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Cystic Pneumatosis in Coeliac Disease Gut: first published as 10.1136/gut.29.6.852 on 1 June 1988. Downloaded from Gut, 1988, 29, 852-855 Case report Cystic pneumatosis in coeliac disease J M SACKIER, E J SMITH, AND C B WOOD From the Royal Postgraduate Medical School, London SUMMARY This report presents a patient with proximal small bowel cystic pneumatosis associated with poorly controlled coeliac disease and pseudo-obstruction. Cystic pneumatosis is rare in the proximal small bowel and we can find no report of its occurrence in association with coeliac disease. The finding of intramural gas in a hollow viscus is well seen to have pneumoperitoneum, but no free fluid. In recognised and has been reported in a large number the small bowel mesentry 5 cm distal to the ligament of conditions, although there is sometimes uncer- of Treitz, there was a collection of gas containing tainty as to aetiology (Table). We wish to present a cysts ranging from 2 to 5 mm in diameter, and case of cystic pneumatosis in a patient with coeliac extending for approximately 100 cm along the small disease, an association not previously recorded. bowel. The mesenteric vessels were all pulsatile, and the bowel appeared healthy. There was no sign of http://gut.bmj.com/ Case report acute or chronic peptic ulceration. No further inter- vention was considered necessary and the abdomen An 83 year old woman was admitted as an emergency was closed. Predictably, she developed a chest with a one week history of abdominal pain, disten- infection which was treated with prolonged oxygen sion, and vomiting. She had suffered with coeliac therapy and antibiotics. The remainder of the post- disease for 40 years, the diagnosis having been operative course was uneventful and on the seventh confirmed by several jejunal biopsies showing sub- day no gas was visible on a plain abdominal radio- on October 1, 2021 by guest. Protected copyright. total villus atrophy. She did not adhere to a gluten graph. She was discharged 11 days after surgery on a free diet and these indiscretions led to repeated gluten free diet and remains well 18 months later. admissions for reinforcement of dietary advice. She had osteoarthrosis and was taking ibuprofen. For Table Reported conditions associated with cystic several weeks before admission she had steatorrhoea pneumatosis but had no bowel action for the two days preceding presentation, although she had passed flatus. On Referetnce examination she was dehydrated but not clinically shocked. The abdomen- was distended with signs of Ulcerative colitis 1 Asthma 2, 3 generalised peritonitis and rectal examination Mesenteric infarction 4. 5 revealed soft, grey, frothy stool. Examination of Ischaemic colitis 6 blood revealed hypokalaemia, hypoalbuninaemia, Scleroderma 7 and a macrocytic anaemia. Arterial blood gases were Colonoscopy (postbiopsy) 8 Crohn's disease 9, 10 normal. An abdominal radiograph (Figure) showed Hirschsprung's discase 10 free intraperitoneal gas, small bowel distension, and Imperforate anus 11 small bowel intramural gas. At laparotomy she was Meconium plugs 11 Leukaemia 12 Whipple's disease 12 Steroid therapy 12 Address for correspondence: Mr J M Sackier, FRCS, Royal Postgraduate Jejunal-ileal bypass 12 Medical School, Hammersmith Hospital, DuCane Road, London W12 (OHS. Chilaiditi's syndrome 7, 13-2t) Received for publication 29 January 1988. 852 Gut: first published as 10.1136/gut.29.6.852 on 1 June 1988. Downloaded from Cysticpneumatosis in coeliac disease 853 http://gut.bmj.com/ on October 1, 2021 by guest. Protected copyright. Figure Plain abdominal radiograph showingfreegas, small bowel distension and intramural gas. Discussion and colon, but are rarely found in the stomach and proximal small bowel.226 The gaseous contents vary, Cystic pneumatosis was first described in 1730 by Du but nitrogen predominates. The cysts probably Vernoy and in 1786 Hunter described the condition represent distended tissue spaces, although cystic in the intestine of a pig, that had been sent to him by lymphatic vessels have also been suggested.27 Giant Jenner. Thus the condition is not unique to man, but cells similar to foreign body giant cells are often seen in animals is usually considered to be of no signi- on histological examination. The aetiology of cyst ficance.2" The cysts range in size from 0-5 mm to formation has variously been thought to be caused by 10 cm, and are most common in the terminal ileum gas forming tumours,> gas forming bacteria,2" dietary Gut: first published as 10.1136/gut.29.6.852 on 1 June 1988. Downloaded from 854 Sackier, Smith, and Wood causes,3" or disturbances of acid based metabolism.3' 4 Scott JR, Miller WT, Urso M, Stadalnik R. Acute In 1965, Colquhoun32 classified the various types mesenteric infarction. Am J Roentgenol Radium Ther of presentation of intramural gas in hollow viscera Nucl Med 1971; 113: 269-79. interstitial 5 Rigler LO, Pogue WL. Roentgen signs of intestinal as: (1) gas-forming infections; (2) necrosis. Am J Roentgenol Radium Ther Nucl Med 1965; emphysema; (3) cystic pneumatosis (pneumatosis 94: 402-9. cystoides intestinalis). The role of gas forming 6 Rosenquist CJ. An unusual pattern of intramural gas in organisms has been extensively documented and will small bowel infarction. Radiology 1971; 99: 337-8. not be discussed further. Interstitial emphysema is 7 Meyers MA, Ghahremani GG, Clements JL Jr, defined by the typical appearance of linear gas Goodman K. Pneumatosis intestinalis. Gastrointest streaks on plain radiographs. There are no cysts Radiol 1977; 2: 91-105. identified and no clinical evidence of gas forming 8 Marshak RH, Blum SD, Eliasoph J. Pneumatosis infection. Therefore cystic pneumatosis is really a involving left side of colon. JAMA 1956; 161: 1626-8. exclusion. 9 Ghahremani GG, Port RB, Beachley MC. Pneumatosis diagnosis of coli in Crohn's disease. Am J Dig Dis 1974; 19: 315-23. In 1952 Koss hypothesised that one required raised 10 Reinhof III WF, Collins NP. Pneumatosis cystoides intraluminal pressure and a break in mucosal intestinalis and regional enteritis. Ann Surg 1959; 149: integrity to develop these cysts, but in 15% of his 593-9. reported cases, neither of these parameters were 11 Robinson AE, Grossman H, Brumley GW. Pneumato- obviously fulfilled.'3 Other authors2 have found the sis intestinalis in the neonate. Am J Roentgenol Radium condition in patients with asthma and proposed a Ther Nucl Med 1974; 120: 333-41. pulmonary origin of the gas. Keyting3 showed that 12 Seaman WB, Fleming RJ, Baker DH. Pneumatosis rupture of an emphysematous bulla could lead to gas intestinalis of the small bowel. Semin Roentgenol 1966; vessels to the root, and 1: 234-242. tracking along blood lung 13 Koss LG. Abdominal gas cysts (pneumatosis cystoides then through the aortic sheath to the mesenteric intestinorum hominis). Analysis with a report of a case vessels. These authors noted that the gas bubbles and a critical review of the literature. Arch Pathol 1952; tended to be grouped around the distribution of the 53: 523-49. major branches of the mesenteric vasculature. 14 Chilaiditi D. Zur Frage der Hepatoptse and Ptose im Cystic pneumatosis has been noted in association Allgemeinen im Anschluss an drei Falle von temporare with mesenteric infarction' where, together with gas partieller Leber verlagerung. Fortschr Geb Roentgenstr in the portal vein, it is a grave prognostic feature, and 1910-1911; 16: 173-208. a similar association exists with ischaemic colitis. 15 Dressler M. Uber die Pneumatosis cystoides intestini. http://gut.bmj.com/ In Helv Med Acta 1939; 6: 229-54. 1933 Jung and Ngai33 documented the formation 16 Moreau P. Sur un cas d'hepatoptase par interposition of subserosal gastric cysts in a patient undergoing avec kystes gaseux de l'intestin. Examen radiologique surgery for a perforated gastric ulcer. The finding of verifie par l'intervention. Arch Elect Med 1917; 27: 393. cystic pneumatosis in perforated ulcers and jejunal 17 Druckman A, Schwartz A, Rabinoui N, Feuchtwanger diverticuli is rare,34 however, which may be due M. Pneumatosis of the intestines. Am J Roentgenol to the lack of raised intraluminal pressure caused by Radium Ther Nucl Med 1961; 86: 911-9. the massive intraperitoneal leak. 18 Forfota E. Die Pneumatosis cystoides intestini im on October 1, 2021 by guest. Protected copyright. Cystic pneumatosis has been seen in a number of Rontgenbild. Fortschr Ger Roentgenstr 1936; 53: 131-9. varying conditions and treatment should always be 19 Kenney JG. Pneumatosis intestinales. Clin Radiol 1963; a 14: 70-6. directed at the primary disease. Should complica- 20 Puschner J. Mesentery filled with air bubbles: Interesting tion occur as a result of the cysts, then surgery may be finding in veterinary meat inspection. Tieraerztl Prax required. The use of hyperbaric oxygen has recently 1973; 1: 379-80. been advocated,35 but such therapy must be pro- 21 Myhre J, Wilson JA. Study on occurrence of pneumo- longed for 48 hours after radiographic dissolution of peritoneum after gastroscopy and observance of inter- the cysts.36 stitial emphysema of stomach. Gastroenterology 1948; 11: 115-9. References 22 Lumsden K. Radiological demonstration of gas in stomach wall. Br J Radiol 1956; 29: 596-600. I Simon M, Shapiro JH, Parker JG, Schein CJ, 23 Cancelmo JJ Jr. Interstitial gastric emphysema, with Weingarten B. The diagnosis and treatment of dilata- report of case. Radiology 1954; 63: 81-3. tion of the colon in severe ulcerative colitis. A diagnostic 24 Schorr S, Marcus M. Case reports. Intramural gastric roentgen sign. Am J Roentgenol 1962; 87: 655-69. emphysema. Br J Radiol 1962; 35: 641-4. 2 Doub HP, Shea JJ. Pneumatosis cystoides intestinalis. 25 Kay-Butler JJ. A case of interstitial gastric emphysema. JAMA 1960; 172: 1238-43. Br J Surg 1962; 50: 99-101.
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