A Pneumonia Patient with Worsening Respiratory Distress by Chilaiditi Syndrome: a Case Report

대한응급의학회지 제 27 권 제 2 호 � 증례� Volume 27, Number 2, April, 2016

Medical A Pneumonia Patient with Worsening Respiratory Distress by Chilaiditi Syndrome: A Case Report

Department of Emergency Medicine, School of Medicine, Eulji University, Daejeon, Korea Chang Ho Lee, M.D., Won Young Sung, M.D., Jang Young Lee, M.D., Sang Won Seo, M.D.

Chilaiditi sign refers to the presence of bowel gas under the nal pain, nausea, vomiting and constipation. Severe cases right diaphragm which is similar in appearance to a pneu- can appear bowel obstruction requiring surgical treat- moperitoneum on radiography, and is caused by abnormal ment, and dyspnea or chest pain may rarely appear1). anatomic positioning of the colon or small bowel between Respiratory distress has been reported in few patients the liver and the diaphragm. When symptoms are present, with Chilaiditi syndrome3-5). In these cases, chronic, this condition is known as Chilaiditi syndrome. The most recurrent dyspnea with gastrointestinal symptoms such common symptoms are gastrointestinal. It has been less as recurrent abdominal pain was caused by Chilaiditi commonly associated with chronic, recurrent respiratory syndrome in adult patients. In pediatric patients, it causes distress. We report acute respiratory distress without gas- recurrent respiratory symptoms and dyspnea. trointestinal symptoms exacerbated by Chilaiditi syndrome In contrast to these cases, we report acute respiratory in a pneumonia patient with no history of chronic respiratory distress without gastrointestinal symptoms, exacerbated disease. by Chilaiditi syndrome in a pneumonia patient without a history of chronic respiratory disease. Key Words: Chilaiditi syndrome, Respiratory distress, Pneumoperitoneum, Diaphragmatic hernia Case Report

Introduction An 81 year old woman admitted to a local hospital 4 days prior presented to our emergency room (ER) with Chilaiditi sign refers to the presence of bowel gas under the right diaphragm that appears similar to a pneumoperitoneum on radiography, and is caused by abnormal anatomic positioning of the colon or small bowel between the liver and the diaphragm. When symptoms are present, this condition is known as Chilaiditi syndrome1). Chilaitditi sign is identified as an incidental radiographic finding and could be mistaken for subphrenic abscess, pneumoperitoneum, or diaphragmatic hernia2). This condition is usually asymptomatic. However gastrointestinal symptoms are common, including abdomi-

책임저자: 성 원 영 대전광역시 서구 둔산동 1306 을지대학교병원 응급의학과 Tel: 042) 611-3256, Fax: 042) 611-3880 E-mail: [email protected] 접수일: 2015년 11월 6일, 1차 교정일: 2015년 11월 17일 Fig. 1. Chest X-ray in the ER shows elevation of the right 게재승인일: 2016년 1월 5일 diaphragm and bowel gas under the diaphragm. 219 220 / 대한응급의학회지: 제 27 권 제 2 호 2016 cough, sputum and general weakness. The patient had L/min of oxygen by mask. undergone surgery for a left femur fracture one year ago On physical examination, the patient was alert, and and was currently on medication for hypertension and chest auscultation revealed coarse breath sounds and arthritis. On admission, pneumonia in the upper left lobe wheezing in both lungs. The abdomen was not distended was found on the chest computed tomography (CT). or tender. Due to wheezing, nebulized bronchodilator Abrupt acute onset of worsening dyspnea began in the was administered. evening on the day of transfer. Chest x-ray showed Arterial blood gas analysis showed pH 7.34, PaCO2 47 extensive bowel gas in the right lung field, and the mmHg, PaO2 54 mmHg, HCO3 25.4 mmol/L, and SaO2 patient was transferred to ER with the diagnosis of 85% with continuous administration of oxygen at 15 diaphragmatic hernia. L/min by mask. Complete blood count revealed white In the ER, the patient complained of severe dyspnea, blood cells 11740/μL, hemoglobin 12.5 g/dL, platelet but no chest pain, abdominal pain, nausea, vomiting or 280,000/μL, no significant changes were found in chemi- diarrhea. Blood pressure was 190/106 mmHg, heart rate cal and electrolyte tests, C-reactive protein level was nor- 138 beats/min, respiratory rate 34 breaths/min, body mal at 0.24 mg/dL, and cardiac enzymes were normal. temperature 36.6�C and oxygen saturation 88% on 15 In the ER, chest x-ray showed elevation of the right

A B

CD Fig. 2. No significant changes in ground glass opacity in the lingual lobe of the left lung are seen the chest CT from the local hospital (A) and our ER (C). The local hospital chest CT shows a chilaiditi sign (B). Chest CT in the ER shows the small bowel interposed along with the colon and compression atelectasis is seen in the lower and middle lobes of the right lung (D). Chang Ho Lee et al.: A Pneumonia Patient with Worsening Respiratory Distress by Chilaiditi Syndrome: A Case Report / 221 diaphragm with a large amount of bowel gas (Fig. 1). To evaluate diaphragmatic hernia and worsening pneumo- Discussion nia, chest and abdominal CT were performed. No significant changes of ground glass opacity in the lingual lobe Chilaiditi sign appears in 0.025~0.28% of the general of the left lung were seen in the CT studies performed at population, and is often incidentally found during a radi- the local hospital and our hospital (Fig. 2A, C). Chest ographic examination. The incidence increases with age CT performed 4 days prior at the local hospital showed and it is less common in children than in adults. It is chilaiditi sign with a colonic loop interposed between the more frequently seen in men6-8). liver and right diaphragm (Fig. 2B). However, unlike To diagnose Chilaiditi sign based on radiologic find- previous imaging studies, much small bowel was inter- ings, the following criteria must be met. The right posed along with the colon and compression atelectasis hemidiaphragm must be adequately elevated above the was seen in the lower and middle lobe of the right lung liver by the intestine, the bowel must be distended by air (Fig. 2D). Signs of mechanical ileus or bowel ischemia to illustrate a pseudo-pneumoperitoneum, and the superi- were not seen on the abdomen CT. or margin of the liver must be depressed below the level Although oxygen was administered, there was no of the left hemidiaphragm9). improvement in respiratory difficulty. Therefore, endo- Important differential diagnoses of the radiographic tracheal intubation was done and the patient was admit- findings include subdiaphragmatic abscess, pneumoperi- ted to the intensive care unit. After admission, treatment toneum, posterior hepatic lesions, right sided diaphrag- for pneumonia was done including mechanical ventila- matic hernias and retroperitoneal masses2). In the present tion and antibiotics. case, the patient was transferred to our ER with the mis- Plain chest x-ray taken on the second day of admission diagnosis of a diaphragmatic hernia. showed decreased bowel gas under the right diaphragm The interposed bowel is most commonly the hepatic (Fig. 3). Mechanical ventilation and intubation were dis- flexure, ascending colon, or transverse colon but continued on the fifth day and the patient was discharged involvement of the small bowel, either alone or in com- without significant complications on the twelfth day. bination with the colon, has been reported10,11). Common etiologies include increased colonic mobility or rebun- dancy, congenital malrotation or malposition of the colon, and elevation of the right hemidiaphragm4). Chilaiditi syndrome is generally asymptomatic and may cause symptoms only in a minority of patients. These range from nonspecific gastrointestinal symptoms such as nausea, anorexia, vomiting, flatulence, and constipation to signs of pseudo-obstruction and rarely to life threatening complications like volvulus or intestinal obstruction, followed by respiratory distress and, less frequently, angina-like chest pain10). Less commonly, Chilaiditi syndrome has been associated with chronic, recurrent respiratory distress3-5). In the present case, the patient had no history of chronic respiratory disease or dyspnea. Moreover, we found no right diaphragm eventration on chest xray and aortic CT performed several years prior at our hospital. Although the definite cause is unknown, we assume that Fig. 3. Follow up chest X-ray on the second day of admis- right diaphragm eventration gradually progresses, and sion shows decreased bowel gas under the right results in a decrease in vital capacity and total lung diaphragm, but the elevation of the right diaphragm capacity12). This patient had a pulmonary infection (pneu- has not disappeared. 222 / 대한응급의학회지: 제 27 권 제 2 호 2016 monia) and decreased respiratory function and thoracic ated Chilaiditi’s syndrome: case report and literature recoil could be expected due to old age. In this situation, review. Am J Gastroenterol. 1996;91:2613-6. the interposition of the increased, dilated bowel loop 03. Santosh K, Gajendra VS, Rakesh S, Devesh PS. Rare between the liver and the diaphragm might be responsi- association B/W respiratory distress and chilaiditi syndrome. Indian J Allergy Asthma Immunol. 2012;26:9-10. ble for decreasing the intrathoracic volume and lung ven- 04. Keles S, Artac H, Reisli I, Alp H, Koc O. Chilaiditi syn- tilation. Due to these complex factors, Chilaiditi syndrome as a cause of respiratory distress. Eur J Pediatr. drome occurred abruptly and worsened the symptoms of 2006;65:367-9. respiratory distress in the present patient. 05. Mesquita MB, Lubetzky R, Cohen S. Chilaiditi Syndrome No treatment is required for an asymptomatic patient as A Cause of Respiratory Distress: A Case Report and with Chilaiditi sign. Initial management of Chilaiditi syn- Review of the Literature. J Gastroint Dig Syst. 2014;4: drome should include bed rest, intravenous fluid therapy, 188. doi:10.4172/2161-069X.1000188. and bowel decompression. If the patient does not respond 06. Alva S, Shetty-Alva N, Longo WE. Image of the month. to initial conservative management, and either the bowel Chilaiditi sign or syndrome. Arch Surg. 2008;143:93-4. obstruction fails to resolve or there is evidence of bowel 07. Elcin CN, Erdem G, TascıI, Dogru T, Sonmez A, Naharci I, et al. Respiratory distress, constipation and acute ischemia, then surgical intervention is indicated1). myocardial infarction in association with chilaiditi’s syn- In the present case, bowel decompression by nasogas- drome: A case report. Anatol J Clin Investig. 2008;2:167- tric tube was not performed in the ER early, but later, 70. after intensive care unit admission. However, a follow up 08. Barrosa Jornet JM, Balaguer A, Escribano J, Pagone F, chest xray before nasogastric tube insertion showed Domonech J, del Castillo D. Chiladiti Syndrome associat- decreased bowel gas under the right diaphragm. We sur- ed with transverse colon first report in a paediatric patient mise that providing positive expiratory pressure and and review of the literature. Eur J Pediatr Surg. 2003;13: recruiting collapsed lung by endotracheal intubation and 425-8. mechanical ventilation resulted in this effect. 09. Lekkas CN, Lentino W. Symptom producing interposition Chilaiditi syndrome is a rare disorder, but must be con- of the colon. Clinical syndrome in mentally deficient adults. JAMA. 1978;240:747-50. sidered in the differential diagnosis of acute respiratory 10. Risaliti A, De Anna D, Terrosu G, Uzzau A, Carcoforo P, distress in a case with air or bowel gas under the right et al. Chilaiditi's syndrome as a surgical and nonsurgical diaphragm. problem. Surg Gynecol Obstet. 1993;176:55-8. 11. Gurvits GE, Lau N, Gualtieri N, Robilotti JG. Air under the right diaphragm: colonoscopy in the setting of REFERENCES Chilaiditi syndrome. Gastrointest Endosc. 2009;69:758-9. 12. Calvinho P, Bastos C, Bernardo JE, Eugenio L, Antunes 01. Moaven O, Hodin RA. Chiladiti syndrome: A Rare Entity MJ. Diaphragmmatic eventration: long-term follow-up and with Important Differential Diagnoses. Gastroenterol results of open-chest plicature. Eur J Cardiothorac Surg. Hepatol (N Y). 2012;8:276-8. 2009;36:883-7. 02. Plorde JJ, Raker EJ. Transverse colon volvulus and associ-