Journal of Pediatric Care

Keywords

Children; Cecal epiploic appendices; Appendix torsion

Introduction

Epiploic appendices of the colon are fatty structures covered with visceral peritoneum, mainly found in sigmoid colon decreasing in number toward cecum and in some cases they are pedunculated that make them predisposed to their torsion. In 1908, Briggs first reported a case of an epiploic appendix torsion mimicking appendicitis [1]. Epiploic appendagitis usually caused from torsion or inflammation of appendices epiploicae occurs most often beyond the fourth decade of life. Only few reports of caecal epiploic appendagitis involving young children have been published till date [2,3]. Although preoperative diagnosis is very difficult because of its rarity and similarity with other pathologies that cause RLQ abdominal pain, recognition of this pathology by pediatric surgeons is essential since it can be treated without surgery. Preoperative diagnosis of this condition is possible with computed tomography (CT) although it is rarely used in children’s with acute abdominal pain.

Torsion or inflammation of appendices epiploicae in the caecum rarely are found as cause of acute abdominal pain (in RLQ) in children and is usually mistaken for acute appendicitis. More often their torsion happened in sigmoid colon which is more mobile and they are much more in number in this part of colon. Depending on the site of occurrence, it can mimic different pathologies causing acute abdominal pain, and as in majority of cases epiploic appendix torsion is misdiagnosed preoperatively.

Case History

A 12 year-old girl Kosovo Albanian nationality referred in our pediatric surgery department because of acute onset abdominal pain in the right lower quadrant that had begun a night before admission. She was treated initially in the morning of next day with analgesics in regional hospital but the pain returned 6 hours later and she was admitted in our department for evaluation on suspicion of acute appendicitis. At admission, she had constant RLQ pain, nausea and anorexia but no vomiting, no elevated temperature and pulse rate was 86 beats per minute. On abdominal examination, localized abdominal guarding, localized rebound tenderness in the right iliac fossa were noted. The white blood cell count was 12.7 10³/mm³, hemoglobin was 12.4 g/dL, hematocrit 34.5% and platelets 143 10³/mm³. Urine and blood biochemical examinations were normal. Abdominal ultrasonography couldn’t visualized vermiform appendix or solid hyperechoic mass, but revealed minimal inter-loop free fluid and few dilated gut loops in the right iliac fossa (Figure 1). A presumptive diagnosis of acute appendicitis was made. The patient was operated on emergency basis and abdomen was accessed through Mcburney ’ s incision. Small amount of serosanguinous fluid was aspirated. The vermiform appendix was not inflamed but around 3 centimeters near to the base of appendix vermiformis on the most distal part of caecum, twisted 1.1 cm² wide and 2,5 cm long pedunculated epiploic appendix was noticed (Figure 2). Mesentery lymph nods were not enlarged, right adnexa, terminal ileum and cecum were grossly normal. Appendectomy and complete removal of the appendix epiploic form its base on cecum and serosa closer were performed. The histological examination of the specimen revealed an ischemic infarct of adipose tissue in the twisted epiploic appendix, while the histological examination of the vermiform appendix was normal (Figure 3). Patient recovered without any early postoperative complications and was discharged the second postoperative day.

Discussion

Figure 1: Minimal free (inter-loop) fluid.

Figure 2: Torsion of appendix epiploic of the caecum with normal appendix vermiform

Figure 3: Ischemic infarct of the appendix epiploic adipose tissue.

Epiploic appendixes are small pedunculated fatty pouches on colons antimesenteric surface. They are composed of adipose tissue and blood vessels arising from serosal of colon surrounded with peritoneum, usually less than 5 cm in length. Their peduncle shape and mobility, make them prone to spontaneous torsion which usually occurs beyond 4th decade of life, predominantly in men [4-7]. Although acute appendicitis is the main cause of acute abdominal pain in children, torsion of epiploic appendix in children it ’ s rarely considered among pediatric surgeons (in pediatric surgery literature) since it rarely is cause of acute abdominal pain especially in pediatric population. Epiploic torsion is rarely seen in patients younger than 19 years and is almost unknown in children, only few case reports of epiploic appendagitis involving caecum in children have been published in literature [2,3,8-12]. In 1908, Briggs first reported a case of an epiploca appendix torsion (appendagitis) mimicking appendicitis [1]. With diagnosis based on clinical manifestations alone, torsion of cecal epiploic appendix is misdiagnosed in the majority of patients. In a series of 1,320 cases of acute abdominal pain by Golash et al. only eight cases were due to acute epiploic appendagitis [13]. The preoperative diagnosis could be suspected in patients who had clinical signs and symptoms of acute appendicitis and a history of appendectomy [14]. Epiploic appendagitis may be primary caused by torsion or spontaneous venous thrombosis or secondary appendagitis caused from inflammation of adjacent organs, such as diverticulitis, appendicitis, cholecystitis. Epiploic appendixes are affected also by calcification due to aseptic fat necrosis, Pericolic abscess and incarceration in hernias [9]. Thomas et al reviewed 197 cases from the literature and 11 of their own cases of acute epiploic appendagitis and classified each according to its cause: torsion and inflammation (73%), hernia incarceration (18%), intestinal obstruction (8%), and intraperitoneal loose body (1%).

Torsion of cecal epiploic appendix is rarely diagnosed preoperatively due to the lack of pathognomonic clinical features which are nonspecific. Most patients manifests with acute onset localized abdominal pain of variable intensity and duration. There can be signs of peritoneal irritation [15]. The pain may be exacerbated by coughing, deep breathing or stretching because the infarcted appendage is adherent to the parietal peritoneum. Nausea, vomiting and loss of appetite are rare symptoms. In most of the cases can be marginally leukocytosis. In general, acute epiploic appendagitis mostly happen in sigmoid colon decreasing in frequency toward descending colon, cecum and ascending colon [9]. Depending on the site of occurrence, they can be confused other intra abdominal pathologies leading to clinical misdiagnosis in most patients. Acute epiploic appendagitis involving sigmoid colon can mimick diverticulitis of sigmoid colon, if proximal part of transverse colon is involved it may mimic acute cholecystitis[15], in the caecum it may mimic acute appendicitis or any other cause of acute pain in the right lower abdomen like regional enteritis, ovarian torsion or a ruptured ovarian cyst, salpingoophoritis, typhlitis and perityphlitis [16,17]. The condition is self limited, and the symptoms usually resolve within 1 week (mean of 4.7 days) without surgical treatment [2,18]. Conservative treatment with non-steroid anti-inflamatory medication is sufficient. Recognition of these conditions on CT images will prevent unnecessary hospital admission, antibiotic therapy, laboratory tests and unnecessary surgery. Typically, the epiploic appendages are visible on CT images when they are inflamed and/or surrounded by fluid. Accurate assessment of the CT features of these conditions is important for selecting the appropriate mode of management and preventing unnecessary hospital admission and surgery. CT images from less than 8% of patients evaluated for exclusion of sigmoid diverticulitis or appendicitis show features of primary acute epiploic appendagitis [7,19]. The presumed diagnosis of this condition is primarily based on the CT features of the oval lesion less than 5 cm in diameter with slightly higher attenuation than peritoneal fat and with surrounding inflammatory changes appearing as a fatty mass, which is connected to the serosal surface in antimesenteric side of colon and has [20-23]. Detection by ultrasound of a solid, well-delineated hyperechoic mass, at the site of maximum tenderness adjacent to the colonic wall and absence or lack of blood flow seen on color Doppler images may suggest the diagnosis [19,20,24]. Magnetic resonance findings include an ovoid mass with a central portion of the mass seen as hyperintense and peripheral as hypointense on T1 and T2- weighted images [2,25].

Nowadays, laparoscopy has been found to be useful for both diagnosis and treatment of cecalepiploica appendix torsion [11,26]. If the diagnosis is made upon exploration, it should be removed and do seromuscular inversion of the affected portion of gut should be performed [3].

Conclusion

In conclusion, torsion of cecal epiploic appendix continue to be difficult to diagnose. Almost all patients with acute abdominal pain caused from torsion of cecal epiploic appendix potentially will be misdiagnosed preoperatively as other causes of acute abdominal pain in RLQ especially with acute appendicitis. Pediatric surgeons should consider it more often as a potential cause of acute abdominal pain in RLQ in children’s especially in appendectomized patients and when associated with lack of laboratory and radiologic futures of acute appendicitis. Ultrasound and especially CT and MRI imaging techniques can potentially diagnose torsion of epiploic appendix. Since they are not used routinely in children with acute abdominal pain, except in very doubtful-confused and severe cases, preoperative diagnosis of cecal epiploic appendix torsion will continue to be difficult and almost all of them will be diagnosed intraoperatively. Since it can be treated conservatively, when suspected, it should be confirmed with CT or MRI examination of abdomen as a best diagnostic tool in order to avoid unnecessary surgical exploration. When encountered during surgical abdominal exploration especially when they are pedunculated and predisposed to torsion it should be removed together with “ no inflamed ” vermiform appendix to avoid potential confusion with other surgical causes of acute abdominal pain in RLQ in the future. It should be removed also in recurrent cases previously diagnosed and treated conservatively.

Authorship

All authors attest that they meet the current ICMJE criteria for Authorship.

Conflict of Interest

The following authors have no financial disclosures: (Isber Ademaj, Fisnik Kurshumliu, Kujtim Ukeperaj).

Authors Contributions

Isber Ademaj carried out design of the study, data collection, drafted the manuscript and was a major contributor in writing the manuscript. Kujtim Ukeperaj performed the literature review and was involved in writing the manuscript. Fisnik Kurshumliu performed histological examination of the epiploic appendage, supervision and analysis of the literature. All authors read and approved the final manuscript.

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