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Journal of Case Reports
Nevus Comedonicus with Trichlemmal Cyst of the Scalp: A Rare Presentation
Nandini Venkatesh1, Vishnukumar Venkatesan2, Kalpana Sridhar1, Madusudhan MR1
1Department of General Surgery, A.J. Institute of Medical Sciences, Mangalore; 2Department of Vascular Surgery, Sri Ramachandra University, Chennai, Tamil Nadu, India.
Corresponding Author:
Dr. Nandini Venkatesh
Email: nan_jc3@hotmail.com
Received: 19-FEB-2016 Accepted: 06-MAY-2016 Published Online: 30-JUN-2016
DOI: http://dx.doi.org/10.17659/01.2016.0072
Abstract
Naevus comedonicus is a developmental abnormality of the skin comprising numerous keratin-filled pits. The lesions are occasionally present at birth, but more often make their initial appearance during childhood or adolescence. We present a case of a rare presentation of nevus comedonicus with trichlemmal cyst of the scalp. The clinical presentation and the surgical management of the patient have been outlined.
Keywords : Cysts, Humans, Keratins, Nevus, Scalp.
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Introduction 

Nevus comedonicus is an uncommon skin condition due to defect in the development of hair follicles. The lesions are occasionally present at birth, but more often make their initial appearance during childhood or adolescence.

Case Report

A 41 year old gentleman, a known epileptic on medications, presented with complaints of a blackish pigmented skin lesion on the scalp for the past 15 years. It has been gradually increasing in size over the past few years and now is associated with itching. There was associated bloody discharge because of incessant scratching of the lesion. On clinical examination, there were multiple brownish-black pigmented irregular nodular skin lesions on the scalp in the occipital region which were around 13x6 cm in size and rectangular in shape. Multiple pus points were seen along with few areas of bleeding due to trauma. There were no other similar lesions noted anywhere else in the body.

    He underwent routine hematological investigations, which were within normal limits. Due to the nature of the lesion, excision biopsy was advocated. The histopathological report came back as verrucous trichelemmal cyst. He received a course of antibiotics and anti-inflammatory drugs to tide over the infection. After obtaining informed consent, he was taken up for surgery. Intraoperative wide local excision of the lesion was performed. The resulting skin defect was covered with a split skin graft from the thigh. The lesion was sent for further histopathological examination as part of institutional protocol. The histopathology report showed nevus comedonicus with trichlemmal cyst.

    Post-operative recovery was uneventful. He was discharged on post-operative day 5. The skin graft had a 100% take. He is currently on follow-up and has not developed lesions till date.


Discussion

Naevus comedonicus is a rare congenital hamartoma of the pilosebaceous unit. The first case of nevus comedonicus was described by Kofmann in 1895 [1]. The undifferentiated epithelium lining the hair follicles produces keratin that forms a laminated plug. This causes expansion within the crypts leading to the clinical appearance of groups of elevated papules with central dark comedones [2]. It is usually unilateral and commonly affects the face, neck, and trunk [3]. Involvement of scalp is quite rare and there have only a handful of case reports describing the condition [4].

    Clinically, naevus comedonicus is characterized by groups of pits filled with black keratinous plugs resembling blackheads. The intervening skin is often normal. It is usually asymptomatic but may become inflamed. Pustules, abscesses and scarring may develop and mimic acne vulgaris. Rarely, developmental defects of the central nervous system, eyes, bone and skin have been associated with naevus comedonicus [5].

    The condition can be managed conservatively with use of topical retinoids and oral isoretinoins. Large lesions can be an eyesore and can be me managed by surgical excision. The resulting defect can be closed primarily or covered with a skin graft or flap. There have been reports of using tissue expanders preoperatively in case of large lesions [6].

References
  1. Kofmann S. A case of rare localization and spreading of Comedones. Arch Dermatol Syphilol. 1895;32:177-178.
  2. Nilles M, de Viragh PA, Mossmann H, Heinrichs C, Eckert F. Naevus follicularis keratosus: clinical aspects, histology and histogenesis. Hautartz. 1992;43:205-209. ?
  3. Woods KA, Larcher VF, Harper JI. Extensive naevus comedo-nicus in a child with Alagille syndrome. Clin Exp Dermatol. 1994;19:163-164.
  4. Suarez TI, Prado A, Cordero AA. Naevus comedonicus. Med Cutan Iber Lat Am. 1985;13:371-376.
  5. Engber PB. The nevus comedonicus syndrome: case report with emphasis on associated internal manifestations.?Int J Dermatol. 1978;17:745-749.
  6. Marcus J, Esterly NB, Bauer BS. Tissue expansion in a patient with extensive naevus comedonicus. Ann Plast Surg. 1992;29:362-366. ?
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Venkatesh N, Venkatesan V, Sridhar K, MR MadusudhanNevus Comedonicus with Trichlemmal Cyst of the Scalp: A Rare Presentation.JCR 2016;6:293-295
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Venkatesh N, Venkatesan V, Sridhar K, MR MadusudhanNevus Comedonicus with Trichlemmal Cyst of the Scalp: A Rare Presentation.JCR [serial online] 2016[cited 2024 Mar 29];6:293-295. Available from: http://www.casereports.in/articles/6/2/Nevus-Comedonicus-with-Trichlemmal-Cyst-of-the-Scalp.html
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