Angiolymphoid hyperplasia with eosinophilia presenting as an axillary artery aneurysm

Abstract

BACKGROUNDThis is a rare cause of an axillary artery aneurysm in a young patient and brings together dermatology, rheumatology and vascular surgery. CASE PRESENTATIONA man in his late 20s presented with a painless swelling in his left axilla. He also had a long history of nocturnal sweating with no history of weight loss or fatigue. On examination, there was a large, well circumscribed lump measuring 3 × 2.5 cm within the apex of the left axilla. The mass was pulsatile, fi rm and non-tender. General examination was otherwise unremarkable; in particular, there were neither skin lesions nor palpable lymphadenopathy. INVESTIGATIONSBiochemical and haematological investigations were normal apart from a mildly raised eosinophil count of 0.5 × 10 9 /l (upper limit of normal 0.4 × 10 9 /l).Doppler ultrasound confi rmed a 2.9 cm long × 1.8 cm wide aneurysm of the distal axillary artery with evidence of an irregular thrombus present within. DIFFERENTIAL DIAGNOSISAngiolymphoid hyperplasia with eosinophilia (ALHE) has been known by a variety of different names, such as epithelioid haemangioma, pseudopyogenic granuloma, infl ammatory angiomatous nodule, papular angioplasia, subcutaneous angioblastic lymphoid hyperplasia with eosinophilia and lymphofolliculosis, intravenous atypical vascular proliferation and histiocytoid haemangioma. 1 This extensive list of nomenclature refl ects the spectral variation in microscopic presentations of ALHE encountered by past investigators and, not least, the divided opinion as to whether the pathogenesis of the lesion is truly neoplastic or a reactive phenomenon.There has been controversy over the exact relationship between Kimura's disease (KD) and ALHE, where the two terms have been used interchangeably in many articles. KD was fi rst described by Chinese authors Kimm and Szeto 2 in 1937 and later made widely recognised by Kimura in 1947. KD is a chronic infl ammatory disease of unknown aetiology usually presenting as solitary or multiple subcutaneous nodules in the head and neck region, often involving the parotid or submandibular salivary glands. KD is associated with regional lymphadenopathy; this may become generalised in longstanding disease, 3 and systemic eosinophilia with raised IgE levels. It is most prevalent in Asians with 85% of cases occurring in males.Although KD has been thought to be integral to the spectrum of ALHE in the past, histological features show that these conditions represent two separate disease entities. 4 KD is characterised by lymphoid nodules with germinal centres which may extend from the dermis to the underlying fascia and muscles. Lesions show a distinct eosinophilic infi ltrate with microabscesses. Vascular proliferation is not always present; however, when seen, there are many canalised capillaries lined by fl at endothelial cells. Systemic eosinophilia is almost always present, seen in approximately 98% of cases in comparison to 20% in ALHE. 5 In contrast to KD, ALHE lesions are superfi cial containing blood vessels of varying luminal sizes, some of w...

Epithelioid hemangioma of brachial artery: report of a case and review of the literature

“…About 22 cases have been reported, involving in order of decreasing frequency: temporal artery [14–20], radial artery [21–24], brachial artery [11, 12], ulnar artery [25, 26], axillary artery [27, 28], subclavian artery [29], facial artery [30], post-auricular artery [31], popliteal artery [32], common carotid artery [33] and occipital artery [12]. The lesion can grow entirely in the lumen of the blood vessel developing occlusive symptoms (such as weakening of the pulse in peripheral arteries) or it can originate from the vascular wall and developing outside with compression of adjacent structures.…”

Angiolymphoid hyperplasia with eosinophilia as simultaneous cutaneous and vascular lesions with radial artery aneurysm

“…The latter appeared as a vascular tumor with aneurysmal dilatation of the radial artery. 1,2 To our knowledge, this is the first described case of simultaneous presentation of skin and vascular lesions in extremities, extending the spectrum of clinical presentation of this rare disease.…”

“…Moreover, previous articles only reported solitary lesions in this area. 1,[5][6][7] Finally, few cases reported an associated aneurysmal dilatation.…”

“…It is known by different names, even sometimes confused with Kimura's disease (KD). [1][2][3] The clinical presentation is variable, usually single lesions located in the head or the neck, and less frequently in the trunk, groin or limbs for which it has always been described as a single injury. It often affects skin and subcutaneous tissue, but vascular lesions have been described.…”

Angiolymphoid hyperplasia with eosinophilia involving the common digital artery of the hand: A case report and classification of upper limb lesions

“…Involvement of the upper limb is extremely rare with only 24 previously reported cases in the world literature [1] , [2] , [3] , [4] , [5] , [6] , [7] , [8] , [9] , [10] , [11] , [12] , [13] , [14] , [15] , [16] , [17] , [18] , [19] , [20] , [21] , [22] , [23] , [24] . In the upper limb, various sites for involvement have been reported such as the skin, the subungual region, the blood vessels, nerves, muscles, and bone [1] , [2] , [3] , [4] , [5] , [6] , [7] , [8] , [9] , [10] , [11] , [12] , [13] , [14] , [15] , [16] , [17] , [18] , [19] , [20] , [21] , [22] , [23] , [24] . As the name implies, the diagnosis is made histologically by the presence of vascular proliferation (angio-hyperplasia), lymphoid follicles (lymphoid hyperplasia) and a prominent eosinophilic infiltrate (eosinophilia).…”