Introduction

Chilaiditi’s sign, or the incidental radiographic finding of bowel positioned between the liver and the right diaphragm, was first documented in 1865[2] . This sign was named in 1910 when Demetrius Chilaiditi reported three cases of asymptomatic patients presenting intra-abdominal free air on routine abdominal or chest x-ray due to the interposition of the bowel between the liver and the right hemidiaphragm [2,3]. Chilaiditi sign is found incidentally in 0.025%–0.28% of chest and abdominal plain films[4] and 1.18%2.4% of abdominal Computed Tomography (CT) scanswith a male predominance (male to female, 4:1) [1,5]. It is more common in the elderly population  where the incidence is approximately 1%. [2]. This report describes a case of a 65 years old female with repeated abdominal pain and acute intestinal obstruction. Chilaiditi Syndrome was diagnosed through radiological finding of interposition of ileal bowel between liver and right diaphragm and treated by a laparoscopic approach.

Case Presentation

A 65-year-old female was admitted to the Emergency Department of the San Leonardo Hospital (Castellammare di Stabia, Napoli) in January 2023 for intermittent upper abdominal pain for more than ten days and inability to pass gas from three days. The symptoms were associated with abdominal distension, nausea and vomiting. Past medical history was significant for hypertension, hypothyroidism and colonic diverticula; past surgical history for one caesarean section and hysteroannexectomy. Patient denied alcohol and illicit drug use or drug allergies. Family history was noncontributory. At the time of clinical evaluation, vital signs were normal: temperature was 36.7°C, blood pressure 145/80 mmHg, pulse 89 beats/minute, respiratory rate 18 beats/minute and oxygen saturation 98% on room air.

Physical examination revealed rebound abdominal tenderness with muscle guarding and hypoactive bowel sounds.

Basic laboratory investigations were ordered. A basic metabolic panel showed that the electrolyte levels were within normal limits except for a low potassium (3.40 mmol/L). Complete blood count and kidney function tests were within normal limits.

Anterioposterior abdominal and chest X-ray showed an air collection under the right diaphragmatic dome (Figure 1). Abdominal contrast enhanced CT scan showed an ileal loop located between the diaphragm and liver corresponding to the subdiaphragmatic air collection detected by X-ray, with normal mural enhancement of the involved intestinal segment, several distended jejunal loops with air-fluid levels and a fair amount of ascitic fluid in the pelvic excavation. An emergency surgery was indicated for suspected acute small bowel obstruction after failure of a conservative management with bowel decompression by a nasogastric tube placement and IV fluid hydratation. (Figure 2).

Figure 1. Abdominal-chest X-ray: air collection under the diaphragm.

Figure 2. Abdominal CT scan: Ileal loop beneath the right diaphragmatic dome

Emergency surgery was performed with a laparoscopic approach. The patient was placed in the supine position with the lower limbs spread apart and the left arm abduced, with the first operator between patient’s legs and second operator to his left. An 12-mm Hasson port was inserted through the periumbilical region using the open technique and pneumoperitoneum was established using dioxide insufflation. Two additional 5-mm trocars were positioned under laparoscopic observation through the right and left upper quadrants. Surgical exploration confimed the presence of several distended bowel loops, a fair amount of ascitic fluid and a closed bowel loop incarcerated by a tick band of scar tissue. Section of the hepatophrenic tick band adhesion constricting the ileal loop was performed with resolution of the adhesive small bowel obstruction (ASBO). No sign of bowel injury were noted. Repositioning of bowel loop, aspiration of the ascitic fluid and drain pelvic placement completed the surgical operation (Figure 3). Laparoscopic procedure and postoperative period were uneventful. The drain was removed on the second post-operative day and the patient was discharged on the fourth post-operative day with no complications.

Figure 3. Entangled ileal bowel from hepatophrenic tick band adhesion.

Discussion

A first case of Chilaiditi syndrome due to ileal bowel interposition was described from a retrospective analysis of works since 1976, where four instances of Chilaiditi syndrome have been found (three males and one female). Interposition of the proximal transverse colon was found in three patients and the small intestine was involved in one patient. [6]. In Chilaiditi’s syndrome, the hepatic flexure is the most common portion of the colon entangled between the diaphragm and the liver, the second most common being the ascending or transverse colon. Only a few cases of small intestine involvement have been reported (3%-5% of cases) [7].

The etiology can be either congenital or acquired. Congenital factors may be an elongated, redundant and hypermobile colon, laxity or absence of suspensory ligaments. Acquired causes may include a small liver due to atrophy in cirrhosis or hepatectomy, presence of ascites, substantial weight loss in obese patients, and an abnormally high diaphragm in conditions such as diaphragmatic muscular degeneration or phrenic nerve injury and, rarely, excessive aerophagia [8,9]. Other factors include enlargement of the lower thoracic cavity (chronic obstructive pulmonary disease), increased intra-abdominal pressure (obesity, multiple pregnancies and ascites), mental retardation and schizophrenia, which are also associated with anatomic abnormalities that result in Chilaiditi sign [10]. Intraperitoneal adhesion caused by previous surgery or widepread tumor local metastasis are also encountered [11]. In addition, psychotropic medication [12] and iatrogenic factors [13], such as endoscopic procedures, have been reported as causative factors.

A characteristic radiological marker of Chilaiditi sign is the observation of air below the diaphragm, with visible haustral folds or valvulae conniventes between the liver and the diaphragmatic surface. In addiction, the location of the air is not changed by altering the posture of the patient.

In patients presenting with Chilaiditi syndrome the most common symptoms are gastrointestinal such as abdominal pain, anorexia, nausea, vomiting and constipation  followed, less frequently, by respiratory distress or cardiac angina [10].

Interventions are not required for asymptomatic patients with Chilaiditi sign and the treatment is usually conservative. Management of non complicated Chilaiditi syndrome in most cases is conservative.

The complications of Chilaiditi syndrome may include intestinal obstruction, volvulus and ischemia/perforation. In rare cases perforated subdiaphragmatic appendicitis may occur as a complication of Chilaiditi syndrome [14,15].

In cases with obstructive complications, the treatment is modulated according to Guidelines for treatment of adhesive small bowel obstruction [16]: non-operative management should be tried in absence of signs of peritonitis, stragulation or bowel ischemia using nihil per os, bowel decompression by a naso-gastric tube or long intestinal tube and intravenous fluid. Water-soluble contrast studies are encouraged because have been shown to reduce the need of surgery as is attributed an active therapeutic role to the contrast.

In case of failure of initial conservative management and either the obstruction fails to resolve or there is evidence of bowel ischemia or other complications, emergency surgical intervention is indicated.

The potential benefits of a laparoscopic approach in the treatment of ASBO include less extensive adhesion formation, earlier post-operative recovery, reduced post-operative pain and short lenght of hospital stay. In an abdomen with very distended loops of bowel and multiple complex adhesions, the risk of severe complications such as enterotomies and delayed diagnosis of perforations could increase [17].

Regarding this issue, Farinella et al. [18] reported that predictor factors for a successful laparoscopic approach to ASBO with reduced complication rate are: ≤ 2 laparotomies, no previous median laparotomy and the presence of a single adhesive band.

In the case reported, the patient was affected by a Chilaiditi syndrome complicated by an intestinal obstruction which has required surgical management. The laparoscopic approach was used in view of the moderate distension of bowel loops and the poor extent of adhesions in the upper abdomen.

Mininvasive surgery allowed the diagnostic confirmation of a band adhesion entangling a small bowel loop and the treatment of the obstructive syndrome without complications, with good outcomes in terms of reduced pain, quick post-operative recovery and short duration of hospital stay.

Conclusions

Chilaiditi sign and Chilaiditi syndrome are rare entities and are therefore often misdiagnosed in clinical practice.

Conservative management is adequate in uncomplicated patients. In complicated cases by volvulus, obstruction and ischemia/perforation, emergency surgery is mandatory. In the case reported, the patient was affected by a Chilaiditi syndrome complicated by an adhesive small bowel obstruction (ASBO) which has required surgical management. The laparoscopic approach allowed the diagnostic confirmation of a band adhesion entangling a small bowel loop and the treatment of the obstructive syndrome without complications with good outcomes in terms of reduced pain, quick post-operative recovery and short lenght of hospital stay.

References

1. Amy X Yin MD,  Gavin H Park BS, Gwendolyn M Garnett MD, John F Balfour MD (2012) Chilaiditi Syndrome Precipitated by Colonoscopy: A Case Report and Review of the Literature Hawaii J Med Public Health 71: 158-162.
2. Farkas R (2008) Chilaiditi’s sign in a blunt trauma patient: a case report and review of the literature. J Trauma 65: 1540-1542.
3. Chilaiditi D (1910) Zur Frage der Hapatoptose und Ptose in allgemeinen im Auschluss an drei FŠlle von temporŠrer partieller Leberverlagerung. Fortschritte auf dem Gebiete der Roentgenstrahlen 16: 173-208.
4. Aldoss IT (2009) Chilaiditi syndrome complicated by cecal perforation. South Med J 102: 841-843.
5. Keles S (2006) Chilaiditi syndrome as a cause of respiratory distress. Eur J Pediatr 165: 367-369.
6. Risaliti A (1993) Chilaiditi’s syndrome as a surgical and nonsurgical problem. Surg Gynecol Obstet 176: 55-58.
7. Nair N, Takieddine Z, Tariq H (2016) Colonic interposition between the liver and diaphragm: “the Chilaiditi sign”.  Can J Gastroenterol Hepatol 2016: 2174704.Farkas R, Moalem J, Hammond J (2008) Chilaiditi’s sign in a blunt trauma patient: a case report and review of the literature.J Trauma 65: 1540-1542.
8. Farkas R, Moalem J, Hammond J (2008) Chilaiditi’s sign in a blunt trauma patient: a case report and review of the literature.J Trauma 65: 1540-1542.
9. Weng WH, Liu DR, Feng CC, Que RS (2014) Colonic interposition between the liver and left diaphragm - management of Chilaiditi syndrome: a case report and literature review.Oncol Lett 7: 1657-1660.
10. Moaven O, Hodin RA (2012) Chilaiditi syndrome: a rare entity with important differential diagnoses. Gastroenterol Hepatol NY 8: 276-278.
11. Hu JL (2007) Diagnosis, differential diagnosis and clinical significance of hepatodiaphragmatic interposition of the colon. Hai Nan Yi Xue Bian Ji Bu 8: 145.
12. Blevins WA, Cafasso DE, Fernandez M, Edwards MJ (2011) Minimally invasive colopexy for pediatric Chilaiditi syndrome. J Pediatr Surg 46: e33-e35.
13. Aldoss IT, Abuzetun JY, Nusair M, Suker M, Porter J (2009) Chilaiditi syndrome complicated by cecal cerforation. South Med J 102: 841843.
14. Lenz M, Kindler M, Schilling M, Pollack T, Schwab W, et al. (2011) Chilaiditi’s syndrome complicated by subdiaphragmatic perforated appendicitis: unusual manifestation of a rare condition. Chirurg 82: 830-833.
15. Kang D, Pan AS, Lopez MA, Buicko JL, Lopez-Viego M (2013) Acute abdominal pain secondary to chilaiditi syndrome. Case Reports in Surgery 2013: 3.
16. Ten Borek RPG, Krielen P, Di Saverio S, Coccolini F, Biffi WL (2018) Bologna guidelines for diagnosis and management of adhesive small bowel obstruction (ASB0): 2017 update of rhe evidence-based guidelines from the world society of emergency surgery ASBO Working Group. World J Emerg Surg 13:24.
17. Strik C, Stommel MW, Ten Broek RP, Van GH (2015) Adhesiolysis in patients undergoing a repeat median laparotomy. Dis Colon Rectum 58:792–8.
18. Farinella E, Cirocchi R, La MF, Morelli U, Cattorini L, Delmonaco P, et al.(2009) Feasibility of laparoscopy for small bowel obstruction. World J Emerg Surg 4:3.