Eosinophilic cholangitis coexisted with idiopathic thrombocytopenic purpura: Report of a case.

Eosinophilic cholangitis is a rare disease of which only 31 cases have been reported. Eosinophilic infiltration causes stricture of the bile duct diffusely or locally, and the imaging of eosinophilic cholangitis resembles primary sclerosing cholangitis or cancer of the bile tract. For eosinophilic cholangitis, treatment with steroid is effective and the prognosis is good. Therefore, its accurate diagnosis is very important. Here, we describe a patient with eosinophilic cholangitis who was also diagnosed with idiopathic thrombocytopenic purpura (ITP). He was treated for ITP using prednisolone, the unexpected sudden interruption of which caused severe deterioration of eosinophilic cholangitis and acute cholecystitis. Cholecystectomy and choledochojejunostomy were performed, and the addition of treatment by prednisolone resulted in a good clinical course. This is the first report on eosinophilic cholangitis coexisting with ITP.

Acute acalculous cholecystitis with eosinophilic infiltration.

We report a case of acute acalculous cholecystitis with eosinophilic infiltration. A previously healthy 6-year-old boy was referred with right abdominal pain. Imaging demonstrated marked thickening of the gallbladder wall and peri-cholecystic effusion. Acute acalculous cholecystitis was diagnosed. Symptoms persisted despite conservative treatment, therefore cholecystectomy was performed. Pathology indicated infiltration of eosinophils into all layers of the gallbladder wall. The postoperative course was uneventful and the patient has had no further symptoms. Eosinophilic cholecystitis is acute acalculous cholecystitis with infiltration of eosinophils. The causes include parasites, gallstones, allergies, and medications. In addition, it may be seen in conjunction with eosinophilic gastroenteritis, eosinophilic pancreatitis, or both. An allergic reaction to abnormal bile is thought to be the underlying cause. The present case did not fulfill the diagnostic criteria of eosinophilic cholecystitis, but this may have been in the process of developing.

A case of eosinophilic cholecystitis: Radiologic-pathologic correlation.

Eosinophilic cholecystitis (EC) is a rare condition that is characterized by eosinophilic infiltration in the gallbladder wall. We report the case of a 35-year-old woman who presented with unremitting right upper quadrant pain for 1 month. Computed tomography showed a strongly enhanced inner layer of the gallbladder wall. Magnetic resonance imaging of the same area showed low signal intensity on T2-weighted imaging. Cholecystectomy was performed, and histological examination of the surgical specimen revealed >100 eosinophils per high-power field in the inner subserosal layer. The area of these histological findings corresponded to the strongly enhanced inner layer of the gallbladder wall identified on computed tomography.

Eosinophilic Cholecystitis as an Atypical Etiology of Biliary Colic: A Case Report and Review of the Literature.

Eosinophilic cholecystitis (EC) is an uncommon cause of acute cholecystitis; the clinical presentation is indistinguishable from other types of cholecystitis, and the diagnosis is made by histopathology study. We present the case of a 73-year-old male patient with right hypochondrial abdominal pain suggestive of symptomatic cholelithiasis. There were no significant findings at the blood workup or physical examination; he underwent a cholecystectomy and was later diagnosed with eosinophilic cholecystitis by histopathology. EC may be associated with some other systemic conditions, such as eosinophilic granulomatosis, eosinophilic ascites, or parasitosis, which will require specific management.

Sustained Eosinophilic Cholangitis Due to a Mite Allergy Mimicking Sclerosing Cholangitis.

Eosinophilic cholangiopathy (EC) presents with thickening and stenosis of the bile duct wall that is histologically characterized by eosinophil infiltration. The diagnosis is often difficult. We herein report a patient who had been followed up with a diagnosis of primary sclerosing cholangitis but had a final diagnosis of EC based on eosinophilia, histological findings of bile duct and liver biopsy specimens, and a review of a previous surgical specimen of the gallbladder. Antigen tests, isolation from her house, and accidental re-exposure to the antigen revealed that the causative antigen was the mite Dermatophagoides pteronyssinus.

Eosinophilic cholecystitis in children: Case series.

INTRODUCTION: Eosinophilic cholecystitis (EC) is rarely seen in the pediatric population. Most of the available literature comes from adult patients, while only anecdotal cases have been reported in children. OBJECTIVE: To describe the clinical course, management, and outcomes of all EC cases treated at a children's hospital. MATERIAL AND METHODS: All cholecystectomy specimens obtained between 2011 and 2017 were retrospectively reviewed. EC was diagnosed when more than 90% of the inflammatory cells in the gallbladder wall were eosinophils, whereas lymphoeosinophilic cholecystitis (LEC) was diagnosed when the percentage of eosinophils was between 50 and 90. We analyzed all clinical aspects of patients with EC and LEC. RESULTS: We identified and reviewed 134 cholecystectomy specimens. Of them, 8 (6.0%) were classified as EC, and 3 (2.2%) as LEC. The mean age at presentation was 12.6 (2-17) years. The female-to-male ratio was 1.5/1. One patient had a history of hereditary spherocytosis, and 3 patients had systemic eosinophilia. All patients presented with clinical and radiological signs of acute cholecystitis and underwent cholecystectomy. Acute cholecystitis was confirmed by histopathology in all cases. All cases of EC and LEC had cholelithiasis. None of the patients required additional treatment. All patients recovered uneventfully. CONCLUSIONS: EC is rarely seen in children, it does not have a specific clinical presentation, and it is always associated with cholelithiasis. The diagnosis is made postoperatively by histopathology. All patients in our study presented with acute cholecystitis. TYPE OF STUDY: Retrospective review. LEVEL OF EVIDENCE: Level IV.

Eosinophilic Cholangitis with Poor Prognosis after Corticosteroid- and Ursodeoxycholic Acid-Related Remission of Peripheral and Peribiliary Eosinophilia.

A 79-year-old man presented with high fever, marked eosinophilia, altered biochemical liver function tests (LFT) with predominance of biliary enzymes, and severe wall thickening of the gallbladder. Magnetic resonance cholangiopancreatography (MRCP) suggested cholecystitis, without signs of biliary strictures. Laparoscopic cholecystectomy and exploratory liver excision revealed eosinophilic cholangitis and cholecystitis, complicated with hepatitis and portal phlebitis. Prednisolone monotherapy rapidly improved peripheral eosinophilia, but not LFT. Liver biopsy showed that infiltrating eosinophils were replaced by lymphocytes and plasma cells. Treatment with ursodeoxycholic acid improved LFT abnormalities. Nevertheless, after 2 months, transaminase-dominant LFT abnormalities appeared. Transient prednisolone dose increase improved LFT, but biliary enzymes' levels re-elevated and jaundice progressed. The second and third MRCP within a 7-month interval showed rapid progression of biliary stricture. The repeated liver biopsy showed lymphocytic, not eosinophilic, peribiliary infiltration and hepatocellular reaction to cholestasis. Eighteen months after the first visit, the patient died of hepatic failure. Autopsy specimen of the liver showed lymphocyte-dominant peribiliary infiltration and bridging fibrosis due to cholestasis. Though eosinophil-induced biliary damage was an initial trigger, repeated biopsy suggested that lymphocytes played a key role in progression of the disease. Further studies are needed to elucidate the relationship between eosinophils and lymphocytes in eosinophilic cholangitis.

An unusual case of a gallbladder polyp.

A 63-year old female patient with a medical history of hypereosinophilic syndrome with neurological and pulmonary involvement presented for a routine follow-up. The patient was asymptomatic but a routine scheduled ultrasound showed a gallbladder polyp of 19mm. One month later this polyp had grown to 36 mm. On magnetic resonance imaging of the liver there was a suspicion of gallbladder cancer and for this reason cholecystectomy was performed. Pathology however showed eosinophilic infiltration. Serum analysis showed an increase in her eosinophil count. The diagnosis of hypereosinophilic syndrome with eosinophilic infiltration of the gallbladder was made. The dose of corticosteroids was augmented and she recovered completely post-operatively with no residual flares of other organ damage during follow up.

A rare case of eosinophilic cholangiopathy.

Eosinophilic cholangiopathy is termed as a rare, benign, and self-limiting disease. Moreover, the interference of malignant tumor to diagnosis and the changing process of disease make the accurate treatment proposal challenging. A significant number of patients require surgery for the definitive diagnosis and resolution of symptoms. We put forward a case of eosinophilic cholangiopathy infiltrating the gallbladder and bile duct with bone marrow involved, coupled with peripheral eosinophilia. The patient underwent a successful treatment using laparoscopic cholecystectomy and steroids, instead of extrahepatic bile duct excision with Roux-en-Y hepaticojejunostomy. The patient gets an accurate treatment in a minimally invasive manner. In conclusion, surgery refers to not only a diagnostic methodology but also a treatment. When the bile duct and gallbladder are involved at the same time, and cannot distinguish benign and malignant diseases, laparoscopic cholecystectomy is feasible, the effect is the same, and the symptoms of eosinophilic cholecystitis are relieved.

Eosinophilic Cholecystitis Associated with Eosinophilic Granulomatosis with Polyangiitis.

We report a case of eosinophilic cholecystitis associated with eosinophilic granulomatosis with polyangiitis (EGPA) complicated by cerebral hemorrhage. A 60-year-old man presented to a local hospital with a diagnosis of acute cholecystitis, with persistent fever and epigastric pain for 2 weeks. His symptoms persisted despite 3-week hospitalization; therefore, he was transferred to our hospital for further evaluation. Laboratory investigations upon admission showed white blood cells 26,300/µL and significant eosinophilia (eosinophils 61%). Abdominal computed tomography revealed no gallbladder enlargement but a circumferentially edematous gallbladder wall. Additional blood test results were negative for antineutrophil cytoplasmic and perinuclear antineutrophil cytoplasmic antibodies; however, immunoglobulin (Ig)G and IgE levels were high at 1,953 mg/dL and 3,040/IU/mL, respectively. He improved following endoscopic transnasal gallbladder drainage for cholecystitis and was diagnosed with EGPA and received corticosteroid and immunosuppressant combination therapy. The eosinophil count decreased immediately after treatment, and abdominal pain and numbness resolved. He returned with left-sided suboccipital hemorrhage likely attributed to EGPA 6 months after discharge. EGPA is characterized by inflammation of small blood vessels and clinically manifests with an allergic presentation of bronchial asthma, as well as renal dysfunction, interstitial pneumonia, enteritis, and cerebral hemorrhage. Few reports have described cholecystitis as a presenting symptom of EGPA. We report a rare case of such a presentation with added considerations.

Hypereosinophilic Syndrome, Multiorgan Involvement and Response to Imatinib.

Hypereosinophilic syndrome (HES) is an uncommon syndrome characterized by peripheral blood eosinophils count of more than 1,500/mm3 with associated tissue damage. It can be either primary or secondary, for example, due to parasitic infections or inflammation. We present a case of a 49-year-old Asian female with recurrent hospital admissions for cholecystitis, gastritis, urinary cystitis, and pancreatitis. Her peripheral blood count showed excessive eosinophils 15,600-19,000/mm3 on different occasions. Pathology of her gallbladder and her gastric biopsies showed eosinophilic infiltration. Her bone marrow biopsy showed a normocellular marrow with active trilineage hematopoiesis, eosinophilia, mild megakaryocytic hyperplasia with a few atypical forms, and mild T-cell lymphocytosis. Flow cytometry showed no evidence of acute leukemia, or T-cell or B-cell lymphoproliferative disorder. On fluorescent in-situ hybridization (FISH), myeloproliferative neoplasms (MPN) testing was negative for platelet-derived growth factor receptor-alpha (PDGFRA), platelet-derived growth factor receptor-beta (PDGFRB), and fibroblast growth factor receptor-1 (FGFR1) rearrangement. Despite not having the FIP1L1-PDGFRA (factor interacting with PAPOLA and CPSF1-platelet-derived growth factor receptor, alpha polypeptide) gene fusion, our patient responded to the treatment with a significant decrease in her absolute eosinophils count and resolution of her symptoms.

Eosinophilic Cholecystitis Occurred in a Patient With Refractory Eosinophilic Airway Inflammation: A Case Report.

BACKGROUND: Eosinophilic cholecystitis (EC) is a rare condition that presents in a manner comparable to acute cholecystitis. The diagnosis is based on classical symptoms of cholecystitis with excessive eosinophilic infiltration within the gallbladder. EC has been reported alone or in combination with manifestations, such as eosinophilic gastrointestinal tract inflammation. However, association with airway inflammation in patients with EC is rare.Case Presentation: We report the case of a 65-year-old man who had refractory eosinophilic chronic rhinosinusitis with bronchial asthma. A second endoscopic sinus surgery (ESS) was performed as treatment for recurrent nasal polyps. EC occurred while inhaled corticosteroids were reduced after ESS. Pathologic examination of the excised gallbladder demonstrated submucosal infiltration with a number of eosinophils. Furthermore, immunohistostaining revealed many galectin-10-positive cells in both the gallbladder mucosa and the paranasal sinus mucosa. Galectin-10 is a major constituent of human eosinophils, also known as the Charcot-Leyden crystal protein, which has been linked with eosinophilic inflammation. Interestingly, nasal polyps were reduced without any additional treatments 1 month after the cholecystectomy. CONCLUSIONS: We experienced a rare case wherein EC onset occurred in a patient with refractory eosinophilic airway inflammation during inhaled corticosteroid tapering. Galectin-10 might help diagnose rare cases of eosinophilic inflammation in multiple organs.

Eosinophilic cholecystitis: a retrospective study spanning a fourteen-year period. / Colecistitis eosinofílica: estudio retrospectivo de 14 años.

INTRODUCTION AND AIMS: Eosinophilic cholecystitis is a rare entity that was first described in 1949 and is clinically indistinguishable from calculous cholecystitis. Histologically, there is transmural inflammatory infiltration of the gallbladder wall, more than 90% of which is composed of eosinophils. The aim of the present article was to review the prevalence of eosinophilic cholecystitis and analyze the clinical and surgical characteristics of patients diagnosed with the disease that were operated on at our hospital. MATERIALS AND METHODS: A retrospective study was conducted on patients that underwent cholecystectomy and whose postoperative histopathologic diagnosis was eosinophilic cholecystitis, within the time frame of January 2000 and August 2014. The demographic, clinical, paraclinical, surgical, and histopathologic variables were described. RESULTS: Over a period of 14 years, a total of 7,494 patients underwent cholecystectomy. Of those patients, 12 had a postoperative histologic diagnosis of eosinophilic cholecystitis. Mean patient age for disease presentation was 39 years (±11 years), and female sex was predominant, with 7 cases. All the patients had concomitant gallstones and 10 patients presented with acute cholecystitis that required urgent cholecystectomy. All the cases were considered idiopathic. We found a prevalence of 0.16%, corresponding to 1 case for every 625 cholecystectomies performed at our hospital. CONCLUSION: We found a low prevalence of eosinophilic cholecystitis (0.16%) in our study population. The clinical manifestations were similar to those of calculous cholecystitis. Cholecystectomy is adequate treatment in patients with idiopathic disease.

Case report: A rare case of eosinophilic cholecystitis presenting after talc pleurodesis for recurrent pneumothorax.

Eosinophilic cholecystitis (EC) is a rare inflammatory condition of the gallbladder, confirmed by a cellular infiltrate comprised of more than 90% eosinophils in the gallbladder wall on histological examination. Although the etiology of EC is largely unknown, local autoimmune reactions within the gallbladder wall to inflammatory mediators from distal sites of inflammation have been hypothesized. Talc pleurodesis (TP) is a common clinical procedure used within respiratory medicine. However, it is associated with activation of systemic acute inflammatory responses including an increase in serum interleukin-8 (IL-8), which is a potent mediator of eosinophil chemotaxis. We report a case of EC following a TP procedure for persistent, secondary pneumothorax.

Peripheral eosinophilia - is it a predictable factor associated with eosinophilic cholecystitis?

BACKGROUNDS/AIMS: The purpose of this study was to evaluate the role of peripheral eosinophilia as a predictable factor associated with Eosinophilic cholecystitis (EC) compared with other forms of cholecystitis in patients who underwent a cholecystectomy. METHODS: Between January 2001 and May 2011, the histopathologic features of 3,539 cholecystectomy specimens were reviewed retrospectively. EC was diagnosed in 30 specimens (0.84%). Data from 30 consecutive patients with EC (eosinophilic cholecystitis group [E-group]) were compared with a retrospective control group of 60 patients (other cholecystitis group [O-group]) during the same period. The two groups were matched for age, gender, and the presence of cholelithiasis. RESULTS: The median absolute eosinophil count 1 day post-operatively was 144 cells/mm(3) (range: 9-801 cells/mm(3)) in the E-group and 93 cells/mm(3) (range: 0-490 cells/mm(3)) in the O-group (p=0.036). Pre-operative peripheral eosinophilia was more common in the E-group than the O-group (20% vs. 3.3%, p=0.015). Multivariate analysis revealed that pre-operative peripheral eosinophilia was an independent significant predictable factor associated with EC (odds ratio=7.250, 1.365 <95% confidence interval<38.494, p=0.020). CONCLUSIONS: In the present study, pre-operative peripheral eosinophilia was shown to be an independent predictable factor associated with EC. Further researches seem to be necessary to confirm this finding.

A Case of Idiopathic Eosinophilic Cholecystitis Treated with Steroids / 대한내과학회지

Eosinophilic cholecystitis is a rare disease characterized by transmural leukocyte infiltration composed of more than 90% eosinophils. Eosinophilic cholecystitis is clinically indistinguishable from ordinary cholecystitis, and as a rule it leads to cholecystectomy. We report a case of idiopathic eosinophilic cholecystitis treated with steroids. A 75-year-old woman presented with a classic history of acute cholecystitis and a peripheral eosinophilia of 41.8%. There was no evidence of allergy or parasitic infestation. An abdominal ultrasonography and computed tomography (CT) scan showed an edematous, thickened gallbladder wall, but no gallstones. There was no evidence of eosinophilic infiltration in other organs. Cholecystectomy was not performed because the patient refused surgical management. However, fever, abdominal pain, and peripheral eosinophilia persisted despite antibiotic and conservative therapy. Therefore, we attempted treatment with prednisolone. A week later, the symptoms disappeared and the peripheral eosinophilia normalized.

Peripheral eosinophilia - is it a predictable factor associated with eosinophilic cholecystitis?

BACKGROUNDS/AIMS: The purpose of this study was to evaluate the role of peripheral eosinophilia as a predictable factor associated with Eosinophilic cholecystitis (EC) compared with other forms of cholecystitis in patients who underwent a cholecystectomy. METHODS: Between January 2001 and May 2011, the histopathologic features of 3,539 cholecystectomy specimens were reviewed retrospectively. EC was diagnosed in 30 specimens (0.84%). Data from 30 consecutive patients with EC (eosinophilic cholecystitis group [E-group]) were compared with a retrospective control group of 60 patients (other cholecystitis group [O-group]) during the same period. The two groups were matched for age, gender, and the presence of cholelithiasis. RESULTS: The median absolute eosinophil count 1 day post-operatively was 144 cells/mm3 (range: 9-801 cells/mm3) in the E-group and 93 cells/mm3 (range: 0-490 cells/mm3) in the O-group (p=0.036). Pre-operative peripheral eosinophilia was more common in the E-group than the O-group (20% vs. 3.3%, p=0.015). Multivariate analysis revealed that pre-operative peripheral eosinophilia was an independent significant predictable factor associated with EC (odds ratio=7.250, 1.365 <95% confidence interval<38.494, p=0.020). CONCLUSIONS: In the present study, pre-operative peripheral eosinophilia was shown to be an independent predictable factor associated with EC. Further researches seem to be necessary to confirm this finding.