A Rare Case of Angiolymphoid Hyperplasia With Eosinophilia With a New Effective Treatment.

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign locally proliferating lesion of unknown etiology, composed of vascular channels lined by endothelial cells, surrounded by lymphocytes and eosinophils. It presents clinically as a cluster of skin to violaceous-colored nodules on the head and neck, particularly in and around the ear. We present the case of a 50-year-old, Pakistani woman with unilateral multiple nodular lesions for eight years in the left ear concha and postauricular area causing complete obliteration of the external auditory meatus with conductive hearing loss of the left ear for seven years. Biopsy showed lymphoid follicles and dilated blood vessels with mixed infiltrate predominantly eosinophils corresponding to the diagnosis of angiolymphoid hyperplasia with eosinophilia. Surgical excision was not feasible, and there was no response to topical steroids. The patient was started on beta blockers. After three months, postauricular lesions completely resolved, and the size of the rest of the nodules decreased markedly; then hearing loss also recovered. Our objective in this study is to emphasize the importance of considering beta blockers for the treatment of ALHE.

A Rare Case Report of Periorbital Pseudolymphoma.

Pseudolymphoma (PSL) of the orbit is a benign lymphoid hyperplasia (LH). It is a rare disease with an extensive range of known causative agents. LH is further classified into "reactive" (RLH) and "atypical" (ALH) types. It clinically presents as a single or a few plaques and/or nodular lesions, particularly on the head, neck, and upper trunk. It must be differentiated from orbital malignant lymphoma. In this report, we present a case of a 58-year-old Pakistani female with an asymptomatic recurrent right periorbital swelling for three years. It was clinically diagnosed as an angiotensin-converting enzyme (ACE) inhibitor-induced angioedema as it responded to stopping the ACE inhibitor; however, after four months, the patient again started to develop right periorbital swelling. An incisional biopsy revealed perivascular and periadnexal infiltration of lymphocytes, plasma cells, and a few neutrophils along with pigmentary incontinence. The formation of multiple lymphoid follicles and infiltration by monomorphic lymphoid cells in deeper skeletal muscle fibers were also observed. Immunohistochemistry (IHC) showed polyclonality and low Ki-67 labeling (20%), corresponding to periorbital RLH. Our objective in this study is to highlight the importance of considering PSL as a differential diagnosis in periorbital swelling. We also suggest that recurrent angioedema may lead to PSL.