Sclerosing encapsulating peritonitis associated with propranolol usage: A case report and review of the literature
2009; Wiley; Volume: 10; Issue: 4 Linguagem: Inglês
10.1111/j.1751-2980.2009.00405.x
ISSN1751-2980
AutoresSumit Kalra, Antwan Atia, Jason D. McKinney, Thomas Borthwick, Roger D. Smalligan,
Tópico(s)Tuberous Sclerosis Complex Research
ResumoJournal of Digestive DiseasesVolume 10, Issue 4 p. 332-335 Free Access Sclerosing encapsulating peritonitis associated with propranolol usage: A case report and review of the literature Sumit KALRA, Corresponding Author Sumit KALRA Department of Internal Medicine andSumit KALRA, Department of Internal Medicine, James H. Quillen VA Medical Center, Johnson City, Mountain Home, TN, 37684, USA. Email: [email protected]Search for more papers by this authorAntwan ATIA, Antwan ATIA Department of Internal Medicine andSearch for more papers by this authorJason McKINNEY, Jason McKINNEY Department of Internal Medicine andSearch for more papers by this authorThomas R BORTHWICK, Thomas R BORTHWICK Gastroenterology, James H. Quillen VA Medical Center, andSearch for more papers by this authorRoger D SMALLIGAN, Roger D SMALLIGAN General Internal Medicine, Quillen College of Medicine, East Tennessee State University, Johnson City, Tennessee, USASearch for more papers by this author Sumit KALRA, Corresponding Author Sumit KALRA Department of Internal Medicine andSumit KALRA, Department of Internal Medicine, James H. Quillen VA Medical Center, Johnson City, Mountain Home, TN, 37684, USA. Email: [email protected]Search for more papers by this authorAntwan ATIA, Antwan ATIA Department of Internal Medicine andSearch for more papers by this authorJason McKINNEY, Jason McKINNEY Department of Internal Medicine andSearch for more papers by this authorThomas R BORTHWICK, Thomas R BORTHWICK Gastroenterology, James H. Quillen VA Medical Center, andSearch for more papers by this authorRoger D SMALLIGAN, Roger D SMALLIGAN General Internal Medicine, Quillen College of Medicine, East Tennessee State University, Johnson City, Tennessee, USASearch for more papers by this author First published: 28 October 2009 https://doi.org/10.1111/j.1751-2980.2009.00405.xCitations: 7 Financial disclosure: None of the authors have any financial conflict of interest related to this manuscript. AboutSectionsPDF ToolsRequest permissionExport citationAdd to favoritesTrack citation ShareShare Give accessShare full text accessShare full-text accessPlease review our Terms and Conditions of Use and check box below to share full-text version of article.I have read and accept the Wiley Online Library Terms and Conditions of UseShareable LinkUse the link below to share a full-text version of this article with your friends and colleagues. Learn more.Copy URL INTRODUCTION Sclerosing encapsulating peritonitis is a rare cause of intestinal obstruction that is characterized by thick, white, fibrous membrane formation in the peritoneum that partially or completely encases the bowel loops (the abdominal cocoon syndrome). The most commonly identified cause of this condition is continuous ambulatory peritoneal dialysis, but some beta adrenergic receptor blocker-associated cases have also been reported in the literature. We present a case and review the literature of sclerosing encapsulating peritonitis associated with propranolol use and discuss the presentation, etiopathogenesis and management of this rare condition. CASE REPORT A 59-year-old man presented with a 5-day history of episodic colicky abdominal pain, abdominal distension and vomiting. This was his ninth admission in the past year with similar symptoms. On each previous occasion he was managed conservatively and recovered, only to have a recurrence a few weeks to months later. His past history was also significant for cryptogenic cirrhosis of the liver with portal hypertension, diabetes mellitus and hypertension. He had no history of abdominal surgery, peritoneal dialysis, or spontaneous bacterial peritonitis. His medications included propranolol, lisinopril, furosemide, spironolactone, insulin, gabapentin, lactulose and sertraline. He was a non-smoker, drank no alcohol and had no history of illicit drug use. On physical examination he was a middle-aged man of average build. His pulse was 78/minute, regular, blood pressure 116/76 mm Hg and he was afebrile. His chest examination revealed clear lungs bilaterally and his heart sounds were normal without any murmurs, gallops or rubs. His abdomen was distended, tympanitic, firm but non-tender. Bowel sounds were present and mildly increased and there was no obvious ascites by clinical examination. Laboratory testing showed a hemoglobin of 10.9 g/dL, white blood cell count 7800 cells/mm3, serum sodium 140 mmol/L, potassium 4.2 mmol/L, chloride 110 mmol/L, bicarbonate 24 mmol/L, glucose 10.6 mmol/L, bilirubin 18.81 μmol/L, aspartate aminotransferase 31 U/L, alanine aminotransferase 32 U/L and alkaline phosphatase 183 U/L. An abdominal computed tomography (CT) scan (Fig. 1) showed dilated loops of the small bowel with air fluid levels and a small amount of ascites but no other significant pathology. He was diagnosed with partial small bowel obstruction and managed conservatively with bowel rest, i.v. fluids, anti-emetics and parenteral nutrition. When the patient failed to improve as quickly as he had on previous admissions, the surgery service was consulted and an exploratory laparotomy was performed. Upon opening the abdominal cavity thick fibrous tissue was found involving the peritoneum that encapsulated the abdominal contents. A biopsy of the peritoneal capsule revealed diffuse fibrosis and inflammatory cells consistent with peritoneal fibrosis and sclerosis (Fig. 2). The patient underwent extensive lysis of the adhesions during the laparotomy had an uneventful recovery and at the time of writing has been symptom free for over 9 months. Figure 1Open in figure viewerPowerPoint Computerd tomography scan of the abdomen shows dilated loops of small bowel with air fluid levels and small amount of ascites. Figure 2Open in figure viewerPowerPoint Biopsy of the peritoneal capsule shows diffuse fibrosis and inflammatory cells. DISCUSSION A partial small bowel obstruction is a clinical problem that internists, gastroenterologists and general surgeons face on a regular basis. While adhesions, malignancy and hernias are the most common causes, sclerosing encapsulating peritonitis is a rare clinical entity that can cause acute, sub-acute or chronic intestinal obstruction. Sclerosing encapsulating peritonitis was first described in 1907 by Owtschinnikow, who used the term ‘peritonitis chronica fibrosa encapsulate’.1 Foo et al. coined the term ‘abdominal cocoon syndrome’ in 1978 due to the cocoon-like appearance of the fibrotic capsule that is encountered when an exploratory laparotomy is performed.2 The condition is characterized by the formation of a thick grayish-white fibrous membrane that encases the small bowel partially or totally.3 A number of hypotheses have been proposed to explain the pathogenesis of sclerosing peritonitis. The condition was described initially in adolescent girls from tropical and subtropical countries.4 This led to the hypothesis that retrograde menstruation2 or a retrograde viral infection through the fallopian tubes was the underlying etiology of sclerosing peritonitis.5 However, since then this condition has also been described in men and in other age groups,6–8 this original theory appears to be unfounded. The proposed etiologies include dietary factors such as ergot-infested pearl millet, greater omentum hypoplasia and mesenteric vessel malformation, though each of these is simply speculative.9–11 There are a number of other conditions and factors associated with its occurrence, the most common of which is continuous ambulatory peritoneal dialysis.12,13 Other associations reported in the literature include ovarian thecomas, tuberculous pelvic inflammatory disease, ventriculoperitoneal shunts, systemic lupus erythematosus, orthotopic liver transplantation, protein S deficiency, ovarian cysts, keratoconjunctivitis sicca syndrome and various beta adrenergic blocking agents.14–21 The exact mechanism by which beta blockers induce sclerosing peritonitis is unknown, but two suggested hypotheses include a possible allergic type reaction to the class of drug or perhaps an excess production of intraperitoneal collagen, which has been shown to be induced by these agents.22,23 The clinical manifestations of sclerosing peritonitis can be nonspecific and include vague, colicky abdominal pain, weight loss, nausea and vomiting, all largely related to the partial small bowel obstruction the condition induces.9 Sclerosing peritonitis can also present with recurrent ascites or as an abdominal mass. Although the diagnosis is most commonly made at laparotomy, occasionally it is suspected on radiological imaging. On CT scan, sclerosing peritonitis can appear as peritoneal thickening or calcification, loculated fluid collections or matting of the small bowel. Thickening of the small bowel loops and calcifications of the liver capsule, spleen, posterior peritoneal wall or bowel can also be seen.24 Ultrasound and magnetic resonance imaging have also been useful in making this diagnosis.25–27 Our patient did not have a history of any of the most commonly associated conditions, but had been taking propranolol for over 4 years at the time of diagnosis. Although he was also taking other medications, none of these have been linked to sclerosing peritonitis. A review of the literature revealed that several beta blockers have been associated with sclerosing encapsulating peritonitis, the first of which was practolol in 1971.21 Practolol was also linked to a diffuse fibrotic disorder called the oculomucocutaneous syndrome, which causes fibrotic changes in the eyes, skin, peritoneal and pleural membranes. The median latency period between usage of practolol and the development of sclerosing peritonitis was 4 years in reported cases.28 These adverse reactions led to practolol being withdrawn from the market in 1975. Since then, case reports involving other beta blockers have been published including timolol, metoprolol, atenolol, oxprenolol, sotalol and propranolol.22,23,29–31 Two such case reports describing the association of propranolol and sclerosing peritonitis are summarized in Table 1.22,31 Each of these cases involved men taking propranolol for 3 months to 2 years for treatment of chronic angina. Table 1. Summary of patients describing association between propranolol and sclerosing peritonitis Patient 1 Patient 2 Patient's age 56 43 Patient's sex Male Male Dose of propranolol 320 mg/day 20 mg q.i.d. Indication for propranolol usage Hypertension and angina pectoris Angina pectoris Duration of propranolol usage 2 years 2 months Clinical picture Discovered during laparotomy for rectal hemorrhage Small bowel obstruction and ascites Diagnostic method During laparotomy During laparotomy Outcome Patient recovered Patient recovered The treatment of sclerosing peritonitis is mainly surgical with lysis of the adhesions at laparotomy. A retrospective analysis of 32 patients with this condition was done to determine the best surgical management comparing membrane resection, enterolysis, partial excision of the membrane, intestinal resection and exploratory laparotomy only. Membrane resection resulted in less patients having persistent bowel obstruction or death and the study recommended this be attempted when feasible. No surgical treatment was recommended in cases of associated ascites, asymptomatic sclerosing peritonitis, or sub-acute intestinal obstruction.32 While this is a rare clinical entity and the causative role of propranolol cannot be established with absolute certainty, such clinical observations build on current knowledge and it is hoped they will eventually contribute to a better understanding of the etiology of this syndrome. Our case reminds physicians to consider the abdominal cocoon syndrome in the differential diagnosis of a patient with recurrent small bowel obstruction, especially in patients on propranolol therapy or with any of the other predisposing conditions mentioned. REFERENCES 1 Owtschinnikow PJ. Peritonitis chronica fibrosa incapsulata. Arch Klin Chir 1907; 83: 623– 34. 2 Foo KT, Ng KC, Rauff A, Foong WC, Sinniah R. Unusual small intestinal obstruction in adolescent girls: the abdominal cocoon. Br J Surg 1978; 65: 427– 30. 3 Sahoo SP, Gangopadhyay AN, Gupta DK, Gopal SC, Sharma SP, Dash RN. Abdominal cocoon in children: a report of four cases. J Pediatr Surg 1996; 31: 987– 88. 4 Eltringham WK, Espiner HJ, Windsor CW et al. Sclerosing peritonitis due to practolol: a report on 9 cases and their surgical management. Br J Surg 1977; 64: 229– 35. 5 Sieck JO, Cowgill R, Larkworthy W. Peritoneal encapsulation and abdominal cocoon. Gastroenterology 1983; 84: 1597– 601. 6 Rajagopal AS, Rajagopal R. Conundrum of the cocoon: report of a case and review of the literature. Dis Colon Rectum 2003; 46: 1141– 43. 7 Masuda C, Fujii Y, Kamiya T et al. Idiopathic sclerosing peritonitis in a man. Intern Med 1993; 32: 552– 55. 8 Burstein M, Galun E, Ben-Chetrit E. Idiopathic sclerosing peritonitis in a man. J Clin Gastroenterol 1990; 12: 698– 01. 9 Xu P, Chen LH, Li YM. 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