Kimura's Disease Involving the Ipsilateral Face and Extraocular Muscles

Kimura's disease (KD) is a rare, chronic inflammatory disorder, which is characterized by tumor-like masses mainly located in the head and neck region. Extraocular muscle involvement in KD is uncommon. We report a case of KD that involved both the extraocular muscles and buccal area. A 13-year-old male presented to our clinic with a two-year history of exophthalmos of the left eye and facial swelling. Facial CT and MRI showed a 1.5 x 1.5 cm2 soft tissue mass located at the left masticator and buccal area, exophthalmos of the left eye, and diffuse thickening of the left extraocular muscles. We performed a lateral rectus muscle incisional biopsy of the left eye. Oral methylprednisolone therapy was initiated and tapered following the incisional biopsy.

Kimura disease with proliferative squamous metaplasia: an unusual finding and a potential diagnostic pitfall.

Kimura disease is a rare form of chronic inflammatory disorder involving subcutaneous tissue, predominantly in the head and neck region and frequently associated with regional lymphadenopathy and/or salivary gland involvement. We present a case of Kimura disease in a 28-year-old male which showed florid squamous metaplasia in the salivary gland ducts and salivary duct inclusions in the intraparotid nodes besides the usual features of Kimura disease. The squamous metaplasia was extensive enough to pose a diagnostic dilemma. We describe this case to highlight the rare histological finding of florid squamous metaplasia in Kimura disease and its diagnostic implications.

Angiolymphoid hyperplasia with follicular mucinosis.

Follicular mucinosis occurring along with angiolymphoid hyperplasia with eosinophils (ALHE) has been described in a 54-year-old female. The patient presented with pruritic erythematous papules on the left frontoparietal scalp. Histopathological examination showed prominent blood vessels in the dermis lined by plump histiocytoid endothelial cells that were surrounded by a dense lymphoid infiltrate with numerous eosinophils; these findings are typical of angiolymphoid hyperplasia with eosinophils. Features of follicular mucinosis were observed in the same section with several hyperplastic follicular infundibula containing pools of mucin in the infundibular epithelium. The concurrent occurrence of these two distinct histopathological patterns in the same biopsy specimen has been described in only three cases to date.