AbstractAmyand’s hernia is a rare type of hernia where the vermiform appendix is within an inguinal hernia sac. Tumors of the appendix are quite uncommon. The coincidence of an Amyand’s hernia with neuroendocrine tumor of the appendix, as in our case, is even more rarely reported.We report the case of an 81-year-old male who presented with an incarcerated right inguinal hernia. After resuscitation, the clinical diagnosis was confirmed by computed tomography. It showed an incarcerated right inguinal hernia which contained the distal ileum, cecum, thickened appendix, as well as a small amount of fluid. Subsequently, the patient was prepared for emergency surgery. During the operation, the hernia sac was found and opened. The appendix was swollen. Therefore, appendectomy was performed. The inguinal defect was repaired using the Modified Bassini Technique. The patient had an uneventful postoperative recovery and surprisingly the histopathology of the appendix revealed a 1.5cm well-differentiated low grade neuroendocrine tumor (carcinoid) of the appendix tip.An incidental finding of neuroendocrine tumor of the appendix in a patient with s hernia is extremely rare. A high index of suspicion is the key to diagnose such a coincidence in order to safely and optimally treat such a condition

Alzahrani, Hassan A.

Alzamel, Hyetham A.

Azzumeea, Fahad

Elbanna, Khaled Y.

International Journal of Surgery Case Reports

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Neuroendocrine tumor of the appendix inside an incarcerated Amyand’s hernia 

CASE  REPORT  –  OPEN  ACCESS
International Journal of Surgery Case Reports 14 (2015) 152–155
Contents lists available at ScienceDirect
International  Journal  of  Surgery  Case  Reports
j ourna l h om epage: www.caserepor ts .com
Neuroendocrine  tumor  of  the  appendix  inside  an  incarcerated
Amyand’s  hernia
Khaled  Y.  Elbannaa, Hassan  A.  Alzahranib,∗, Fahad  Azzumeeaa, Hyetham  A.  Alzamelc
a Medical Imaging Department, King Fahad National Guard Hospital, King Abdulaziz Medical City (KAMC), Riyadh 11426, Saudi Arabia
b Department of Surgery, Faculty of Medicine, King Khalid University (KKU), Abha 61421, Saudi Arabia
c Department of Surgery, King Fahad National Guard Hospital, King Abdulaziz Medical City (KAMC), Riyadh 11426, Saudi Arabia
a  r  t  i  c  l e  i  n  f  o
Article history:
Received 22 April 2015
Received in revised form 29 July 2015
Accepted 30 July 2015
Available online 5 August 2015
Keywords:
Neuroendocrine
Carcinoid
Amyand
Hernia
a  b  s  t  r  a  c  t
Amyand’s  hernia  is a rare  type  of  hernia  where  the  vermiform  appendix  is  within  an  inguinal  her-
nia  sac.  Tumors  of the  appendix  are  quite  uncommon.  The  coincidence  of  an  Amyand’s  hernia  with
neuroendocrine  tumor  of the  appendix,  as in  our  case,  is  even  more  rarely  reported.
We  report  the  case  of  an  81-year-old  male  who  presented  with  an  incarcerated  right  inguinal  hernia.
After  resuscitation,  the  clinical  diagnosis  was conﬁrmed  by computed  tomography.  It  showed  an  incar-
cerated  right  inguinal  hernia  which  contained  the  distal ileum,  cecum,  thickened  appendix,  as  well  as  a
small  amount  of ﬂuid.  Subsequently,  the  patient  was  prepared  for emergency  surgery.  During the  oper-
ation, the hernia  sac  was  found  and  opened.  The  appendix  was  swollen.  Therefore,  appendectomy  was
performed.  The  inguinal  defect  was  repaired  using  the Modiﬁed  Bassini  Technique.  The  patient  had  an
uneventful  postoperative  recovery  and surprisingly  the  histopathology  of  the  appendix  revealed  a  1.5 cm
well-differentiated  low  grade  neuroendocrine  tumor  (carcinoid)  of  the  appendix  tip.
An  incidental  ﬁnding  of neuroendocrine  tumor  of  the appendix  in  a patient  with  s  hernia  is extremely
rare.  A  high  index of  suspicion  is  the  key  to  diagnose  such  a coincidence  in  order  to safely  and  optimally
treat  such a condition.
©  2015  The  Authors.  Published  by  Elsevier  Ltd. on behalf  of Surgical  Associates  Ltd.  This  is an  open
access  article  under  the CC BY-NC-ND  license  (http://creativecommons.org/licenses/by-nc-nd/4.0/).
1. Background
An Amyand’s hernia is a rare occurrence where the vermiform
appendix is found within an inguinal hernia sac [1]. It was ﬁrst
described by a distinguished French surgeon, Claudius Amyand
(1660–1740). Amyand’s hernia is classically described as occurring
in 1% of all inguinal hernias. Appendicitis in an Amyand’s hernia
accounts for 0.1% of all cases of appendicitis. Its clinical presentation
is similar to that of an incarcerated hernia, and thus it is difﬁcult to
diagnose clinically. It can be diagnosed preoperatively using ultra-
sound and computed tomography. The appendix can remain within
the hernia sac without symptoms throughout a patient’s lifetime.
It is possible that strangulation can occur at the neck of the hernia
resulting in vascular obstruction, inﬂammation and even perfora-
tion of the appendix [2].
Neoplasms of the appendix are quite uncommon, accounting for
only 0.4% of gastrointestinal tumors [3]. The occurrence of these
two conditions together is even more rarely reported [4]. Here
we present a case with the incidental ﬁnding of a neuroendocrine
tumor of the appendix within an incarcerated Amyand’s hernia.
∗ Corresponding author.
E-mail address: hassanali304@hotmail.com (H.A. Alzahrani).
2. Case report
An 81-year-old male presented to our emergency department
complaining of painful right groin swelling for 12 h. He had a history
of reducible right inguinal hernia for the previous 8 years. He com-
plained that the pain was progressively worsening, but he denied
any history of vomiting or constipation. The patient was known
to have chronic obstructive pulmonary disease, Alzheimer disease
and benign prostatic hyperplasia.
On physical examination the patient was distressed with tem-
perature of 36.5 ◦C, respiratory rate of 20/min, heart rate of 67/min
and blood pressure of 84/59 mmHg. The abdominal examination
revealed a 10 × 8 cm right inguinal swelling, which was  tender and
irreducible. The rest of the abdomen was  soft. Digital rectal exam-
ination revealed an enlarged, ﬁrm prostate.
On laboratory workup, he was  found to have WBC  of 7.3/L,
hemoglobin of 11.8 g/L, platelet count of 471 × 109/L, Sodium
135 mmol/L, Potassium 5.1 mmol/L, and Creatinine 66 mol/L.
The patient had a rapid resuscitation with intravenous ﬂuids
and analgesia. Computed tomography of the abdomen and pelvis
was done after only intravenous contrast injection as the patient
couldn’t tolerate oral contrast. The images demonstrated incarcer-
ated right inguinal hernia containing the distal ileum, cecum, and
appendix together with a small amount of ﬂuid within the hernia
http://dx.doi.org/10.1016/j.ijscr.2015.07.038
2210-2612/© 2015 The Authors. Published by Elsevier Ltd. on behalf of Surgical Associates Ltd. This is an open access article under the CC BY-NC-ND license (http://
creativecommons.org/licenses/by-nc-nd/4.0/).
CASE  REPORT  –  OPEN  ACCESS
K.Y. Elbanna et al. / International Journal of Surgery Case Reports 14 (2015) 152–155 153
Fig. 1. Axial image; the right inguinal hernia sac containing cecum.
Fig. 2. Reconstructed coronal image showing the hernia.
sac. The appendix was  slightly edematous; however, no appen-
dicular masses were identiﬁed and there were no signs of bowel
obstruction or ischemia (Figs. 1 and 2). Our radiological diagnosis
was an incarcerated Amyand’s hernia.
The patient was taken to the operating room. An emergency
right anterior inguinal exploration was performed and revealed an
indirect inguinal hernia. The hernia sac contained cecum, appendix
and a small amount of free ﬂuid. There was no bowel ischemia
or gangrene, however the appendix was inﬂamed (Figs. 3 and 4).
Therefore, an appendectomy was done and the cecum was  freed
from adhesion to the sac and reduced into the abdominal cavity.
The hernia sac was ligated and excised then the inguinal defect was
repaired using the Modiﬁed Bassini Technique. The patient had an
uneventful postoperative recovery and was discharged one week
after surgery.
Histopathology revealed a 1.5 cm well-differentiated, low grade
neuroendocrine tumor (carcinoid) of the appendix tip. It was  invad-
ing the submucosa, muscularis propria and subserosa but not the
visceral peritoneum or the mesoappendix. There was no lympho-
vascular invasion and resection margin was free from tumor.
Fig. 3. Intraoperative picture of the hernia sac and healthy cecum.
Fig. 4. Intraoperative picture of the appendix looking unhealthy.
3. Discussion
Inguinal hernia may  contain the omentum or small bowel. Cer-
tain unusual contents may  be encountered, such as the bladder, a
Meckel’s diverticulum (Littre’s hernia) [5], or a portion of the cir-
cumference of the bowel (Richter’s hernia) [6]. When an appendix
was found in a hernia sac, it was  named Amyand’s hernia [1].
Classically, it accounts for 1% of all inguinal hernias or even less
(between 0.4% and 0.6% according to two recent studies by Psar-
ras et al., and Gurer et al.,) [7,8]. Amyand’s hernia is, as expected,
more common in male patients and presents as a bimodal age dis-
tribution in neonates and in patients above 70 years. Coexistence
of Amyand’s hernia with a benign or malignant condition has been
described rarely in the literature [2].
CASE  REPORT  –  OPEN  ACCESS
154 K.Y. Elbanna et al. / International Journal of Surgery Case Reports 14 (2015) 152–155
According to a report published by the National Cancer Institute
appendiceal neoplasms account for approximately 0.4% of gastroin-
testinal tumors [3]. The most common tumors of the appendix are
neuroendocrine tumors (NETs). Appendiceal NETs make up to 20%
of all NETs of the gastrointestinal tract. They usually present in the
3rd to 5th decade. Females are more frequently affected than males.
Most NETs of the appendix are found incidentally in appendectomy
specimens from patients with appendicitis [9]. The gold standard
treatment is surgical treatment by resection of the whole appendix
for carcinoids located around the tip. In cases where the tumor is
larger than 2 cm or located at the base of the appendix, a wider
resection has to be performed with right hemicolectomy [10] [11].
After reviewing the English literature, there is only one reported
case of such a rare coincidence (by Wu,  C. and Yu, C.). The case
was diagnosed clinically as incarcerated inguinal hernia. Therefore,
emergency hernioplasty was arranged without preoperative radi-
ological investigations for the hernia. The histologic ﬁnding was
unexpected, as in our case, and revealed adenocarcinoid tumor
of the appendix. The tumor measured about 2.5 cm in length and
about 1 cm in diameter with free resection margin at the base of
the appendix [4].
Preoperative clinical diagnosis of Amyand’s hernia is difﬁcult
without speciﬁc signs or symptoms. The most common presen-
tation is irreducible painful hernia (83%) [12]. Using ultrasound
and Computed tomography (CT) make preoperative diagnosis of
Amyand’s hernia feasible [2]. The ultrasound image is that of a
blind-ended tubular structure with thickened wall in connection
with the cecum within the hernia sac [13] [14]. CT allows direct
visualization of the appendix inside the inguinal canal and even if
this is not possible, location of the cecum in proximity to the hernia
sac is indicative of Amyand’s hernia [15] [16]. Amyand’s hernia has
also been diagnosed incidentally with barium enema [17].
Preoperative imaging allows some understanding of the situa-
tion of the appendix inside the hernia: ﬂuid in the right scrotum,
heterogeneous tissue edema on ultrasound, and gas on CT are prob-
ably indicative of perforation. Use of these modalities has allowed
a satisfactory percentage of preoperative diagnosis. However, the
preoperative imaging does not change the surgical management of
Amyand’s hernia [2].
The surgical management of Amyand’s hernia is based on the
Losanoff and Basson classiﬁcation [18]. A type 1 Amyand hernia
has a normal appendix. A hernia reduction and mesh repair with-
out appendectomy is done. For type 2 with acute appendicitis but
no abdominal sepsis, an appendectomy is performed through the
hernia and also a primary repair with no mesh. Type 3 with acute
appendicitis and abdominal wall or peritoneal sepsis requires a
laparotomy, appendectomy, and primary repair of hernia. For type
4, where acute appendicitis is associated with a related or unrelated
pathology, the management is the same as for types 2 and 3 her-
nias but the second pathology should be investigated and treated
appropriately.
Today prosthetic repairs are accepted to be superior to “non-
mesh” suture repairs. A recent meta-analysis revealed that the
Shouldice herniorrhaphy is the best non-mesh technique in terms
of recurrence, though it is more time consuming and needs a
slightly longer postoperative hospital stay. Nevertheless, the use
of mesh is associated with a lower rate of recurrence [19].
The use of mesh in emergency repair of complicated hernias is
debatable. Recent evidences is in favor of mesh use in cases with
incarceration. However, prosthetic repair creates a risk for surgical
site infection in cases where a gangrenous intestine is met  and a
resection-anastomosis is required [20]. The biologic meshes may
gain importance in the future because they may  be used in con-
taminated areas. However, they are extremely expensive which is
the main factor limiting their use [21].
4. Conclusion
The incidental ﬁnding of an appendiceal neuroendocrine tumor
in a patient with Amyand’s hernia is extremely rare, High index
of suspicion is the key to diagnose such a coincidence, in order to
safely and optimally treat such a condition.
Conﬂict of interest
The authors declare that there is no conﬂict of interest regarding
the publication of this paper.
Sources of funding
The source of fund is self-funded by the authors.
Ethical approval
This paper was ethically approved by the IRB of the King Abdu-
laziz Medical City.
Consent
The patient was  appropriately consented and agreed to publi-
cation of this paper.
Author contribution
1. Khaled Y. Elbanna — Contributed in data collection and writing
the paper.
2. Fahad Azzumeea — Contributed in data interpretation.
3. Hyetham A. Alzamel — Contributed in study concept.
4. Hassan A. Alzahrani — Contributed in data collection and writ-
ing the paper.
Guarantor
Hassan A. Alzahrani: Assistant Professor of Surgery King Khalid
University, P.O. Box 641, Abha 61421, Kingdom of Saudi Arabia.
Acknowledgement
The authors would like to thank all the colleagues who con-
tributed to management of this patient. We appreciate Dr. John
Patrick O’Brien for his contribution.
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Neuroendocrine tumor of the appendix inside an incarcerated Amyand’s hernia

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