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• W4206487106 abstract "To the Editor: A 20-year-old woman presented with multiple asymptomatic rough skin lesions in a linear distribution over the left forearm and arm since birth. She did not have any systemic complaints, and her family history was unremarkable. Cutaneous examination revealed multiple follicular and nonfollicular keratotic papules and comedones in a linear distribution over the left forearm and arm (Fig. 1). Other mucocutaneous, general, and systemic examinations were within normal limits. Differential diagnosis of nevus comedonicus (NC), comedonal Darier disease, and porokeratotic eccrine ostial and dermal duct nevus was considered. Histology revealed multiple dilated follicular infundibulum with keratotic plugs. The infundibular epithelium showed features of vacuolar and granular degeneration with normal interfollicular epidermis. Perinuclear eosinophilic inclusion, dyskeratotic cells, and acrosyringeal epidermolytic hyperkeratosis (EHK) were also noticed (Fig. 2). The diagnosis of linear comedonal epidermolytic nevus was made.FIGURE 1.: A and B, Multiple keratotic, both follicular and nonfollicular papules and comedones (blue arrow) in a linear distribution.FIGURE 2.: A and B, Histology showing dilated follicular infundibulum with a keratotic plug and EHK (H&E, ×50, ×100). C, Acrosyringeal (blue arrow) EHK (H&E, ×50). D, The presence of dyskeratotic cells (red arrow) and perinuclear eosinophilic inclusion (black arrow) can be noticed (H&E, ×100).EHK, a histopathological pattern of abnormal keratinization, is characterized by the presence of compact hyperkeratosis and granular and vacuolar degeneration of the cells of the stratum spinosum and granulosum. It can be noticed in different clinical settings, both congenital and acquired, ranging from a severe keratinization disorder such as epidermolytic ichthyosis to an incidental histopathological finding in various nevi. The importance lies in detecting the epidermolytic variant of linear verrucous epidermal nevus, which may manifest as bullous ichthyosis in the offspring.1 The EHK of the follicular epithelium is a rare entity. The condition has acquired various names since its first report. It was originally described by Montgomery as “nevus comedonicus with eosinophilic granular degeneration of the prickle cells and cells in the stratum granulosum extending almost to the basal cell layer of the epidermis.”1 Plewig et al, in 1975, called the same entity as “nevoid follicular EHK,” in a 22-year-old woman who presented with multiple follicular keratotic papules that looked like open and closed comedones in the left side of the upper part of the body. The pathology showed features of EHK in hair follicles, sebaceous ducts, and acrosyringium.2 Barsky et al1 reported 4 cases of EHK of the follicular epithelium as NC with EHK. They postulated a link between the 2 conditions, and epithelial nevi can affect appendages, that is, hair follicles. We decided to use the term “linear comedonal epidermolytic nevus” because the cases reported before suggest that the follicular EHK associated with the comedones, as in our case, falls in the spectrum of epidermolytic nevus rather than in NC. The following are supporting data. The occurrence of generalized EHK (epidermolytic ichthyosis) in a child with a father having NC with EHK endorses the fact that comedones with EHK are different from comedones of NC.3 It is probably the comedonal counterpart of the linear epidermolytic verrucous epidermal nevus. Similarly, Schecter et al reported a case of linear NC with EHK in a woman who had a dark brown, linear, cribriform plaque with follicular plugging since childhood and showed EHK on histopathology.4 We believe this may be a case of a linear verrucous epidermal nevus with comedonal EHK. They also suggested looking for the presence of EHK in NC to identify the patients who may be at a risk of transmitting generalized EHK, a well-known risk associated with linear epidermolytic verrucous epidermal nevus, not with NC. A report of coexistence of linear epidermolytic nevus and epidermolytic NC in a patient with keratin 10 mutation further adds to the evidence. Besides, the histopathology image provided by the authors for the lesion of epidermolytic NC shows the presence of EHK both in the epidermis and the follicular infundibulum, suggesting that the primary pathology is EHK that involved both the cutaneous and follicular epithelium.5 Similarly, there is no report of the presence of commonly associated secondary inflammatory changes such as papules, pustules, nodules, cysts, and scarring, or any syndromic associations of NC in NC with EHK. Besides, we observed mostly open comedones without any dark-colored keratotic plug, characteristic of NC, in the index case.6 The various histopathological association of NC has not been reported to involve the comedones, except in familial dyskeratotic comedone, that shows dyskeratotic changes in the comedones.1 In conclusion, we are reporting a rare case of linear comedonal epidermolytic nevus. We believe it belongs to the spectrum of epidermolytic nevus rather than part of NC, although larger studies will define the exact nature of this rare entity." @default.
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• W4206487106 date "2020-11-03" @default.
• W4206487106 modified "2023-10-16" @default.
• W4206487106 title "Linear Comedonal Epidermolytic Nevus: A Rare Entity" @default.
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• W4206487106 doi "https://doi.org/10.1097/dad.0000000000001836" @default.
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