The cases of solitary fibrofolliculoma are extremely rare, since being first reported in 1984 . Only eight prior papers, involving 12 cases, have previously been published worldwide [3-10]. We reviewed the previously published cases and found that most lesions were found on the face, with only 2 around the eyes, one on the eyelid and the other on the eyebrow (Table 1). Among all 13 cases, including the present one, solitary fibrofolliculoma occurs more frequently in women (5 men and 8 women), with the mean age at presentation being 51 years. The duration of symptom varying from several months to many years. Solitary fibrofolliculomas share the clinical appearance of multiple fibrofolliculomas, yellowish to flesh-colored, dome-shaped papules.
Histologically, fibrofolliculoma has both an epithelial and a mesenchymal origin, showing distinctive and characteristic features with minor variation . The center of the lesion presents a hair follicle and consists of an expansion of the fibrous root sheath, which typically surrounds the hair follicle, along with proliferating bands or ribbons of perifollicular connective tissue. Cesinaro and coauthors found immunohistochemical expression of factor XIIIa in the bizarre perifollicular cells in a background of CD34-positive spindle cells, which aids for better characterize the nature of the lesion .
Since solitary fibrofolliculoma is extremely infrequent and definitively diagnosed only by histological results, it can be easily overlooked or clinically misdiagnosed. The 37-year female patient reported by Chang and coauthors was previously misdiagnosed as chalazion and received incision and curettage only. For years, her condition had not improved, recurring several times . Fortunately, fibrofolliculoma rarely develops to malignancy. Once this type of lesion in the eyelid is observed, a diagnosis of fibrofolliculoma should be considered. As the lesion of the presented case was located on the eyelid, it also should be included as a differential diagnosis from malignant conditions such as basal cell carcinoma and squamous cell carcinomas, which are the most common malignant eyelid tumors. Basal cell carcinomas appear as a translucent, waxy papule with a rolled, pearly border, and telangiectasia. As it enlarges, central ulceration usually develops. Squamous cell carcinomas appear as a painless, elevated, nodular, or plaque-like lesion with chronic scaling and fissuring of the skin. The characteristic features of squamous cell carcinomas also include pearly irregular borders and a tendency to develop ulceration with irregular rolled edges.
Sharing the characteristics of BHD syndrome, fibrofolliculoma is considered to be hamartomas, composed of both connective tissue and follicular epithelial components . Most fibrofolliculoma may have some common histogenesis such as abnormal function of hair follicle bulge cells, and differential diagnosis should be considered in the histopathological exam.
Surgical excision is usually chosen for the skin fibrofolliculomas in the first operation for pathological diagnosis. The CO2 laser, or erbium-doped YAG laser, might be a better choice for multiple fibrofolliculomas or recurrent lesions [12-13]. Currently, there are no uniform standards for eyelid lesions. Surgical treatment for an eyelid lesion should be individualized based on the size, growth rate, invasion, and interference with eyelid function and aesthetics [14-15]. Benign eyelid lesions can be treated with less invasive techniques, such as shaving biopsy, and simple excision. For the benign tumors on lid margin, shave excision including a 1-mm margin of normal tissue should be considered [16,17]. Also, postoperative follow-up is necessary to demonstrate early recurrence, and to improve facial appearance as required.