Correspondence: Nicholas Stewart, BSc., MBBS, Dermatology Registrar, Concord Hospital, PO Box 316, Rozelle, Sydney, NSW 2039, Australia. Email: firstname.lastname@example.org
Angiolymphoid hyperplasia with eosinophilia (ALHE), also known as epithelioid hemangioma, is a benign vascular proliferation of uncertain etiology. Herein, we present the first documented case of ALHE following venipuncture and review the published work regarding the relationship of this entity to preceding trauma.
The presumed pathogenesis, associations and treatment options of angiolymphoid hyperplasia with eosinophilia (ALHE) are highlighted in the context of the following case report. The pre-existing notion that immunisation may occasionally result in ALHE as a reactive phenomenon is also explored and challenged.
An 87-year-old woman presented with five flesh-colored, subcutaneous nodules measuring up to 10 mm in diameter on the volar surfaces of both wrists and in both cubital fossae (Fig. 1). The nodules appeared within 1 month of discharge from hospital following an admission for the investigation of anemia. Each lesion was situated over a superficial vein and corresponded to a site of recent venipuncture or cannulation.
The patient's medical history included: osteoporosis (oral biphosphonates), hypertension (beta-blocker) and chronic endogenous eczema (prednisolone [5 mg daily], topical steroids and moisturizers).
Two lesions were excised and submitted for histopathology. Sections demonstrated a marked nodular dermal inflammatory infiltrate composed of lymphocytes and moderate numbers of eosinophils, surrounding numerous vascular channels lined by epithelioid endothelial cells (Fig. 2). These changes were separated from the overlying normal epidermis by a Grenz zone. No arteriovenous malformation was identified and no large caliber vessels were seen on the sections examined. The lymphoid cells were composed of a population of interstitial CD3 positive T cells and occasional aggregates of CD20-positive B cells. The epithelioid endothelial cells stained positive for both CD31 and CD34 (See Fig. 3).
A diagnosis of ALHE secondary to venipuncture trauma was made. The remaining nodules were treated with intralesional triamcinolone acetonide (10 mg/mL) and resolved completely after one injection (Fig. 4). There has been no recurrence after 12 months of follow up. To the authors' knowledge, this represents the first reported case of ALHE occurring post-venipuncture.
Angiolymphoid hyperplasia with eosinophilia is a benign vascular proliferation presenting as tan, brown or dull-red papules and nodules commonly involving the periauricular area and scalp, although many other cutaneous sites[1-6] and involvement of other organs (e.g. parotid, bone, colon)[4, 7, 8] have also been reported. Typical cutaneous lesions vary from asymptomatic to pruritic, painful and/or pulsatile [1, 4, 7] The clinical differential diagnosis includes lymphoid hyperplasia, lymphoma cutis, sarcoidosis, cutaneous metastases and an insect bite reaction.
Angiolymphoid hyperplasia with eosinophilia was first described in 1969 and originally thought to represent late-stage Kimura's disease, but this is now widely regarded as a separate entity.[4, 7, 10] The etiology is idiopathic, however, trauma, pregnancy and immunizations have been implicated in a minority of cases.[1, 4] The pathogenesis favors a reactive over a neoplastic process, with an arteriovenous malformation postulated to represent the earliest abnormality in lesion development.[1, 11]
The largest study of ALHE to date consists of a retrospective cohort of 116 histologically confirmed cases. Of these, 10 cases (9%) were associated with antecedent trauma (frostbite, surgery, laceration, frictional trauma, blunt trauma and other unspecified). The time course between injury and the onset of lesions ranged from 7 months to 20 years (median, 30 months). Similar results were observed in another retrospective cohort consisting of 96 cases, which documented preceding trauma in a total of 12 (12.5%). Unfortunately, further information regarding the nature of the inciting trauma and the time course over which lesions developed was not included. Anecdotal case reports have documented ALHE development following a welding burn, blunt trauma to the scalp, scabetic infestation and otitis externa.
The evidence to support a role of immunization in the etiology of ALHE is less clear. The documented association appears to have arisen from a case series which detailed the development of histologically typical lesions at sites of recent immunization in five young children. The authors concluded that ALHE may have arisen as a result of an allergic/reactive phenomenon secondary to the constituents of the immunization. However, several different vaccines were implicated across these cases, making allergy to a single agent less likely. In addition, the evidence regarding the importance of arteriovenous malformation development in the early pathogenesis[2, 11] suggests that penetrating trauma may have been a more important etiological factor. This would certainly be consistent with the assumed mechanism of lesion development in the present case.
Angiolymphoid hyperplasia with eosinophilia spontaneously regresses in the majority of cases after several months, however, topical and intralesional corticosteroids, imiquimod, pentoxifylline, vinblastine, systemic retinoids, interferon, surgery (including Mohs micrographic surgery), embolization, radiotherapy and laser treatment (CO2, neodymium:yttrium–aluminum–garnet, pulsed dye laser) have all been reported as therapeutic options.[4, 7, 8] The lesions in this case cleared after a single treatment with intralesional triamcinolone acetonide (10 mg/mL), exceeding the documented efficacy of this treatment from a recent case report. However, ongoing follow up to monitor for recurrence is continuing.
Drs Stewart, Zagarella and Mann had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis. Study concept and design: Stewart, Zagarella, Mann. Acquisition of data: Stewart, Zagarella, Mann. Analysis and interpretation of data: Stewart, Zagarella, Mann. Drafting of the manuscript: Stewart, Zagarella, Mann. Critical revision of the manuscript for important intellectual content: Stewart, Zagarella, Mann. Statistical analysis: Stewart, Zagarella, Mann. Obtained funding: Stewart, Zagarella, Mann. Administrative, technical or material support: Stewart, Zagarella, Mann. Study supervision: Stewart, Zagarella, Mann.