Trichilemmoma arising in a sebaceous nevus treated with Cryo

introduction

The sebaceous nevus of Jadassohn (NSJ) is a benign congenital verrucous hamartoma composed of follicular, sebaceous and apocrine appendages, varying from a few millimeters to several centimeters in diameter. The solitary lesion is usually present at birth near the top of the scalp and face as a subtle or slightly raised lesion, which gradually thickens throughout life, becoming warty in adulthood.1.2 Various secondary neoplasms, mainly of adnexal origin, can occur in the sebaceous nevus. Removal of the TPC gene has been identified in the sebaceous nevus and may be responsible for the predisposition to the development of basal cell carcinoma and other neoplasms,3 thus requiring local excision,4 carbon dioxide (CO2) ablative laser therapy5 or argon and erbium yttrium‐aluminum‐garnet (Er:YAG).6.7

After reviewing the literature, we found no reports of NSJ being treated with cryotherapy. We report a case of NSJ associated with trichilemmoma successfully treated with cryotherapy.

Case report

A 16-year-old woman presented to the dermatology clinic at King Fahd University Hospital, Saudi Arabia, with a history of rough lesions on her upper lip from infancy. Initially, the lesion consisted of raised, skin-colored papules, which had gradually thickened over the past 3 years in a linear configuration. Dermatological examination revealed a 3 × 1 cm flesh-colored linear verrucous plaque on the left upper lip (Figure 1). Systemic examination was unremarkable. The differential diagnosis includes epidermal nevus, sebaceous nevus and common wart.

Figure 1 NSJ before treatment.

Figure 2 Continued.

Figure 2 (A) Sebaceous nevus with overlying trichilemmoma: proliferation of sebaceous glands in the dermis with overlying papillomatous projection of eosinophilic basaloid cells (H&E, ×40). (B) Sebaceous hyperplasia with vacuolated cytoplasm (H&E, ×200). (VS) Exophytic trichilemmoma papillomatous projection of clear eosinophilic cuboid palisade cells (H&E, ×100).

Skin biopsy revealed hyperkeratosis and papillomatosis with exophytic papillary projections of pale eosinophilic basaloid cells. The dermis showed a proliferation of immature sebaceous glands with vacuolated cytoplasm. Collectively, these are features of sebaceous nevus with coexisting trichilemmoma (Figure 2A–C).

A local excision was proposed to the patient because of the risk of malignant transformation. However, she opted to undergo cryotherapy rather than local excision due to cosmetic concerns about scarring that may result from the surgery. Thus, the patient underwent eight sessions of cryotherapy with liquid nitrogen in the form of three freeze-thaw cycles of 20 to 30 seconds per session over a period of 2 months, with a good aesthetic result and softening of the lesions ( Figure 3). The patient has just completed 1 year of follow-up and is currently under active surveillance for any concerning changes.

picture 3 NSJ after eight cryotherapy sessions.

Discussion

NSJ is a benign congenital hamartoma composed of follicular and adnexal appendages and epidermal hyperplasia. It usually presents at birth as a well-circumscribed yellowish lesion that can vary in size. It usually affects the scalp and the face. The lesions are usually slightly raised, with a hairless, velvety surface at an earlier age. However, as the pilosebaceous structures mature during puberty, the lesions may gradually become verrucous and thickened.1.2

Various secondary neoplasms can arise in the sebaceous nevus, which are mainly of adnexal origin. A retrospective study by Idriss and Elston reviewed 707 cases of sebaceous nevi and reported the occurrence of secondary neoplasms in 21.4% of their cases. Benign tumors accounted for 18.9% of their cases, compared to 2.5% of malignant tumor cases. The most common secondary benign tumor was trichoblastoma (34.7%), followed by syringocystadenoma papillifera (24.7%). Trichilemmomas accounted for 5.3% of neoplasms. The most common malignant neoplasm was basal cell carcinoma, which accounted for 5.3% of all neoplasms.8

There have been different approaches in the management of sebaceous nevus, especially since no consensus has been reached regarding the need for prophylactic excision.9–11 Yet Rosen et al recommended that all sebaceous lesions of the nevus be excised because of the risk of malignant transformation, although they pointed out the morbidity associated with anesthesia and excision.9 Kamyab-Hesari et al, on the other hand, recommended only close follow-up for all children with sebaceous nevus.ten Many other authors have recommended an individualized approach, especially for patients who have visible lesions that can be easily followed.11

As the benefits of surgical excision are still debatable and the risk of malignant transformation is low, we found cryotherapy to be an effective and satisfactory therapy with acceptable cosmetic results, especially in young adolescents who wish to avoid a more invasive procedure. .

Conclusion

Cryotherapy is an optional treatment modality in NSJ, especially for patients who present with visible lesions that can be easily followed, with acceptable cosmetic results. Further systemic studies should be conducted to verify the efficacy of cryotherapy in the treatment of NSJ with long-term follow-up.

Ethics and Declaration of Consent

Signed consent has been obtained from the family member for the release of case details and accompanying images. Institutional approval was not required to release case details.

Acknowledgement

We would like to thank Dr. Mohammed J. Alyousef for the histopathology images.

Author’s contributions

Both authors contributed to data analysis, writing or editing the article, agreed on the journal to which the article was submitted, gave final approval to the version to be published and agree to be responsible for all aspects of the work.

Disclosure

The authors report no conflict of interest in this work.

The references

1. Bolognia J, Cerroni L, Schaffer JV. Dermatology. 4th ed. Philadelphia: Elsevier; 2018.

2. James WD, Elston DM, Berger TG. Andrews Diseases of the Skin: Clinical Dermatology. 12th ed. Philadelphia, PA: Elsevier; 2016.

3. Xin H, Matt D, Qin JZ, Burg G, Böni R. The sebaceous nevus: a nevus with deletions of the PTCH gene. Res. Cancer. 1999;59(8):1834–1836.

4. Margulis A, Bauer BS, Corcoran JF. Surgical management of cutaneous manifestations of linear sebaceous nevus syndrome. Plast Reconstr Surg. 2003;111:104. doi:10.1097/01.PRS.0000046246.50517.A6

5. Ashinoff R. Linear sebaceous nevus of Jadassohn treated with carbon dioxide laser. Pediatr Dermatol. 1993;10:189–191.

6. Ginsbach G. Sebaceous nevus: argon laser treatment. Hautarzt. 1980;31:338–339.

7. Aithal V. Sebaceous nevus: response to erbium YAG laser ablation. Indian J Plast Surg. 2005;38:48–50. doi:10.4103/0970-0358.16497

8. Idriss MH, Elston DM. Secondary tumors associated with the sebaceous nevus of Jadassohn: a study of 707 cases. J Am Acad Dermatol. 2014;70(2):332–337. doi:10.1016/j.jaad.2013.10.004

9. Rosen H, Schmidt B, Lam HP, et al. Management of sebaceous nevus and risk of basal cell carcinoma: an 18-year review. Pediatr Dermatol. 2009;26(6):676–681. doi:10.1111/j.1525-1470.2009.00939.x

10. Kamyab-Hesari K, Seirafi H, Jahan S, et al. Sebaceous nevus: clinicopathologic study of 168 cases and review of the literature. Int J Dermatol. 2016;55(2):193–200. doi:10.1111/ijd.12845

11. Moody MN, Landau JM, Goldberg LH. Sebaceous nevus revisited. Pediatr Dermatol. 2012;29(1):15–23. doi:10.1111/j.1525-1470.2011.01562.x

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