International Journal of Surgery Case Reports 114 (2024) 109182

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International Journal of Surgery Case Reports

journal homepage: www.elsevier.com/locate/ijscr

Case report

Umbilical Littre hernia: A rare case report of an acute abdomen

Mohamed Ben Khalifa *, Amel Ben Belaid, Mossaab Ghannouchi, Karim Nacef, Mahmoud Fodha,
Moez Boudokhane
General surgery Department Tahar sfar hospital Mahdia, Faculty of Medecine, University of Monastir Tunisia, Tunisia

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction: Littre's hernia (LH) is due to the presence of a Meckel's diverticulum (MD) in a hernial sac. It is an
Case report extremely rare condition in less than 1 % of all MD cases. It is often asymptomatic and is diagnosed incidentally
Littre hernia during routine surgery for hernia repair. Surgery is the main treatment. Although the management of uncom­
Small bowel obstruction
plicated MD remains controversial, MD's management in the setting of either bowel obstruction or of Littre's
Surgery
hernia is done according to the clinical principles combined with the availability of local expertise.
Presentation of case: We report a case of an 11-year-old boy presented to the emergency room with a diagnosis of
acute obstructive syndrome. Radiological exams were inconclusive. Intra-operatively findings showed a stran­
gulated MD trapped in a small umbilical hernia. A simple wedge resection, followed by a primary closure of the
remaining defect was performed. During 12 months of observation the patient remains in good condition.
Discussion: LH is an uncommon type of abdominal wall hernia. Preoperative diagnosis is difficult.
Even abdominal ultrasound and computed tomography (CT) cannot reveal the right diagnosis and it is generally
performed intraoperatively. The main treatment is surgery. Repair of a Littre hernia requires both management
of Meckel's diverticulum and repair of the hernia with sutures or mesh.
Conclusion: LH is a very rare type of hernia. Diagnosis is very difficult. All surgeons should be aware of this type
of hernia to avoid life-threatening complications. The application of hernia repair recommendations for children
may anticipate the happening of complicated LH.

1. Introduction
Hernia (LH) is due to the presence of a Meckel's diverticulum (MD) in
a hernial sac [1]. LH usually result in an inguinal or femoral hernia and
rarely in an umbilical hernia. It's an extremely rare condition with an
unknown incidence [2]. A recent systematic review identified only 53 of
these hernias published in the literature, most commonly in the groin
(73 %) [3]. It is often asymptomatic and is diagnosed incidentally during
routine surgery for hernia repair. When complications arise, LH may
present as a small bowel obstruction, hernial strangulation, or acute
abdomen. Preoperative diagnosis is difficult and there are no specific
radiological signs [1]. Surgery is the main treatment. Although the
management of uncomplicated MD remains controversial, MD‘s man­
aging in the setting of either bowel obstruction or of Littre's hernia is
done according to clinical principles combined with the availability of
local expertise [2,4]. In this work, a case of umbilical LH discovered
during emergency management of obstructive syndrome, in an 11-year-
old boy, was presented. This case is reported according to the 2020
SCARE guidelines [5].
2. Presentation of case
An 11-year-old boy presented to the emergency room with a diag­
nosis of acute obstructive syndrome resulting in vomiting, abdominal
bloating, and abdominal pain for two days. He had no history of hernia
or other illness. On physical examination, he had normal vital signs, a
fever of 38.5 ◦ C, umbilical swelling of 1.5 cm without evident hernia,
abdominal distention, and mild tenderness in the lower right abdomen
with no herniation or obvious peritoneal signs. There were no inflam­
matory signs, edema, or bump in the umbilicus except mild pain. Blood
tests showed a white blood cell count of 21 K cells/mm3 and a C-reactive
protein of 71 mg/dl. Post-ileal Appendicitis was initially suspected, and
abdominal ultrasound was performed without any conclusive results. An
abdominal computed tomography (CT) scan was ordered and showed

* Corresponding author at: General Surgery Department Tahar sfar hospital Mahdia, Faculty of Medicine, University of Monastir Tunisia, Route Skanes avenue Ali
Bourguiba, 5000 Monastir, Tunisia.
E-mail address: mbk.surg@gmail.com (M.B. Khalifa).

https://doi.org/10.1016/j.ijscr.2023.109182
Received 1 November 2023; Received in revised form 14 December 2023; Accepted 16 December 2023
Available online 21 December 2023
2210-2612/© 2023 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
M.B. Khalifa et al.
distension of the small intestine and a doubtful presence of a right
paramedian feces sign with no information about appendix. The patient
underwent surgery by midline approach. Cefazolin was administered to
cover the surgery. Explorations found a liquid in the peritoneal cavity
with a strangulated MD distended and inflamed in a small umbilical
hernia sac (Fig. 1). The content was mobilized out of the sac; the
diverticulum measured 4 cm and was located on the anti-mesenteric
edge approximately in 50 cm from the ileocecal junction (Fig. 2). The
diverticulum was managed by a simple wedge resection, followed by a
primary closure of the remaining defect. The hernia which measured 3
cm was closed as the midline by continues slowly absorbable sutures.
The postoperative course was uneventful, and the patient was dis­
charged on the fourth postoperative day. The histopathological evalu­
ation revealed a diverticulum with a necrotic apex and an inflamed base
without masses or lesions inside the lumen. During 12 months of
observation the patient remains in good condition.
Fig. 1. Strangulated MD (a) founded in a small hernia sac (b) through the umbilical ring (black arrow).
2
International Journal of Surgery Case Reports 114 (2024) 109182
3. Discussion
LH is an exceedingly rare complication of MD, first reported by
French surgeon Alexis de Littré in the 1700s [2]. It results from the
passage of an MD that could be associated with intestinal loops through
a hernial defect. In fact, MD is due to the absence of complete closure of
the omphalo-mesenteric duct connecting the midgut with the yolk sac
during embryonic life, which should normally be done during the 8th
week of gestation. It occurs in 0.3 % to 3 % of the general population and
represents the most common congenital malformation of the gastroin­
testinal tract [3]. It generally sits on the anti-mesenteric edge of the
ileum, generally located between 30 and 100 cm from the ileocecal
valve. MD may contain heterogeneous mucosa (stomach, duodenum,
pancreas, or colon). About 80–85 % of the ectopic tissue in MD is gastric.
Its mean length is 5 cm with extremes ranging from 0.5 cm to 85 cm.
These diverticulum's characters are crucial given their role in the choice
of treatment procedure [6,7]. Moreover, two distinct subtypes of LH can
be defined: the more common “true LH” contains only MD and should be
distinguished from Richter's hernia, where part of the intestinal wall is
M.B. Khalifa et al.
Fig. 2. Liberated MD before wedge resection.
strangulated in the hernial sac, but no MD is involved. A “mixed LH”
containing a segment of small intestine in addition to a MD is less
frequently reported [2,3]. Its overall incidence is not yet precise, but it is
reported that 1 % of patients with diverticulum will develop Littre's
hernia. When MD is symptomatic (hemorrhage, diverticulitis, perfora­
tion), it mainly appear on men, but it is more frequent in women when it
is trapped in a hernial sac. Furthermore, LH can occur in both adults and
children. In the adult population, they may present as an inguinal her­
nia, commonly on the right side (50 %), femoral (20 %) or umbilical (20
%) [1,8]. In children, they are thought to occur more frequently in
umbilical hernias [2]. In this respect, the incidence of umbilical hernia
occurring in children varies depending on the ethnicity and age. It is
found in 20 % of Caucasian children at birth and in 5 % of 6 years-old
children. It is more common in premature or low birth weight newborns
and in African children. It is sometimes “syndromic”, occurring in the
context of Down syndrome, mucopolysaccharidoses, congenital hypo­
thyroidism or Wiedemann-Beckwith syndrome [9].Preoperative diag­
nosis of LH is extremely difficult to establish and is generally performed
intraoperatively. In fact, less than 10 % are diagnosed before surgery.
The clinical signs are either absent if the MD is not complicated or
atypical and non-specific. Regardless of the location of LH or age of
patient, symptoms, including abdominal pain, distention, nausea, and
vomiting, make headway more gradually than those of other compli­
cated hernias. Sometimes there is a bump next to the hernia site due to a
local inflammatory response [6,7,10]. In strangulated HL, obstruction is
not always present. It only happens when the diverticulum causes cur­
vature of the intestine. If only the MD is “trapped” and the rest of the
intestine is free, the obstruction would therefore be absent. Peritonitis is
very rare but diverticulum perforation leading to a fecal fistula which
can progress to an enterocutaneous fistula remains possible [3,6]. In
emergency cases, even abdominal ultrasound, and computed
3
International Journal of Surgery Case Reports 114 (2024) 109182
tomography (CT) cannot reveal the right diagnosis. They can only show
signs of intestinal obstruction and its complications. In a recent sys­
tematic review including 53 patients, ultrasound was used in eight cases
and CT scan in ten cases to support the diagnostic procedure. However,
the presence of MD in the hernia sac was confirmed during surgery in all
cases [3]. The most perfect diagnostic and therapeutic procedure is
laparoscopy in doubtful cases. However, this modality is more invasive
than conventional imaging methods and therefore not recommended as
an initial step for diagnosis. The main treatment is surgery. Although the
management of uncomplicated MD remains controversial, MD's man­
agement in the setting of either bowel obstruction or of Littre's hernia is
done according to clinical principles combined with the availability of
local expertise.
Laparoscopic exploration methods either with laparoscopic only or
laparoscopically assisted methods in children with MD are safe, feasible,
and effective. For MDs presenting with an infection or intestinal
obstruction, the indication for laparoscopic exploration followed the
indications for general appendicitis and intestinal obstruction. It is
therefore very important to decide according to the patient's case
regarding the indications for laparoscopic exploration [11].Given that
midline laparotomy is the most commonly used emergency approach for
patients with unknown origin of bowel obstruction, our patient who
presented signs of intestinal obstruction did not benefit from the lapa­
roscopic intervention.
In fact, LH treatment requires both management of Meckel's diver­
ticulum and repair of the hernia. The absence of guidelines of LH
treatment results from being under reported entity. Thus, it's recom­
mended, that all authors should systematically publish more cases in
order to improve the available literature regarding the best clinical
management of Littre hernia. Generally, management of MD requires
resection in all symptomatic patients regardless of age. The controversy
M.B. Khalifa et al.
concerns the management of MD sometimes discovered accidentally on
imaging or intraoperatively in patients with asymptomatic umbilical
hernia [12]. The clinical characteristics, the risk of future complications
and the anatomical particularities of the diverticulum must be consid­
ered. A recent review of literature suggests resection for accidental MD
only in patients with a higher risk of complications: male gender, age
less than 50 years, diverticulum length greater than 2 cm and macro­
scopic presence of abnormal tissue at the time of surgery [3]. The MD
should be resected with transverse closure of the ileum if there is no
edema or inflammation at the base of the diverticulum that could cause
postoperative ileal stenosis. A segmental resection of the small intestine
with anastomosis should be performed if there are signs of ischemia,
inflammation perforation or macroscopic ectopic tissue at the base of
the MD [1,13]. Concerning our patient, we identified the MD which had
a strangulated part twisted in the umbilical ring with edema and ne­
crosis. The diverticulum was managed by a simple wedge resection,
followed by a primary closure of the remaining intestine.
Considering Hernia repair, the use of a mesh is a gold standard
regarding LH occurring in adults. In cases where the small bowel is
ischemic or perforated, mesh must be avoided due to the risk of mesh
infection [2,13]. Differently, the management of umbilical hernias in
children is very specific. The procedure is a simple parietal repair,
without the use of prosthetic material. It differs from that of inguinal
hernias in children since the intervention is not systematically indicated
at the time of diagnosis. It is most often postponed until the age of 4 due
to the large number of spontaneous regressions and the very rare
occurrence of strangulation. It also differs from umbilical hernias in
adults because its repair is carried out without prosthetic material [9].
The defect reparation can be assured with interrupted or continuous
absorbable sutures [14]. For our patient, hernia defect was closed as the
midline by continuous slowly absorbable sutures. Accordingly, a sys­
tematic literature review showed that despite the high prevalence of
pediatric umbilical hernias, there is a paucity of high-quality data to
guide management. The literature does suggest that expectant man­
agement of asymptomatic hernias until age 4–5 years, regardless of size
of hernia defect, is both safe and the standard practice of many pediatric
hospitals [15].
Finally, if we followed the hernia repair recommendations, the
complicated LH occurring in our 11-year-old patient should have been
avoidable. Unfortunately, Both the very small defect which made the
hernia almost undetectable on physical examination, and the patient's
low socio-economic level which prevented him from being examined
several times by a pediatrician dismissed the detection of umbilical
hernia and so surgery at the right time.
4. Conclusion
LH is a very rare type of hernia whose preoperative diagnosis is
extremely difficult due to non-specific semiology. Even abdominal ul­
trasound and computed tomography (CT) cannot reveal the right diag­
nosis. LH treatment requires both management of MD and repair of the
hernia. Treating umbilical hernia in children at the right time may
anticipate complicated LH happening.
Ethical approval
Patient's parents gave their informed consent for this publication.
It is exempt from ethical approval because it is an observation report.
Funding
No funding.
Author contribution
MBK, MG, KN=Study concept, Data collection, and surgical therapy
International Journal of Surgery Case Reports 114 (2024) 109182
for the patient.
MBK, ABB, MF=Writing- original draft preparation.
MBK =Editing and writing.
MB = senior author and manuscript reviewer.
Guarantor
Mohamed Ben Khalifa
Research registration number
Not applicable.
Conflict of interest statement
No conflict of interest.
Acknowledgments
NA.
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