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Zebra-like Hyperpigmentation in an Infant With Multiple Congenital Defects

Zebra-like Hyperpigmentation in an Infant With Multiple Congenital Defects Abstract An unusual pattern of zebra-like hyperpigmentation was observed in a black male infant with congenital defects that included an atrial septal defect, dextrocardia, auricular atresia, deafness, and growth retardation. The hyperpigmentation was strikingly linear and symmetrical and followed a dermatomal distribution, with involvement of the trunk and extremities. Results of histological, histochemical, and electron microscopic studies were normal except for an increase in the number of melanocytes in the bands of hyperpigmentation. This pigmentary anomaly appears to be transient and has faded to 50% of its original intensity. A review of the literature has not disclosed any similar cases. (Arch Dermatol 115:878-881, 1979) References 1. Staricco RJ, Pinkus H: Quantitative and qualitative data on the pigment cells of adult human epidermis . J Invest Dermatol 28:33-45, 1957. 2. Hori Y, Toda K, Pathak M, et al: A fine structure study of human epidermal melanosome complex and its acid phosphatase activity . J Ultrastruct Res 25:109-120, 1968.Crossref 3. Carney RG: Incontinentia pigmenti: A world statistical analysis . Arch Dermatol 112:535-542, 1976.Crossref 4. Morgan JD: Incontinentia pigmenti (Bloch-Sulzberger syndrome) . Am J Dis Child 122:294-300, 1971.Crossref 5. Curth HO, Warburton D: The genetics of incontinentia pigmenti . Arch Dermatol 92:229-235, 1965.Crossref 6. Whiting DA: Naegeli's reticular pigmented dermatosis . Br J Dermatol 85( (suppl 7) ):71-72, 1971.Crossref 7. Chernosky ME, Anderson DE, Chang JP, et al: Familial progressive hyperpigmentation . Arch Dermatol 103:581-598, 1971.Crossref 8. Furuya T, Mishima Y: Progressive pigmentary disorder in Japanese child . Arch Dermatol 86:412-418, 1962.Crossref 9. Gibbs RC: Unusual striped hyperpigmentation of the torso . Arch Dermatol 95:385-386, 1967.Crossref 10. Ment L, Alper J, Sirota RL, et al: Infant with abnormal pigmentation, malformations, and immune deficiency . Arch Dermatol 114:1043-1044, 1978.Crossref 11. Esterly NB, Solomon LM: Neonatal dermatology: III. Pigmentary lesions and hemangiomas . J Pediatr 81:1003-1013, 1972.Crossref 12. Osmundsen PE: Pigmented contact dermatitis . Br J Dermatol 83:296-300, 1970.Crossref 13. Edidin DB, Esterly NB, Bamzai AK, et al: Chondrodysplasia punctata (Conradi-Hunnerman syndrome) . Arch Dermatol 113:1431-1434, 1977.Crossref 14. Konigsmark BW: Hereditary childhood hearing loss and integumentary system disease . J Pediatr 80:909-919, 1972.Crossref 15. Schnyder UW: Genetics and dermatology . Dermatologica 150:129-135, 1975.Crossref 16. Nordling CO: A new theory on cancer-inducing mechanism . Br J Cancer 7:68-72, 1953.Crossref 17. Nicholls EM: Genetic susceptibility and somatic mutation in the production of freckles, birthmarks and moles . Lancet 1:71-73, 1978. 18. Whimster LW: An experimental approach to the problem of spottiness . Br J Dermatol 79:397-420, 1965. 19. Selmanovitz VJ, Krivo JM: Pigmentary demarcation lines: Comparison of Negroes and Japanese . Br J Dermatol 93:371-377, 1975.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Dermatology American Medical Association

Zebra-like Hyperpigmentation in an Infant With Multiple Congenital Defects

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Publisher
American Medical Association
Copyright
Copyright © 1979 American Medical Association. All Rights Reserved.
ISSN
0003-987X
eISSN
1538-3652
DOI
10.1001/archderm.1979.04010070054026
Publisher site
See Article on Publisher Site

Abstract

Abstract An unusual pattern of zebra-like hyperpigmentation was observed in a black male infant with congenital defects that included an atrial septal defect, dextrocardia, auricular atresia, deafness, and growth retardation. The hyperpigmentation was strikingly linear and symmetrical and followed a dermatomal distribution, with involvement of the trunk and extremities. Results of histological, histochemical, and electron microscopic studies were normal except for an increase in the number of melanocytes in the bands of hyperpigmentation. This pigmentary anomaly appears to be transient and has faded to 50% of its original intensity. A review of the literature has not disclosed any similar cases. (Arch Dermatol 115:878-881, 1979) References 1. Staricco RJ, Pinkus H: Quantitative and qualitative data on the pigment cells of adult human epidermis . J Invest Dermatol 28:33-45, 1957. 2. Hori Y, Toda K, Pathak M, et al: A fine structure study of human epidermal melanosome complex and its acid phosphatase activity . J Ultrastruct Res 25:109-120, 1968.Crossref 3. Carney RG: Incontinentia pigmenti: A world statistical analysis . Arch Dermatol 112:535-542, 1976.Crossref 4. Morgan JD: Incontinentia pigmenti (Bloch-Sulzberger syndrome) . Am J Dis Child 122:294-300, 1971.Crossref 5. Curth HO, Warburton D: The genetics of incontinentia pigmenti . Arch Dermatol 92:229-235, 1965.Crossref 6. Whiting DA: Naegeli's reticular pigmented dermatosis . Br J Dermatol 85( (suppl 7) ):71-72, 1971.Crossref 7. Chernosky ME, Anderson DE, Chang JP, et al: Familial progressive hyperpigmentation . Arch Dermatol 103:581-598, 1971.Crossref 8. Furuya T, Mishima Y: Progressive pigmentary disorder in Japanese child . Arch Dermatol 86:412-418, 1962.Crossref 9. Gibbs RC: Unusual striped hyperpigmentation of the torso . Arch Dermatol 95:385-386, 1967.Crossref 10. Ment L, Alper J, Sirota RL, et al: Infant with abnormal pigmentation, malformations, and immune deficiency . Arch Dermatol 114:1043-1044, 1978.Crossref 11. Esterly NB, Solomon LM: Neonatal dermatology: III. Pigmentary lesions and hemangiomas . J Pediatr 81:1003-1013, 1972.Crossref 12. Osmundsen PE: Pigmented contact dermatitis . Br J Dermatol 83:296-300, 1970.Crossref 13. Edidin DB, Esterly NB, Bamzai AK, et al: Chondrodysplasia punctata (Conradi-Hunnerman syndrome) . Arch Dermatol 113:1431-1434, 1977.Crossref 14. Konigsmark BW: Hereditary childhood hearing loss and integumentary system disease . J Pediatr 80:909-919, 1972.Crossref 15. Schnyder UW: Genetics and dermatology . Dermatologica 150:129-135, 1975.Crossref 16. Nordling CO: A new theory on cancer-inducing mechanism . Br J Cancer 7:68-72, 1953.Crossref 17. Nicholls EM: Genetic susceptibility and somatic mutation in the production of freckles, birthmarks and moles . Lancet 1:71-73, 1978. 18. Whimster LW: An experimental approach to the problem of spottiness . Br J Dermatol 79:397-420, 1965. 19. Selmanovitz VJ, Krivo JM: Pigmentary demarcation lines: Comparison of Negroes and Japanese . Br J Dermatol 93:371-377, 1975.Crossref

Journal

Archives of DermatologyAmerican Medical Association

Published: Jul 1, 1979

References