A rare case of syringocystadenoma papilliferum: clinical and histopathological insights

  • Raden Roro Rini Andayani Department of Dermatology, Venereology, and Aesthetic, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah/Dr. Sardjito General Hospital, Yogyakarta, Indonesia
  • Satiti Retno Pudjiati Department of Dermatology, Venereology, and Aesthetic, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah/Dr. Sardjito General Hospital, Yogyakarta, Indonesia
  • Erliana Tantri Harsono Department of Dermatology, Venereology, and Aesthetic, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah/Dr. Sardjito General Hospital, Yogyakarta, Indonesia
  • Dwinanda Almira Rizkiani Department of Dermatology, Venereology, and Aesthetic, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah/Dr. Sardjito General Hospital, Yogyakarta, Indonesia
  • Hanggoro Tri Rinonce Department of Anatomical Pathology, Faculty of Medicine, Public Health, and Nursing, Universitas Gadjah Mada, Yogyakarta, Indonesia
  • Rita Cempaka Department of Anatomical Pathology, Faculty of Medicine, Public Health, and Nursing, Universitas Gadjah Mada, Yogyakarta, Indonesia
  • Indra Wahono Department of Anatomical Pathology, Faculty of Medicine, Public Health, and Nursing, Universitas Gadjah Mada, Yogyakarta, Indonesia
  • Siti Isya Wahdini Department of Surgery, Faculty of Medicine, Public Health, and Nursing, Universitas Gadjah Mada, Yogyakarta, Indonesia
  • Dyah Ayu Mira Oktarina Department of Dermatology, Venereology, and Aesthetic, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah/Dr. Sardjito General Hospital, Yogyakarta, Indonesia
Keywords: syringocystadenoma papilliferum, adnexal tumor, histopathology, dermoscopy, verruca vulgaris

Abstract

Syringocystadenoma papilliferum (SCAP) is a rare benign adnexal neoplasm originating from apocrine or eccrine sweat glands, first reported by Stokes in 1917. Clinically, SCAP manifests in various forms such as nodules, plaques, or warts, predominantly affecting the scalp in adolescents or children. Histopathologically, it is characterized by papillary and ductal invaginations lined with a two-layered epithelium. This condition can be present at birth or emerge during puberty. We present a 19-year-old female with a congenital, pinkish-brown, wart-like lesion on her scalp that progressively enlarged and spread to the face. Physical examination revealed a verrucous plaque with central erosion, extending from the temporal scalp to the lateral face. Dermoscopy showed papillary structures with defined lesion borders, while histopathological findings confirmed SCAP. The patient underwent surgical excision, and no new lesions were detected during a 3 mo follow-up. Syringocystadenoma papilliferum is often confused with other neoplasms such as sebaceous nevus or verruca vulgaris, but its definitive diagnosis relies on histopathology. The tumor’s characteristic features include cystic invaginations into the dermis with a two-layered epithelium, differentiating it from other conditions. Surgical excision remains the treatment of choice to prevent potential malignant transformation and improve cosmetic outcomes. Syringocystadenoma papilliferum, though rare, can be effectively managed through timely diagnosis and surgical excision. In this case, the patient’s postoperative outcome was favorable, with no recurrence of the lesion. Histopathological evaluation is crucial for accurate diagnosis and guiding treatment strategies.

References

Foreman RK, Duncan LM. Appendage tumors of the skin. In: Kang S (ed) Fitzpatrick's Dermatology 9th ed. New York: Mc Graw Hill Education; 2019. 1820-56.

Yao M, Rao L. Linear synngocystadenoma papilliferum of the limb: a rare localization of an uncommon tumour. Anais Brasileiros de Dermatologia, 2023; 98(6):858-60.

https://doi.org/10.1016/j.abd.2021.09.024

Vazmitel M, Michal M, Mukensnabl P, Kazakov DV. Syringocystadenoma papilliferum with sebaceous differentiation in an intradermal tubular apocrine component. Report of a case. Am J Dermatopathol, 2008; 30(1):51-3.

https://doi.org/10.1097/DAD.0b013e31815c4cf4

C. Zouboulis C. Skin glands: sebaceous, eccrine, and apocrine glands. In: Kang Sewon (ed) Fitzpatrick's Dermatology 9th ed. New York: Mc Graw Hill Education; 2021. 71-88.

Monaco M, González VM, Vigovich FA, Larralde M. Syringocystadenoma papilliferum in the scalp, with a linear presentation. An Bras Dermatol, 2023; 98(3):406-9.

https://doi.org/10.1016/j.abd.2021.11.011

Goyal P, Meena D, Kumar A. A rare tumor at an unusual site with a rare association. Indian Dermatol Online J, 2024 Feb 13;15(2):363-5.

https://doi.org/10.4103/idoj.idoj_466_23

Yamamoto O, Doi Y, Hamada T, Hisaoka M, Sasaguri Y. An immunohistochemical and ultrastructural study of syringocystadenoma papilliferum. Br J Dermatol, 2002; 147(5):936-45.

https://doi.org/10.1046/j.1365-2133.2002.05027.x

Chen G, Liu W, Liao X, Meng J, Leng X. Ultrasonographic and pathological findings of syringocystadenoma papilliferum in the skin. Med Ultrason, 2023; 25(4):472-3.

https://doi.org/10.11152/mu-4292

Léda LDSB, Lins MDSVS, Leite EJDS, Cardoso AEC, Houly RLS. Syringocystadenoma papilliferum combined with a tubular apocrine adenoma. An Bras Dermatol, 2017; 92(5):721-3.

https://doi.org/10.1590/abd1806-4841.20174580

Candrawinata VS, Koerniawan HS, Prasetiyo PD, Baskoro BA. Syringocystadenoma papilliferum in a 20-year-old adult: a case report and literature review. J Surg Case Rep, 2022; 2022(10):rjac470.

https://doi.org/10.1093/jscr/rjac470

Duran J, Plaza JA. Update on immunohistochemical techniques in dermatopathology. A focus on adnexal neoplasms. Semin Diagn Pathol, 2022; 39(4):265-87.

https://doi.org/10.1053/j.semdp.2022.01.001

Liu L, Wang H, Dong J. Classification of and the latest photoelectric therapy for epidermal nevi Chinese. J Plastic Reconstr Surg, 2022; 4(2):91-6.

https://doi.org/10.1016/j.cjprs.2022.04.003

Cuda JD, Rangwala S, Taube JM. Benign epithelial tumors, hamartomas, and hyperplasias. In: Kang sewon (ed) Fitzpatrick's Dermatology. New York: Mc Graw Hill Education; 2019. 1799-819.

Jiang J, Chen Y, He Q, Yang J, Zhang Z, Yang H, et al. Syringocystadenoma papilliferum and basal cell carcinoma arising in nevus sebaceous. Clin Cosmet Investig Dermatol, 2022; 15:2021-6.

https://doi.org/10.2147/CCID.S378746

Garcias-Ladaria J, Cuadrado Rosón M, Pascual-López M. Epidermal nevi and related syndromes -- part 1: keratinocytic nevi. Actas Dermosifiliogr (Engl Ed), 2018; 109(8):677-86.

https://doi.org/10.1016/j.adengl.2018.07.019

Published
2025-08-11
How to Cite
1.
Andayani RRR, Pudjiati SR, Harsono ET, Rizkiani DA, Hanggoro Tri Rinonce, Rita Cempaka, Indra Wahono, Siti Isya Wahdini, Dyah Ayu Mira Oktarina. A rare case of syringocystadenoma papilliferum: clinical and histopathological insights. InaJBCS [Internet]. 2025Aug.11 [cited 2025Aug.25];57(3). Available from: https://journal.ugm.ac.id/v3/InaJBCS/article/view/17146
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