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A rare development of basal cell carcinoma on trichoepithelioma in a chemical burn scar tissue

A case report

Kim, Seong Hwan MDa; Kim, Dong Wan MDb; Hwang, Jae Ha MD, PhDb; Kim, Kwang Seog MD, PhDb; Lee, Sam Yong MD, PhDb,*

Section Editor(s): NA.,

doi: 10.1097/MD.0000000000012252
Research Article: Clinical Case Report
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Rationale: Trichoepithelioma (TE) is a rare benign skin tumor that originates from the hair follicle epithelium. Although skin lesions arising from scar tissues are mostly malignant, the development of a benign tumor such as TE is a rare event.

Patient concerns: A 28-year-old male patient who had a scar on the left cheek which arose 10 years ago because of a chemical burn visited our hospital. Scar revision was performed under local anesthesia.

Diagnosis: Histological examination identified the specimen as a TE with malignant foci suspicious of basal cell carcinoma (BCC) at the lateral margin of the specimen.

Interventions: A second stage operation was planned to excise the BCC with a 4 mm margin of the normal skin around the previous lesion.

Outcomes: No complication or recurrence was noted during the 1-year follow-up period after surgery, and the appearance of the scar improved.

Lessons: The correct differential diagnosis between TE and BCC is very important. So follow up at regular intervals is recommended for evaluation of recurrence or transformation into BCC in patients with tumors arising from chemical burn scars. And the concomitant development of TE and BCC should also be considered from a chemical burn scar.

aDepartment of Plastic and Reconstructive Surgery, Kangnam Sacred Heart Hospital, College of Medicine, Hallym University, Seoul

bDepartment of Plastic and Reconstructive Surgery, Chonnam National University Medical School, Gwangju, Korea.

Correspondence: Sam Yong Lee, Department of Plastic and Reconstructive Surgery, Chonnam National University Medical School, 42 Jebong-ro, Dong-gu, Gwang Ju 61469, Korea (e-mail: sylee@chonnam.ac.kr).

Abbreviations: BCC = basal cell carcinoma, TE = trichoepithelioma.

The authors have no conflicts of interest to disclose.

This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0

Received May 15, 2018

Accepted August 14, 2018

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1 Introduction

Trichoepitheliomas (TEs) are regarded as poorly differentiated hamartomas of the hair germ follicle, located mainly on the face, nasolabial folds, forehead, upper lip, and scalp. There are 2 clinical forms: hereditary multiple and nonhereditary solitary.[1] The solitary types do not show hereditary transition. Hereditary TE is seen in younger people with multiple millimetrical nodules.[2]

TE is a benign tumor but occasionally can undergo transformation to malignant neoplasms more commonly as basal cell carcinoma (BCC). Malignant transformation of such lesions is quite rare. The correct differential diagnosis between these tumors is very important because BCC is a locally aggressive neoplasm and requires total surgical excision with wide margins while TE only needs simple excision. In this article, we report a case of BCC on TE which developed after chemical burn.

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2 Case report

A 28-year-old male patient visited our hospital for an examination of a scar on the left cheek which arose 10 years ago because of a chemical burn that had enlarged during the last 5 months. The scar was a 12-mm diagonally lined hypertrophic scar (Fig. 1). The patient did not report any pain or pruritus; however, he experienced slight tenderness on palpation. Scar revision was performed under local anesthesia for aesthetic purpose. On gross examination, a mass lesion was located within the scar tissue. Histological examination identified the mass as a TE located within the scar tissue that showed the typical appearances of lobules of basaloid cells and keratosis. However, a malignant focus suspicious of BCC was noted at the lateral margin of the specimen. A second stage operation was planned to excise the BCC with a 4 mm margin of the normal skin around the previous lesion. Any suspicious lateral or deep margin was confirmed to be tumor-free by frozen section biopsies during the operation. The pathological examination of the specimen revealed a 12×3 mm sized BCC at the margin of the previous scar tissue. The tumor cells showed 10 mitotic figures per 10 high-power fields and perineural invasion (Fig. 2). No complication or recurrence was noted during the 1-year follow-up period after surgery, and the appearance of the scar improved (Fig. 3). We obtained the patient's medical records and reviewed the related literature. Informed consent to participate in the study was obtained from the patient.

Figure 1

Figure 1

Figure 2

Figure 2

Figure 3

Figure 3

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3 Discussion

TEs are benign skin tumors developed from skin annexes showing differentiation toward hair follicles. The lesion is usually a round-oval shaped papule or nodule, about 2 to 8 mm in diameter. The majority of the cases are found on the forehead, nasolabial groove, upper lip, and the cervical region due to the high number of sebaceous glands in these areas.[3] It may also be found on the neck, chest, upper arm, and thigh.[4] Three clinical forms of TE have been reported; a small solitary form, a small multiple forms with autosomal dominant hereditary characteristic, and a rare giant solitary form.[5,6] The management of TE by surgical excision or curettage is usually adequate due to its benign nature.

TE is histologically characterized by a well-circumscribed dermal tumor composed of uniform basaloid cells in a cellular fibrous stroma. The histopathological differentiation of TE from BCC is sometimes difficult as both neoplasms are composed of nests of basaloid cells with follicular differentiation. TE may have epithelial structures resembling hair follicle, small keratocysts lined by stratified squamous epithelium and foci of calcification while BCC consists of basaloid tumor island with peripheral palisading, stromal retraction, increased mitotic activity, and necrosis.[7,8] In some cases, the differentiation of BCC with follicular differentiation and TE may be impossible with hematoxylin and eosin staining. Immunohistochemical staining with Bcl-2, CD10, CD34, transforming growth factor-β, Ki-67 proliferative index, pleckstrin homology-like domain, family A, member 1 (PHLDA1) may assist in distinguishing BCC from TE.[8–11]

Tumors arising from an old scar have been reported to be squamous cell carcinoma, BCC, leiomyosarcoma, malignant melanoma, clear cell adenocarcinoma, giant cell fibroblastoma, and cutaneous lymphoma.[12–14] The development of benign tumor such as TE from a scar tissue is a rare event that has been reported by Lee et al.[15] We describe a rare case of TE arising from a chemical burn scar tissue with the concomitant finding of BCC, which to our knowledge is the first to be documented. There have been reports of TE undergoing transformation to BCC usually in the setting of multiple TEs.[8,9] Sahin et al report a case of giant solitary TE which transformed into BCC.[16] However, in the case described here, the patient is a 28-year-old male with a single, small-sized TE. The age of the patient and the single lesion at presentation are not typical findings that raise suspicions for a malignant tumor. Fortunately, the patient noticed a growth in the size of the tumor in the last 5 months which can be a clinical cue to suspect a malignant lesion. Our case is unique and important in that it presents the concomitant finding of a benign and malignant tumors arising from a chemical burn scar. Follow up at regular intervals should be considered for evaluation of recurrence or transformation into BCC.[17]

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4 Conclusion

Although development of a malignant tumor is a well-known complication of a chronic burn scar, benign tumor such as TE can also occur. Even in such cases, the concomitant development of BCC should also be considered. Follow up at regular intervals for evaluation of recurrence or transformation of TE to BCC is recommended in patients with tumors arising from chemical burn scars.

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Author contributions

Conceptualization: Sam Yong Lee.

Data curation: Dong Wan Kim.

Investigation: Seong Hwan Kim.

Methodology: Seong Hwan Kim.

Resources: Dong Wan Kim.

Supervision: Sam Yong Lee.

Writing – original draft: Seong Hwan Kim.

Writing – review & editing: Jae Ha Hwang, Kwang Seog Kim, Sam Yong Lee.

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References

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[2]. Genc S, Sirin Ugur S, Arslan IB, et al. A giant solitary trichoepithelioma originating from the auricle. Dermatol Surg 2012;38:1527–8.
[3]. Stoica LE, Dascalu RC, Patrascu V, et al. Solitary trichoepithelioma: clinical, dermatoscopic and histopathological findings. Rom J Morphol Embryol 2015;56:827–32.
[4]. Ardigo M, Zieff J, Scope A, et al. Dermoscopic and reflectance confocal microscope findings of trichoepithelioma. Dermatology 2007;215:354–8.
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[6]. PHM. Pathology of the Skin with Clinical Correlations. 1989;Philadelphia: JB Lippincott Co, 15–26.
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[10]. Poniecka AW, Alexis JB. An immunohistochemical study of basal cell carcinoma and trichoepithelioma. Am J Dermatopathol 1999;21:332–6.
[11]. Swanson PE, Fitzpatrick MM, Ritter JH, et al. Immunohistologic differential diagnosis of basal cell carcinoma, squamous cell carcinoma, and trichoepithelioma in small cutaneous biopsy specimens. J Cutan Pathol 1998;25:153–9.
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[13]. Shalin SC, Haws AL, Carter DG, et al. Clear cell adenocarcinoma arising from endometriosis in abdominal wall cesarean section scar: a case report and review of the literature. J Cutan Pathol 2012;39:1035–41.
[14]. Wallingford SC, Olsen CM, Plasmeijer E, et al. Skin cancer arising in scars: a systematic review. Dermatol Surg 2011;37:1239–44.
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[16]. Sahin MT, Demir MA, Kaya Y, et al. Inguinal keratotic Basal cell carcinoma mimicking giant solitary trichoepithelioma. J Dermatol 2003;30:395–9.
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Keywords:

basal cell carcinoma; chemical burn; trichoepithelioma

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