A case of juvenile eosinophilic cholangitis: Rapid peripheral blood hypereosinophilia after admission leading to diagnosis

Fukatsu, Shusaku; Kitamura, Katsuya; Asai, Yasufumi; Nagai, Kazumasa; Kikuchi, Miho; Asano, Koichiro; Tadokoro, Kenichi; Yamanishi, Fumito; Tomita, Yusuke; Abé, Masakazu; Wada, Takuya; Matsue, Yubu; Nutahara, Daisuke; Taira, Junichi; Nakamura, Hironori; Itoi, Takao

doi:10.1002/jgh3.12454

Cited by 3 publications

(5 citation statements)

References 7 publications

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“…Matsumoto et al advocated that the EC bile duct image is reversible by treatment with CS (2), but bile duct stenosis often remains after treatment, as in the present case. Among individuals with EC, 30%-40% do not show eosinophilia (13)(14)(15). Our patient also showed no eosinophilia (eosinophil count >1,500/mm 3 ) for approximately 3 years of follow-up at our hospital and the previous hospital.…”

Section: Discussionmentioning

confidence: 49%

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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Section: Discussionmentioning

confidence: 49%

Sustained Eosinophilic Cholangitis Due to a Mite Allergy Mimicking Sclerosing Cholangitis

et al. 2022

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“…Furthermore, the number of infiltrating eosinophils in bile duct biopsies has not been well reported (only three cases), and the number of infiltrating eosinophils ranged from 4 to 60 per HPF. 8 Two biopsies were taken from the thickened bile duct wall in this case, and the eosinophil counts were greater than 50 and 200/HPF, respectively. There are no established criteria for eosinophil count in the histological diagnosis of EC.…”

Section: F I G U R Ementioning

confidence: 83%

“…6 Seow-En et al 7 proceeded with surgery for EC with bile duct stenosis, taking into consideration the exclusion of malignancy, recurrence with medical treatment, and side effects of steroid therapy, but it must be noted that EC is a benign disease and surgery is a highly invasive procedure. According to Fukatsu et al, 8 there are only seven reported cases of EC that were diagnosed by bile duct biopsy or cytology.…”

Section: Discussionmentioning

confidence: 99%

“…proceeded with surgery for EC with bile duct stenosis, taking into consideration the exclusion of malignancy, recurrence with medical treatment, and side effects of steroid therapy, but it must be noted that EC is a benign disease and surgery is a highly invasive procedure. According to Fukatsu et al., 8 there are only seven reported cases of EC that were diagnosed by bile duct biopsy or cytology. Furthermore, the number of infiltrating eosinophils in bile duct biopsies has not been well reported (only three cases), and the number of infiltrating eosinophils ranged from 4 to 60 per HPF 8 .…”

Section: Discussionmentioning

confidence: 99%

“…According to Fukatsu et al., 8 there are only seven reported cases of EC that were diagnosed by bile duct biopsy or cytology. Furthermore, the number of infiltrating eosinophils in bile duct biopsies has not been well reported (only three cases), and the number of infiltrating eosinophils ranged from 4 to 60 per HPF 8 . Two biopsies were taken from the thickened bile duct wall in this case, and the eosinophil counts were greater than 50 and 200/HPF, respectively.…”

Section: Discussionmentioning

confidence: 99%

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A case of eosinophilic cholangitis without bile duct stenosis diagnosed by bile duct biopsy

et al. 2022

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Eosinophilic cholangitis (EC) is a rare benign disease that is often misdiagnosed as a malignancy due to the development of biliary stricture. This disease is generally diagnosed by liver biopsy or surgery. Herein, we report a case of EC diagnosed in an 86‐year‐old Japanese woman, who presented with fever, elevated eosinophil count, and elevated liver enzyme level, based on intraductal ultrasound evaluation showing bile duct wall thickening and bile duct biopsy of the same site. We diagnosed this case as EC based on the triad of wall thickening of the biliary system, histopathological findings of eosinophilic infiltration of the biliary tract, and reversibility of biliary abnormalities without treatment. Bile duct biopsy during endoscopic retrograde cholangiopancreatography (ERCP) is rarely used to confirm the diagnosis of EC without bile duct stenosis. For EC and cholecystitis associated with eosinophilia, bile duct biopsy under ERCP, which is less invasive, should be considered. This patient was older than the previously reported patients, and the value of a minimally invasive diagnosis was high.

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Case report: Rare benign obstructive disease of the biliary tract—a rare case of eosinophilic cholangitis

Huang,

Fang,

Zhong

et al. 2024

Front. Med.

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ObjectiveEosinophilic cholangitis (EC) is an uncommon form of benign biliary obstruction. It frequently eludes accurate clinical diagnosis, leading to inappropriate treatment methods. It is our aspiration that this clinical report will impart comprehensive insights into EC and, specifically, the critical role of tomographic examination.Case summaryA 34-year-old man was urgently admitted to the hospital due to excruciating abdominal distress persisting for several hours. Following a six-day course of anti-inflammatory therapy, his symptoms displayed marginal improvement, prompting his discharge. He returned to the hospital a month later for re-examination on doctor’s orders. Based on the results of the re-examination, the patient refused steroid hormone shock therapy and subsequently underwent laparoscopic left-lateral hepatic lobectomy in order to confirm the diagnosis. The preoperative absolute counts of eosinophils in the peripheral blood were documented as 2.3 × 109/L, 3.06 × 109/L, and 1.50 × 109/L consecutively; concurrently, the corresponding percentages of eosinophils were quantified at levels of 21.90%, 30.70%, and 19.20%. The subsequent postoperative pathological assessment unveiled EC as the definitive diagnosis. The patient has since remained free from disease recurrence and is presently alive.ConclusionWhen encountering a patient presenting with persistent elevation in absolute eosinophil count in peripheral blood, coupled with imaging manifestations suggestive of intrahepatic periductal inflammation, diagnosis of EC should be highly suspected. The most optimal diagnostic and therapeutic workflow for EC could entail CT-guided liver lesion biopsy, ensued by glucocorticoid pulse therapy, and finally, short-term monitoring utilizing CT or MRI (including T1WI, T2WI, DWI, CEMRI) techniques.

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A case of juvenile eosinophilic cholangitis: Rapid peripheral blood hypereosinophilia after admission leading to diagnosis

Cited by 3 publications

References 7 publications

Sustained Eosinophilic Cholangitis Due to a Mite Allergy Mimicking Sclerosing Cholangitis

Sustained Eosinophilic Cholangitis Due to a Mite Allergy Mimicking Sclerosing Cholangitis

A case of eosinophilic cholangitis without bile duct stenosis diagnosed by bile duct biopsy

Case report: Rare benign obstructive disease of the biliary tract—a rare case of eosinophilic cholangitis

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