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�B�BB/(eB/((�*�*B������i����k%��*./�/0�/�*�4'�4�*�*��4��+�ee(eeeeeh/h/(eee�/eeee���������������������������������������������������������������������4eeeeeeeeen	|:Exophthalmia as a first manifestation of epitheliod hemangioma
Introduction�:
Angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon, benign vascular disease. The aetiology of ALHE is obscure. The most common lesions seen in this entity are dermal or subcutaneous nodules of the face, scalp, neck and particularly around the ears. Involvement of the ocular adnexa is rare and uncommon compared with lesions of other sites. Orbital involvement  is unusual and a rare clinical entity. We report a case of Orbital involvement of ALHE revealed by an exophthalmia.
Case report�:
A 25-year-old  man was referred to the ophthalmolgy department  with a one-month history of a slowly progressive exophthalmia of the left eye, fluctuating swelling of the eyelids, and diplopia. He had an unremarkable medical history and no history of trauma. 
Ophthalmologic examination confirmed a minimal exophthalmos of his left eye (Fig.1) with displace eye downward and slight limitation of the elevation. A poorly defined soft mass in the upper eyelid could be palped below the superior orbital rim with associated pain and inflammatory signs. 
Examination of anterior and posterior segment of both eyes revealed no abnormality.  Intraocular pressure was normal and best corrected visual acuity was 10/10 (Snellen shart) in both eyes.
There were no skin lesions on the body and no regional lymphadenopathy.
Computed tomography (CT) demonstrated soft-tissue mass occupying superior extraconal space of left orbit with homogenous contrast enhancement (Fig. 2). 
The patient was treated for an infectious orbital cellulitis with systemic antibiotics (Cefotaxime and Fosfomycin) for 14 days. Faced with the worsening of symptoms, orbitofrontal brain magnetic resonance imaging (MRI) was performed.
The MRI axial T1-weighted image postcontrast showed  radiological progression (exophthalmos grade 1)(Fig. 3). Coronal and sagital T1-weighted images postcontrast with fat suppression showed the posterior extension of the inflammatory mass and its close contact with the superior rectus muscle and the eyeball (Fig. 4,5).
For diagnosis purpose, a transpalpebral biopsy was performed and histopathological examination of biopsy specimen stained with hematoxylin-eosin showed a diffuse inflammatory infiltrate with lymphocytes, plasma cells and abundant eosinophils surrounded the endothelial cell proliferations. No mitoses or atypical cells were observed (Fig. 6). These histopathological findings were compatible with a diagnosis of ALHE of the orbit or epithelioid hemangioma.
Laboratory investigations were performed: blood account, urinalysis and whole body scan were normal.
Corticosteroids at dose of 1mg/Kg of wieght/day have been used with complete  resolution within one week of treatment (Fig. 7). Regular follow up was performed and no evidence of recurrence was observed.
Discussion :
Angiolymphoid hyperplasia with eosinophilia (ALHE) was first described by Wells and Whimster in1969 [1]. ALHE is somewhat more common in females, however, a male predominance has been noted in selected Asian studies. It presents most commonly in patients aged 20�50 years, with a mean onset of 30�33 years�[2]. Clinically, ALHE presents as single or multiple pink to reddish-brown papules or subcutaneous nodules that are usually located on the head and neck, and especially in the preauricular region [3]. 
Rarely, these lesions have also been reported to develop in extracutaneous sites, including the colon, orbit, bone, parapharyngeal space, lip [4] and tongue�[5]. ALHE is uncommon in the periocular area. ALHE has been described rarely in the orbit, eyelids, lacrimal gland [6] and  conjunctiva [7]. Orbital involvement is an unusual clinical entity [8]. Although it is usually superficial in nature, some authors have reported muscular and bony involvement [8].�Features of orbital involvement may include proptosis, ocular dysmotility, watering, pruritus around the eyes and blurring of peripheral vision [6]. To our knowledge, it is the first case report of exophtalmia as a first manifestation of ALHE.
 ALHE is a relatively rare and uncommon benign vascular disease of uncertain pathogenesis. Many theories exist to explain the aetiology of ALHE. Inflammatory processes such as allergic reactions, traumas, infection or autoimmune disorders are identified as possible causes [9]. Histologically, it is a benign vascular lesion that's characterized by the proliferation of atypical endothelial cells in combination with an eosinophilic infiltrate and lymphoid aggregates. Lymphoid follicles showed proliferation of blood vessels lined with plump endothelial cells and vessels in the intervening stroma, with intense infiltration by eosinophils. [2].The density of both the lymphoid cells and eosinophils is highly variable [10]. 
The clinical characteristics and histopathology of epithelioid hemangioma allow differentiation with Kimura disease, angiosarcoma, intravascular papillary endothelial�hyperplasia, and other vascular tumors that share the common feature of an abnormal proliferation of endothelial cells. Although epithelioid hemangioma is extremely rare, it should be considered in the differential diagnosis of orbital tumors [11]. The main differential diagnosis is Kimura disease. ALHE and Kimura�s disease are known to have similar clinical, laboratory and histopathological findings.  Whether the two diseases are distinct or variations of the same disease have been discussed for many years. The main distinguishing feature between these two diseases is histopathological findings. Both diseases have an infiltrate of mostly lymphocytes and eosinophils, but ALHE is characterized by proliferating blood vessels lined by plump epithelioid endothelial cells, compared with the flat endothelial cells in Kimura�s disease. Moreover, elevations of blood eosinophils and serum immunoglobulin E in laboratory examinations characterize Kimura�s disease. Our case was diagnosed as ALHE instead of Kimura�s disease based on the differential features. In our patient, the absence of lymphadenopathy on physical examination and imaging CT thoraco-abdomino-pelvic and the normality of the biopsy of the salivary glands were in favor of AHLE. This diagnosis was supported by the absence of blood eosinophilia and normal IgE levels. Histologically, the diagnostic of HALE was retained by the absence of lymphoid follicles, of germinal centers and predominant fibrosis in the infiltration, two elements characterizing Kimura's disease.
The most effective treatment seems complete surgical excision but recurrence rate after this procedure is approximatively 33% [12].  In fact, the lesions are often multilobulated, poorly delineated and often impossible to remove completely. For our patient, therapy based on corticosteroids was effective through a long-term monitoring. Systemic corticosteroids are often effective but with a high frequency of recurrence, estimated at 33% [13-15]. Other treatment options like radiotherapy, cryotherapy, corticosteroids (topical, systemic, intralesional administration), laser therapy, interferon alpha-2a, Methotrexate, and chemtherpaeutic agents such as vinblastine, mepolizumab and imiquimod have been used with success [16-20]. 
Conclusion
Epithelioid hemangioma is a rare benign vascular disease that can be revealed exceptionnaly by an exophtalmia. The main differential diagnoses are Kimura's disease and orbital tumors. Despite the multiple treatments proposed for the mangement of this disease, recurrences are frequent and regular follow-up is required. 

Consent
Written informed consent was obtained from the patient for publication of this Case report and any accompanying images.
































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[20] Redondo P, Del Olmo J, Idoate M (2004). Angiolymphoid hyperplasia with eosinophilia successfully treated with imiquimod. Br J Dermatol 151:1110�1.
























































































                                 























































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