Eosinophilic cystitis and idiopathic hypereosinophilic syndrome in an eight-year-old girl

Derakhshan, Dorna; Ilkhanipoor, Homa; Derakhshan, Ali

doi:10.4103/1319-2442.144272

Cited by 2 publications

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“…Only a high-dose steroid was effective (oral prednisolone 2 mg/kg/day), and cyclosporine was added to treat recurrent abdominal pain. In particular, Dorna et al 7) reported a similar case to ours. An 8 year old girl presented with a history of dysuria, increased urinary frequency, and suprapubic pain.…”

Section: Discussionsupporting

confidence: 85%

A Case of Hypereosinophilic Syndrome with Bladder Involvement in a 7-Year-Old Boy

2015

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Hypereosinophilic syndrome (HES) is characterized by the presense of hypereosinophilia with evidence of target organ damage. We report a patient diagnosed with eosinophilic cystitis and HES. A 7 year old boy had hematuria, dysuria, and increased urinary frequency for 1 day. Laboratory examinations revealed hypereosinophilia (eosinophils, 2,058/µL), hematuria, and proteinuria. Abdominal sonography revealed diffuse and severe wall thickening of the bladder. The patient was treated initially with antibiotics. However, his symptoms did not improve after 7 days. A computed tomography scan demonstrated severe wall thickening of the bladder and the hypereosinophilia persisted (eosinophils, 2,985/µL). The patient complained of chest discomfort, dyspnea, epigastric pain, and vomiting on hospital day 10. Parasitic, allergic, malignancy, rheumatologic, and immune workups revealed no abnormal findings. Chest X-rays, electrocardiography, and a pulmonary function test were normal; however, the hypereosinophilia was aggravated (eosinophils, 3,934/µL). Oral deflazacort was administered. A cystoscopic biopsy showed chronic inflammation with eosinophilic infiltration. The patient's respiratory, gastrointestinal, and urinary symptoms improved after 6 days of steroids, and he was discharged. The eosinophil count decreased dramatically (182/µL). The hypereosinophilia waxed and waned for 7 months, and the oral steroids were tapered and stopped. This case describes a patient diagnosed with eosinophilic cystitis and HES.

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Section: Discussionsupporting

confidence: 85%

A Case of Hypereosinophilic Syndrome with Bladder Involvement in a 7-Year-Old Boy

2015

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“…Cases have also been reported in association with idiopathic hypereosinophilic syndrome, such as observed in the present case. 13,14 Peripheral eosinophilia is detected in about 40% of cases. Radiologic testing by intravenous urography, ultrasonography, or CT scan often detects bladder wall thickening or solitary tumor-like lesions that can mimic bladder cancer.…”

Section: Discussionmentioning

confidence: 99%

BRAFmutation as a novel driver of eosinophilic cystitis

Choi

Tsigelny

Boichard

et al. 2017

Cancer Biology & Therapy

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Eosinophilic cystitis is a rare manifestation of hypereosinophilia and a cause of morbidity, including dysuria and hematuria. Although some cases can be attributed to infection or allergy, most cases are assessed to be idiopathic and treated with corticosteroids. However, hypereosinophilia can also be due to actionable clonal molecular alterations in the haematopoietic cells, similar to other myeloproliferative neoplasms. Common mutations associated with eosonophilic syndromes are of platelet-derived growth factor receptor α or β or c-kit, though other pathogenic mutations have been found by next generation sequencing. Determination of a specific mutation may therefore identify clonality and refine treatment of some cases. Here we review the molecular features of eosinophilic disorders. We also describe the use of a liquid biopsy of circulating cell-free DNA in the workup of a case of eosinophilic cystitis in which next generation sequencing of cell-free DNA showed a BRAF I463T mutation. In silico modeling supports the functional impact and potential clinical relevance of BRAF I463T.

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Eosinophilic cystitis and idiopathic hypereosinophilic syndrome in an eight-year-old girl

Cited by 2 publications

References 0 publications

A Case of Hypereosinophilic Syndrome with Bladder Involvement in a 7-Year-Old Boy

A Case of Hypereosinophilic Syndrome with Bladder Involvement in a 7-Year-Old Boy

BRAFmutation as a novel driver of eosinophilic cystitis

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