Dermatology Reports 2020; volume 12:8791

A case of post-inflammatory warty dyskeratoma of the Case Report Correspondence: Vito Ingordo, Outpatients’ Department of Dermatology, District n. 6, chest: Other dermoscopic A small erythemato-papular lesion Local Health Centre Taranto, Taranto, 74121, features superficially eroded occurred on the hairy Italy region of the chest in a 53-year-old Tel.: +39-099-7793821 Caucasian man. He was on the whole in E-mail: [email protected] Vito Ingordo,1 Gerardo Ferrara,2 3 4 good health; he was affected by a mild Irene Ingordo, Giuseppe Argenziano hypertension which was under control with Key words: Warty dyskeratoma, dermoscopy; 1Outpatients’ Department of an ACE-inhibitor drug. No other skin dermatoscopy; etiopathogenesis; histopathol- ogy. Dermatology, District n. 6, Local Health lesions were observed. No history of 2 Darier’s disease was reported. After a one- Centre Taranto, Taranto; Anatomic Conflict of interest: the authors declare no Pathology Unit, Hospital of Macerata, week topical corticosteroid-antibiotic thera- potential conflict of interests. Macerata; 3Private practice, Milano; py, the lesion appeared as a skin-coloured 4Dermatology Unit, University of and umbilicated with a central kera- Received for publication: 12 July 2020. totic plug (Figure 1A). Dermoscopy (per- Campania “L. Vanvitelli”, Napoli, Italy Accepted for publication: 20 July 2020. formed with Molemax II, Derma Medical Systems, Vienna, Austria) revealed an This work is licensed under a Creative eight-shape whitish collarette surrounded Commons Attribution-NonCommercial 4.0 Abstract by light brown pigmentation. A central International License (CC BY-NC 4.0). white structureless area with an adjacent Warty Dyskeratoma (WD) is a rare con- ©Copyright: the Author(s), 2020 rosette were observed. Some small rust- Licensee PAGEPress, Italy dition consisting in single or multiple papu- coloured blood crusts were also observed in lar or nodular lesions of the skin or of the Dermatology Reports 2020; 12:8791 the centre of the lesion; no prominent vas- doi:10.4081/dr.2020.8791 oral mucosamucosa. Histologically, a cup- cular pattern was detected (Figure 1B). shaped epidermal invagination centred by a Histology featured a cup-shaped epidermal only plug of epidermal hyperparakeratosis with invagination (Figure 1C) filled with a WD, whereas cystic and cup-shaped lesions suprabasal and is hyperkeratotic and parakeratotic plug, con- are relatively well-developed lesions. Most typically observed. A case of post-inflam- taining numerous grains and corps ronds authors believe that cutaneous WD is matory WD, which was also observed by (Figure 1D). The base of invagination usecon- derived from the hair follicle because of its dermoscopy, is described. Dermoscopy sisted of hyperplastic epithelium with digi- closeness or clear-cut connection to pilose- showed an eight-shape whitish collarette tate projections (villi). Based on these baceous unit,3,5-7,9 thereby suggesting that surrounded by light brown pigmentation. A microscopic features, a diagnosis of WD “follicular dyskeratoma” might be a better central white structureless area with an 6 was made. term for this entity. A study performed with adjacent rosette were observed. Some small human keratin antibodies, specific for cor- rust-coloured blood crusts were also tex and inner root sheet of normal human observed in the centre of the lesion; no hair,8 and a survey carried out searching the prominent vascular pattern was detected. Discussion expression of cytokeratins CK1, CK5, Ck10, The etiopathogenesis of this benign neo- 5 CK17, CK19 confirmed this hypothesis. WD plasm could be multifactorial. Dermoscopy Cutaneous WD is a rare condition;5,6 it arising in regions devoid of follicular struc- of WD is not specific but may help to ruling appears in middle-aged patients as a cuta- tures (oral mucosa12 and nail bed13, 14) may out other skin tumors. neous elevated nodule or cyst with a raised represent examples of isolated FAD sec- border and a somewhat umbilicated or pore- ondary to a local inflammation or a neoplas- like centre. Most of the lesions are brown, 5,6,9,12 but some are described as skin-coloured, tic proliferation. Therefore, Allon et al. 3,5,6 suggested for oral lesions the term “isolated Introduction yellow, red, black, tan, or grey. Usually a 12 Non-commercialsingle lesion is observed,3,5-7, but cases with FAD” rather WD. After the seminal description by Helwig in multiple lesions have been described.8-10 The etiology of cutaneous WD is 1 1954 as “isolated Darier’s disease”, Warty Head and are mainly affected.3,5-7 unknown. Ultraviolet light, autoimmunity, Dyskeratoma (WD) was described as a distinc- Trunk and limbs are interested in a minority viral infections, and chemical carcinogens 2,3,6,7,9,15 tive clinic-pathological entity by Szymanski in of patients;3,5-7 a few cases of vulvar11 and have been postulated to play a role. 2 1957.2 Shortly after, Graham and Helwig3 pro- oral12 WD are described. Szymanski showed the presence of viral posed the name “isolated dyskeratosis follicu- Patients affected by cutaneous WD are DNA and RNA in the epithelial cells lining 6 laris”, because their cases exhibited a pattern usually middle-aged.3,5-7 In some case series the villi, but Kaddu et al. failed to find of dyskeratosis and acantholysis similar to that the male sex was prevalent,3,7 in another HPV-DNA in wide number of HPV in their described in Darier’s disease. Ackerman4 sug- series women were more affected than case series. In oral WD, smoking seems to gested that WD should be included within the men,6 but according to other authors both play the main etiologic role. In this spectrum of dermatoses with Focal sexes were interested in the same propor- instance, ill-fitting dentures, alcohol use, Acantholytic Dyskeratosis (FAD): he pro- tion.5 Mucosal WD seems instead more check biting, sniff use, extraction site, and posed that the term WD should be restricted to prevalent in middle-aged males.12 immunosuppression were also suggested lesions clinically presenting as a single nodule, Kaddu et al.6 described three architec- how etiologic factors.12 whereas papular lesions should be best tural patterns of cutaneous WD, namely, It was proposed that WD represents a labelled as “papular form of FAD”. To date, cup-shaped, cystic, and nodular. They sporadic localized error in epithelial matu- however, the term WD is commonly used for hypothesized that nodular lesions may rep- ration and cohesiveness.16 In this regard it is both papular and nodular lesions.5,6 resent an early stage in the occurrence of interesting to emphasize that Ackerman

[page 53] [Dermatology Reports 2020; 12:8791] Case Report classified solitary FAD in three forms: i) a garis, Hailey-Hailey disease, syringocys- clinically nonapparent type (histologically toadenoma papilliferum.3,6,7,11,17 Conclusions incidental lesions); ii) a papular type (focal Noteworthy, the presence of histopathologi- WD is a rare condition and its develop- acantholytic dyskeratoma) and iii) a nodular cally clear surgical margins virtually 4 ment could be multifactorial, including also type (warty dyskeratoma). “Incidental excludes the inflammatory dermatoses list- a post-inflammatory pathogenesis. FAD” has also been observed in many skin ed above. According to patterns reported in lesions (i.e. epithelial lesions, fibrohistio- Dermoscopy of WD has been per- Appendix, the dermoscopic features of WD cytic lesions, inflammatory conditions, formed in a limited number of cases 20 16-20 are not specific; however, dermoscopy melanocytic neoplasms and miscellaneous (Appendix). of acantholytic may help rule out the above-mentioned con- lesions). The pathologic changes either dyskeratotic , acantholytic occurred within the lesion or in the “clini- dyskeratoses, and Grover’s disease display ditions, thereby making WD a clinical diag- cally normal-appearing” epithelium imme- a central yellowish to brown star-like pat- nosis which can be raised by exclusion. diately adjacent to the lesion 15. tern overlying a pinkish homogeneous Accordingly, as proposed by Griffiths et structure-less area.16 Syringocystoadenoma al.,8 the development of WD at various papilliferum reveals a polymorphous vascu- anatomical sites could be multifactorial. lar pattern on a pinkish-white background.19 References This hypothesis is in agreement with the Papules of Darier’s disease show a centrally 1. Helwig EB. Proceedings of the 20th post-inflammatory onset of our case. located yellowish/brownish area surround- 17 seminar on skin neoplasms and der- Clinical differential diagnosis of cuta- ed by a thin whitish halo. Dermoscopy of matoses, International Congress of neous and mucosal WD includes basal cell and squamous cell carci- Clinical Pathologists, 1954 Sept, , epidermal cyst, seborrheic ker- is characterized by keratin masses, Washington, DC, USA. Am Soc Clin atosis, actinic , squamous cell car- white structures (white circles, white clods, Pathol 1955;53-6. cinoma, melanocytic , keratoacan- and white structureless areas), and hairpin 17,20 2. Szymanski FJ. Warty dyskeratoma: a thoma, common , condyloma acumina- vessels. In viral wart, the most frequent benign cutaneous tumor resembling tum, , sebaceous hyper- dermoscopic findings are the mosaic pattern Darier’s disease microscopically. Arch plasia, adnexal tumour, oral papilloma or and the exophytic keratotic projection pat- only 5,6,11,17 Dermatol 1957;75:567-72. . tern (dotted, linear, or hairpin vessels, often 3. Graham JH, Helwig EB. Isolated Histological differential diagnosis must surrounded by a whitish halo), often with 20 dyskeratosis follicularis. AMA Arch be made with Darier’s disease, Grover’s microhemorrages. use Derm 1958;77:377-89 disease, acantholytic squamous cell carci- 4. Ackerman AB. Focal acantholytic noma, keratoacanthoma, vul- dyskeratosis. Arch Dermatol 1972;106:702-6. 5. Diallo M, Cribier B, Scrivener Y. Warty dyskeratoma: infundibular histogenesis. Anatomoclinical study of 43 cases. Ann Dermatol Venereol 2007;134:633-6. 6. Kaddu S, Dong H, Mayer G, et al. Warty dyskeratoma – “follicular dyskeratoma”: analysis of clinicopatho- logic features of a distinctive follicular adnexal neoplasm. J Am Acad Dermatol 2002;47:423-8. 7. Tanay A, Mehregan AH. Warty dysker- atoma. Dermatologica 1969;138:155- 64. Non-commercial 8. Griffiths TW, Hashimoto K, Sharata HH, Ellis CN. Multiple warty dysker- atomas of the . Clin Exp Dermatol 1997;22:189-91. 9. Martorell-Calatayud A, Sanmartin- Jimenez O, Traves V, Guillen C. Numerous umbilicated papules on the trunk: multiple warty dyskeratoma. Am J Dermatopathol 2012;34:674-5. 10. Ugras N, Adim SB, Kilicoglu M, Baskan EB. Multiple warty dysker- Figure 1. A) Clinical picture featuring a skin-coloured, umbilicated nodule with a central atomas: case report. Iran J Public keratotic plug. B) Dermoscopy of WD, typified by an eight-shape whitish collarette sur- Health 2014;43:1145-7. rounded by light brown pigmentation; a central white structureless area with an adjacent 11. Tiangco Torres KM, Junkins-Hopkins rosette; some small rust-coloured blood crusts at the centre of the lesion. C) JM. Cystic acantholytic dyskeratosis of Histopathologic picture disclosing a cup-shaped epidermal invagination filled with ker- atinous hyper-parakeratotic plug. D) A closer histologic view with numerous grains and the : an unusual presentation of a corps ronds. follicular adnexal neoplasm. Indian Dermatol Online J 2016;7:272-4. 12. Allon I, Buchner A. Warty dysker-

[Dermatology Reports 2020; 12:8791] [page 54] Case Report

atoma/focal acantholytic dyskeratosis- 10.1155/2017/3613109. 18. Lencastre A, Campos S, Cabete J. an update on a rare oral lesion. J Oral 15. DiMaio DJ, Cohen PR. Incidental focal Warty dyskeratoma. J Am Acad Pathol Med 2012;41:261-7. acantholytic dyskeratosis. J Am Acad Dermatol 2016;75:e97-8. 13. Baran R, Perrin C. Focal subungual Dermatol 1998;38:243-7. 19. Isaka Y, Matsuda H, Ogita A, et al. Case warty dyskeratoma. Dermatol 16. Salerni G, Alonso C, Calligaris M, et al. of warty dyskeratoma on the anterior 1997;195:278-80. Dermoscopy of multiple warty dysker- chest: the relationship between its der- 14. Vargas-Laguna E, Imbernón-Moya, atoma. Dermatol Pract Concept moscopic and histopathological find- Aguillar-Martinez A, Burgos F. An 2017;7:47-9. ings. J Dermatol 2018;45:e171-2. unusual location of subungual warty 17. Nakagawa T, Umekage K, Komatsu S, 20. Di Brizzi EV, Russo T, Piccolo V, et al. dyskeratoma: a case report and review et al. Case of warty dyskeratoma with Warty dyskeratomas: clinical and der- of the literature. Case Rep Dermatol unique dermoscopic features. J moscopic features. Int J Dermatol Med 2017;2017:3613109. doi: Dermatol 2018;45:e74-5. 2019;58:e229-31.

only use

Non-commercial

[page 55] [Dermatology Reports 2020; 12:8791]