PubMed · 14606173
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Hong Chen, Lester D Thompson. 2003. Kimura disease.. https://pubmed.ncbi.nlm.nih.gov/14606173/
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We report a case of angiolymphoid hyperplasia with eosinophilia (epithelioid haemangioma) involving multiple deep mediastinal, abdominal and intramammary lymph nodes in a 52-year-old woman with weight loss and raised CA-125 levels. The unusual clinical presentation with raised CA-125 levels and its occurrence within multiple deep visceral lymph nodes has never been reported in literature.
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.
Angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon benign, idiopathic vasculoproliferative condition manifested by multiple or solitary subcutaneous nodules, usually in the head and neck region of young adults. It is very rare in the extremities especially in the hands. Although recurrence is common, surgical excision is still regarded as the treatment of choice. We report an 11-year-old girl with histologically confirmed ALHE on the dorsum of her left hand, which underwent complete spontaneous regression within 10 weeks. Angiolymphoid hyperplasia is a benign disease and it is important to recognise this condition so that early superficial lesions can be observed for 3-6 months pending spontaneous regression.