Isolated granulomatous noncaseating pancreatitis is a rare condition exceptionally described in human population. We demonstrate a case of the a 71-years-old female patient suffering from recent diabetes mellitus, generalized atherosclerosis and hypertension who died due to pulmonary embolism and terminal bronchopneumonia. Lipomatosis of pancreatic tissue was observed during the postmortem examination. Histological examination of pancreatic tissue discovered multiple small noncaseating epithelioid cell and giant cell granulomas, partly replacing the islets of Langerhans. To our knowledge, our case represents the first description of noninfectious granulomatous pancreatitis associated with acute manifested insulin-dependent diabetes mellitus. 1. Introduction Noncaseating granulomatous inflammation confined to the pancreas has been only exceptionally described in human patients. Infections like tuberculosis and syphilis, exogenous noxes, autoimmunity, and systemic granulomatous diseases are the most frequent causes of granuloma formation within the pancreatic tissue [1]. Abdominal pain or epigastric discomfort, diarrhoea, weight loss, and obstructive jaundice are listed among the clinical symptoms of granulomatous pancreatitis [1–3]. We report a case of a patient who clinically presented with acute manifested diabetes mellitus associated with isolated granulomatous pancreatitis discovered in the postmortem examination and we present a review of the available literature. 2. Clinical History A 71-year-old obese woman was admitted with the recent onset of diabetes mellitus manifested as hyperglycaemic ketoacidotic precoma. The past medical history was unremarkable. Recently, arterial hypertension was discovered. Her body weight was 110？kg, BMI 38. The plasma glucose level ranged from 3.1 to 15.1？mmol/L. The patient was treated with intensified insulin regime. The status of the patient was complicated by intermittent fever and several antibiotics were repeatedly administered. Terminally, clinical signs of septic shock and multiorgan failure appeared and the patient died. Postmortem examination performed 11 hours after death discovered signs of septic shock with activation of spleen pulp and terminal bronchopneumonia. Thromboemboli were found in several peripheral branches of the pulmonary artery. Hypertrophy of the heart (545？g), predominantly of the left ventricle, was also observed. The pancreas showed a macroscopically lobular arrangement and lipomatosis; other macroscopic changes were not visible. Lungs, thoracic lymph nodes, and other organs did not show
References
[1]
J. Stürmer and V. Becker, “Granulomatous pancreatitis: granulomas in chronic pancreatitis,” Virchows Archiv, vol. 410, no. 4, pp. 327–338, 1987.
[2]
W. G. Choi, C. W. Lee, H. J. Park et al., “A case of granulomatous pancreatitis caused by syphilis,” Korean Journal of Medicine, vol. 52, pp. 696–701, 1997.
[3]
A. R. Essop, J. Posen, I. Segal, and D. Pantanowitz, “Isolated granulomatous pancreatitis,” Journal of Clinical Gastroenterology, vol. 6, no. 1, pp. 61–64, 1984.
[4]
C. S. Pramesh, A. A. Heroor, P. J. Shukla, P. M. Jagannath, and L. J. De Souza, “Pancreatic tuberculosis,” Tropical Gastroenterology, vol. 23, no. 3, pp. 142–143, 2002.
[5]
S. Hari, A. Seith, D. N. Srivastava, G. Makharia, and S. Pal, “Isolated tuberculosis of the pancreas diagnosed with needle aspiration: a case report and review of the literature,” Tropical Gastroenterology, vol. 26, no. 3, pp. 141–143, 2005.
[6]
N. V. Adsay, O. Basturk, D. S. Klimstra, and G. Kl？ppel, “Pancreatic pseudotumors: non-neoplastic solid lesions of the pancreas that clinically mimic pancreas cancer,” Seminars in Diagnostic Pathology, vol. 21, no. 4, pp. 260–267, 2004.
[7]
M. Caceres, M. S. Sabbaghian, R. Braud, S. Wilks, and M. Boyle, “Pancreatic sarcoidosis: unusual presentation resembling a periampullary malignancy,” Current Surgery, vol. 63, no. 3, pp. 179–185, 2006.
[8]
M. Shukla, M. F. Hassan, V. Toor, J. Kaur, C. Solomon, and H. Cohen, “Symptomatic pancreatic sarcoidosis: case report and review of literature,” Journal of the Pancreas, vol. 8, no. 6, pp. 770–774, 2007.
[9]
A. L. Mayne, Ahmad j, M. Loughrey, and M. A. Taylor, “Sarcoidosis of the pancreas mimicking adenocarcinoma,” BMJ Case Reports, 2013.
[10]
S. Gaur, “Sarcoidosis manifested as hypercalcemic pancreatitis,” Southern Medical Journal, vol. 94, no. 9, pp. 939–940, 2001.
[11]
R. Sánchez-Lozada, J. Soriano-Rosas, and R. Gutiérrez-Vega, “Acute pancreatitis, diabetes, and sarcoidosis: case report and review of the literature,” Gaceta Médica de México, vol. 140, no. 3, pp. 343–346, 2004.
[12]
M. Gschwantler, G. Kogelbauer, W. Klose, B. Bibus, D. Tscholakoff, and W. Weiss, “The pancreas as a site of granulomatous inflammation in Crohn's disease,” Gastroenterology, vol. 108, no. 4, pp. 1246–1249, 1995.
[13]
D. S. Strayer and J. Kapp, “Insulin-specific antibodies and insulitis in mice immunized with purified porcine insulin,” Clinical and Experimental Immunology, vol. 47, no. 1, pp. 77–84, 1982.
[14]
V. Deshpande, M. Mino-Kenudson, W. Brugge, and G. Y. Lauwers, “Autoimmune pancreatitis: More than just a pancreatic disease? A contemporary review of its pathology,” Archives of Pathology and Laboratory Medicine, vol. 129, no. 9, pp. 1148–1154, 2005.
[15]
R. G. Naik, B. M. Brooks-Worrell, and J. P. Palmer, “Latent autoimmune diabetes in adults,” Journal of Clinical Endocrinology and Metabolism, vol. 94, no. 12, pp. 4635–4644, 2009.
[16]
R. D. G. Leslie, H. Kolb, N. C. Schloot et al., “Diabetes classification: grey zones, sound and smoke: action LADA 1,” Diabetes/Metabolism Research and Reviews, vol. 24, no. 7, pp. 511–519, 2008.
[17]
J. Wright, A. Yates, H. Sharma, and P. Thibert, “Histopathological lesions in the pancreas of the BB Wistar rat as a function of age and duration of diabetes,” Journal of Comparative Pathology, vol. 95, no. 1, pp. 7–14, 1985.
