ALHE is a nodular skin tumor associated with eosinophilia and vascular proliferation. Most cases occur on the auricle, forehead, and scalp, whereas rare cases, such as those involving the hand, account for only 4% of all ALHEs [
2]. Table
2 describes the documented cases of ALHE involving hands. ALHE is difficult to cure [
3]; the gold standard for treatment of ALHE is complete surgical resection. Due to the possibility of recurrence in some cases, complete resection of the tumor is necessary. Furthermore, functional reconstruction in conjunction with complete resection is essential for the treatment of ALHEs of the hand.
Table 2
Reported instances of angiolymphoid hyperplasia with eosinophilia in the hand
James M. Swinehart et al., 1979 [ 20] | 24 | M | Lt.palm, subcutaneous | single | NA | NA | resection | local reccurence | + | 8 months |
Arnold M et al., 1999 [ 21] | 20 | F | left arm and hand subcutaneous | multiple | 5-10 mm | bleeding, pain | resection | no local reccurence, but another site | + | 24 months |
B. D. Krapohl et al., 2003 [ 22] | 33 | F | Rt.palm and ring finger subcutaneous | multiple | NA | discoloration | resection | no recurrence in 3 months | NA | 18 months |
C Conill et al., 2004 [ 23] | 32 | F | Rt.index and middle fingers subcutaneous and bone | multiple | NA | deformation of the nail, pain | radiation therapy | no recurrence in 9 years | – | NA |
| 11 | F | Rt.dorsum of hand subcutaneous | single | 20 mm | itching | spontaneous resolution | no recurrence in 12 months | – | a month |
H Ozcanli et al., 2007 [ 25] | 42 | F | Rt.palm, middle and ring fingers subcutaneous | multiple | 30-80 mm | itching, pain, disturbance of sensation | resection, laser treatment | no recurrence in 2 years | + | 12 months |
Nick Pappas et al., 2010 [ 26] | 18 | F | Rt.palm subcutaneous | single | 15-10 mm | no pain | resection | no recurrence in 1 year | – | a month |
Mohammad M. Al-qattan et al., 2017 [ 27] | 32 | F | Lt.palm subcutaneous | single | NA | NA | resection | no recurrence in 1 year | + | 6 months |
On clinical examination, it is important to differentiate between ALHE and Kimura’s disease, using both laboratory and histological tests. Similar to AHLE, Kimura’s disease is classified as a type of eosinophilic dermatitis. Although eosinophilia was observed in the blood of the patient in the current report, systemic eosinophilia is generally rare. In addition, ALHE exhibits a slow growth rate and rarely presents with lymphadenopathy (5–20% of cases) [
4]. Moreover, unlike in Kimura’s disease, the itching sensation is strong in AHLE. In many cases, immunoglobulin levels, such as that of immunoglobulin E, are normal. ALHE often involves neoangiogenesis and, accordingly, presents with plump, epithelioid endothelial cells and arteriovenous shunts [
4]. Often, ALHE lesions are located in the dermis. Kimura’s disease is probably an allergic or autoimmune response, whereas ALHE is the result of a benign neoplasm of endothelial cells caused by inflammation or stimulation [
5]. The cause of ALHE is unknown [
6]. Some clinicians speculate that it develops in response to various infections, such as herpes virus 8 or human papillomavirus-6, or injury, such as excessive contact with the rim of eye glasses [
7,
8]. Meanwhile, tumor growth is reported to occur in response to changes in hormone levels during pregnancy; however, there are no clear differences between men and women [
9‐
12]. In the present case report, our patient had numerous skin injuries on her fingers other than that affected by the lesion. She reported that she frequently fed wild cats near her home and had received several scratches from the animals in the past, which may be related to the onset of ALHE in this case.
Is it necessary to treat benign ALHE tumors?
ALHE does not develop complications during conservative therapy, and no cases of malignant transformation have been reported so far [
5]. Yet, ALHE can be exacerbated by surgical stress; hence, refraining from excision without careful consideration is critical. Some cases of ALHE also recognize genetic mutation in T cell. According to Kempf
et al., ALHE is not a vascular lesion, but a type of CD4-positive T cell lymphoma [
13‐
15]. Accordingly, careful observation is necessary, even in cases not involving surgical procedures. Early symptoms include itching and a throbbing sensation, but bleeding and pain are only observed as the tumor grows in size. Surgical resection may be indicated in the presence of more serious symptoms, such as pain, and if the patient wishes to undergo resection.
What is critical for success in surgical resection?
Pathologically, ALHE involves vascular damage characterized by the presence of an unstable basement membrane and arteriovenous shunts [
18,
19]. The removal of these abnormal vessels may be important during surgical resection. Furthermore, difficulties in identifying the margins of these lesions are believed to contribute to their high recurrence rate. Abnormal blood vessels related to ALHE often exist in the dermis; therefore, it is advisable to use natural anatomic barriers, such as the aponeurosis and fascia, as the resection margin. Several clinicians believe that ALHE resection should mirror the resection margin of malignant soft tissue tumors. It is important to examine the preoperative image before surgical treatment. In the present case study, both abnormal vessels and aponeurosis at the bottom of the tumor were excised. Recurrence is less likely when a wide resection with margins involving the dermis is performed. Reconstruction using a skin flap or artificial epithelium is recommended in cases where the deformity is large.