Eosinophilic cholangitis with eosinophilic granulomatosis with polyangiitis: A case report and review of the literature

Yoshihara, Risa; Komai, Takashi; Nagafuchi, Yasuo; Tsuchida, Yoshiaki; Shoda, Hirofumi; Tanaka, Mariko; Ushiku, Tetsuo; Fujio, Keishi

doi:10.1016/j.alit.2019.08.002

Cited by 5 publications

(4 citation statements)

References 8 publications

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“…The prognosis and frequency of eosinophilic cholangiopathy with EGPA remain elusive. Previously, only 19 cases of EGPA in conjunction with eosinophilic cholecystitis have been reported [ 6 – 23 ], while only 2 studies reported EGPA together with eosinophilic cholangitis [ 24 , 25 ]. Most of the reported cases had a histopathological diagnosis, but in the present case, the contemporaneous appearance of EGPA and cholecystitis was diagnosed based on the patient’s symptoms, laboratory test results, and imaging findings, without a pathological diagnosis.…”

Section: Discussionmentioning

confidence: 99%

“…In general, EGPA is mainly treated with PDN; the EGPA practice guidelines recommend the use of PDN or cyclophosphamide for remission induction therapy and AZA for remission maintenance therapy [ 32 ]. In a recent case report, eosinophilic cholangitis, in conjunction with EGPA, was successfully treated by combining PDN and mepolizumab [ 24 ]. In the present case, PDN was administered in monotherapy, without the addition of cyclophosphamide or mepolizumab, which was sufficient to induce remission, while AZA was added to maintain it.…”

Section: Discussionmentioning

confidence: 99%

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Eosinophilic Granulomatosis with Polyangiitis Presenting as Steroid-Responsive Sclerosing Cholangitis and Cholecystitis: A Rare Case Report

2023

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Patient: Male, 47-year-old Final Diagnosis: Eosinophilic cholangitis • eosinophilic cholecystitis • eosinophilic granulomatosis with polyangiitis Symptoms: Epigastralgia • fatigue • fever • weigh loss Clinical Procedure: — Specialty: Rheumatology Objective: Unusual clinical course Background: Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare systemic vasculitic condition characterized by bronchial asthma and eosinophilia. While biliary involvement is uncommon in EGPA, we present a unique case of EGPA presenting as steroid-responsive sclerosing cholangitis and cholecystitis. This case highlights the importance of considering EGPA in the differential diagnosis of biliary diseases, especially in patients with a history of bronchial asthma. Case Report: A 47-year-old man with a history of bronchial asthma presented with fatigue, weight loss, and epigastralgia. Blood tests revealed eosinophilia and elevated inflammatory markers, leading to the diagnosis of EGPA. Further imaging studies, including magnetic resonance cholangiopancreatography and contrast-enhanced computed tomography, confirmed the presence of sclerosing cholangitis and cholecystitis, a rare manifestation of EGPA. Conclusions: Prompt treatment with prednisolone and azathioprine resulted in remission of symptoms and resolution of cholangitis and cholecystitis in this case. Our findings emphasize the importance of early recognition and appropriate management of EGPA-associated biliary involvement. Increased awareness of this rare manifestation may facilitate timely diagnosis and improve patient outcomes.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Eosinophilic Granulomatosis with Polyangiitis Presenting as Steroid-Responsive Sclerosing Cholangitis and Cholecystitis: A Rare Case Report

2023

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“…Generally, an allergic response to any antigen is assumed to be the cause. Nevertheless, the causative agent in each case has not always been identified (5)(6)(7)(8)(9)(10)(11).…”

Section: Discussionmentioning

confidence: 99%

Sustained Eosinophilic Cholangitis Due to a Mite Allergy Mimicking Sclerosing Cholangitis

et al. 2022

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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.

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“…12 Involvement of the hepatobiliary system in EGPA is very rare but has been reported. 13 The pathogenesis of EGPA remains largely unknown, but abnormal eosinophilic proliferation and tissue toxicity from eosinophil-derived proteins are thought to play major roles. 7 Eosinophils are complex cells that produce an array of molecules with diverse functions.…”

Section: Discussionmentioning

confidence: 99%

Budd–Chiari syndrome as a complication of eosinophilic granulomatosis with polyangiitis in a young Chinese man: a case report

Shi

Ding

et al. 2020

J Int Med Res

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Hepatic vein thrombosis is a rare occurrence in the clinical course of eosinophilic granulomatosis with polyangiitis (EGPA). The major mechanism of thrombosis has been postulated to involve the release of toxic proteins from eosinophils. A 36-year-old man with EGPA was admitted to our hospital in July 2018 with hematemesis and melena. Findings on physical examination included ascites and pigmentation of the lower extremities. Ultrasonography of the hepatic vein and inferior vena cava showed an obstruction of the hepatic vein. Magnetic resonance imaging showed low enhancement in the right hepatic vein region. At 34 years of age, the patient’s EGPA had initially presented as asthma with eosinophilia (white blood cell count of 11.46 × 1012/L with 14.6% eosinophils). His skin biopsy showed infiltration of inflammatory cells and eosinophils, especially around medium-sized vessels, which was consistent with EGPA. The patient was thus diagnosed with Budd–Chiari syndrome associated with EGPA.

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Eosinophilic cholangitis with eosinophilic granulomatosis with polyangiitis: A case report and review of the literature

Cited by 5 publications

References 8 publications

Eosinophilic Granulomatosis with Polyangiitis Presenting as Steroid-Responsive Sclerosing Cholangitis and Cholecystitis: A Rare Case Report

Eosinophilic Granulomatosis with Polyangiitis Presenting as Steroid-Responsive Sclerosing Cholangitis and Cholecystitis: A Rare Case Report

Sustained Eosinophilic Cholangitis Due to a Mite Allergy Mimicking Sclerosing Cholangitis

Budd–Chiari syndrome as a complication of eosinophilic granulomatosis with polyangiitis in a young Chinese man: a case report

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