Annals of Clinical & Laboratory Science 37:182-185 (2007)
© 2007 Association of Clinical Scientists
Eosinophilic Cholecystitis, with a Review of the Literature
Rada Shakov,
Gilbert Simoni,
Aquilles Villacin and
Walid Baddoura
Departments of Medicine and Pathology, St. Joseph’s Regional Medical Center, Paterson, New Jersey, and School of Graduate Medical Education, Seton Hall University, South Orange, New Jersey
Address correspondence to Rada Shakov, M.D., Department of Medicine, St. Joseph’s Regional Medical Center, Paterson, NJ 07503, USA; tel 718 915 2706; fax 718 996 3848; e-mail: radaroze{at}yahoo.com.
 |
Abstract
|
|---|
Eosinophilic cholecystitis (EC) is a rare entity that presents in a manner comparable to acute cholecystitis. The diagnosis is based on classical symptoms of cholecystitis with the presence of >90% eosinophilic infiltration within the gallbladder. We report the case of a 29-yr-old man who presented with unremitting right upper quadrant pain, chills, and loss of appetite. After confirmation of the diagnosis with ultrasound and hepatobiliary scan (HIDA), a cholecystectomy was performed. Pathologic examination of the excised gallbladder demonstrated submucosal infiltration with eosinophils, consistent with EC. Peripheral eosinophilia was not observed; the subject’s blood eosinophil count did not exceed 3% during the period of observation. The patient was discharged with no further symptoms. Cases of EC have been infrequently reported since its original description in 1949. EC has been reported alone or in combination with manifestations such as eosinophilic cholangitis, hypereosinophilic syndromes (HES), and parasitic infestations. The patient described herein gave no indications of such previously suggested causes of EC. This report illustrates an authentic case of idiopathic EC.
Keywords: eosinophilic cholecystitis, gallbladder disease
|
Introduction
|
|---|
Eosinophilic cholecystitis (EC) is an uncommon condition that was first described in 1949 [1]. EC has a clinical presentation similar to that of typical cholecystitis, with right upper quadrant (RUQ) pain and an elicited Murphy’s sign. (With firm palpation of the RUQ at the costal margin, the patient is asked to breathe deeply. Upon inhalation, if the patient stops suddenly or catches his or her breath, this is considered a positive Murphy’s sign. Upon descent the inflamed gallbladder makes contact with the examiner’s fingertips and produces this reaction). Histologically, EC is distinguished by a dense transmural infiltration of eosinophils composing 90% or more of the leukocytes [2,3].
The etiology of EC is unclear, but suggested origins include allergies, local diathesis involving gallstones, parasites, acalculous cholecystitis, HES (where the eosinophils invade tissues such as brain, heart, lung, liver, and gallbladder), and eosinophilic gastroenteritis (EGE) [2,4–10].
We report a case of EC in a man who presented with RUQ pain. Laboratory and diagnostic studies validated the diagnosis of acute cholecystitis. A search of the literature revealed 24 individual accounts of EC since 1949 (Table 1
, see page 184). The majority of these cases was associated with EGE, HES, and eosinophilic cholangiopathy, while the remaining cases involved either parasitic infestation or acalculous cholecystitis [3,4,8,10,11]. Another 3 reports reviewed resected gallbladders in large sample populations [5,13,14].
In a series by Fox and Mainwaring [13], 16 of 625 gallbladders surgically removed during a 2-yr period exhibited eosinophilic infiltration. Three of the 16 were marked by pure eosinophilic infiltration, In the series published by Dabbs [5], 217 gallbladders were reviewed over a 2-yr period and 14 instances (6.5%) of EC were observed. A study reported by Muhlberger [14] revealed 55 of 660 resected gallbladders (8.3%) that had 
25% eosinophilic infiltration.
|
Case Report
|
|---|
The patient was a 29-yr-old obese Hispanic man who presented with 2 days of unremitting RUQ pain associated with chills and loss of appetite. The patient had been medically treated for cholelithiasis at another institution prior to admission.
Past medical history was significant for gastric bypass surgery 10 yr prior and an allergy to latex. No drug allergies were reported. The patient was maintained on cephalosporins during the hospital stay. He denied any illicit drug use and admitted to social smoking and daily alcohol consumption. Physical examination was pertinent for RUQ tenderness and a positive Murphy’s sign.
The initial laboratory tests showed a blood white blood cell count of 11,800 (reference interval, RI, 4.5–11 x103/mm3) with 354 eosinophils. Serum total bilirubin was 1.4 mg/dl, (RI 0.4–2 mg/dl), aspartate aminotransferase (AST) 341 U/L, (RI 12–54 U/L), alanine aminotransferase (ALT) 225 U/L, (RI 17–63 U/L), and alkaline phosphatase (ALP) 257 U/L, (RI 38–126 U/L).
Ultrasound of the abdomen demonstrated multiple gallstones and an 8 mm common bile duct. HIDA scan disclosed findings consistent with cholecystitis and a patent common bile duct without evidence of cystic duct obstruction. Endoscopic retrograde cholangiopancreatography could not be undertaken because of the patient’s history of gastric bypass surgery. Magnetic resonance cholangiopancreatography (MRCP) was done and revealed an enlarged gallbladder with numerous gallstones in the neck and a small amount of sludge. The common duct had a small filling defect proximally, possibly representing a gallstone. Pancreatic duct dilatation was not present.
The patient underwent an open cholecystectomy. An inflamed gallbladder with a thickened wall and numerous small stones was removed. There was no evidence of cysts or thrombosis of the vasculature. Pathologic examination revealed >90% eosinophilic infiltration in the submucosa as illustrated in Fig. 1.
|
Discussion
|
|---|
In the majority of cases of cholecystitis, gallstone impaction in the neck of the gallbladder or cystic duct sets off the inflammatory process. The bile concentrates, which leads to chemical irritation and may facilitate bacterial incursion [10]. Lymphocytes, macrophages, and neutrophils comprise the inflammatory infiltrate along with mucosal edema, vascular congestion, and regions of necrosis. When the infiltrate is made up of an abundant amount of eosinophils, there are signs of inflammation, granulation tissue, and fibroblast propagation [13,14,15].
Eosinophilic infiltration of the gastrointestinal tract may occur not only as EC, HES (including eosinophilic cholangiopathy), or EGE, but also as eosinophilic granulomatous hepatitis and eosinophilic ascites [12]. The symptoms observed vary as to the location and depth of the gastrointestinal tract affected [16]. When there is mucosal invasion, post-prandial nausea and vomiting, weight loss, iron deficiency anemia, steatorrhea, malabsorption, and protein-losing enteropathy are observed. In muscularis propria invasion, nausea and vomiting also occur with abdominal distention and intermittent bowel obstruction. With subserosal invasion, abdominal pain, distention, pleural effusion, and ascites occur [17,18].
In a study of 625 surgically removed gallbladders in England, 16 had eosinophilic infiltration [13]. The examined gallbladders were divided into 2 categories based upon different histologic features of the inflammatory infiltrate [13]. The majority of these gallbladders was included in the first category, with eosinophils comprising up to 50% of the cells. The eosinophilic infiltrate had been most evident in the subserosal and muscularis, sparing the mucosa in 3 of the gallbladders [19].
Fifteen of the reported cases of EC were from Korea. The age of these patients ranged from 17–58 yr, with idiopathic etiology in most cases [3]. Two cases were due to parasitic infestation, one with Ecchinococcus and the other due to Clonorchis sinensis [3,4]. There were only two patients who had a positive history for allergy, specifically to penicillin and pethidine. Peripheral eosinophilia was noted in only 4 of the 15 cases. When peripheral eosinophilia is observed, the EC may be an expression of HES [2]. In cases without peripheral eosinophilia, a local reaction to bile or gallstones may be responsible [2].
Acalculous cholecystitis has also been associated with eosinophilic cholecystitis, with invasion of the mucosal surface by eosinophils [19]. Causes of acute acalculous cholecystitis include herbal medicine [7], critical illness, decreased motility of the gastrointestinal tract, major burns, and hyperalimentation [19]. Other causes include polyarteritis nodosa, lupus erythematosus, Crohn’s disease, sarcoidosis, and Sjogren’s syndrome [10].
In the absence of any apparent precipitating etiology, the case described herein is considered one of idiopathic EC. Pathologic examination of the gallbladder revealed >90% eosinophilic infiltration without evidence of parasitic infestation. Although a cephalosporin was used in this patient, he had no known allergies to cephalosporins or penicillin. Non-latex gloves were used during the hospital stay and operation. The patient had an unremarkable recovery and was discharged to home without any complications. Significant peripheral eosinophilia was not noted during the hospital course.
Treatment for EC, which oftentimes cannot be distinguished from cholecystitis, consists of surgical removal of the diseased gallbladder (cholecystectomy), comparable to the treatment of standard cholecystitis. Medical management may depend on the severity and disease location. Considerable infiltration of the muscularis by eosinophils with vascular occlusion entails surgery, whereas involvement of the mucosa or adventitia without vascular changes responds well to steroids (11). In our patient, definitive therapy with surgery led to full recovery.
In summary, idiopathic cholecystitis is significant because it is not apparent solely through laboratory tests. Considering peripheral eosinophilia, one must think of the number of entities that may cause such findings and rule out these causes before the diagnosis of idiopathic EC can be made with certainty. Idiopathic EC is a rare condition of which gastroenterologists, pathologists, and surgeons need to be cognizant. Although the presentation may be similar to general cholecystitis, the underlying etiology must be defined because if a causative factor is discovered, treatment may warrant more than a cholecystectomy or may lead to findings of other organ infiltration.
|
References
|
|---|
- Albot G, Poilleux, Olivier C, Libaude H, Cascarigny P. Les cholecystites a eosinophils. Presse Med 1949;39: 558–559.
- Felman RH, Sutherland DB, Conklin JL, Mitros FA. Eosinophilic cholecystitis, appendiceal inflammation, pericarditis, and cephalosporin-associated eosinophilia. Digest Dis Sci 1994;39:418–422.[Medline]
- Kim YH. Eosinophilic cholecystitis in association with Clonorchis sinensis infestation in the common bile duct. Clin Radiol 1999;54:552–554.[Medline]
- Russel COH, Dowling JP, Marshall RD. Acute eosinophilic cholecystitis in association with hepatic echinococcosis. Gastroenterology 1979;77:758–760.[Medline]
- Dabbs DJ. Eosinophilic and lymphoeosinophilic cholecystitis. Am J Surg Pathol 1993;17:497–501.[Medline]
- Rosengart TK, Rotterdam H, Ranson JHC. Eosinophilic cholangitis: a self-limited cause of extrahepatic biliary obstruction. Am J Gastroenterol 1990;85:582–585.[Medline]
- Adusumilli PS, Lee B, Parekh K, Farrelly P. Acalculous eosinophilic cholecystitis from herbal medicine: a review of adverse effects of herbal medicine in surgical patients. Surgery 2002;131:352–356.[Medline]
- Tenner S, Roston A, Lichtenstein D, Brooks D, Herlihy E, Carr-Locke D. Eosinophilic cholangiopathy. Gastrointest Endosc 1997;45:307–309.[Medline]
- Al-Abdulla NA, Schulick RD, Regan F. Hypereosinophilic sclerosing cholangitis: findings using half-Fourier magnetic resonance imaging. Hepatogastroenterology 2000;47:359–361.[Medline]
- Hepburn A, Coady A, Livingstone J, Pandit N. Eosinophilic cholecystitis as a possible late manifestation of the eosinophiliamyalgia syndrome. Clinical Rheumatology 2000;19:470–472.[Medline]
- Tajima K, Katagiri T. Deposits of eosinophil granule proteins in eosinophilic cholecystitis and eosinophilic colitis associated with hypereosinophilic syndrome. Digest Dis Sci 1996;41:282–288.[Medline]
- Butler TW, Feintuch TA, Caine WP. Eosinophilic cholangitis, lymphadenopathy, and peripheral eosinophilia: a case report. Am J Gastroenterol 1985;80:572–574.[Medline]
- Fox H, Mainwaring AR. Eosinophilic infiltration of the gallbladder. Gastroenterology 1972;63:1049–1052.[Medline]
- Muhlberger VF. Zur Morphologie der eosinophilen cholecystitis und die frage ihrer allergischen genese. Int Arch Allergy 1954;5:434–448.
- Hellstrom HR. Eosinophilic and lymphoeosinophilic cholecystitis. Am J Surg Pathol 1994:18:215.[Medline]
- Kerstein MD, Sheahan DG, Gudjonsson LJ. Eosinophilic cholecystitis. Am J Gastroenterol 1976; 66:349–352.[Medline]
- Rodriguez HE, Djoham RS, Cahill WJ, Connolly MM, Podbielski FJ. Laparoendoscopic diagnosis of eosinophilic enteritis. JSLS 1998;2:181–184.[Medline]
- Stringel G, Mercer S, Sharpe D, Shipman R, Jimenenez C. Eosinophilic gastroenteritis. Canad J Surg 1984;27: 182–183.[Medline]
- Parry SW, Pelias ME, Browder W. Acalculous hypersensitivity cholecystitis: hypothesis of a new clinicopathologic entity. Surgery 1988;911–916.
- Leegaard M. Eosinophilic cholecystitis. Acta Chir Scand 1980;146:295–296.[Medline]
- Steffen RM, Wyllie R, Petras RE, Caulfield ME, Michener WM, Firor HV, Norris DG. The spectrum of eosinophilic gastroenteritis: report of six pediatric cases and review of the literature. Clin Pediat 1991;30:404–411.[Medline]
Top
Abstract
Introduction
