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Angiolymphoid hyperplasia with eosinophilia is seldom diagnosed preoperatively. When excising what seems to be a sebaceous cyst, the surgeon can be caught out by unexpected haemorrhage.
An Asian man of 29 sought advice about an occipital lump that had been present for 5 years. It was occasionally itchy but painless. There were no other lumps and he gave no history of trauma to the site. On examination the lump was spherical (2 cm in diameter), soft and smooth; it was attached to the skin, which was hair-bearing; there was no regional lymphadenopathy. The preoperative clinical diagnosis was a sebaceous cyst.
The lump was excised with an ellipse of skin. Underlying the skin was a solid multilobated yellowish white mass measuring 1.0 × 1.5 × 1.3 cm. The surgical field was bloody, and about 100 mL of blood was lost before haemostasis was achieved. Microscopy showed a multi-nodular but ill-defined proliferation of vascular structures associated with lymphoid aggregates and numerous eosinophils (Figure 1). The features were consistent with angiolymphoid hyperplasia with eosinophilia. The patient was followed up for 2 years without evidence of recurrence.
In a patient with what seems to be a sebaceous cyst in the head and neck region, other diagnoses to be considered are metastases from tumours elsewhere, low-grade angiosarcoma, lymphocytoma, Kaposi’s sarcoma, angiofollicular hyperplasia and tuberculosis. Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign condition of unknown cause usually diagnosed in the third to fifth decades of life.1 The lesions may be single or multiple,2 and patients may report pruritus (as did ours) or haemorrhage.3 Clinically the condition resembles Kimura’s disease, though the lesions tend to be more superficial and lymph node involvement is less common.3 Microscopically they are clearly different: Kimura’s disease is characterized by the presence of numerous lymphoid follicles with distinct germinal centres and an abundance of dendritic cells. IgE has been identified in the germinal centres of Kimura’s disease but not ALHE.4
Local medical treatments have been used in ALHE but most patients have the lesion excised surgically. Recurrence is unusual. For surgeons who believe they are operating on a simple sebaceous cyst a hazard, as illustrated here, is that they will find themselves suddenly having to deal with a very bloody operating field.