Year: 2009
Type: article
Abstract: Editor, A 44-year-old Japanese woman presented with a 1-year history of a painless, subcutaneous mass in the left medial canthal region; there was no redness or itching sensation in the area. The tumour showed a repetitive cycle of enlargement and diminishment. The patient had no history of systemic disease, ocular trauma or eyelid surgery. Macroscopic examination revealed the presence of an elastically hard, smooth, non-mobile, oval-shaped, subcutaneous tumour measuring 25 × 10 mm in the left medial canthus (Fig. 1A). A syringe test demonstrated that there was no obstruction in the lacrimal passage. Physical examination revealed lymphadenopathy in the neck. Computed tomography showed a homogeneous mass with indistinct demarcation located above the left lacrimal sac (Fig. 1B). Blood samples showed eosinophilia (7.9%; normal range 0.3–6.0%) and a mild elevation in the serum immunoglobulin (Ig) E level (335.4 U/ml; normal range < 269.1 U/ml). (A) Facial photography shows a smooth, oval-shaped, non-reddish, subcutaneous tumour measuring 25 × 10 mm in the left medial canthal region (arrow). (B) Computed tomography shows a homogeneous mass (arrow) adjoining the left medial orbital rim. No bone erosion is visible. The demarcation between the mass and the left lacrimal sac is indistinct. We performed tumour excision. Histopathological examination of the excised tissue revealed prominent fibrosis and inflammation consisting of abundant lymphocytes and eosinophils (Fig. 2A, B). Numerous lymphoid follicles with germinal centres and eosinophilic abscesses were also present. A proliferation of capillary vessels, composed of endothelial cells without nuclear atypia or abundant cytoplasm, was noted. The shape of the endothelial cells was flat. The resection margins were not tumour-free and the skin layer of the lesion was not found. A syringe test showed normal passage and the patient did not develop epiphora. No additional therapy was administered. The tumour had not recurred at 6 months after the operation. Pathological findings. (A) Prominent fibrosis and infiltration of inflammatory cells throughout the lesion. Lymphoid follicles are observed to have prominent germinal centres (arrows). (Haematoxylin and eosin; original magnification × 40.) (B) Lymphoid follicle showing eosinophilic infiltration with fibrosis and capillary proliferation. Capillary vessels are observed to be lined with normal endothelial cells (arrows). (Haematoxylin and eosin; original magnification × 400.) Kimura’s disease, characterized by the presence of single or multiple subcutaneous nodules, generally occurs in the head and neck regions and rarely in the periocular regions (Buggage et al. 1999). Although only three cases involving the medial canthus have been described, two of them were determined to represent cases of angiolymphoid hyperplasia with eosinophilia (ALHE) (Hidayat et al. 1983; Mariatos et al. 2001), which had previously been considered to represent the same condition as Kimura’s disease, but which is now believed to differ both clinically and histopathologically (Buggage et al. 1999; Seregard 2001). To the best of our knowledge, only one case of Kimura’s disease in the medial canthus has been reported to date (Sahu et al. 2007); thus, this is an extremely rare entity. When Kimura’s disease occurs in the medial canthus, its manifestation may mimic various features of lacrimal sac diseases. In the ALHE case mentioned above, the tumour involved the lacrimal passage and caused its obstruction (Mariatos et al. 2001). Because Kimura’s disease extends to deeper tissues more easily than ALHE (Buggage et al. 1999), it is likely to extend to the lacrimal pathway. Furthermore, it is difficult to differentiate Kimura’s disease from diseases of the lacrimal sac solely by the findings of imaging studies because lesions in the former are not distinctly demarcated (Seregard 2001). As the medial canthus is an extremely rare site for Kimura’s disease, this condition has not previously been included in the differential diagnosis of lacrimal sac tumours and dacryocystitis. However, efforts should be made to understand this rare condition because adequate awareness will benefit its pre- and intraoperative management.
Source: Acta Ophthalmologica
Institutions: Seirei Hamamatsu General Hospital, Osaka City University
Language: English
Subfield: Oncology
Field: Medicine
Domain: Health Sciences
Open Access status: bronze
OpenAlex ID: W2014533465