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Chilaiditi Syndrome: A Case of Recurrent Respiratory Distress be Multiply Misdiagnosed Gao Yong-shun, Zhang Yun-fei, Feng Hai-lin, Zhang Yu-kun Keywords Chilaiditi syndrome, Respiratory distress, Diagnostic errors A 61-year-old woman was admitted to the First Affiliated Hospital Vasculocardiology Department of Zhengzhou University with recurrent respiratory distress. Her symptoms generally worsen at night when the patient lies supine, and which was partly alleviated when the patient had a seat or stand a few minutes, without fever, cough, phlegm, chest pain and hemoptysis, which had persisted during the preceding 2 weeks. 2 weeks ago, the patient was admitted to the local hospital and diagnosed as “1.Coronary Disease, 2.Chronic Hepatitis B, 3.Stage Ⅱ Hypertension” and was administered with conservative treatment. However, her symptoms were not significantly alleviated. The patient was then referred to our hospital in September 2012 for further evaluation and treatment. Her past medical history included hypertension two years earlier, and her regular medications included compound kendir lenves (one tablets daily) and reserpine (one tablets daily). Her family history was negative. The patient’s height 163cm, weight 78kg, BMI 29.35. On physical examination, the patient had normal vital signs and oxygen saturation of Yong-Shun Gao, Yun-Fei Zhang, Hai-Lin Feng, Yu-Kun Zhang. Department of gastrointestinal surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450052, China. Correspondence to: Dr. Gao Yong-Shun,Department of gastrointestinal surgery, The First Affiliated Hospital of Zhengzhou China.(E-mail:[email protected],13803899721). University, Zhengzhou 450052, 95%-100%on 2 litres of oxygen and seat. Body position was orthopnea. The breath sounds were decreased at the right base compared with the left. There was no other physical finding. Contagion test showed HBsAg(+), HBeAg(+), Anti-HBc(+), WBC:12.5×109 /L. Biochemistry measurements were found to be normal. The patient was conservatively treated with expanding coronary artery and act blood therapy according to the diagnosis as “Coronary Disease”, but her symptom wasn’t significantly alleviated. ECG and Heart Doppler ultrasound were found to be normal, heart disease was ruled out the possibility. Then the patient went to the Respiratory Medicine Department, and was conservatively treated with antibiotic therapy according to the diagnosis as “pulmonary inflammatory”, because of her WBC:12.5×109 /L and the breath sounds were decreased at the right base, but her symptom still existed. The CT scan revealed the air below the right diaphragm accidentally, with the aim of it was to observe the condition of pulmonary inflammatory (Fig. 1). Afterwards the patient went to the Department of Gastrointestinal Surgery. The barium enema examination revealed the hepatodiaphragmatic interposition of the colon (Fig. 2), the diagnosis of “Chilaiditi syndrome” was made. The patient underwent reduction of interposed colon and greater omentum companion with conventional open surgery on October 10, 2012. Intra-operation finding included liver volume reduced, surface distribution of multiple nodules varies in size, and its substantial touch was a little hard. The hepatic flexure of colon and the great mass of greater omentum were in between the liver and the right hemi diaphragm. Ascending colon fixed good, and there was no redundant colon with long mesentery. After the operation, the patient reported complete resolution of her symptoms, and X-ray showed the colon was fixed in right location on the 4 day after surgery. The patient was discharged to home on October 22, 2012. She remained symptom free at 8 months follow up. Respiratory distress was frequently found in diseases of cardiovascular and respiratory system, such as bronchial asthma, interstitial pneumonia, cardiac tamponade and cardiac failure resulting from different causes. This patient’s symptoms presented similar to paroxysmal nocturnal dyspnea caused by acute left heart failure. The etiology of Chilaiditi syndrome is very complex. Any condition leading to an enlarged right subdiaphragmatic space, hypermobility of the colon or higher intra-abdominal pressure can predispose patients to Chilaiditi syndrome [4-4]. In the case reported here, the patient had an enlarged right subdiaphragmatic space due to cirrhosis, and a higher intra-abdominal pressure due to obesity and omentum fatty disposition, which were likely the etiology of Chilaiditi syndrome. Most patients with Chilaiditi syndrome are asymptomatic, be diagnosed with an incidented radiologic finding. The others patients may present with a wide range of complaints which can be misleading to the clinician. Symptoms associated with Chilaiditi syndrome always occur as gastrointestinal discomfort, cluding abdominal pain, distention, bloating, nausea, vomiting, flatulence, and changes in intestinal habits as well as more unusual chest manifestations, such as substernal pain, cardiac arrhythmias, dyspnea, and respiratory distress. Moreover, chest symptoms are always accompanies with the emergence of gastrointestinal symptoms and appear. Reviewed of the literature, the respiratory distress caused by Chilaitidi syndrome always occurred in children or the elderly [1-4], and accompanies with the abdominal pain, distention, nausea, and hiccup. To our knowledge, this is the first case which only is associated with recurrent respiratory distress without gastrointestinal symptoms. Therefore, physicians often ignore the possibility of Chilaiditi syndrome. When the patient underwent CT scan to observe the condition of pulmonary inflammatory, the CT showed an incidental radiologic finding of the air below the right diaphragm. After that, the patient had a definite diagnose. The diagnosis of Chilaiditi syndrome need to be combined with radiographic findings. The air below the right diaphragm on X-ray or CT scan appears mostly in Chilaiditi syndrome, ture pneumoperitoneum and subphrenic abscess. The presence of visible haustral folds adjacent to the diaphragm, the Chilaiditi syndrome’s characteristic finding, makes recognition of this phenomenon critical in distinguishing it from subphrenic abscess and true pneumoperitoneum. If an X-ray or CT scan doesn’t show visible haustral folds adjacent to the diaphragm, a barium enema examination is recommended to establish an accurate diagnosis. Due to the interposed colonic loop moved down obviously, result of the erect chest X-ray was negative, but the horizontal position CT scan showed the air below the right diaphragm. The diagnostic value of ultrasound in Chilaiditi syndrome is limited[5]. After the patient sitting a few minutes, her symptoms of respiratory distress still cannot relieve completely. Considering the causes as though the colon pressure was lifted, but Pulmonary reexpansion and Pulmonary ventilation flow ratio still needs some time to return to normal. The treatment of Chilaiditi syndrome is generally conservative. Surgical treatment may be required in case of intestinal obstruction, ischemia, or perforation. Because of the respiratory distress wasn’t significantly alleviated after conservative treatment, this patient required for surgical treatment. After the operation, the patient reported complete resolution of her symptoms, and X-ray showed the colon was fixed in right location. This syndrome is a rare disorder with recurrent respiratory distress and we conclude that this entity should be kept in mind in patients with recurrent respiratory distress. References: 1. Keles S, Artac H, Reisli I, et al. Chilaiditi syndrome as a cause of respiratory distress. Eur J Pediatr, 2006;165: 367–369 PMID:16489467 2. Dogu F, Reisli I, Ikinciogullari A, et al. Unusual cause of respiratory distress: Chilaiditi syndrome. Pediatr Int, 2004; 46:188–190 PMID:23530798 3. Amy X, Gavin H, Gwendolyn M, et al. Chilaiditi Syndrome Precipitated by Colonoscopy: A Case Report and Review of the Literature. HAWAI‘I Journal Of Medicine & Public Health, 2012, 71(6): 158-162 PMID:22787564 4. Sheikh Z, Khan A, Khan S. Chilaiditi’s syndrome: colonic interposition in a young patient with abdominal pain. Postgrad Med J, 2011;87: 239 PMID:21257995 5. Moaven O, Richard A. Chilaiditi Syndrome: A Rare Entity with Important Differential Diagnoses. Gastroenterology Hepatology, 2012,8(4):276-278 PMID:22723763 & Fig 1 The CT scan revealed the air below the right diaphragm. A B Fig 2A The barium enema examination revealed the hepatodiaphragmatic interposition of the colon; 2B The X-ray showed the colon was fixed in right location on the 4 day after surgery.