Indian J Gastroenterol DOI 10.1007/s12664-014-0466-y
ORIGINAL ARTICLE
Primary epiploic appendagitis: Reconciling CT and clinical challenges Jamel Saad & Hussein Ali Mustafa & Asem Mohamed Elsani & Fawaz Alharbi & Saad Alghamdi
Received: 15 January 2014 / Accepted: 6 May 2014 # Indian Society of Gastroenterology 2014
Abstract Objectives Our aim was to explain the spectrum of clinical and CT findings in 18 patients with acute epiploic appendagitis. Methods and Materials We reviewed the clinical records and CT pictures of 18 consecutive patients seen in Nejran Armed Forces Hospital, Nejran, Kingdom of Saudi Arabia and Sohag University Hospital, Sohag, Egypt between July 2006 and June 2013 with a diagnosis of primary epiploic appendagitis. Results The age ranged between 22 and 64 years old with a mean of 44.3. Males were affected in 72 % of cases. Eleven patients had acute pain in left lower quadrant. Nausea and vomiting were encountered in four patients. No fever was recorded in all cases. Leukocytosis was found in four patients and C-reactive protein (CRP) was high in five. CT scan diagnosed primary epiploic appendagitis (PEA) in 15 patients, while 3 patients were diagnosed intraoperatively. The left colon was affected in 11 patients. All patients except one
J. Saad (*) Department of Radiology, Nejran Armed Forces Hospital, Alfahd Street, Nejran 1002, Saudi Arabia e-mail:
[email protected] H. A. Mustafa Deparment of Surgery, Nejran Armed Forces Hospital, Alfahd Street, Nejran 1002, Saudi Arabia A. M. Elsani Deparment of Surgey, Sohag Hospital, Sohag hospital, Sohag, Egypt F. Alharbi Deparment of Internal Medicine, Nejran Armed Forces Hospital, Alfahd Street, Nejran 1002, Saudi Arabia S. Alghamdi Department of Family Medicine, Nejran Armed Forces Hospital, Alfahd Street, Nejran 1002, Saudi Arabia
had a central fatty core surrounded by inflammation with size ranging between 1.5 and 3.5 cm in length. Fifteen patients were treated conservatively, and three cases underwent surgical exploration. Ten patients (55.5 %) completed the follow up schedule. Complete resolution was noted in three patients at 2 weeks, in six patients at 3 months, and only one patient had residual changes at 6 months. Conclusion PEA is a challenging clinical diagnosis, and CT scan is extremely necessary in diagnosing this disease accurately. The awareness of the surgeons concerning this rare occasion will avoid unnecessary hospital admission and operative treatment. Keywords Acute abdomen . Diagnosis . Management
Introduction Epiploic appendagitis is primary or secondary inflammation of the epiploic appendages. Primary epiploic appendagitis (PEA), which is the subject of this study, is the result of torsion with subsequent ischemia or is attributable to spontaneous phlebothrombosis without torsion [1]. Secondary epiploic appendagitis is the inflammation of the epiploic appendages caused by nearby pathological processes, like diverticulitis, appendicitis, or cholecystitis [1, 2]. It is hard to diagnose clinically due to the absence of specific clinical features since it is characterized by sudden onset of sharp localized pain within the left or right iliac fossa with mild gastrointestinal symptoms, which might simulate a surgical emergency. Clinically, it can be mistaken for acute diverticulitis or acute appendicitis as these are the most common causes of acute abdominal pain [3]. In the past and before employing a CT scan in evaluation of patients with acute abdomen, most of these cases were discovered at laparotomy.
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CT scan plays a key role in confirmation of diagnosis of PEA as it has specific features [3]. Awareness of general surgeons and radiologists concerning the clinical presentations and imaging findings of this entity is vital to hit a correct diagnosis and to avoid unnecessary hospital admission and surgery. The purpose of this study is to investigate the clinical options, CT findings, and management of 18 cases of PEA admitted at Nejran Armed Forces Hospital, Kingdom of Saudi Arabia (KSA) and Sohag University Hospital, Egypt in the period from 2006 to 2013.
Material and Methods We reviewed the clinical records and CT pictures of 18 consecutive patients seen in Nejran Armed Forces Hospital, KSA and Sohag University Hospital, Egypt between July 2006 and June 2013 with a designation of primary epiploic appendagitis. Patients with secondary epiploic appendagitis were excluded. All patients were bestowed to the emergency and surgical department of our hospital for a suspicion of acute abdomen. Complete history and clinical examination were obtained from every patient. Routine laboratory investigations including complete blood count, C-reactive protein (CRP), urine analysis, blood sugar, urea, and creatinine were requested for each patient. Imaging investigations were done for each patient including plain X-ray, abdominal ultrasonography (US), and abdominal CT scan (spiral CT machine, Siemens from July 2006 to 2010 and multidetector CT machine 64 slices, GE Medical Systems from May 2010 to 2013). CT examinations were evaluated for colon wall thickening; focal fatty center; inflammatory changes; location, size, and presence or absence of central high density within the fat; and any associated pathology. After confirmation of diagnosis of PEA, patients were treated conservatively by nonsteroid antiinflammatory drugs for 1–2 weeks. Patients, who were not responding to conservative treatment, suspected to possess complications or with no clear-cut diagnosis were subjected to surgical exploration. Each patient was followed up clinically and by CT scan after 2 weeks and 3 and 6 months with comparison with the previous imaging findings.
Results There were 18 patients, 72 % (13/18) were males. The age range of the patients was 22–64 years old (mean 44.3 years). The body mass index (BMI) was ranging from 23.5 to 39 with a mean of 30.7. All patients had abdominal pain of abrupt onset, localized to left iliac fossa in 11 cases, right iliac fossa in 4
cases, and epigastrium and right hypochondrium in 3 cases. Nausea and vomiting were reported in four cases (22 %). None of the patients was febrile at the time of presentation. Abdominal tenderness supported in seven cases; however, no rigidity or rebound tenderness was present in any studied patient. Twenty-two percent (4/18) had mild leukocytosis (<10,000/ mL) and CRP was high in 28 % (5/18). The presumptive clinical diagnoses after medical history and physical examination were diverticulitis (eight cases, 44.4 %), acute appendicitis (five cases, 28 %), cholecystitis (one case), intestinal obstruction (one case), left ureteric stone (one case), and epiploic appendagitis (2 cases), (Table 1). Plain X-ray was normal in all except two cases, the first one showed calcification in the right hypochondrium and the second suggesting adhesive intestinal obstruction in a patient who underwent appendectomy 20 years past. Abdominal US was performed in all patients. It was suggestive of acute appendicitis in one case, chronic cholecystitis with porcelain gallbladder in one case, acute epiploic appendagitis in four cases, and unremarkable in the remaining 12 cases. CT scan showed an ovoid fatty mass with hyperattenuated ring and disproportionate adjacent fat stranding on the antimesenteric border of the colon in 14 cases (Fig. 1a, b) and on the mesenteric side in one case. Three cases were missed by CT scan. Diagnosis was suggestive of acute appendicitis in one patient, cholecystitis with porcelain gallbladder in the second patient (Fig. 2), and adhesive intestinal obstruction in the third patient. The dimension of the fatty central core was between 1.5 and 3.5 cm in length. The foremost common parts of the colon affected, in decreasing order of frequency, were the sigmoid colon (n=7) (Fig. 3), descending colon (n=4) (Fig. 1), cecum (n=2) (Fig. 4), and ascending colon (n=2). The most common positions of inflammatory changes in relationship to the colonic lumen, in decreasing order of frequency, were as follows: anterior (n=11), lateral (n=5), inferior (n=1), and medial (n=1). Associated thickening of the adjacent colonic wall was detected in 11 % (2/18) of the patients (Fig. 5). The central high attenuation dot was seen in 61 % (11/18) of the cases (Fig. 1). Fifteen patients were diagnosed accurately by CT scan and treated conservatively. Three cases were diagnosed after laparotomy and were treated by excision of the affected ischemic epiploicae. One patient was diagnosed as adhesive small bowel obstruction after appendectomy 20 years past found to have had ischemic epiploic appendagitis of the left colon. The second patient was diagnosed as cholecystitis with porcelain gallbladder, and it was absolutely simply a torsion of calcified epiploic appendages that was adherent to the gallbladder, and the third case was diagnosed as acute appendicitis found to be inflamed epiploic appendages located medial to the cecum.
Indian J Gastroenterol Table 1 The characteristics of the 18 studied cases No. Age Sex BMI Presenting complaint
Site
Abdominal examination
CBC (per mL) CRP Provisional diagnosis
CT finding
Treatment
1
60
M
32.5 Abdominal pain LIF
Normal
8,000
0
Diverticulitis
PEA
Conservative
2
50
M
30.2 Abdominal pain LIF
Normal
10,000
2
Diverticulitis
PEA
Conservative
3
45
M
Abdomen tenderness 10,000
100
Acute appendicitis
PEA
Surgical
4
26
F
24.3 Abdominal pain RIF and RHP and vomiting 26.7 Abdominal pain RIF
Normal
7,000
4
Acute appendicitis
PEA
Conservative
5
53
M
35.6 Abdominal pain LIF
Normal
8,000
45
Diverticulitis
PEA
Conservative
6
45
M
28
Abdominal pain LIF
Normal
5,000
3
Diverticulitis
PEA
Conservative
7
64
F
33.5 Abdominal pain LIF
Normal
5,000
0
Diverticulitis
PEA
Conservative
8
50
F
18
9
30
M
28.4 Abdominal pain Left and right side Abdomen tenderness 10,000 and vomiting 27.2 Abdominal pain RIF Normal 6,000
10
55
M
38
Adhesive obstruction
Surgical
4
Acute appendicitis
PEA
Conservative
Abdomen tenderness
5,000
1
Diverticulitis
PEA
Conservative
Normal
7,000
2
Diverticulitis
PEA
Conservative
Abdomen tenderness
6,000
0
Appendagitis
PEA
Conservative
11
56
M
Abdominal pain LIF and vomiting 30.5 Abdominal pain LIF
12
23
M
23.5 Abdominal pain RIF
13
34
M
27.5 Abdominal pain RIF
Normal
8,000
0
Acute appendicitis
PEA
Conservative
14
60
M
36.2 Abdominal pain LIF
Normal
5,000
2
Diverticulitis
PEA
Conservative
15
28
F
Normal
7,000
12
Acute appendicitis
PEA
Conservative
16
56
M
34.8 Abdominal pain RIF and vomiting 31.5 Abdominal pain LIF
Abdomen tenderness
7,000
0
Appendagitis
PEA
Conservative
17
40
F
39
Abdomen tenderness
6,000
50
Acute cholecystitis
18
22
M
25.6 Abdominal pain LIF
Tenderness
7,600
0
Left ureteral stone
Calcified Surgical PEA PEA Conservative
Abdominal pain RHP
BMI body mass index, CBC complete blood count, CRP C-reactive protein, LIF left iliac fossa, PEA primary epiploic appendagitis, RIF right iliac fossa, RHP right hypochondrium
All patients whether or not treated conservatively or surgically showed clinical improvement with the absence of abdominal pain before discharge from the hospital and on follow up visits. Only ten out of 18 patients (55.5 %) completed the follow up schedule (2 weeks and 3 and 6 months). Follow up CT scan showed complete resolution of the signs of PEA at 2 weeks in three patients (Fig. 6), at 3 months in six patients (Fig. 7), and one patient showed residual signs at 6 months. Fig. 1 Plain axial (a) and coronal (b) CT scan images of the abdomen showing an oval fat density lesion adjacent to the left colon on the anti-mesenteric side (arrow) with hyperattenuated ring and disproportionate adjacent fat stranding. A central hyperattenuating focus is noted, representing the target sign (head arrow)
Discussion Epiploic appendages are serous membrane outpouchings that arise from the serosal surface of the colon, contain fat and vessels, and are between 0.5 and 5-cm long. Each had one or two arterioles and a venule. The pedunculated form, free vary of movement and tortuous nature of their blood supply, provides appendices epiploicae vulnerable to torsion or ischemia [1, 2].
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Fig. 2 Plain axial CT scan of the abdomen showing a calcified mass in the right flank adjacent to right colon diagnosed as porcelain gallbladder (arrow) Fig. 4 Plain axial CT image shows fat lesion (arrow) with hyperattenuating center and surrounding inflammation that abuts the cecum
Epiploic appendagitis is a rare, inflammatory lesion that results from torsion or phlebothrombosis of the epiploic appendages [1]. In some studies, patients are typically obese people in the second to fifth decades of life [4]. Our patients matched with these studies as 83 % were obese, between the second and sixth decades of age with male preponderance. In agreement with most of the literature, the majority of our patients experienced sudden onset of localized abdominal pain mostly in the left or right lower abdomen quadrant, not associated with gastrointestinal symptoms except in few patients (22 %) [3]. Some studies [6─8] stated that, the site of the pain varies according to the location of the inflammation, but the right or left lower quadrant is the most common site. The patient can often localize and point directly to the involved area. Left lower quadrant pain is the more commonly encountered localization [4, 5]. This is going with our study as 61 %
of our patients experienced pain on the left side of the abdomen with a provisional clinical diagnosis of acute diverticulitis. Clinically, we can suspect the diagnosis of acute diverticulitis as it usually occurs in elderly patients and manifested with evenly distributed lower abdominal pain associated with nausea, vomiting, fever, rebound tenderness, and leukocytosis [6, 7]. In contrast with PEA where the pain is usually localized and not associated with nausea, vomiting, anorexia, fever, change in bowel function, leukocytosis, or other signs of a serious systemic illness. In our series, only 22 % of our patients had associated gastrointestinal symptoms and mild leukocytosis. The laboratory tests are often non-specific. Abnormal laboratory results may include slightly elevated serum levels of CRP which is concordant with our results as 28 % presented mildly high CRP results. The white blood cell count (WBC) is
Fig. 3 Axial CT scan section of the pelvis after rectal opacification showing an oval fat density lesion adjacent to the sigmoid colon on the anti-mesenteric side (arrow) with hyperattenuated ring and disproportionate adjacent fat stranding. No hyperattenuating center seen
Fig. 5 Axial CT image shows the inflamed appendix epiploica (long arrow) anterior to the sigmoid colon with hyperattenuating center surrounded by fat stranding. Wall thickening of the ascending colon noted adjacent to the inflamed epiploic appendix (short yellow arrow)
Indian J Gastroenterol Fig. 6 Follow up CT scan performed 2 weeks later shows complete resolution of the lesion (arrows)
usually normal or only moderately elevated [6]. In the current study, WBC of the patients with PEA was elevated only in 22 % of patients. Patients presenting with acute abdominal pain, localized tenderness without associated symptoms, or leukocytosis should raise a high index of suspicion for PEA [8–10]. The common presumptive clinical diagnosis for patients with PEA before radiologic interventions was either diverticulitis or appendicitis. Mollà et al. [9] reported that 7.1 % of patients investigated to exclude sigmoid diverticulitis had radiologic findings of PEA. In the current study, diverticulitis accounted for 44.4 % of the presumptive diagnosis in patients Fig. 7 Follow up CT scan done 2 weeks later demonstrates increased density and size of the lesion. Six months later, CT scan shows complete resolution of the inflammation (arrows)
with PEA followed by acute appendicitis 28 %. Only two of our patients were suspected of having PEA. This could be explained by the absence of awareness of clinicians regarding this rare disease. PEA can be diagnosed on abdominal ultrasonography in both gray-scale and color Doppler. Ultrasound has the advantage of correlating the location of the lesion with the location of maximum tenderness. The inflamed appendage appears as a non-compressible, solid, hyperechoic ovoid mass with a subtle peripheral hypoechoic rim located at the point of maximal tenderness [8]. In our study, abdominal US was performed in all patients and was diagnostic of PEA only in four
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patients. It was suggestive of acute appendicitis in one case, chronic cholecystitis with porcelain gallbladder in one case, and was unremarkable in the remaining 12 cases. This could be explained by the lack of awareness of surgeon as well as radiologists concerning this rare disease and to patient’s morphology, as the majority of our patients were obese making the ultrasound study difficult. Although ultrasound has the advantage of correlating the location of the lesion with the location of maximum tenderness, this approach probably is best suited for patients with a thin body habitus and requires experienced radiologist. Acute appendagitis had characteristic CT features including a pericolic ovale fatty core surrounded by a thin dense rim due to surrounding inflammation and containing a “target lesion” with a central region of hyperattenuation [1, 3]. The most common CT appearance seen in our study was the presence of 1.5 to 3.5 cm fat density lesion with surrounding inflammatory changes usually abutting the anterior wall of colon which was concordant with the study of Singh et al. [5]. Although the presence of a central high-attenuation focus (target sign) within the fat is a helpful finding in making the diagnosis, its absence does not exclude the diagnosis [5]. The target sign was noted in only 50 % of the patients in our study. In agreement with the study by Hiller et al. [11] and Singh et al. [5], we noted colon wall thickening in only two patients in our study (12.5 %). This point is important in the differentiation of acute appendagitis from acute diverticulitis. The location of an acute appendix epiploica anterior to the colon is also useful in making a confident diagnosis [4, 12]. Only one patient of our series had an atypical presentation on CT scan examination which demonstrated a calcified cystic mass in the gallbladder fossa with a picture similar to porcelain gallbladder. The definitive diagnosis was made only after surgical resection and histopathological examination. The CT features of primary epiploic appendagitis are characteristic and even pathognomonic while excluding other causes of abdominal pain. The classic CT finding is a two to three, 5-cm, oval-shaped, paracolic fat density, surrounded by a peripheral thickened hyperattenuating rim with the absence of adjacent colonic wall thickening is virtually pathognomonic for PEA [1, 3, 5, 11, 13]. A high-attenuated central dot within the inflamed appendage that corresponds to a thrombosed draining appendageal vessel supports further the diagnosis but is not constantly seen [11, 13]. The CT changes of acute epiploic appendagitis in our study completely resolved in all except one patient who underwent follow up CT at 6 months after the acute presentation. However, the CT changes were present to a variable degree in majority of patients in whom CT was performed within 2 weeks or 3 months. It is important to be aware of the evolutionary follow up CT findings of acute appendagitis because these findings may persist for several months in spite of clinical improvement and mimic the diagnosis of acute
epiploic appendagitis in the absence of a prior comparison study or suggestive clinical history [5, 11, 14]. The alternative diagnoses of PEA on CT include mesenteric panniculitis, acute diverticulitis, trauma, omental infarction, or neoplasm such as a liposarcoma. The diagnosis of this condition is based primarily on the typical CT features and, to a lesser extent, on the clinical features such as focal abdominal pain in the absence of gastrointestinal symptoms and leukocytosis [6, 15]. Approximately 7.1 % of patients investigated to exclude sigmoid diverticulitis have imaging findings of primary epiploic appendagitis [16]. Acute epiploic appendagitis may be mistaken clinically for acute appendicitis when it is involving the cecum. Although an omental infarction can have a CT picture similar to that of epiploic appendagitis, it lacks the dense peripheral ring that is seen in epiploic appendagitis and demonstrates an oval heterogeneous fatty mass with linear fat stranding [5, 13]. According to Jalaguier et al. [17], CT is accurate for distinguishing of left acute colonic diverticulitisassociated secondary epiploic appendagitis from PEA. A variety of complications can follow PEA [18, 19]. Accompanying surrounding inflammation can trigger adhesions with multiple secondary symptoms [18]. Other possible complications include local abscess formation, intussusception, bowel obstruction, and peritonitis [13, 20, 21]. In our study, no complication was reported. Our case of adhesive intestinal obstruction was believed to be due to previous appendectomy rather than a PEA complication. Therapy of epiploic appendagitis is a topic of some debate. It is described by some authors as a self-limiting condition with patients recovering in less than 10 days with oral anti-inflammatory medication [13, 16]. Most of the surgical literature favors a conservative therapy regiment [16]. However, Sand et al. [22] showed 40 % recurrence rate in PEA. They believed that conservative treatment had a high recurrence rate and surgical interventions may be necessary for recurrent cases. Laparoscopic removal of the involved appendage is occasionally required and consists of simple ligation and excision of the inflamed appendage [23]. In the current study, most patients (83, 3 %) were treated conservatively. Only three patients were treated surgically, the first one was an adhesive intestinal obstruction, the second was presenting an atypical CT appearance with calcified mass located near the bed of gallbladder giving a picture of porcelain gallbladder, and the third was missed on CT study and diagnosed as acute appendicitis. All three cases were proved intraoperatively PEA. No recurrence was noted during the follow up period. Our opinion regarding the treatment of this rare disease is, to favor a conservative treatment for non-complicated cases and keep surgery, particularly laparoscopy, for rare cases not
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responding to conservative management and complicated cases such as adhesive intestinal obstruction or abscess collection. The weakness in this study and other more recent studies in the literature is the lack of pathologic confirmation of acute epiploic appendagitis; in addition to some limitations to this study, such as being a relatively small series and retrospective analysis, further prospective, larger studies of PEA are needed.
Conclusion We can conclude that PEA is a challenging clinical diagnosis and CT scan is extremely necessary in diagnosing this disease accurately. With awareness of the surgeons as well as the radiologists regarding this rare occasion; we are able to avoid supernumerary hospitalization and surgical intervention as most of the cases answer conservative management satisfactorily.
Conflict of interest JS, HAM, AME, FA, and SA all declare that they have no conflict of interest. Ethical statement The authors declare that the study was performed in a manner to conform with the Helsinki Declaration of 1975, as revised in 2000 and 2008 concerning Human and Animal Rights, and the authors followed the policy concerning Informed Consent as shown on Springer.com.
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