Elevated Numbers of Cells Producing Interleukin-5 and Interleukin-10 in a Boy with Kimura Disease

Hosoki, Koa; Hirayama, Masahiro; Kephart, Gail M.; Kita, Hirohito; Nagao, Mizuho; Uchizono, Hiromasa; Toyoda, Hidemi; Senba, Yuko; Yu, I.; Komada, Yoshihiro; Ihara, Toshiaki; Fujisawa, Takao

doi:10.1159/000337777

Cited by 14 publications

(12 citation statements)

References 49 publications

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“…In our study, dermatologists diagnosed most patients, a fact that may account for the larger number of dermatological past conditions reported. These cutaneous manifestations may result from a physiopathological process similar to KD, characterized by a predominant expression of Th2 cytokines, stimulating eosinophils and IgE production [10,22] as in atopic dermatitis. The history of unspecific dermatitis was not associated with recurrence in our study though it appeared more frequently in patients with recurrence (69%) than in patients without (33%).…”

Section: Discussionmentioning

confidence: 99%

Retrospective Multicentric Study of 25 Kimura Disease Patients: Emphasis on Therapeutics and Shared Features with Cutaneous IgG4-Related Disease

Kottler

Barète

Quéreux³

et al. 2015

Dermatology

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Background: Kimura disease (KD) is a rare lymphoproliferative inflammatory disease of unknown etiology. Data regarding therapeutic modalities and pathophysiology are scarce. Objectives: Analyze therapeutic and follow-up data and compare KD with cutaneous IgG4-related disease (IgG4-RD). Methods: Multicentric retrospective study of 25 KD patients with analysis of treatment, follow-up and IgG4 immunostaining. Comparison with published cases of cutaneous IgG4-RD. Results: Patients were mostly male (84%), median-aged 42 years with lymph node, lacrimal/salivary gland and kidney involvements in 45, 24 and 12%, respectively. Surgical excision had 100% complete response and 60% relapse. Oral corticosteroids had 100% response with 50% relapse. Thalidomide, cyclosporine or interferon-α had 100% response, but 100, 20 and 50% relapse, respectively. KD showed clinicopathological similarities with 27 published cases of cutaneous IgG4-RD. Conclusion: Surgery may be used in resectable KD cases, whereas cyclosporine or thalidomide may represent interesting alternatives to oral corticosteroids in other cases. KD shares features with cutaneous IgG4-RD.

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Section: Discussionmentioning

confidence: 99%

Retrospective Multicentric Study of 25 Kimura Disease Patients: Emphasis on Therapeutics and Shared Features with Cutaneous IgG4-Related Disease

Kottler

Barète

Quéreux³

et al. 2015

Dermatology

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“…Association between Kimura's disease and renal involvement remains unclear. Previous studies suggest that the predominance of Th2 and Tc1 cells and certain cytokines including IL-5 as an eosinophil-active cytokine and IL-10 may contributes to the pathogenesis of Kimura's disease, but further research is warranted [13][14][15].…”

Section: Discussionmentioning

confidence: 99%

Eosinophilic peritonitis and nephrotic syndrome in Kimura’s disease: a case report and literature review

Yang

Song

et al. 2020

BMC Nephrol

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Background: Eosinophilic peritonitis is a relatively rare entity. Kimura's disease is a rare chronic inflammatory disorder of unknown etiology, characterized by subcutaneous nodules mainly in the head and neck region, regional lymphadenopathy and occasional involvement of kidney. There is currently no report of eosinophilic peritonitis in Kimura's disease. Case presentation: A 44-year-old Chinese man presented with abdominal distention, nausea, vomiting and edema in lower limbs for 1 month. Laboratory data showed elevated eosinophils in peripheral blood and ascites, nephrotic syndrome with progressively renal dysfunction, and elevated IgE. Ultrasonography of lymph nodes showed multiple lymphadenopathy in bilateral inguinal regions. Surgical excision was performed for one of the enlarged lymph nodes and histopathology revealed diagnosis of Kimura's disease. Renal biopsy indicated focal segmental glomerulosclerosis (FSGS) and acute tubulointerstitial nephritis with infiltration of eosinophils in renal interstitium. The patient was prescribed with oral prednisolone therapy (30 mg/day), and underwent continuous ambulatory peritoneal dialysis (CAPD). The peripheral and peritoneal eosinophil count decreased rapidly and normalized within 2 days. Forty-five days after prednisolone therapy, partial remission of nephrotic syndrome and decrease of serum creatinine were achieved while peritoneal dialysis dosage had decreased. Inguinal lymph nodes gradually shrunk in size. The overall conditions remain stable afterwards. Conclusions: This rare case highlighted the clinical conundrum of a patient presenting with eosinophilic peritonitis, lymphadenopathy, nephrotic syndrome and renal failure associated with Kimura's disease. The remarkable eosinophilia, pathology of lymph node and kidney, as well as significant response to steroids should guide towards the diagnosis.

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“…12 Additional studies have shown elevated granulocyte-macrophage stimulating-factor (GM-CSF), tumor necrosis factor-α (TNF-α), soluble interleukin (IL)-2 receptor (sIL-2R), IL-4, IL-5, IL-10, and IL-13. 13,14 Another study indicated that the activation of the IL-21/pERK1/2 pathway is a component of Kimura disease immunopathogenesis and pERK1/2 could be a potential prognostic indicator of the disease. 15 These findings may help lay the groundwork for elucidating the underlying pathophysiology of Kimura disease.…”

Section: Discussionmentioning

confidence: 99%

A rare association of Kimura’s disease with chronic pulmonary aspergillosis

et al. 2018

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Kimura’s disease is a rare chronic benign inflammatory condition characterised by nodules of skin and soft tissue and lymphnodes. Eosinophilic infiltration is a prominent finding. It has been reported in association with various conditions like nephrotic syndrome, bronchial asthma, ulcerative colitis and aortitis syndrome. Hereby we present a case in medical literature to the best of our knowledge in Kimura’s disease in association with chronic cavitary pulmonary aspergillosis, which has never been reported. He was treated with antifungals, after which he responded clinically, eosinophilia has subsided and he is under regular follow up. Now This case report suggest possible role of chronic aspergillosis as a cause for Kimura’s disease.

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Elevated Numbers of Cells Producing Interleukin-5 and Interleukin-10 in a Boy with Kimura Disease

Cited by 14 publications

References 49 publications

Retrospective Multicentric Study of 25 Kimura Disease Patients: Emphasis on Therapeutics and Shared Features with Cutaneous IgG4-Related Disease

Retrospective Multicentric Study of 25 Kimura Disease Patients: Emphasis on Therapeutics and Shared Features with Cutaneous IgG4-Related Disease

Eosinophilic peritonitis and nephrotic syndrome in Kimura’s disease: a case report and literature review

A rare association of Kimura’s disease with chronic pulmonary aspergillosis

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