of the surgical procedure. In these cases, high-­volume
fistula output and consequent skin damage are a great challenge for
the health-­care team. We describe a new nonoperative technique
for the transient management of a lateral high-­output ECF, involving
the insertion of an occlusive device in order to redirect intestinal
content to the distal bowel, reducing the fistula output.

2  |  C A S E D E S C R I P TI O N

A 12-­year-­old boy with chronic intestinal failure secondary to short

bowel syndrome underwent an isolated small bowel transplant. The
graft was resected four months after transplantation, due to severe
F I G U R E 2  Scheme of the device inserted inside the fistula in its
rejection. Bowel continuity was achieved by a side-­to-­side anasto-
final position. A, The bottle nipple coming out through the fistula.
mosis between the native duodenum and the transverse colon. Early B, Bottle nipple's base against the bowel wall to ensure occlusion.
anastomotic leak was treated by reoperation, but the anastomotic C, Foley catheter pulling up the bottle nipple to ensure adequate
dehiscence recurred, and nonoperative treatment with VAC was in- occlusion pressure. D, Patent flow of enteric content through the
dicated. Closure of the leak was not achieved by this method, result- fistula site
ing in a high-­output ECF (1000 –­1500 ml per day) located on the
lateral side of the duodeno-­colic anastomosis with a complete dehis-
cence of the surgical incision (Figure 4a). After multiple unsuccessful
attempts to manage the fluid output and the severely damaged skin,
a novel occlusive system was designed, using a silicone bottle nipple
(BN) that was inserted in the fistula (Figure 1).
A silicon BN and a 6-­Fr silicone Foley catheter were used to
design a handmade system to be introduced in the fistula opening
without occluding the distal bowel end. It was inserted through the
fistula, with the wider side of the BN inside the bowel. After giving
a gentle traction, a complete occlusion was achieved (Figure 2). The
Foley catheter was inserted through a small opening at the tip of
the BN, and the balloon inflated inside it (Figure 1). Traction of the
Foley tube allows the BN to be constantly pulled against the abdom-
inal wall to ensure persistent occlusion. Additionally, the device is
fastened with non-­absorbable suture through the BN and fixed to
the skin with adhesive tape. VAC may be maintained (Figure 3) to
enhance wound and skin recovery (Figure 4b).
After a few days, the wound VAC system was no longer needed as
adequate sealing was obtained, with no need of replacing the device F I G U R E 3  Immediate postinsertion view, the Foley tube (white
for 4 weeks. As a result, fistula output dropped to zero, and distal arrow) with the VAC (black arrow) placed. GJ tube on the left (head
bowel transit was restored, with 3 to 4 daily stools at last follow-­up of arrow)
OLIVA R et al.
F I G U R E 4  A, Postoperative wound
dehiscence with enteroatmospheric fistula (A)
(white arrow), surrounded by granulation
tissue. B, One month later, complete
healing of the wound and skin recovery
with absence of fluid leakage from the
fistula is observed
visit. Oral feeding was re-­started with significant weight gain and
a total recovery of the damaged skin. The patient was discharged
home after six months, with both enteral and parenteral nutrition. A
clear improvement of quality of life was achieved. After one month
of its placement, the patient developed a small lateral decubitus fis-
tula. At that moment, reconstructive surgery was performed, and
both fistulas were successfully closed.
3  |   D I S C U S S I O N
This case represented a challenge for the team, and after having tried
all the available options, we used a low-­cost technique designed for
obturation of lateral ECF in patients with high intestinal fluid output.
There is a direct relationship between high location of the fistula
with sepsis, malnutrition, and severe electrolyte disbalance, duode-
nal fistulas being the most difficult to treat. Requirement of surgical
closure and a high patient morbidity and mortality are the rule 6.
Evenson in 2006 reported that spontaneous closure occurs
only in 30% of ECF 5, but 90–­95% of those fistulas that will resolve
without surgery will do so within the first 4 to 5 weeks 1. Different
unsuccessful treatment strategies were used in our patient, with
progressive clinical worsening. The management continued to be
challenging until the newly designed device provided a temporary
solution, allowing the damaged skin to heal and restarting enteral
feedings and contributing to weight gain.
Vacuum-­assisted closure is a frequently used strategy to enable
nonoperative closure, providing distal bowel patency can be demon-
strated 7. However, this strategy was not enough to manage the high
output fistula in our case.
Specially after failed operative fistula closure procedures, imme-
diate reoperation may not be recommended, depending on the pa-
tient's clinical status. While waiting for these conditions to improve,
daily care has a significant impact on the patient's quality of life.
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(B)
As mentioned, a wide variety of treatment options have been
described to overcome these challenges. These strategies are based
on two main objectives: reduction in the volume of fluid production
through pharmacologic treatment and suspension of oral feedings,
and diversion of the fistula output to a reservoir. The introduction
of drugs like proton pump inhibitors, glycopyrrolate, or somatosta-
tin is useful for overcoming the spoliation of fluid and electrolytes
8
. Although many of these alternatives have proved to be effective
in this setting, they are often not enough to achieve clinical stabil-
ity. Furthermore, diversion or collection of the fistula output, over
a scarred belly and a persistently ulcerated skin, may be difficult to
sustain and requires a significant effort of the health-­care team.
The previously reported techniques have shown promising re-
sults in different situations, but most of them are not universally
applicable. In our opinion, identification of the best strategy in
every individual case is key for a successful outcome. The proposed
technique here has been applied in a very unique situation, but this
principle may be useful to be applied in similar ECF or other clinical
settings. The main benefit of this nonoperative technique is the abil-
ity to occlude a high-­output fistula in patients who did not respond
to other treatments, allowing the distal flow to be restored and re-
ducing abdominal wall damage.
A potential risk for this device could be contact ulceration in the
adjacent bowel mucosa, as occurred in our patient. Probably it could
have been avoided if the device had been replaced or repositioned
in order to reduce continuous pressure on a particular contact zone
with the intestinal mucosa.
Another possible complication could be the BN migration inside
the bowel lumen; a double secure mechanism (the Foley tube and
the fixation suture) was used to prevent this.
An additional disadvantage is the temporary nature of the solu-
tion provided. However, this device can help delay the need for sur-
gery, allowing the patient to recover a good clinical and nutritional
condition and reducing postoperative complications of fistula closure.
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4 of 4       OLIVA R et al.
We believe that this technique provides a useful option for sur-
geons dealing with difficult cases of ECF. Additionally, this may be an
option to manage patients with a hostile abdomen while on waiting
list for intestinal transplantation.
AC K N OW L E D G E M E N T S
We would like to express our very great appreciation to Prof.
Daniele Alberti for his valuable and constructive suggestions in the
manuscript.
C O N FL I C T O F I N T E R E S T
None.
AU T H O R C O N T R I B U T I O N S
Oliva Agustina R conceived of the presented idea. Oliva Agustina R,
Violo G. Paula, and Lerendegui Luciana developed the theory and
performed the computations. Lerendegui Luciana and Lobos Pablo
verified the analytical methods. Sanchez Clariá Rodrigo, Moldes
Juan, and Lobos Pablo encouraged Oliva Agustina R to investigate
different techniques for enterocutaneous fistula management and
supervised the findings of this work. All authors discussed the re-
sults and contributed to the final manuscript.
DATA AVA I L A B I L I T Y S TAT E M E N T
There is no data statement.
ORCID
Agustina R Oliva  https://orcid.org/0000-0002-6147-9567
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How to cite this article: Oliva AR, Violo Gonzalez P,
Lerendegui L, Sanchez Clariá R, Moldes J, Lobos P. A novel
technique for large lateral enterocutaneous fistula
management after intestinal transplantation. Pediatr
Transplant. 2021;00:e14105. https://doi.org/10.1111/
petr.14105