17. Angiolymphoid hyperplasia with eosinophilia mimicking temporal arteritis

INTRODUCTION: A 32-year-old man presented to rheumatology clinic with a 2-year history of progressively worsening right temporal headache, a painful hard lump on his right temple and pre-auricular tenderness not responding to conventional analgesics. Patient denied any jaw and tongue claudication. His vision was not compromised and patient denied weight loss, night sweats and arthralgia. He was fit and well otherwise. CASE DESCRIPTION: Examination revealed a pulsatile mass with a length of 1 cm along the superficial temporal artery. Temporal artery ultrasound revealed lump corresponds to the superficial artery with thickened wall and halo formation raising the possibility of temporal arteritis. MRI head scan suggested inflammation of the superficial temporal artery. DISCUSSION: Because of the atypical features of young age and normal inflammatory markers, corticosteroid treatment was not commenced. A temporal artery biopsy showed no evidence of vasculitis, but revealed reactive lymphoid follicles with eosinophil infiltrates in the surrounding soft tissue. The diagnosis of angiolymphoid hyperplasia with eosinophilia (ALHE) was made. The patient’s headaches have completely resolved after the temporal biopsy. Unfortunately, three months later the lump and the headaches recurred. The symptoms improved with topical tacrolimus 0.1% ointment and he underwent for complete surgical excision of the lump. Patient is asymptomatic and remained well. KEY LEARNING POINTS: We present a case of angiolymphoid hyperplasia with eosinophilia which can mimic temporal arteritis presentation as in our case and has totally different approach to management. ALHE, which is a benign, locally proliferative condition with predilection to the periauricular and scalp area with no definitive treatment however, complete excision can be curative. It may recur in up to one-third of cases in the absence of complete surgical excision as in our patient. CONFLICT OF INTEREST: The authors declare no conflicts of interest.