Angiolymphoid hyperplasia with eosinophilia and entrapment of the ulnar nerve

Abstract

Background:The angiolymphoid hyperplasia with eosinophilia (ALHE) is a sporadic vasoproliferative lesion of uncertain etiology involving the skin and the subcutaneous tissue. Occasionally, it involves also the large arteries compressing the near nerves. ALHE is commonly confused with Kimura's disease because of their clinical and histological similarities.Case Description:We report a case of a 52-year-old female suffering from a 6-month pain and paresthesias in the fourth and fifth finger of the right hand. The angiography showed a pseudoaneurysm in the proximal third of the right ulnar artery. A complete surgical excision of the vascular lesion was undertaken. The lesion forced the right ulnar nerve. The histopathological diagnosis deposed for ALHE.Conclusion:Up to now, literature has described 8 cases of ALHE involving the arteries, and only one case originating from the ulnar nerve. The authors report a case of a female with ALHE involving the ulnar artery that compressed the ulnar nerve. Clinical aspects, radiological features, surgical treatment, and operative findings are discussed reviewing the pertinent literature.

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

“…Our review of the literature revealed 24 well documented cases [1] , [2] , [3] , [4] , [5] , [6] , [7] , [8] , [9] , [10] , [11] , [12] , [13] , [14] , [15] , [16] , [17] , [18] , [19] , [20] , [21] , [22] , [23] , [24] . We did not include cases with poor documentation [27] .…”

“…The ALHE lesion in our patient involved the common digital artery and there were two other previously reported cases with common digital artery involvement [1] , [23] . Involvement of the radial artery [10] , [20] , ulnar artery [8] , [9] , [14] , brachial artery [2] , [11] , and axillary artery [12] , [13] have also been reported. Total excision of the involved artery is curative; and reconstruction with vein grafts is only mandatory after axillary and brachial artery excisions.…”

“…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).…”