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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 17  |  Issue : 4  |  Page : 277-279

Nevus comedonicus associated with epidermoid cyst


1 Department of Otorhinolaryngology-Head and Neck Surgery, All Institute of Medical Sciences, Raipur, Chhattisgarh, India
2 Department of Dermatology, Venereology and Leprology, All Institute of Medical Sciences, Raipur, Chhattisgarh, India
3 Department of Pathology, All Institute of Medical Sciences, Raipur, Chhattisgarh, India

Date of Web Publication7-Oct-2016

Correspondence Address:
Neel Prabha
Department of Dermatology, Venereology and Leprology, All India Institute of Medical Sciences, Raipur, Chhattisgarh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2319-7250.184329

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  Abstract 

Nevus comedonicus (NC) is a type of an epidermal nevus which was first described in 1895 by Kofmann. Clinically, it is characterized by linear lesion comprising numerous dilated follicular openings with keratinous plugs resembling classical comedones, seen mainly on the head and neck area followed by trunk and upper arm. Treatment options of NC include topical therapy, laser, and surgery. NC associated with epidermoid cyst is rare. We report here a case of NC affecting middle part of the neck over left sternocleidomastoid muscle with epidermoid cysts which was surgically treated.

Keywords: Comedones, epidermoid cyst, nevus comedonicus


How to cite this article:
Arora R, Nagarkar NM, Prabha N, Hussain N. Nevus comedonicus associated with epidermoid cyst. Indian J Paediatr Dermatol 2016;17:277-9

How to cite this URL:
Arora R, Nagarkar NM, Prabha N, Hussain N. Nevus comedonicus associated with epidermoid cyst. Indian J Paediatr Dermatol [serial online] 2016 [cited 2019 Dec 16];17:277-9. Available from: http://www.ijpd.in/text.asp?2016/17/4/277/184329


  Introduction Top


Nevus comedonicus (NC) is a type of an epidermal nevus which was first described in 1895 by Kofmann.[1] Clinically, it is characterized by linear lesion comprising numerous dilated follicular openings with keratinous plugs resembling classical comedones, seen mainly on the head and neck area followed by trunk and upper arm. Cases may also occur at other sites including palms, soles,[2],[3],[4],[5] and glans penis.[6] They may be present at birth or develop throughout adult life. The prevalence of NC has been estimated from 1 in 45,000 to 1 in 100,000, with no gender or racial preference.[7],[8] Epidermoid cyst, on the other hand, is common and most frequently affects young- and middle-aged person. It results either from inflammation of pilosebaceous follicles or from deep implantation of a fragment of epidermis by a blunt penetrating injury or due to developmental defect. NC associated with epidermoid cyst is rare.[9],[10],[11],[12] We report here a case of NC affecting middle part of the neck over left sternocleidomastoid muscle with epidermoid cysts which was surgically treated.


  Case Report Top


A 4-year-old male child presented with comedo-like lesions over the left side of the neck since birth. He developed nodular swellings scattered within the area of these comedone-like lesions for the last 1-year. The lesions were asymptomatic. There was no family history of similar complaints. Cutaneous examination revealed groups of dilated follicular openings filled with black, hard plugs distributed over the left side of neck over sternocleidomastoid muscle 2 cm below angle of mandible in 5 cm × 2 cm area with multiple nodulocystic swellings of the size 0.2–0.4 cm in diameter with scarring and fibrous tracts scattered in between [Figure 1]. General physical and systemic examination was normal. Routine laboratory investigations including complete blood count, blood chemistry, and urinalysis were within normal limits. Contrast-enhanced computed tomography neck showed cutaneous and subcutaneous lesion overlying the left sternocleidomastoid muscle. Diagnosis of NC was made on the basis of history and clinical examination. Under general anesthesia, the patient underwent a surgical procedure in which the lesion was resected using one elliptical incision. For excising the NC, the incision line was drawn to take wide margins to avoid recurrence. External jugular vein was ligated, and lesion was lying superficial to sternocleidomastoid muscle. Lesion was excised in toto without rupture; this procedure prevented the possibility of recurrence, and the resection was successfully completed. The sites of resection were primarily sutured.
Figure 1: Multiple comedones like lesions, nodulocystic swellings, and fibrous tracts scattered in between over left side of neck

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The histopathology of the comedo-like lesions revealed dilated hair follicles containing keratinous debris, consistent with NC [Figure 2]. The interfollicular epidermis was normal. Sections from nodulocystic swelling showed cyst wall lined by stratified squamous epithelium with granular layer and lamellated keratin, consistent with epidermoid cyst [Figure 3]. Accordingly, a diagnosis of NC with epidermoid cysts was rendered. The patient had no recurrences or other complications during a 3-month postoperative follow-up period [Figure 4].
Figure 2: The histopathology of the comedo-like lesions revealed dilated hair follicles containing keratinous debris (H and E, ×10)

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Figure 3: Cyst lined by stratified squamous epithelium (H and E, ×10)

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Figure 4: Postoperative clinical photo after 3 months showed a mild depressed and erythematous scar without recurrence

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  Discussion Top


NC can present in two clinical entities. The first is the asymptomatic comedo-like lesions and the second is the presence of cysts, scars, fistulas, and abscesses due to inflammation and infection of the comedo-like lesions. The case described here is of the second group. NC may be linear, interrupted, unilateral, bilateral, along the lines of Blaschko or segmental. The characteristic histological feature of NC is an aggregation of deep, wide invaginations of acanthotic epidermis, filled with concentric lamellae of keratin.[7] The interfollicular epidermis may be histologically normal, acanthotic, hyperkeratotic,[13] and/or epidermolytic.[14] There have been occasional reports of associated developmental anomalies including ipsilateral cataract,[15] epidermal nevus,[13] and trichilemmal cyst.[16] Engbers [17] reported NC syndrome in which NC was associated with other skin lesions or internal abnormalities. NC syndrome belongs to the large category of epidermal nevus syndromes in which epidermal nevus is associated with abnormalities of other body organs such as neurological deficits (epilepsy and electrocardiogram abnormalities), skeletal abnormalities (scoliosis and spina bifida), and eye problems (congenital cataracts).[17] Physical examination and other laboratory test results indicated that the current case was not NC syndrome. The present case had epidermoid cysts along the distribution of NC, which is a rare association. The histopathological findings of cystic lesions in this case were almost identical to epidermoid cyst. Differential diagnosis of NC is porokeratotic eccrine ostial, dermal duct nevus, atrophoderma vermiculata, and keratosis pilaris atrophicans.

Being benign, NC does not require aggressive treatment, except for esthetic reasons or in complicated cases. However, for those with the inflamed entity, proper treatment is needed to prevent complications of the recurrent infection and inflammation. Conservative options include extraction of comedones, and the use of topical agents such as retinoic acid, urea, tretinoin, ammonium lactate lotion, tacalcitol, tazarotene, and calcipotriene. Conservative treatment may improve cosmesis in some patients but cannot prevent recurrence. Erbium-YAG laser and carbon dioxide lasers have also been reported to be successful.[18],[19] Localized NC can be removed by surgical excision followed by primary suture with good esthetic results. Cases with larger affected areas require staged excision or skin grafting. In some cases, reconstruction using a tissue expander would be helpful.[20] We treated our patient with surgical excision.


  Conclusion Top


Here, we report a case of NC associated with epidermoid cysts with successful treatment with surgical excision.

Financial Support and Sponsorship

Nil.

Conflicts of Interest

There are no conflicts of interest.

 
  References Top

1.
Kofmann S. A case of rare localization and distribution of comedones. Arch Dermatol Syphilol 1895;32:177-8.  Back to cited text no. 1
    
2.
Harper KE, Spielvogel RL. Nevus comedonicus of the palm and wrist. Case report with review of five previously reported cases. J Am Acad Dermatol 1985;12(1 Pt 2):185-8.  Back to cited text no. 2
    
3.
Leppard B, Marks R. Comedone naevus. A report of nine cases. Trans St Johns Hosp Dermatol Soc 1973;59:45-51.  Back to cited text no. 3
    
4.
Wood MG, Thew MA. Nevus comedonicus. A case with palmar involvement and review of the literature. Arch Dermatol 1968;98:111-6.  Back to cited text no. 4
    
5.
Marsden RA, Fleming K, Dawber RP. Comedo naevus of the palm – A sweat duct naevus? Br J Dermatol 1979;101:717-22.  Back to cited text no. 5
    
6.
Abdel-Aal H, Abdel-Aziz AM. Nevus comedonicus. Report of three cases localized on glans penis. Acta Derm Venereol 1975;55:78-80.  Back to cited text no. 6
    
7.
Nabai H, Mehregan AH. Nevus comedonicus. A review of the literature and report of twelve cases. Acta Derm Venereol 1973;53:71-4.  Back to cited text no. 7
    
8.
Happle R. The group of epidermal nevus syndromes part I. Well defined phenotypes. J Am Acad Dermatol 2010;63:1-22.  Back to cited text no. 8
    
9.
Mathur DK, Bhargava P, Agarwal US, Mathur SK, Singh R, Bhargava R. Naevus comedonicus associated with epidermoid cyst. Indian J Dermatol Venereol Leprol 1996;62:250-1.  Back to cited text no. 9
[PUBMED]  Medknow Journal  
10.
Kim YJ, Hong CY, Lee JR. Nevus comedonicus with multiple cyst. J Korean Cleft Palate Craniofac Assoc 2009;10:135-7.  Back to cited text no. 10
    
11.
Chhabra N, Pandhi D, Verma P, Sharma S. Inflammatory nevus comedonicus with epidermoid cyst. Indian J Dermatol 2014;59:422.  Back to cited text no. 11
[PUBMED]  Medknow Journal  
12.
Jeong HS, Lee HK, Lee SH, Kim HS, Yi SY. Multiple large cysts arising from nevus comedonicus. Arch Plast Surg 2012;39:63-6.  Back to cited text no. 12
    
13.
Kim SC, Kang WH. Nevus comedonicus associated with epidermal nevus. J Am Acad Dermatol 1989;21 (5 Pt 2):1085-8.  Back to cited text no. 13
    
14.
Aloi FG, Molinero A. Nevus comedonicus with epidermolytic hyperkeratosis. Dermatologica 1987;174:140-3.  Back to cited text no. 14
    
15.
Suite M, Mahabir R. Unilateral comedo nevus associated with ipsilateral congenital cataract. Int J Dermatol 1994;33:390-1.  Back to cited text no. 15
    
16.
Leppard BJ. Trichilemmal cysts arising in an extensive comedo naevus. Br J Dermatol 1977;96:545-8.  Back to cited text no. 16
    
17.
Engber PB. The nevus comedonicus syndrome: A case report with emphasis on associated internal manifestations. Int J Dermatol 1978;17:745-9.  Back to cited text no. 17
    
18.
Caers SJ, Van der Geer S, Beverdam EG, Krekels GA, Ostertag JU. Successful treatment of nevus comedonicus with the use of the erbium yag laser. J Eur Acad Dermatol Venereol 2008;22:375-7.  Back to cited text no. 18
    
19.
Sardana K, Garg VK. Successful treatment of nevus comedonicus with ultrapulse CO2 laser. Indian J Dermatol Venereol Leprol 2009;75:534-5.  Back to cited text no. 19
[PUBMED]  Medknow Journal  
20.
Marcus J, Esterly NB, Bauer BS. Tissue expansion in a patient with extensive nevus comedonicus. Ann Plast Surg 1992;29:362-6.  Back to cited text no. 20
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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