SESD is a rare and benign neoplasm that can affect different body areas [
1‐
9], first described by Rothko
et al. in 1980 as six cutaneous tumors affecting multiple body areas of a 48-year-old man without any systemic disease or family history [
1]. Subsequent case reports and series show that it favors the head, face and trunk of middle-to-old-age individuals regardless of sex [
1‐
10]. On clinical examination, these lesions can grow as rapidly as a few months or slowly over 1 to 3 years and may present as nonspecific papules, nodules or plaques of varying sizes, consistency and color [
2,
3,
5]. This broad clinical presentation gives wide differential diagnoses ranging from totally benign lesions, such as sebaceous hyperplasia and seborrheic keratosis, to highly malignant lesions, such as sebaceous carcinoma and basal cell carcinoma [
3]. The histopathological features of the lesion also give rise to wide differential diagnoses, mainly that of sebaceous differentiation including, but not limited to, seborrheic keratosis with sebaceous differentiation (SKSD), sebaceous hyperplasia, sebaceous adenoma, sebaceoma, sebaceous carcinoma and basal cell carcinoma with sebaceous differentiation (Table
1) [
2]. SESD can be very difficult to distinguish from SKSD histopathologically. However, the presence of squamous like cells arranged in whorls (squamous eddies) that are occasionally found around wide infundibular spaces filled with corneocytes (pseudohorn cysts) is more common in SKSD [
3,
4,
11]. Basal cell carcinoma with sebaceous differentiation usually presents with an aggressive behavior and tend to arrange in a lobular pattern rather than the plate-like configuration in SESD [
4]. To the best of our knowledge, ocular involvement of SESD has been reported twice in the literature as eyelid lesions in two patients (Table
2) [
2,
5].
Table 1
Differential diagnosis of superficial epithelioma with sebaceous differentiation
1 | Seborrheic keratosis with sebaceous differentiation |
2 | Sebaceous hyperplasia |
3 | Sebaceous adenoma |
4 | Tumors of the follicular infundibulum with sebaceous differentiation |
5 | Sebaceous carcinoma |
6 | Basal cell carcinoma with sebaceous differentiation |
Table 2
Ocular cases of superficial epithelioma with sebaceous differentiation
| 1987 | 57 | M | Eyelid | 4mm pearly papule | No recurrence with 8-months f/u |
| 1992 | 38 | F | Right upper lid | 6mm yellow plaque | No recurrence with 6-months f/u |
Our report | 2014 | 46 | M | Right upper lid | 4mm tan-colored papule | No recurrence with 5-months f/u |
Currently, the literature is full of different terms describing tumors with foci of sebaceous differentiation. The difficulty in diagnosing these lesions has led to the use of inappropriate terms and, as a consequence, cases of SESD are underdiagnosed cases. Reticulated acanthoma with sebaceous differentiation, SKSD, acanthomatous superficial sebaceous hamartoma, sebocrine adenoma, sebomatricoma and poroma with sebaceous differentiation are all names for SESD in the literature. Despite the different nomenclature, the unifying architecture of different cases of SESD, specifically the broad plate-like outgrowth, favors SESD as the correct description [
3,
10,
12]. In addition, different immunohistochemical studies can differentiate this tumor from others [
2,
3,
10].
Different reports show a lack of association between SESD and Muir–Torre syndrome (MTS) [
1‐
3,
5,
12,
13]. Although our patient had orchiectomy for a testicular seminoma, the expression of mismatch repair proteins MLH1 and MSH2 was positive, which excludes any microsatellite instability and supports the lack of an association with MTS.
The histogenesis of the tumor is not yet known. Friedman
et al. [
5] speculated that it originates from the pilosebaceous unit. This speculation is supported by Kawachi
et al. [
10] and their immunohistochemical studies, in which they discovered its differentiation toward the sebofollicular epithelium. On the basis of the immaturity of the sebaceous cells in the lesion and the negative staining for epithelial membrane antigen and carbonic anhydrase-2, Kato and Ueno [
2] suspected the epidermal pluripotential cells as the origin of SESD and not the pilosebaceous unit.