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Home , Chilaiditi syndrome, Diaphragmatic hernia

Eur opean Rev iew for Med ical and Pharmacol ogical Sci ences 2014; 18: 1694-1697 “Left-sided” Chilaiditi sign? A large gastric perforation with secondary

S. RAI, S. YAHIA, G. GRAVANTE, A. PALIT 1, K. MARIMUTHU, G. MATHEW

Department of Colorectal Surgery, George Eliot Hospital, Nuneaton, United Kingdom 1Department of Radiology, George Eliot Hospital, Nuneaton, United Kingdom

Abstract. – BACKGROUND: Although the nosed as a and leads to un - Chilaiditi sign correctly underestimates the radi - necessary surgical interventions. Chilaiditi syn - ological presence of air-under-the-diaphragm, in drome is recognised as the association of the ra - few cases it has lead to delays in the diagnosis diological sign with complications such as ab - of underlying pneumoperitoneum. In this article 3 4 5,6 we report the case of a young lady presenting dominal pain , or respiratory distress . with pancreatitis and radiographic find - Chilaiditi sign has been reportedly linked to ings of “left-sided” Chilaiditi sign with underly - anatomical, pathological factors and certain dis - ing a large gastric perforation. eases. Anatomical factors range from malfixation CASE REPORT: A 35 year old Caucasian fe - or malposition of bowel, redundant or elongated male presented to the Emergency Department 7-9 with a twenty-four hour history of severe epigas - bowel, laxity of hepatic suspensory ligaments . tric pain radiating into her back. Stable observa - In all these cases the underlying anatomical vari - tions, mildly raised white cell count, increased ant allows for augmentation of the space between amylase and subdiaphragmatic radiolucency the and the diaphragm, thus, allowing for in - were interpreted as with Chi - terposition of bowel in this space. Pathological laiditi sign and treated accordingly. Twenty-four factors include conditions such as liver , hours later the deterioration of the clinical condi - tions required a CT scan and an emergency la - pregnancy and chronic obstructive airways dis - parotomy that lead to the diagnosis of a large ease as these result in reduced liver volume. An gastric perforation. However, after several days increased abdominal pressure or enlargement of she died of a disseminated intravascular coagu - the lower thoracic cavity are also postulated lation in the Intensive Care Unit. mechanisms indicated in the predisposition to CONCLUSIONS: In our case the young age of 7-9 the patient and lack of underlying comorbidities Chilaiditi sign . More recently, Chilaiditi sign initially compensated the severity of the perfora - has been described in a case of acute pancreatitis tion until it became evident from the absent res - probably due to the paralytic often associat - olution of the pancreatitis. Bearing this in mind, ed with this disease (“sentinel loop”) and the con - radiological appearances similar to Chilaiditi sequent bowel dilatation and eventual interposi - sign with significant symptoms or signs should tion between the diaphragm and the liver 10 . In this be investigated with a CT scan in order to reach promptly a correct diagnosis. article we report the case of a young lady present - ing with a clinical picture of acute pancreatitis Key Words: and radiographic findings similar to the Chilaiditi Chilaiditi sign, Gastric perforation, Acute pancreatitis . sign that masked a large gastric perforation.

Case Report Introduction A 35 year old Caucasian female presented to the Emergency Department with a twenty-four The incidental radiographic finding of subdi - hour history of severe epigastric pain radiating aphragmatic radiolucency due to bowel inter - into her back. She had complained of feeling posed between the liver and the right hemidi - nauseous for one day and had two episodes of aphragm is named Chilaiditi sign, first described prior to attending the Emergency De - by Demetrius Chilaiditi in 1910 when he was partment. working in Vienna 1,2 . The occurrence is reported Since the summer of 2011 she had noted as 0.025-0.28% of the general population 2 and its symptoms of early satiety and inability to take importance lies in the fact that it can be misdiag - liquids first thing in the morning. She noted that

1694 Corresponding Author: Gianpiero Gravante, MD; e-mail: [email protected] “Left-sided” Chilaiditi sign? A large gastric perforation with secondary pancreatitis shortly after ingesting solids she would begin to vomit. Occasionally she described waking in the morning with a sensation of vomit in her mouth. Over a period of six months from October 2 011, she had lost four stones. On March 2012 she was admitted to the Accident and Emergency Depart - ment for multiple episodes of vomiting and re - ceived an esophogastroduodenoscopy that showed significant looping in the stomach and inability to pass the scope into the , signs suspicious for paraoesophageal and . She was discharged home with the request for an urgent barium meal. This was done few days later and showed the antrum locat - ed anteriorly and superiorly and the posterior sur - face of the stomach anteriorly, in keeping with a mesentero axial volvulus. One week later she presented to the Emergency Department with re - current vomiting being unable to tolerate solids or liquid. She underwent an emergency laparoto - Figure 1. Chest X-ray on admission. Although diagnosis my for an incarcerated con - on admission was mistaken for Chilaiditi sign, a postopera - taining the fundus of the stomach. It was noted tive review of the images found an elevated left hemidi - there was a congenital opening in the diaphragm aphragm and free air under the left hemidiaphragm consis - tent with perforation of the bowel. approximately 6 × 6 cm which was closed using Ethilon sutures. A Nissen fundoplication was car - ried out and feeding jejunostomy tube inserted. Post-procedure she progressed well with removal She also manifested severe and of the feeding tube and discharged home two abdominal examination showed generalised weeks post-operatively. One month after the op - guarding with a distended . An arterial eration she was seen in an Outpatient Clinic and blood gas revealed a pH 7.17, PaCO 2 6.5 kPa, stated that she had on eating quickly and PaO 2 14.4 kPa, Na 130 mmol/L, K 4.6 mmol/L, had to avoid certain foods. lactate 4.7 mmol/L, BE –11.3 mmol/L, HCO 3 Six months after her surgery, on the present 17.5 mmol/L. A CT abdomen and pelvis scan re - admission, she had significant amounts of ab - vealed extensive and free intraperitoneal dominal pain, located in the epigastrium and ra - air, indicative of bowel perforation and features diating into her back. Her abdomen was tender in suggestive of pancreatitis. the epigastrium, otherwise soft and non-distend - She was taken to the operating room for an ed in the remaining quadrants . The blood tests re - emergency exploratory laparotomy. Laparotomy vealed an elevated amylase 652 IU/L (normal < revealed an acute gastric perforation of approxi - 101 iU/L), a mildly elevated white cell count mately 10 cm x 4 cm on the fundus of the stom - 9 9 11.78 × 10 /L (normal 4.00-11.00 × 10 /L) and ach towards the greater curvature with significant neutrophils count 9.54 10 9/L (normal 2.00-7.00 extravasation of gastric contents. The gastric per - 9 × × 10 /L). Chest X-ray showed “left-sided” Chi - foration appeared necrotic in origin without any laiditi sign and abdominal X-ray no specific other gastric pathology. An emergency sleeve bowel pattern (Figure 1); therefore, she was diag - gastrectomy was performed and the abdomen nosed with acute pancreatitis and scored mild us - was washed out and closed. Post-operatively she ing the Glasgow prognostic criteria. Initial treat - remained on the intensive care unit (ICU) for ment consisted in supplemental oxygen adminis - three days where she was supported with maxi - tered via nasal cannula , fluid resuscitation with mum dose inotropes and intravenous antibiotics. IV fluids, catheterisation with urine output moni - She remained septic with temperature of 40.0°C toring, and patient -controlled -analgesia (PCA) and eventually became anuric and developed using morphine. Her condition deteriorated DIC disseminated intravascular coagulation (not - overnight when she developed sinus tachycardia, ed on post mortem examination). Three days lat - tachypnoea and a raised temperature (39.0 °C ). er she died.

1695 S. Rai, S. Yahia, G. Gravante, A. Palit, K. Marimuthu, G. Mathew

Discussion well as any anatomical abnormality that may ex - plain her sequence of events. However , no defini - Hepatodiaphragmatic interposition of the tive outcome was reached. colon can be confused with pneumoperitoneum One potential explanation for the gastric perfo - and subphrenic abscesses radiologically and lead ration and acute pancreatitis is the occurrence of to unnecessary surgical intervention if not recog - closed loop obstruction on a background of the nised correctly. There are several points of dis - Nissen fundoplication and possible co-existence tinction to note between this condition and that of superior mesenteric artery (SMA) syndrome. of a pneumoperitoneum or subphrenic abscess. The syndrome is characterised by compression of Haustral markings in the subdiaphragmatic air the third, or transverse portion of the duodenum are present and the positional orientation of the against the aorta by the SMA secondary to a nar - patient will not change the location of the subdi - rowing of the angle between these two vessels 19 . aphragmatic air in the hepatodiaphragmatic in - It leads to acute or chronic intermittent partial or terposition of the colon 11,12 . complete duodenal obstruction 20,21 with an ap - In our patient the showed proximate incidence of 0.013-0.3% 22,23 . However, subdiaphragmatic radiolucency on a clinical at post-mortem examination, no such evidence background of stable vital parameters, raised was found supporting this diagnosis in our pa - amylase and mildly raised white cell count. This tient. lead to the diagnosis of acute pancreatitis with “left-sided” Chilaiditi sign, as previously re - ported 10 . The Chilaiditi sign has been intro - Conclusions duced to correctly interpret the clinical meaning of the radiological sign of air-under-the- In this patient the young age and lack of under - diaphragm on the right side and avoid an erro - lying comorbidities initially compensated the neous diagnosis of perforation . However, there severity of the perforation until it became evident have been various cases where an underlying from the absent resolution of the pancreatitis. Bear - pathology was not promptly detected 1,13-15 . The ing this in mind, radiological appearances similar presence of normal vital parameters does not to Chilaiditi sign with significant symptoms or necessarily exclude bowel perforation and the signs should be investigated with a CT scan in or - per se in certain patients der to reach promptly a correct diagnosis. may mask an underlying significant pathology. Where in doubt, suspicious symptoms should ––––––––––––– –– ––– –– require further evaluations to rule out underly - Conflict of Interest ing perforations. The Authors declare that they have no conflict of interests. Our patient had a previous laparotomy where a Nissen fundoplication was carried out to repair a congenital incarcerated diaphragmatic hernia , in - References crease the lower oesophageal sphincter pressures and decrease episodes of reflux 16 . The Nissen 1) ANTONACCI N, D I SAVERIO S, B ISCARDI A, G IORGINI E, procedure has low post-operative complication VILLANI S, T UGNOLI G. Dyspnea and large bowel ob - 17 struction: a misleading Chilaiditi syndrome. Am J rates as well as a low morbidity and mortality Surg 2011; 202: e45-47. but there have been cases where gastric or oe - 2) ALVA S, S HETTY -A LVA N, L ONGO WE . Image of the sophageal perforations have resulted during a month. Chilaiditi sign or syndrome. Arch Surg Nissen fundoplication. A review by Perkidis et 2008; 143: 93-94. 18 al highlighted 25 patients (1%) with an intraop - 3) GLATTER RD, A PRIL RS, M ISKOVITZ P, N EISTADT LD. Se - erative gastric perforation and 15 patients (0.6%) vere recurrent abdominal pain: an anatomical requiring conversion from a laparoscopic to an variant of Chilaiditi's syndrome. MedGenMed open procedure out of 2453 cases of patients un - 2007; 9: 67. dergoing a Nissen fundoplication. No study or 4) PLORDE JJ, R AKER EJ. volvulus and associated Chilaiditi's syndrome: case report and review literature was found identifying gastric literature review. Am J Gastroenterol 1996; 91: perforation as a late post-operative complication. 2613-2616. During post-mortem examination of our patient, 5) KELES S, A RTAC H, R EISLI I, A LP H, K OC O. Chilaiditi attempts were made to look at the previous di - syndrome as a cause of respiratory distress. Eur aphragmatic repair and Nissen fundoplication as J Pediatr 2006; 165: 367-369.

1696 “Left-sided” Chilaiditi sign? A large gastric perforation with secondary pancreatitis

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