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Soonchunhyang Med Sci > Volume 18(2); 2012 > Article
Cho, Lee, Kim, Jang, Jung, and Woo: Chilaiditi’s Sign in the Colon of Adult Patients: A Case Series and Literature Review Including 55 Cases

ABSTRACT

Chilaiditi’s sign refers to a condition in which the right colon or, rarely, the small intestine is interposed between the liver and the right hemidiaphragm. Recognizing this sign is important, because it may present with a variety of abdominal symptoms and may be confused with a surgical abdomen leading to unnecessary surgical intervention. Management is usually conservative unless complications such as volvulus or obstruction occur. Previous reports focused on the complications and surgical management of Chilaiditi’s sign. We present a report of three cases of Chilaiditi’s sign that were managed non-surgically, together with an updated review of the English literature.

INTRODUCTION

Normal anatomy between the liver and colon does not permit hepatodiaphragmatic interposition of the colon [1,2]. Nevertheless, it does occur in rare conditions and is called Chilaiditi’s sign. This condition was first described by Beclere in 1899; the term “Chilaiditi’s sign” has been used since Chiliditi reported three patients in 1910 [2]. When accompanied by symptoms it is classified as Chilaiditi’s syndrome [3].
Recognizing this sign is important because it can be easily mistaken for a pneumoperitoneum leading to unnecessary surgical intervention [4]. Data on Chilaiditi’s sign or syndrome in the literature was presented mostly in the form of case or imaging report. Moreover, previous reports have focused on the complications and surgical management.
We experienced three cases of Chilaiditi’s sign that did not require surgery. In this report, we present three cases of non-surgically managed Chilaiditi’s sign of the colon and provide a review of the English literature.

CASE REPORTS

1. Case 1

A 76-year-old man presented with generalized edema, abdominal distention and vomiting for the past month. He had suffered from diabetes mellitus for 24 years and had no surgical history. Physical examination revealed a distended abdomen with right upper quadrant tenderness. No peritoneal irritation signs were present. A complete blood count showed: white blood cells, 3,400/mm3; hemoglobin, 7.5 g/dL; hematocrit, 22.1%; and platelets, 60,000/mm3; morphological characteristics of a peripheral blood smear demonstrated macrocytic anemia. While his folate level was normal, vitamin B12 was depressed to 105 pg/mL (reference range, 200 to 950 pg/mL). Chest radiograph showed an air-distended contour under the right hemidiaphragm with dilated bowel loops (Fig. 1A). Abdominal computed tomography (CT) scan demonstrated a distended large bowel between the diaphragm and liver without an obstructive lesion (Fig. 1B). Further biochemical and instrumental investigations, including colonoscopy, revealed no underlying causes. With typical clinical manifestation, supported by radiologic findings and after exclusion of organic lesions occluding the gut lumen, he was finally diagnosed Chilaiditi’s syndrome with chronic intestinal pseudo-obstruction and vitamin B12 deficient anemia. Along with vitamin B12 supplementation, he underwent conservative treatment including nihil per os (NPO), nasogastric tube, repeated enemas, and taking prokinetics. Ileus and the patient’s general condition improved simultaneously. He recovered well, proceeded to an oral diet, and was discharged from the hospital without any sequele 23 days after admission.

2. Case 2

A 64-year-old man with hepatitis B viral liver cirrhosis, presented with anorexia while taking amoxicillin/clavulanate for TB lymphadenitis. His physical examination was unremarkable. Chest radiography revealed elevation of the right hemidiaphragm and the presence of subphrenic interposition of the colon between the liver and diaphragm. CT scan confirmed this sign, in which the colon was observed in front of the liver. Amoxicillin/clavulanate was changed to isonizid, and all symptoms improved concurrently. It was finally shown that the patient’s present symptoms were unrelated to the colonic interposition. Because the cause of his symptoms was medication-related, we would use the term Chilaiditi’s sign instead of Chilaiditi’s syndrome for this patient’s diagnosis. He is currently under outpatient follow-up without any symptoms or complications.

3. Case 3

A 67-year-old man with hepatitis C viral liver cirrhosis and hepatocellular carcinoma admitted due to hepatic encephalopathy. His abdomen was distended with ascites. No tenderness or guarding was present. Chest radiography and CT scan revealed colon air shadow in the space between the right diaphragm and the liver. His symptoms were irrelevant to the radiologic findings, thus our impression was an incidental finding of Chilaiditi’s sign secondary to advanced liver cirrhosis and ascites. He was managed conservatively for the ascites and cancer, but expired due to hepatic failure.

DISCUSSION

The frequency of Chilaiditi’s sign varies in the general population from 0.025% to 0.28% based on upright chest radiographs [2]. It is known to increase in incidence with age and has a male predominance [5].
The PubMed database was searched for articles that reported Chilaiditi’s sign or syndrome (Fig. 2) using the following search criteria: Chilaiditi’s sign OR Chilaiditi’s syndrome OR hepatodiaphragmatic interposition. We limited our searches to studies in humans that were >19 years old and were published in English between January, 1945 and March, 2012. Studies were excluded if they described surgically managed or complicated Chilaiditi’s syndrome or if treatment modalities were not available. We finally summarized the details of 55 non-surgically managed patients along with the current three cases with emphasis on clinical presentation, diagnosis, treatment, and outcome (Table 1).
Thirty six (62%) out of fifty eight patients were male. The mean age was 60.3 years with a range of 20 to 86 years. Forty-one (70.6%) patients had relevant symptoms. Abdominal pain, constipation, nausea, and vomiting were common symptoms (53.4%, 31%, 24.1%, respectively). Seventeen (29.3%) asymptomatic patients didn’t require any treatment for the Chilaiditi’s sign. Thirty-eight and (65.5%) patients with symptoms were managed conservatively including bowel decompression, intravenous hydration, stool softeners, and prokinetics. Three patients (5.1%) underwent exploratory operation for incorrect diagnosis. All patients who underwent medical treatment achieved symptomatic improvement excluding two cases that didn’t state the patient’s clinical outcome. Documentation about the radiological outcomes was available for eight patients, and three of these had radiological resolution along with symptomatic improvement.
Chilaiditi’s sign can be temporary or permanent. Even if the symptoms disappear, displacement may persist [6]. Predisposing factors are hypothesized to relate to conditions that increase colonic mobility in conjunction with an enlarged potential suprahepatic space [5,7] These can be categorized by intestinal, diaphragmatic, and hepatic contributing factors [8]. Intestinal factors include a redundant colon with long mesentery or partial obstruction. A possible diaphragmatic factor include abnormal upright positioning of the diaphragm due to phrenic nerve injury and changes in intrathoracic pressure, as in cases of emphysema [2,5]. Hepatic factors include a small liver, such as that due to cirrhosis [5,7,9]. Some reports mention that neurodevelopmental abnormalities, such as schizophrenia and mental retardation, may cause interposition due to the tendency toward constipation and meteorism [5,8]. Although it is difficult to prove a causal relationship, cases 2 and 3 had liver cirrhosis which is a well known risk factor. However, case 1 presented in a patient with chronic intestinal pseudo-obstruction due to bowel habit change. To our knowledge, this is the second case of chronic intestinal pseudo-obstruction as a cause of Chilaiditi’s sign [10] reported to-date. Such rare conditions can make the diagnosis and treatment difficult and therefore, it is important for the physician to understand that Chilaiditi’s sign may occur without typical risk factors.
On plain radiography, Chilaiditi’s sign presents as “air under the diaphragm” which can simulate a pneumoperitoneum from a perforated viscous [7]. Indeed, our PubMed search indicated three patients who were initially misdiagnosed with a surgical abdomen and underwent an exploratory operation [1113]. Additionally, one patient recovered completely with conservative treatment because he refused surgery [14]. Another important condition to distinguish is a subphrenic abscess, but the differential diagnosis can be made by simple radiography. Pneumoperitoneum is usually observed on both sides below the diaphragm, and the air shadows move according to body position [7]. A subphrenic abscess usually presents as a relatively small air-fluid level under the unilateral side of the diaphragm, without intestinal haustral marking, and is often accompanied by pleural effusion on the same side [7,15]. In cases of doubt, an abdominal CT scan will help confirm the diagnosis [7].
Treatment is required when symptoms occur. The hallmark of therapy is conservative, including fluid supplementation, nasogastric decompression, and enemas [5]. Surgical management is rarely needed for complications such as volvulus or obstruction [1,9]. In our cases, one patient (case 1) achieved both clinical and radiologic improvement by symptomatic measures, whereas the other two patients (cases 2 and 3) presented with the sign as an incidental finding and, hence no specific treatment was implemented. In the latter two cases, the sign itself persisted but relevant symptom did not occur.
Currently, there is no known pharmacologic or mechanical strategies to prevent the recurrence. Controlling the predisposing factor such as emphysema or liver cirrhosis maybe helpful. If the risk factor is not modifiable, preventive measures to reduce additional risk factor such as constipation should be taken.
We emphasize that recognizing this sign is important. First, the failure to recognize this sign may give rise to unnecessary examinations or, even worse, emergency surgery. Second, identifying this sign is mandatory before a percutaneous transhepatic procedure to avoid intestinal injury leading to pneumoperitoneum. Last, Chilaiditi’s sign can be a cause of potential abdominal symptoms but rarely a complication that requires emergency surgical management.

Fig. 1
Chilaiditi’s syndrome associated with a chronic intestinal pseudo-obstruction. (A) Chest X-ray showing an air-distended viscous under the right hemidiaphragm with dilated bowel loops. (B) Abdominal computed tomography scan showing the large bowel between the diaphragm and liver with a paralytic ileus without an obstructive lesion.
sms-18-2-13-f1.tif
Fig. 2
Flow of case reports through the search and selection process.
sms-18-2-13-f2.tif
Table 1
Summary of the clinical features and outcome of cases with Chilaiditi’s sign
Characteristic Value
Sex (n= 58) Male 36 (62)
Age (yr, n= 58) Mean (range) 60.3 (20–86)
Age over 60 38 (67)
Underlying disease according to contributing factors (n= 58) Intestinal factora) 4 (6.8)
Diaphragmatic factorb) 4 (6.8)
Neurological factorc) 18 (31)
Hepatic factord) 5 (15.6)
Procedure relatee) 1 (3.1)
Not available 26 (44)
Symptom onset (n= 41) < 7 day 11 (26.8)
> 7 day 9 (21.9)
Not available 21 (51.2)
Presenting symptoms (n= 58) Asymptomatic 17 (29.3)
Abdomen pain 31 (53.4)
Constipation 18 (31)
Nausea/vomiting 14 (24.1)
Respiratory distress 6 (10.3)
Anorexia 4 (6.8)
Physical sign (n= 58) Normal 14 (24)
Abdomen distension 22 (37.9)
Right upper quadrant tenderness 13 (22.4)
Decreased bowel sound 4 (6.8)
Not available 12 (20.6)
Final diagnosis (n= 58) Chilaiditi’s sign 17 (29.3)
Chilaiditi’s syndrome 41 (70.6)
Management Observation 17 (29.3)
Conservative treatmentf) 38 (65.5)
Exploratory operation due to incorrect diagnosis 3 (5.1)
Radiologic outcome (n= 8) Documentation of radiologic outcome available 8
Radiologic resolution 3 (3.7)
Outcome of symptomatic patients (n= 41) Not available 2 (7)
Symptom relief 39 (90.2)

Values are presented as number (%).

a) Intestinal factors included were gastrointestinal tract malignancy and pseudo-obstruction.

b) Diaphragmatic factors included were diaphragmatic paralysis, eventuation of right hemidiaphragm, and emphysema.

c) Neurologic factors included were Parkinson’s disease, Alzheimer’s disease, schizophrenia, and mental retardation.

d) Hepatic factors were hypoplastic right lobe of liver and liver cirrhosis.

e) Procedure related case was associated with enteral feeding tube inserted with endoscopy.

f) Conservative measures included bowel decompression, nihil per os, enemas, nasogastric tube, intravenous hydration, stool softeners, prokinetics, and pain killers.

REFERENCES

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