|Year : 1990 | Volume
| Issue : 6 | Page : 448-449
Nevus unilateralis comedonicus
B Mohan Gharpuray, Sharad Mutalik
B Mohan Gharpuray
Source of Support: None, Conflict of Interest: None
A five months old female child with a rare condition, nevus unilateralis comedonicus is reported. Treatment with topical tretinoin cream resulted in partial improvement.
Keywords: Nevus unilateralis comedonicus
|How to cite this article:|
Gharpuray B M, Mutalik S. Nevus unilateralis comedonicus. Indian J Dermatol Venereol Leprol 1990;56:448-9
Nevus unilateralis comedonicus is a rare condition; approximately 100 cases have been reported There are many synonyms for this condition, viz. nevus acneiformis, systematized sebaceous gland nevus, zoniform nevus with comedones.
There is no sexual or racial predisposition. Approximately half the cases are present at birth; and other half start before age of 15. No familial cases have been reported. This disorder is recognized as a developmental or nevoid condition, similar to the more common epidermal nevus. It is distinguished from linear epidermal nevi only by its peculiar tendency to form follicular keratin plugs or comedones.1 The basic defect is a developmental abnormality in the formation of hairs by the primary epithelial germ.
There is no preference to site. It usually occurs as a single lesion, although occasionally it shows bilateral linear lesions or randomly distributed rather than linear lesions. There are groups of 20 to 50 comedones which tend to grow as the child matures. The large comedones extend above the surface of surrounding skin and give a `Nutmeg crater' feeling. Comedones can be shelled out freely, leaving craters in the normal skin. In about half the cases there is secondary infection of the keratin cysts, leading to suppuration and residual scarring. An association with Epidermolytic hyperkeratosis is also documented.
In histopathological examination; each comedo is represented by a wide and deep invagination. These invaginations resemble dilated hair follicles. One occasionally finds at their base one or even several hair shafts. Also one or two small sebaceous gland lobules may be seen opening into the lower pole of the invaginations. Secondary inflammatory changes with acute or chronic cellular infiltrates around the cyst also occur.
Complications are very rare. Suppuration and residual scarring as a result of secondary infection can be prevented by timely and appropriated use of antibiotics. Development of basal cell tumour has been reported in one case.
| Case Report|| |
A five months old female child was seen for the lesions over the left lower extremity; present since birth. The lesion was asymptomatic except for minor oozing at few places. This was the first child born of nonconsanguineous marriage.
Dermatological examination revealed groups of comedones and pitted lesions; occupying left thigh and leg [Figure - 1], arranged in a sharply demarcated band like fashion. Pits were filled with keratin at places and few pustules were also seen. Comedones could be shelled out easily. Regional lymph nodes were not enlarged. The child was otherwise healthy.
Skin biopsy was not done as the parents refused to give consent. From the typical clinical presentation we diagnosed this case as `Nevus unilateralis comedonicus'. Secondary infection was treated with erythromycin suspension (50 mg/kg/day) orally and fusidic acid cream (2% cream B.D.) topically. After controlling the secondary infection, we switched over to Tretinoin cream (0.025% H.S.) topically which resulted in partial improvement; with flattening of the comedones. [Figure - 2]
| Comments|| |
A rare disorder is presented which poses a purely cosmetic problem and for which no satisfactory treatment is available. Our patient has shown partial improvement with Tretinoin, but the defect being developmental, question about maintenance therapy remains unanswered. Role of oral etretinate in this condition is also not documented. Newer and more potent retinoids which can be applied locally may help in this disorder.
| References|| |
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|7.||Barsky S et al : Nevus comedonicus with epidermolytic hyperkeratosis, Arch Dermatol, 1981, 117 : 86-88. |
|8.||Lever WF and Shaumberg Lever G : Histopathology of the skin, 5th ed, JB Lippincott Company, Philadelphia, 1975; p 452. |
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[Figure - 1], [Figure - 2]