Abstract
A 75-year-old man presented with abdominal distension, hypoproteinemia, ascites and a 35-mm mass in the small bowel mesentery. Laparotomy was performed, and he was diagnosed with sclerosing mesenteritis. His clinical condition improved, with computed tomography (CT) showing tumor shrinkage and decreasing ascites after administration of prednisolone; however, on drug withdrawal, abdominal fullness recurred and CT revealed an enlarging tumor and increasing ascites. Capsule endoscopy (CE) and double-balloon enteroscopy (DBE) were performed to further investigate hypoalbuminemia, which revealed white villi, white nodules, white debris, and mucosal edema in the jejunum. Biopsies from the jejunal mucosa demonstrated infiltration by chronic inflammatory cells consisting mostly of lymphocytes and plasma cells, with marked lymphangiectasia of the lamina propria and submucosa. A fecal alpha-1-antitrypsin clearance test revealed abnormal leakage from the gastrointestinal tract, confirming that hypoalbuminemia was secondary to protein-losing enteropathy (PLE). The incidence of sclerosing mesenteritis accompanied by PLE is very rare. Only six cases have been reported so far. CE and DBE were helpful for diagnosing this condition, and should be performed in patients in whom the cause of hypoalbuminemia is unknown, and in those with PLE.
Similar content being viewed by others
References
Emory TS, Monihan JM, Carr NJ, et al. Sclerosing mesenteritis, mesenteric panniculitis and mesenteric lipodystrophy: A single entity? Am J Surg. 1997;21:392–8.
Akram S, Pardi DS, Schaffner JA, et al. Sclerosing mesenteritis: clinical features, treatment, and outcome in ninty-two patient. Clin Gastroenterol Hepatol. 2007;5:589–96.
HÖring E, Hingel T, Hens K, et al. Protein-losing enteropathy: first manifestation of sclerosing mesenteritis. Eur J Gastroenterol Hepatol. 1995;7:481–3.
Rajendran B, Duerksen DR. Retractile mesenteritis presenting as protein-losing gastroenteropathy. Can J Gastroenterol. 2006;20:787–9.
Kida T, Suzuki K, Matsuyama T, et al. Sclerosing mesenteritis presenting as protein-losing enteropathy: a fatal case. Intern Med. 2011;50:2845–9.
Endo K, Moroi R, Sugimura M, et al. Refractory sclerosing mesenteritis involving the small intestinal mesentery: a case report and literature review. Intern Med. 2014;53:1419–27.
Rispo A, Sica M, Bucci L, et al. Protein-loosing enteropathy in sclerosing mesenteritis. Eur Rev Med Pharmacol Sci. 2015;19:477–80.
Nishiya D, Mikami T, et al. A case of suspected mesenteric panniculitis with a large amount of chylous ascites. Nihon Shoukakibyougakkai Zasshi. 2007;104:1212–17.
Jura V. Sulla mesenterite e sclerosante. Policlinico. 1924;31:575–81.
Durst A, Freund H, Rosenmann E, et al. Mesenteric panniculitis: review of the literature and presentation of cases. Surgery. 1977;81:203–11.
Sabate J, Torrubia S, Maideu J, et al. Sclerosing mesenteritis: imaging findings in 17 patients. Am J Roentgenol. 1999;172:625–9.
Daskalogiannaki M, Voloudaki A, Prassopoulos P, et al. CT evaluation of mesenteric panniculitis: prevalence and associated disease. Am J Roentgenol. 2000;174:427–31.
Horton K, Lawler L, Fishman E, et al. CT findings in sclerosing mesenteritis: spectrum of disease. Radiographics. 2003;23:1561–7.
Van Breda A, Schuttevaer H, Coerkamp E, et al. Mesenteric panniculitis: US and CT features. Eur Radiol. 2004;14:2242–8.
Ghanem N, Pache G, Bley T, et al. MR findings in a rare case of sclerosing mesenteritis of the mesocolon. J Magn Reson Imaging. 2005;21:632–6.
Tytgat G, Roozendaal K, Winter W, et al. Successful treatment of a patient with retractile mesenteritis with prednisone and azathioprine. Gastroenterology. 1980;79:352–6.
Bush R, Hammar S, Rudolph R. Response to Cyclophosphamide. Arch Intern Med. 1986;146:503–5.
Kikiros C, Edis A. Mesenteric panniculitis resulting in bowel obstruction: response to steroids. J Surg. 1989;59:287–90.
Genereau T, Bellin M, Wechsler B, et al. Demonstration of efficacy of combining corticosteroids and colchicine in two patients with idiopathic sclerosing mesenteritis. Dig Dis Sci. 1996;41:684–8.
Koornstra J, Olfen G, Noort G. Retractile mesenteritis: to treat or not to treat. Hepatogastroenterology. 1997;44:408–10.
Mazure R, Fernandez P, Niveloni S, et al. Successful treatment of refractile mesenteritis with oral progesterone. Am Gastroenterol. 1998;114:1313–7.
Bala A, Coderre S, Johnson D, et al. Treatment of sclerosing mesenteritis with corticosteroids and azathioprine. Can J Gastroenterol. 2001;15:533–5.
Issa I, Baydoun H. Mesenteric panniculitis: various presentations and treatment regimens. World J Gastroenterol. 2009;14:3827–30.
Acknowledgements
The authors would like to thank Haruo Fujinaga and Yasuharu Kaizaki for their helpful comments and information.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Human animal rights
All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1964 and its later amendments.
Informed consent
Informed consent was obtained from all patients for being included in the study.
Rights and permissions
About this article
Cite this article
Saito, Y., Hiramatsu, K., Nosaka, T. et al. A case of protein-losing enteropathy caused by sclerosing mesenteritis diagnosed with capsule endoscopy and double-balloon endoscopy. Clin J Gastroenterol 10, 351–356 (2017). https://doi.org/10.1007/s12328-017-0755-y
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12328-017-0755-y