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Chronic intermittent abdominal pain in young woman with intestinal malrotation, Fitz-Hugh-Curtis Syndrome and appendiceal neuroendocrine tumor: a rare case report and literature review.

ABSTRACT: There are a lot of different causes of abdominal pain; in this case, a young woman suffers from three diseases with similar symptoms. Adult intestinal mal-rotation is a rare condition of deviation from the normal 270° counter clockwise rotation of the midgut resulting in, not only mal-position of the small intestine, but also mal-fixation of the mesentery. Fitz-Hugh-Curtis syndrome is a rare complication of pelvic inflammatory disease; it involves liver capsule inflammation associated with genital tract infection, which is usually caused by Neisseria gonorrhoea and Chlamydia trachomatis. Neuroendocrine tumors are enterochromaffin cell neoplasms that arise from cells of the endocrine (hormonal) and nervous systems; the appendicular one is the most common primary malignant lesion of these tumors, it's incidence is about 0.3 - 0.9% of appendectomies done. Just for knowledge, this is the first described case of concomitant presence of all these diseases with clinical symptoms attributable to each one.40-years-old woman suffers from acute abdominal pain, predominantly on the right quadrants, without abdominal distension, no guarding nor rigidity and normal intestinal peristalsis. She has a long history of abdominal intermittent pain, with cramps every 30-40 min, resolving spontaneously. She was diagnosed as intestinal mal-rotation through computed tomography scan which has evidenced a mobilized intra--peritoneal duodenum with cecum/ascending colon predominately lying on the left side and the small intestine almost entirely lying on the right side of abdomen, without evidence of effusion, edema or signs of intestinal ischemia or infarction. Exploratory laparoscopy demonstrated an inflammatory process in the hepatic-renal space, with bloody adhesions above the liver capsule; this is additional to the typical pelvic inflammatory disease signs (Fitz-Hugh-Curtis syndrome). Appendectomy was performed with histological analysis resulting in appendicular neuroendocrine tumor.Although the patient has an intestinal mal-rotation which could explain the abdominal painful symptoms, it is not possible to exclude other concomitant causes, such as perihepatitis on pelvic inflammatory disease or neuroendocrine tumors. Even if all these diseases are rarely seen in daily clinical practice, they should be considered in the differential diagnosis of chronic intermittent abdominal pain in a young woman.

SUBMITTER: Cusimano A

PROVIDER: S-EPMC4715868 | biostudies-other | 2016 Jan

REPOSITORIES: biostudies-other

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Project description:Introductionand importance: Caecal volvulus represents 30% of colonic volvulus. It happens due to torsion or hyperflexion of a hypermobile caecum. Usually it is secondary to an axial rotation of the caecum and the ileum around the mesentery. On the other hand Intestinal malrotation occurs due to incomplete or faulty rotation and fixation of the gut during fetal life. The occurrence of these two anomalies together is scarse which makes this case report interesting.Case presentationA 75 year old man with medical history of terminal kidney failure, presented to the emergency room with an intestinal obstruction syndrome. On examination the patient had a distended abdomen with tenderness in the left upper quadrant. Biology found an important biological inflammatory syndrome with hyperleukocytosis and elevated CRP. Plain X-ray of the abdomen in erect posture showed an air fluid colonic level in the left hypochondrium. CT scan showed signs of caecal volvulus with intestinal malrotation. A brief reanimation and nasogastric aspiration couldn't solve the problem therefore emergency laparotomy was needed ileocaecal resection was performed associated with LADD's procedure in order to treat both anomalies and prevent further gut volvulus.Clinical discussionDespite it's rareness, caecul volvulus represents the second cause of large bowel volvulus just behind sigmoid volvulus. Intestinal malrotation in adults subjects is estimated to occur in 0.2-0.5%.The uniqueness of our case is that these two anomalies were associated in such a way that it made both the diagnosis and the therapy even more difficult. Abdominal CT has become mandatory for pre-operative diagnosis of intestinal volvulus. Surgery is the gold standard treatment for caecal volvulus. The usual options are manual detorsion, carcopexy, caecostomy and colectomy.ConclusionThis case reports a rare association of a caecum volvulus with intestinal malrotation that emphasis the place of modern technologies such as CT scan in order to achieve correct preoperative diagnosis. We also describe our approach to this uncommon surgical emergency in order to provide an efficient treatement.

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Chronic intermittent abdominal pain in young woman with intestinal malrotation, Fitz-Hugh-Curtis Syndrome and appendiceal neuroendocrine tumor: a rare case report and literature review.

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