Syringocystadenoma Papilliferum at an Unusual Location: a Case Report

Case Report | DOI: https://doi.org/10.31579/2578-8949/086

Syringocystadenoma Papilliferum at an Unusual Location: a Case Report

  • Anirudha Gulanikar* 1
  • Omkar S. Kulkarni 2

1Associate professor, Department of Dermatology, MGM medical college and hospital, Aurangabad, India

2Junior resident, Department of Dermatology, MGM medical college and hospital, Aurangabad, India

*Corresponding Author: Anirudha Gulanikar, Associate professor, Department of Dermatology, MGM medical college and hospital, Aurangabad, India.

Citation: A Gulanikar, Omkar S. Kulkarni. (2021) Syringocystadenoma papilliferum at an unusual location: a case report. Dermatology and Dermatitis. 6(4); Doi:10.31579/2578-8949/086

Copyright: ©2021 Anirudha Gulanikar, This is an open-access article distributed under the terms of The Creative Commons. Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Received: 27 September 2021 | Accepted: 04 October 2021 | Published: 08 October 2021

Keywords: syringocystadenoma papilliferum; adnexal tumours; benign cutaneous tumours

Abstract

A case of 15year old female presented with lesion over back since childhood, with occasional bleeding and oozing from lesion without any associated systemic complaints. There were multiple verrucous coalescing papules forming plaque with overlying erosion present over left lower back- diagnosed provisionally as angiokeratoma circumscriptum and was biopsied. Histopathology revealed findings consistent with Syringocystadenoma papilliferum. Surgical excision was done and closed with rotation flap. Syringocystadenoma is benign cutaneous adnexal tumor presenting clinically with many morphologies such as warty papules, nodules, plaques with oozing of serous material. Lesion is usually seen in head and neck area in most cases however can also occur on extremities, buttocks, anogenital region. It is characterized by multiple invaginations from skin surface in association with hair follicles lined by cuboidal to columnar epithelium on luminal aspect and myoepithelial cells on outside. There is papillary architecture and dermal ductal component. 

Introduction

Skin appendageal tumours are rare tumours which either arise from or differentiate towards pilosebaceous apparatus, eccrine or apocrine morphology. These are more predominantly found in scalp and anogenital region given more density of pilosebaceous apparatus there. Pilar component predominates over scalp whereas glandular component tends to predominate over face. These present with vague clinical features and diagnosed only after histopathological characterization. 

Case report

15year old female came with complaint of lesion over back since childhood. It was asymptomatic and pea sized to begin with, gradually increased in size over last 10-12 years. There is history of occasional bleeding and oozing from lesion. There are no systemic symptoms. On examination, patient had multiple skin-coloured papules coalescing to form plaque that was well circumscribed and had few eroded areas on lower left back. Few areas showed signs of spontaneous resorption. She was provisionally diagnosed as? angiokeratoma circumscriptum? lymphangioma circumscriptum. She was investigated further – here hemogram and blood biochemistry were unremarkable. Punch biopsy was taken and histopathological examination showed crateriform invaginations connected with surface epidermis that are lined by double layer of epithelium – luminal epithelium being apocrine and basal being cuboidal. On the floor of invagination, there were many papillary projections forming solid areas. Surrounding dermal tissue showed mixed infiltrate of lymphocytes, plasma cells and neutrophils.  Thus histopathological diagnosis of Syringocystadenoma papilliferum was made and excision was planned in coordination with plastic surgery department. Lesion was excised and wound was closed with triangular rotation flap. The excised lesion measured 5X3X1 cm and on histopathology, confirmed diagnosis of Syringocystadenoma papilliferum without any cancerous changes. 

Figure 1: Preoperative picture of patient showing multiple verrucous papules coalescing to form plaque with erosion and crusting over surface. Areas showing spontaneous resolution also seen
Figure 2: Postoperative clinical picture of patient
Figure 3: Excised specimen measuring approximately 5X3X2 cm
Figure 4: Histopathology showing invagination from epidermis with apocrine morphology with papillary projections toward base and few base and few solid areas. There is inflammatory infiltrate composed of plasma cells, neutrophils surrounding the investigation.

Discussion

Syringocystadenoma papilliferum is a benign adnexal tumor that arises from glandular ductal epithelium. Its origin is uncertain- it’s considered to arise from pluripotent appendageal cells or apoeccrine glands [1]. It can occur as an isolated tumor or as a secondary growth arising in Nevus sebaceous [2]. It occurs commonly in children and adolescents as acquired lesion, but can be congenital in rare cases [3]. In 75

References

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