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Case Reports
. 2023 Sep 28;11(10):2667.
doi: 10.3390/biomedicines11102667.

Bleomycin Electrochemotherapy of Dermal Cylindroma as an Alternative Treatment in a Rare Adnexal Neoplasm: A Case Report and Literature Review

Antonio Bonadies  1 , Alessandra Iorio  2 , Vitaliano Silipo  2 , Carlo Cota  3 , Flavio Andrea Govoni  4 , Michela Battista  5 , Tiziano Pallara  1 , Emilia Migliano  1
Affiliations
Case Reports

Bleomycin Electrochemotherapy of Dermal Cylindroma as an Alternative Treatment in a Rare Adnexal Neoplasm: A Case Report and Literature Review

Antonio Bonadies et al. Biomedicines. .

Abstract

Background: Brooke-Spiegler syndrome is a rare autosomal dominant disorder characterized by the continuous development of multiple benign skin appendage tumors. It is treated usually by repeated standard surgery. Here, we present a case study where electrochemotherapy (ECT) with bleomycin was used as an effective alternative approach in treating advanced dermal cylindromatosis of the head and neck in a patient with Brooke-Spiegler syndrome.

Patients and methods: A 45-year-old woman presented with multiple recurrent dermal cylindroma lesions on her scalp. Previous treatment consisted of several surgical excisions that resulted in psychological deterioration due to the formation of numerous scars and extensive alopecic areas. ECT was offered to provide tumor removal and disease control and to improve the patient's quality of life.

Results: The treatment was well tolerated, and a significant reduction in neoplastic tissue was achieved. Importantly, scalp skin condition significantly improved, regaining a fair follicular density on the margins.

Conclusion: This report suggests the feasibility of bleomycin ECT as a less invasive alternative option for controlling multiple scalp cylindroma lesions with cosmetically acceptable results, and improving quality of life.

Keywords: Brooke–Spiegler syndrome; adnexal neoplasm; bleomycin; dermal cylindroma; electrochemotherapy; rare skin tumor.

PubMed Disclaimer

Conflict of interest statement

M.B. is an employee of IGEA S.p.A IGEA Spa, Carpi, Mo, Italy.

Figures

Figure 1
Figure 1
Histopathological analysis of the scalp lesion before ECT treatment (H&E stain). The dermal proliferation of basaloid cells arranged in a nest with the typical “jigsaw” puzzle pattern compatible with cylindroma (×40) (A). Higher magnification of the dotted rectangle region (×200) shows that each nest is composed of a palisading peripheral lining of smaller basophilic cells (black arrows), an inner population with larger and more differentiated paler cells (stars), and shows the presence of eosinophilic hyaline-like globules (yellow arrows). The nests are surrounded by a thickened eosinophilic hyalinized basement membrane material (arrowheads) (B).
Figure 2
Figure 2
Multiple cylindroma lesions localized on the scalp treated with bleomycin ECT. Baseline presentation before starting bleomycin ECT (A); at 1 year after bleomycin ECT (B).
See this image and copyright information in PMC

References

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