10.5826/dpc.1302a128
text/xml
2160-9381
2832
en
Giuseppe Argenziano
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
Multiple Pigmented Trichoepitheliomas: A Case Report
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Letter to the Editor
2023-04-29
2022-09-09
2023-04-29
2023-06-05
e2023128
Dermatol Pract Concept
Giuseppe Argenziano
2023/04/29
10.5826/dpc.1302a128
e2023128
Dermatology Unit, Department of Mental and Physical Health and Preventive Medicine, University of Campania Luigi Vanvitelli, Naples, Italy
2160-9381
en
e2023128
Supekar BB, Rambhia KD, Tomar SS, Singh RP, Multiple Familial Trichoepithelioma with Varied Malignancies. Indian J Dermatol. 2021 Mar-Apr;66(2):226. doi: 10.4103/ijd.IJD_647_18. PMID: 34188306; PMCID: PMC8208248.
Multiple Pigmented Trichoepitheliomas: A Case Report
https://dpcj.org/index.php/dpc/article/download/2832/2104
                 Multiple Pigmented Trichoepitheliomas:
                             A Case Report
      Stefano Caccavale1, Carmine Fiorentino1, Maria Pia Boccellino1, Francesca Pagliuca2,
                         Ruzica Jurakic Toncic3, Giuseppe Argenziano1


1 Dermatology Unit, Department of Mental and Physical Health and Preventive Medicine, University of Campania Luigi Vanvitelli,
  Naples, Italy
2 Division of Pathology, Department of Mental and Physical Health and Preventive Medicine, University of Campania Luigi Vanvitelli,
  Naples, Italy
3 Department of Dermatology and Venereology, University of Zagreb, Zagreb, Croatia



Citation: Caccavale S, Fiorentino C, Boccellino MP, Pagliuca F, Jurakic Toncic R, Argenziano G. Multiple Pigmented Trichoepitheliomas:
A Case Report. Dermatol Pract Concept. 2023;13(2):e2023128. DOI: https://doi.org/10.5826/dpc.1302a128
Accepted: November 2, 2022; Published: April 2023
Copyright: ©2023 Caccavale et al. This is an open-access article distributed under the terms of the Creative Commons
Attribution-NonCommercial License (BY-NC-4.0), https://creativecommons.org/licenses/by-nc/4.0/, which permits unrestricted
noncommercial use, distribution, and reproduction in any medium, provided the original authors and source are credited.
Funding: None.
Competing Interests: None.
Authorship: All authors have contributed significantly to this publication.
Corresponding Author: Stefano Caccavale, MD, Dermatology Unit, Department of Mental and Physical Health and Preventive Medicine,
University of Campania Luigi Vanvitelli, Via Sergio Pansini, 5, 80131 Naples, Italy; phone: +39 3336365526; fax: +39 0815468759;
E-mail: stefano85med@libero.it




Dear Editor,                                                                  A 70-year-old-man, Fitzpatrick photo-type 4, pre-
                                                                         sented to our attention for Zoon balanitis. During the clini-
    Trichoepitheliomas       are   uncommon       benign    tumors       cal examination of the total body surface we found multiple
with infundibulocystic differentiation. They most often occur            nodular pigmented lesions of the face, trunk and lower limbs.
in women, presenting themselves as translucent round pap-                     We identified 5 pigmented, non-ulcerated, smooth,
ules or nodules mainly localized at the face or scalp [1]. They          brown/blue papules and nodules of 0.5-0.7 cm of diameter.
may appear as solitary or multiple lesions in ­non-familial or           Two lesions were localized on the head, respectively at the
­familial forms (including multiple familial trichoepitheliomas,         superciliary arch and the occiput. The other three lesions
Brooke-Spiegler syndrome and familial c­ ylindromatosis) [1,2].          were localized at the posterior thigh, the superior back and
­Familial forms are often due to mutations in CYLD gene, on              the chest.
chromosome 9, with autosomal dominant transmission. This                      On dermoscopy (Figure 1, A-F), the lesions were
gene has incomplete penetration in males, so the prevalence              highly pigmented and showed basalioma-like features,
of familial forms is higher in women. In these cases, the onset          such as brown-blue globules and nests, blue-black blotches
of trichoepitheliomas is earlier compared to sporadic forms.             and leaf-like areas together with less specific patterns
    To the best of our knowledge, we report the first case               (shiny-white lines, milia-like cysts and gray-blue veil). Four
of multiple pigmented trichoepitheliomas discussed in liter-             of them presented bright vessels. The lesion localized at the
ature so far.                                                            posterior thigh presented a blue lacunar-like pattern.



Letter to Editor | Dermatol Pract Concept. 2023;13(2):e2023128                                                                           1
Figure 1. (A-F) Clinical presentation on admission (A,C,E). Dermoscopic images (B,D,F) revealed: bright vessels (red arrows in B,D,F),
milia-like cyst (blue arrow in B), leaf-like areas (D), brown-blue globules and nests (D), lacunar-like pattern (F), shiny white lines (F),
blue-white veil (B,F), brown-blue blotches (B,F).




    Since the dermoscopic clues were suggestive for pig-               dermoscopically very difficult to distinguish from pigmented
mented basal cell carcinomas, we decided to excise all le-             basal cell carcinomas, similarly to non-pigmented ones. At
sions. The histopathology of the biopsy samples showed                 date, the histopathological examination still represents the
islands and nests of follicular germinative cells, without stro-       gold standard to the final diagnosis. More studies will be
mal retraction spaces, immersed in a conspicuous fibrocytic            needed to understand if there may be a correlation between
stroma, with high cellularity. The well-circumscribed tumors           the photo-type and predisposition to develop pigmented
were focal connected with the overlying epidermis. In addi-            rather than non-pigmented trichoepitheliomas.
tion, multiple horn cysts were detected in some lesions. All
these features suggested overall the diagnosis of trichoepithe-
                                                                       References
liomas and not of basal cell carcinomas.
    Due to the presence of multiple pigmented trichoepitheli-           1. Karimzadeh I, Namazi MR, Karimzadeh A. ­Trichoepithelioma:
                                                                           A Comprehensive Review. Acta Dermatovenerol Croat. 2018;
omas, a DNA analysis to exclude a possible genetic etiology
                                                                           26(2):162-165. PMID: 29989874.
was proposed to the patient, but he refused.
                                                                        2. Supekar BB, Rambhia KD, Tomar SS, Singh RP, Multiple Famil-
    In conclusion, this was the first case of multiple pig-                ial Trichoepithelioma with Varied Malignancies. Indian J Der-
mented trichoepitheliomas described so far in litera-                      matol. 2021;66(2):226. DOI: 10.4103/ijd.IJD_647_18. PMID:
ture to our knowledge. These lesions are clinically and                    34188306. PMCID: PMC8208248.




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