Kimura's Disease Associated with Bronchial Asthma Presenting Eosinophilia and Hyperimmunoglobulinemia E which were Attenuated by Suplatast Tosilate (IPD-1151T).

Abstract

A 29-year-old man developed atopic bronchial asthma in association with eosinophilia and hyperimmunoglobulinemia E (hyper-IgE). A biopsy specimen from an inguinal lymph node showed changes consistent with Kimura's disease. IPD-1151T (suplatast tosilate), an anti-allergy drug, attenuated eosinophilia and hyper-IgE as well as the serum level ofeosinophil cationic protein (ECP). The drug, however, did not affect the positivity for specific IgE antibodies against common allergens or the bronchial hyperresponsiveness to acetylcholine. Interleukin (IL)-2, IL-4, IL-5, interferon (IFN)-y, and tumor necrosis factor (TNF)-oc were measured to be undetectable in serum before or during therapy. However, the expressions of mRNAsfor IL-2, IL-4, IL-5, IFN-y, and TNF-a in peripheral blood T-lymphocytes and the expression of IL-5 mRNA in peripheral blood eosinophils were detected before and during therapy, which were unchanged by therapy with IPD-1151T. The present results suggest that different mechanisms other than the predominance of type 2 helper (TH2)-like T-lymphocytes may underlie Kimura's disease and atopic bronchial asthma regarding the findings of eosinophilia and hyper-IgE, which could be modulated by IPD-1151T.

Kimura's Disease with Generalized LymphadenopathyDemonstrated by Positron Emission Tomography Scan

“…First, the association of peripheral eosinophilia and nephrotic syndrome (12,13) has been frequently discussed in Kimura's disease. Second, many different regional lymph nodes (14) or extremities (15) involvement at presentation have been reported. Third, the rate of recurrence may be as high as 257 5% after surgical excision (11,16) and 11~17% after local radiotherapy (11,17 …”

Eosinophilic Lung Disease Complicated by Kimura's Disease: A Case Report and Literature Review

“…A high prevalence of nephropathy has been reported in patients with KD (8)(9)(10), and nephrotic syndrome is seen in the majority of reported cases, with most patients displaying mild histological changes on renal biopsies and many responding well to steroid therapy. The histological findings are mild to moderate, with membranous nephropathy, mini- There are some case reports of bronchial asthma occurring in association with KD (11,12), such as those described by Tsukagoshi et al (11). In their report, suplatast tosilate reduced the number of peripheral blood eosinophils and the serum levels of IgE and eosinophilic cationic proteins (ECPs), thereby improving asthma symptoms.…”

“…Complications such as nephrotic syndrome (8-10), bronchial asthma (11,12), atopic dermatitis, urticaria, ulcerative colitis (13) and aortitis syndrome (14) have been reported. In a review of 429 Japanese cases of KD, Ishii identified five cases associated with bronchial asthma, three cases associated with allergic rhinitis, two cases associated with atopic dermatitis and five cases associated with urticaria, suggesting that an allergic mechanism may contribute to KD (2).…”

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

“…Surgical excision, systemic corticosteroids and local radiation are often performed [7]. Treatments with immunosuppressive agents [2,23], cytotoxic therapy [24], antiallergic drugs [5,25] and all-trans-retinoic acid [26] have also been reported. Surgical resection and radiotherapy were not indicated for our case because there were multiple tumors.…”