Steroid-sensitive nephrotic syndrome associated with Kimura disease

Nakahara, C; Wada, Takahito; Kusakari, Jun; Kanemoto, Katsuyoshi; Kinugasa, Hideyo; Sibasaki, M.; Nagata, Michio; Matsui, Akira

doi:10.1007/s004670050798

Cited by 47 publications

(40 citation statements)

References 20 publications

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“…The Th2 cell, which is the main factor in the late-phase response in the allergic process, plays the major role in the production of cytokines, including IL-4 and IL-13, both of which regulate IgE and IL-5 [14]. Nakahara et al [15] reported elevated IL-5 levels in a patient having steroid-sensitive nephrotic syndrome. These authors concluded that the Th2 cell should be the major factor for the development of Kimura's disease.…”

Section: Discussionmentioning

confidence: 99%

“…Concerning the steroid response, especially in pediatric patients, Nakahara et al [15] reported recently that a steroid-sensitive nephrotic syndrome is associated with Kimura's disease, and Rajpoot et al [3], also recently, reported steroid-resistant nephrotic syndromes in children who developeded end-stage renal failure later and in whom kidney histopathology revealed mild to moderate mesangial hypercellularity with an increase in mesangial matrix. Reports of adult patients also revealed various histopathologies of the kidney in those who had nephrotic syndrome associated with Kimura's disease.…”

Section: Discussionmentioning

confidence: 99%

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Steroid-Resistant Nephrotic Syndrome Associated with Kimura’s Disease

Chartapisak¹,

Opastirakul²

2002

Am J Nephrol

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Kimura’s disease is a chronic inflammatory disease characterized by tumor-like lesions in the soft tissue and lymph nodes of head and neck area or parotid gland. It has a high frequency of an association with nephrotic syndrome. Reported cases of nephrotic syndrome and Kimura’s disease were mostly from adult patients with only 5 children mentioned. This study reports the case of a 15-year-old-boy who manifested with steroid-resistant nephrotic syndrome for 4 years. Pathological examination of the kidney revealed mild mesangial proliferation. Subsequently, he developed a soft-tissue mass in the parotid gland area. Histopathological investigation of the mass revealed eosinophilic infiltration together with plasma cells and mast cells which is a main characteristic of Kimura’s disease. The patient, however, continued to have nephrotic-range proteinuria after removing the subcutaneous mass.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Steroid-Resistant Nephrotic Syndrome Associated with Kimura’s Disease

Chartapisak¹,

Opastirakul²

2002

Am J Nephrol

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“…Low-dose cyclosporine combined with low-dose prednisolone is a recent trend in the treatment of nephrotic syndrome. 17,20 Renal biopsy demonstrated minimal glomerular change on light microscopy and no glomerular deposition by fluorescent immunohistochemistry in the present patient. Glomerular IgE deposition in this patient remains unknown because routine fluorescent immunohistochemical staining did not include IgE.…”

Section: Discussionmentioning

confidence: 86%

Nephrotic syndrome during the tapering of oral steroids after pathological diagnosis of Kimura disease from a lacrimal gland mass: case report and review of 10 Japanese patients

Matsuo

Tanaka

Kinomura

2017

JCEH

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JC EH lin xp ematopathol Case Study INTRODUCTIONThe ocular adnexa are supporting tissues for the eye globe, comprising the eyelid, lacrimal gland and sac, extraocular muscles, and orbital interstitial tissue. These sites are frequently affected by lymphoma 1 and inflammatory diseases such as IgG4-related disease.2 Magnetic resonance imaging of ocular adnexal masses cannot differentiate inflammatory diseases from lymphoma. Therefore, excisional biopsy is required to establish the diagnosis and decide the treatment strategy.Kimura disease predominantly occurs in young Asian males and presents as soft subcutaneous granulomas, which are frequently seen in the head and neck. Lymphoid follicular hyperplasia and marked eosinophilic infiltration are the pathological characteristics of Kimura disease, and blood examination commonly reveals eosinophilia and high serum immunoglobulin E (IgE).3-6 Kimura disease rarely manifests as orbital tumors such as lacrimal gland masses. [7][8][9] Pathologically, it remains controversial whether Kimura disease in Asian reports and angiolymphoid hyperplasia with eosinophilia in Western reports are the same entity. 6 In this study, we report a patient with Kimura disease who initially presented with a lacrimal gland mass and then developed nephrotic syndrome one month after the tapering of oral prednisolone. We also reviewed 10 reported Japanese patients with Kimura disease associated with proteinuria. 10-18 CASE REPORTA 42-year-old man noticed upper eyelid swelling on the left side that subsided spontaneously 5 months ago. He again developed left eyelid swelling with left exophthalmos 2 months ago, and was referred to an ophthalmologist. In his past history, he underwent bilateral tonsil extirpation due to otitis media at the age of 5 years. He developed systemic urticaria lasting a week after eating sashimi 3 years ago, and Nephrotic syndrome during the tapering of oral steroids after pathological diagnosis of Kimura disease from a lacrimal gland mass: case report and review of 10 Japanese patients A 42-year-old man with eosinophilia and high serum immunoglobulin E (IgE) developed a lacrimal gland mass on the left side. Excisional biopsy revealed hyperplasia of lymphoid follicles, and infiltration with lymphocytes and eosinophils around lacrimal gland acini, leading to the pathological diagnosis of Kimura disease. IgE-positive cells were mainly found along follicular dendritic cells, and a small number of IgG4-positive cells was present. One month after oral prednisolone was started at 40 mg daily and tapered to 10 mg daily, he developed lower leg edema on both sides and marked proteinuria (10.8 g/day). Renal biopsy showed no glomerular abnormalities, no immunoglobulin deposition, and no tubulointerstitial infiltration with eosinophils, leading to the diagnosis of minimal change nephrotic syndrome. Proteinuria subsided in response to an increased dose of prednisolone to 30 mg daily. Proteinuria relapsed three times in the following 5 years when oral prednisolone was tapered. In conclus...

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“…Local irradiation has been successful in controlling local disease, with few recurrences [19], but has been used mainly when other methods have failed to give control, and is not recommended in young patients [20]. Cyclosporin A has also been used successfully [21]. Treatment needs to be undertaken with the knowledge that the clinical course is benign, and no malignant transformation of Kimura disease has been described.…”

Section: Discussionmentioning

confidence: 98%

Vincristine treatment of nephrotic syndrome complicated by Kimura disease

et al. 2005

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Kimura disease is a rare inflammatory condition of unknown aetiology. It typically presents in young Asian males with the triad of non-tender subcutaneous swellings in the head and neck region, peripheral eosinophilia and raised serum IgE. About 16% of cases have associated renal disease. We present the case of a 10-year-old boy with a past history of steroid responsive, frequently relapsing nephrotic syndrome who developed a right submandibular swelling and eosinophilia. Kimura disease was diagnosed on the basis of clinical and histological findings. The condition recurred during relapses of nephrotic syndrome. Because of poor adherence with oral medication, our patient was treated with intravenous vincristine with synchronous remissions of his nephrotic syndrome and Kimura swellings on each occasion.

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Steroid-sensitive nephrotic syndrome associated with Kimura disease

Cited by 47 publications

References 20 publications

Steroid-Resistant Nephrotic Syndrome Associated with Kimura’s Disease

Steroid-Resistant Nephrotic Syndrome Associated with Kimura’s Disease

Nephrotic syndrome during the tapering of oral steroids after pathological diagnosis of Kimura disease from a lacrimal gland mass: case report and review of 10 Japanese patients

Vincristine treatment of nephrotic syndrome complicated by Kimura disease

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