Department of Ophthalmology, Pusan National University College of Medicine, Busan, South Korea.
Biomedical Research Institute, Pusan National University Hospital, Busan, South Korea.
Am J Case Rep. 2024 Jan 29;25:e942753. doi: 10.12659/AJCR.942753.
BACKGROUND Kimura disease is a rare, chronic inflammatory disorder typically presenting as a painless mass in the head or neck and associated with elevated serum immunoglobulin E and blood and tissue eosinophilia. Generally benign, its management is not well-defined, but corticosteroids are a common initial treatment. We detail a case of refractory Kimura disease successfully managed with CVP (Cyclophosphamide, Vincristine, Prednisone) chemotherapy and no recurrence during 6 rounds of treatment. CASE REPORT A 64-year-old woman, previously diagnosed with Kimura disease, returned to the hospital with upper eyelid ptosis. Upon examination, a solid mass was palpable in her left upper eyelid. Peripheral blood tests confirmed elevated IgE levels at 356.0 IU/ml. An excisional biopsy showed infiltration of lymphocytes and eosinophils, consistent with Kimura disease. Despite undergoing corticosteroid treatment, surgical debulking, radiation, and immunosuppressant therapy, her condition worsened. Concerns were raised due to imaging features suggestive of lymphoma, although no malignancy was evident in subsequent biopsies. It was decided to manage the disease using CVP chemotherapy, leading to significant symptom improvement. There have been no recurrences during the 12-month follow-up period. CONCLUSIONS Kimura disease is typically benign and responsive to treatment, but it often recurs and can progress. When symptoms are not controlled with conventional treatments, including corticosteroids, immunosuppressants, radiation, and surgical debulking, chemotherapy may be a reasonable option even when no definite signs of malignancy is identified. Further research is needed to explore the utility of CHOP and CVP in managing uncontrolled Kimura disease.
木村病是一种罕见的慢性炎症性疾病,通常表现为头部或颈部无痛性肿块,并伴有血清免疫球蛋白 E 升高以及血液和组织嗜酸性粒细胞增多。该病通常为良性,但治疗方法尚未明确,皮质类固醇是常见的初始治疗方法。我们详细介绍了一例难治性木村病患者,经 CVP(环磷酰胺、长春新碱、泼尼松)化疗成功治疗,6 个疗程后无复发。
一名 64 岁女性,曾被诊断为木村病,因上眼睑下垂返回医院。检查时,可触及其左眼上眼睑有一实性肿块。外周血检查证实 IgE 水平升高至 356.0 IU/ml。切除活检显示淋巴细胞和嗜酸性粒细胞浸润,符合木村病。尽管接受了皮质类固醇治疗、手术去瘤、放疗和免疫抑制剂治疗,但病情仍恶化。由于影像学特征提示淋巴瘤,引起了关注,尽管随后的活检未发现恶性肿瘤。决定使用 CVP 化疗来治疗这种疾病,导致症状显著改善。在 12 个月的随访期间,没有复发。
木村病通常为良性且对治疗有反应,但常复发且可能进展。当常规治疗(包括皮质类固醇、免疫抑制剂、放疗和手术去瘤)无法控制症状时,即使没有明确的恶性肿瘤迹象,化疗也可能是一种合理的选择。需要进一步研究来探讨 CHOP 和 CVP 在治疗未控制的木村病中的应用。