Kimura Disease: A Case Report and Review of the Literature with A New Management Protocol

Fouda, Mohamed A.; Gheith, Osama; Refaie, Ayman; El-Saeed, Mohamed; Bakr, Mohamed A.; Wafa, Ehab W.; Abdelraheem, Mona; Sobh, Mohamed

doi:10.4061/2010/673908

Cited by 65 publications

(106 citation statements)

References 10 publications

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“…20 Since the year 2000, nephrotic syndrome with Kimura disease has been frequently reported in Chinese patients. [21][22][23][24][25][26] In the review of 10 Japanese patients with Kimura disease associated with proteinuria, renal biopsy confirmed glomerular IgE deposition in only one patient (Case 1 in Table 1) who exhibited a low level of proteinuria (0.6 g/day), and glomerular endocapillary and mesangial proliferation. In this patient (Case 1), interstitial lymphoid follicles with eosinophilic infiltration were also noted.…”

Section: Discussionmentioning

confidence: 99%

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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Section: Discussionmentioning

confidence: 99%

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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“…Cyclosporine, due to its effects on Th2 lymphocytes has been used as a therapy for Kimura's disease [11]. M A Fouda et al [12] have tried single-pulse intra-venous cyclophosphamide and subsequently maintained on mycophenolate mofetil (MMF) and high dose systemic steroid with a satisfactory response. Considered as an inflammatory process, the disease has an excellent prognosis, although it may recur locally and wax and wane over time.…”

Section: Discussionmentioning

confidence: 99%

Kimura’s Disease: Report of a Case & Review of Literature

Kar

Sethi

2012

J. Maxillofac. Oral Surg.

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Kimura's disease is an uncommon allergic disorder of unknown etiology affecting sporadic populations. A male patient in his third decade presented with a swelling in his left facial region. His investigations showed a raised eosinophil count. The lesion was excised surgically. He was put on a short term steroid therapy. Biopsy revealed it as Kimura's disease. Subsequent follow-ups have been satisfactory with no recurrences.

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“…Most cases reported to date involve young Asian males, with most patients being aged between 20 and 40 years [2]. The incidence of KD with coexisting renal disease ranges from 10% to 60% [3,4]. Herein, we report an atypical manifestation of KD accompanied with nephrotic syndrome (NS) occurring in a 45-year-old man.…”

Section: Letter To the Editormentioning

confidence: 99%

“…Proteinuria may occur in 12–16% of patients and 59–78% of them have a NS [3,4]. The renal pathologies reported in China have included minimal change disease, mesangioproliferative and membranous nephropathy, while a wider spectrum of histological lesions have been reported in other countries [5].…”

Section: Letter To the Editormentioning

confidence: 99%

Kimura disease accompanied with Nephrotic syndrome in a 45-year-old male

Gong

Labh

et al. 2015

Diagn Pathol

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Kimura disease (KD) is an uncommon chronic inflammatory disorder of unknown etiology, occurs mainly in Asian young males, presenting as subcutaneous growing masses, with a predilection for head and neck, with or without satellite lymphadenopathy. Herein, we report a case of an atypical manifestation of KD accompanied with NS in a middle-aged man, though the patient was clinically misdiagnosed previously. The diagnosis of KD can be difficult and misleading, so we must explore the main points of KD so as to prevent misdiagnosis.

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Kimura Disease: A Case Report and Review of the Literature with A New Management Protocol

Cited by 65 publications

References 10 publications

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

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

Kimura’s Disease: Report of a Case & Review of Literature

Kimura disease accompanied with Nephrotic syndrome in a 45-year-old male

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