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Is Spontaneous Disappearance of Nevus Depigmentosus Possible?

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Is Spontaneous Disappearance of Nevus Depigmentosus Possible?

Vol. 24, No. 1, 2012 109

Received December 2, 2010, Revised May 16, 2011, Accepted for publication May 23, 2011

Corresponding author: Hee Young Kang, M.D., Ph.D., Department of Dermatology, Ajou University School of Medicine, San 5 Woncheon- dong, Yeongtong-gu, Suwon 443-721, Korea. Tel: 82-31-219-5190, Fax: 82-31-219-5189, E-mail: hykang@ajou.ac.kr

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://

creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Ann Dermatol Vol. 24, No. 1, 2012 http://dx.doi.org/10.5021/ad.2012.24.1.109

LETTER TO THE EDITOR

Fig. 1. Spontaneous disappearance of hypopigmented macules and patches. (a) A 10-month-old male presented with hypo- pigmented macules and patches on his right thigh in linear configuration since birth. A diffuse bluish patch that was sus- pected to be an aberrant Mongolian spot surrounded the hypo- pigmented areas. The holes were made by punch biopsy. (b) After 6 years, complete disappearance of the previous hypo- pigmented macules and patches and the surrounding aberrant Mongolian spot was noted.

Is Spontaneous Disappearance of Nevus Depigmentosus Possible?

Dong Jun Lee, M.D., Hee Young Kang, M.D., Ph.D.

Department of Dermatology, Ajou University School of Medicine, Suwon, Korea

Dear Editor:

Nevus depigmentosus (ND) is characterized by a conge- nital hypopigmented macule or patch. The pathophysi- ology of ND is poorly understood, but likely associated with a functional defect in melanosome transfer from melanocytes to keratinocytes1. ND is known to remain generally stable in size and distribution throughout life1. Herein, we describe a case of a 6-year-old boy whose ND disappeared, suggesting the possibility of spontaneous re- solution of ND.

A 10-month-old male was brought to our clinic because of whitish patches on his right leg that had been present since his birth. Physical examination revealed hypopig- mented patches on the medial side of right thigh (Fig. 1a).

A diffuse bluish patch that was suspected to be an aber- rant Mongolian spot surrounded the hypopigmented areas.

A 2-mm punch biopsy of the hypopigmented patches and adjacent perilesional normal skin at 1 cm from margin was performed. There were no remarkable findings on H&E staining (Fig. 2a). Fontana-Masson staining showed de- creased epidermal pigmentation in the lesional skin com- pared to the perilesional normal skin (Fig. 2b). There was no difference in the number of NKI/beteb positive mela- nocytes between the lesional and perilesional normal skin (Fig. 2c). These findings were consistent with ND. The patient did not receive any treatment and was lost to

follow-up until his recent revisit 6 years later. At that time, he returned with a facial mobiliform eruption, accom- panied by fever and conjunctival injection, and was diag- nosed with erythema infectiosum. Interestingly, during physical examination, it was noted that the previous ND and the surrounding aberrant Mongolian spot had com- pletely disappeared (Fig. 1b). The patient’s parents did not recall the time of the ND disappearance.

Spontaneous resolution of ND has never been reported.

However, it should be noted that there has been no epidemiological study regarding the fate of ND. More- over, there have been several reports suggesting the possibility that the functional defect of the melanocytes in ND is reversible2,3. Bardazzi et al.2 described the devel- opment of lentigines within ND after narrow band ul-

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DJ Lee and HY Kang

110 Ann Dermatol

Fig. 2. Histological findings. (a) There were no remarkable findings on H&E staining. (b) Fontana-Masson staining showed decreased epidermal pigmentation in the lesional skin compared to the perilesional normal skin. (c) There was no difference in the number of NKI/beteb positive melanocytes between the lesional and perilesional normal skin. Original magnification was ×200.

traviolet B exposure. They suggested the possibility of par- tial activation of melanosome transfer, resulting in over- production of melanin within ND. Oiso et al.3 reported a case of acquired junctional melanocytic nevus develop- ment in ND. These cases suggest that melanocytes in ND could transform from a functionally defective stage to an active stage or shift to other forms such as nevus cells.

Interestingly, in our case, the ND and Mongolian spots were coexistent at the initial visit and both had di- sappeared at the next visit. The possible relationship bet- ween Mongolian spots and spontaneous regression of ND could be suspected.

There was no confirmative diagnosis of ND. It is important to consider the other hypopigmenting conditions, such as postinflammatory hypopigmentation or hypomelanosis of Ito4. Because the lesion was present from birth and he did not have any history of inflammation on the hypopig- mented lesion, postinflammatory hypopigmentation could be differed from our case. Hypomelanosis of Ito consists of hypopigmented zones or spots with irregular borders, whorls, patches, or linear streaks with various patterns of distribution5. It follows the lines of Blaschko that represent a peculiar pattern of distribution. In addition, hypomel- anosis of Ito usually encounter the extracutaneous mani-

festation. Reviewed in aspect of distribution of lesions and extracutaneous symptoms, we thought that hypomelanosis of Ito was less likely in our case. Therefore, considering the congenital hypopigmented macules, distribution, and other clinical manifestations, the hypopigmented macules and patches were most likely ND.

In conclusion, our case suggests the possibility that ND could disappear. An epidemiological study of ND with long term follow up is further needed to know if ND could disappear, as in our case, or is persistent throughout life.

REFERENCES

1. Kim SK, Kang HY, Lee ES, Kim YC. Clinical and histo- pathologic characteristics of nevus depigmentosus. J Am Acad Dermatol 2006;55:423-428.

2. Bardazzi F, Balestri R, Antonucci A, Spadola G. Lentigines within nevus depigmentosus: a rare collateral effect of UVB therapy? Pediatr Dermatol 2008;25:272-274.

3. Oiso N, Kawara S, Kawada A. Acquired melanocytic naevus in naevus depigmentosus. Clin Exp Dermatol 2009;34:

e311-312.

4. Lee HS, Chun YS, Hann SK. Nevus depigmentosus: clinical features and histopathologic characteristics in 67 patients. J

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Is Spontaneous Disappearance of Nevus Depigmentosus Possible?

Vol. 24, No. 1, 2012 111 Am Acad Dermatol 1999;40:21-26.

5. Assogba K, Ferlazzo E, Striano P, Calarese T, Villeneuve N, Ivanov I, et al. Heterogeneous seizure manifestations in Hy-

pomelanosis of Ito: report of four new cases and review of the literature. Neurol Sci 2010;31:9-16.

수치

Fig. 1. Spontaneous disappearance of hypopigmented macules  and patches. (a) A 10-month-old male presented with  hypo-pigmented macules and patches on his right thigh in linear  configuration since birth
Fig. 2. Histological findings. (a) There were no remarkable findings on H&E staining

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