Korean J Ophthalmol.  2009 Sep;23(3):219-223. 10.3341/kjo.2009.23.3.219.

Kimura's Disease Involving the Ipsilateral Face and Extraocular Muscles

• Lee SJ
• Song JH
• Kim SD

• KMID: 1115758
• DOI: http://doi.org/10.3341/kjo.2009.23.3.219

Abstract

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.

Keyword

Exophthalmos; Extraocular muscle; Eyelid swelling; Kimura's disease

MeSH Terms

Administration, Oral
Adolescent
Angiolymphoid Hyperplasia with Eosinophilia/complications/*diagnosis/drug therapy/surgery
Cheek/*pathology
Edema/etiology
Exophthalmos/etiology
Face
Glucocorticoids/administration & dosage
Humans
Magnetic Resonance Imaging
Male
Methylprednisolone/administration & dosage
Oculomotor Muscles/*pathology/surgery
Postoperative Care
Treatment Outcome

Figure

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  Fig. 1 Photograph of patient's face prior to surgery showing exophthalmos of the left eye and swelling of the left upper eyelid and buccal area.

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  Fig. 2 T2-weighted MRI scan with enhancement showed exophthalmos of the left eye and diffuse thickening of the left extraocular muscles (A, B) and a relatively well-defined enhancing homogenous mass at the left masseter muscle, left temporalis muscle, and left medial pterygoid muscle (C).

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  Fig. 3 A&B, Histopathological examination of an incisional biopsy of the left lateral rectus muscle showed lymphoid cell aggregation and abnormal vascular proliferation with marked eosinophilic infiltration in hypertrophied collagenous tissue. (A: Hematoxylin & Eosin ×100, B: Hematoxylin & Eosin ×200).

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  Fig. 4 After two months of oral steroid therapy, the exophthalmos of the left eye and facial swelling dramatically decreased, as documented by this photograph of the patient's face following therapy.

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  Fig. 5 A&B, Histopathological examination of the excised facial mass showed follicular hyperplasia and abnormal vascular proliferation with massive mature eosionphilic infiltration in hypertrophied collagenous tissue. (A: Hematoxylin & Eosin ×40, B: Hematoxylin & Eosin ×200).

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