Adult Intussusception of Appendicular Mucinous Cystoadenoma

Abstract

Intussusception is a rare cause of adult intestinal obstruction. Clinical manifestations are not specific, which makes a preoperative diagnosis difficult to establish and often causes delay. We report a case of acute intestinal obstruction due to ileocolic intussusception. An emergency laparoscopy was performed revealing an obstructive mass at the ileocecal region resulting from an appendicular tumor. A right hemicolectomy was conducted. The pathologic examination of the resected sample concluded the mass comprised an appendicular mucinous cystoadenoma. We discuss the clinical features, preoperative diagnosis, and surgical strategies of adult intussusception, as well as the uncommon finding of an appendicular mucinous tumor as its causative lesion, with a review of the available literature.Key words: Adult intussusception -Appendiceal mucocele -Intestinal obstruction I ntussusception is the invagination of a proximal segment of bowel (intussusceptum) into the lumen of the adjacent distal segment (intussuscipiens). Pediatric patients account for 95% of all cases of intussusception, in whom it is usually idiopathic or the result of a viral process; whereas adult patients account for only 1% to 5%, and an organic lesion is found in up to 90% of the adult cases. 1,2Acute bowel obstruction due to intussusception is rare in adults and accounts for 0.003% to 0.02% of hospital admissions and for only 1% of all intestinal obstructions.1 Preoperative diagnosis is a real challenge for physicians because of its unspecific and extremely variable presentation and requires a high index of suspicion. Therefore, the initial diagnosis is usually missed or delayed and may only be established during surgery. [3][4][5] We present a very uncommon case of acute intestinal obstruction resulting from an ileocolic intussusception secondary to a mucinous tumor of the appendix and review the literature about this finding as a lead point. Case ReportA 57-year-old female presented to the emergency department with a 2-to 3-month history of intermittent and worsening abdominal pain, great- est in the right lower quadrant, which had increased gradually over the past 3 days. It was associated with dizziness and diarrhea but she denied any history of fever, nausea, vomiting, bloody stools, or weight loss. The patient's medical history included a groin hernia surgery and gastroesophageal reflux on oral treatment.Initial vital signs were within normal limits. Abdominal examination revealed diffuse tenderness on palpation, above all in the right lower quadrant, but without rebound or voluntary guarding. There was no palpable mass, and the auscultation found struggle bowel sounds. Blood tests were normal as was urine analysis.Abdominal X-ray exam was not demonstrative of intestinal obstruction. Computed tomography (CT) of the abdomen and pelvis showed a pathologic mass at the ileocolic region, of cystic appearance with parietal calcifications, with edematous pericolonic fat stranding suggesting of ileocolic intussusception causing an intestinal obstr...

Multiple intestinal hemangioma concurrent with low-grade appendiceal mucinous neoplasm presenting as intussusception—a case report and literature review

“…Based on the literature search using the term “appendiceal mucinous tumor” and “intussusception” from the articles in the PubMed database between January 2000 and February 2021, 29 articles [ 19 – 47 ] were obtained, among which 20 [ 19 – 21 , 23 , 24 , 26 – 40 ] were eligible after excluding 9 articles including 2 [ 22 , 47 ] in Japanese language and 1[ 25 ] in Dutch, 2 [ 41 , 42 ] involved the cases with no appendiceal mucinous tumor, 2 [ 44 , 46 ] with appendiceal mucinous tumor with no intussusception, 1 [ 45 ] with sigmoidorectal intussusception caused by a mucinous adenocarcinoma of the sigmoid colon, as well as 1 study [ 43 ] focused on the histological and imaging features. Among the remaining 20 articles, one article [ 26 ] mentioned 3 related cases in the literature review including 1 with LAMNs induced intussusception, 1 with intussusception caused by appendiceal mucocele [ 48 ], and 1 with intussusception caused by appendiceal mucocele combined with schistosomiasis [ 49 ]. Mucocele was non-neoplastic mucinous lesion characterized by markedly thinned or denuded mucosa combined with luminal dilatation, without atypia and hyperplasia.…”

Intususcepción apendicular por foco endometriósico. Un caso clínico.

“…La intususcepción de las vísceras huecas corresponde a la invaginación del segmento proximal del intestino dentro del segmento distal adyacente de la misma porción de intestino 1 . Cerca del 95% de todos los casos de intususcepción ocurre dentro de la población pediátrica, describiendo hasta un 5% de presentación en adultos, pudiendo encontrar alguna lesión orgánica como etiología en el 90% de los casos 6 .…”

“…La intususcepción del apéndice cecal es una condición en extremo rara en la medicina, con estimaciones de incidencia variadas en la población gene-ral, lo más aceptado es una cifra cercana al 0,01% 1 . Las patologías benignas son la principal causa de intususcepción apendicular (77%) 2 , dentro de este grupo la patología más frecuente es la endometriosis, dando origen a casi un tercio de los casos 3 .…”

Gastrointestinal hemorrhage caused by adult intussusception secondary to small intestinal tumors

“…Small intestinal intussusceptions are mainly caused by lesions in or outside the intestine, including adenomatous polyps, lipomas, stromal tumors, lymphoma, intestinal inflammation, Michael diverticulum, postoperative intestinal adhesions, adenocarcinoma, and metastatic carcinoma. Some rare lesions also occur, such as a neuroendocrine tumor, [ 9 ] angiolipoma, [ 21 ] myoepithelial hamartoma, [ 22 ] and appendiceal mucinous cystadenoma, [ 23 ] which have only been described in case reports. Adenocarcinoma accounts for approximately 30% of small intestinal intussusceptions, whereas colonic intussusceptions are mainly caused by malignant tumors.…”