Linear and Whorled Nevoid Hypermelanosis in a Nigerian Child: A Case Report and Literature Review
Linear and whorled nevoid hypermelanosis (LWNH) is a rare skin condition characterized by linear streaks and swirls of macular hyperpigmentation along the lines of Blaschko. The disease may occasionally have associated extracutaneous manifestations. The exact pathogenesis is unknown and no satisfactory treatment modalities for LWNH are currently available. There are few case reports of the disease in literature worldwide and none has been previously reported in Nigeria. We are reporting one such rare case of linear and whorled nevoid hypermelanosis in a 9 month old Nigerian child. We report a 9 month old female who presented with abnormal dark skin pigmentation noticed at birth suggestive of LWNH with extracutaneous presentation including microcephaly, turricephaly, anisocoria, nystagmus and delayed developmental milestones. This shows the existence of this rare disease in Nigeria. Linear and whorled nevoid hypermelanosis though uncommon, is associated with significant morbidity especially among those with extracutaneous lesions. No effective treatment is available for this disorder.
Linear, Whorled, Nevoid, Hypermelanosis, Nigerian
Mehta V, Vasanth V, Balachandran C, Mathew M. Linear and whorled nevoid hypermelanosis. Int J Dermatol 2011; 50: 491-492.
Akiyama M, Aranami A, Sasaki Y, Ebihara T, Sugiura M. Familial linear and whorled nevoid hypermelanosis. J Am Acad Dermatol 1994; 30: 831-3.
Kalter DC, Griffiths WA, Atherton DJ. Linear and whorled nevoid hypermelanosis. J Am Acad Dermatol. 1988; 19: 1037-1044.
Port M, Courniotes J, Podwal M. Zosteriform lentiginous naevus with ipsilateral rigid cavus foot. Br J Dermatol 1978; 98: 693–698.
Alimurung FM, Lapenas D, Willis I, Lang P. Zebra-like hyperpigmentation in an infant with multiple congenital defects. Arch Dermatol 1979; 115: 878–881.
Iijima S, Naito Y, Naito S, Uyeno K. Reticulate hyperpigmentation distributed in a zosteriform fashion: a new clinical type of hyperpigmentation. Br J Dermatol 1987; 117: 503–510.
Patrizi A, Di Lernia V, Varotti C. Reticulate hyperpigmentation distributed in a zosteriform fashion. Br J Dermatol 1989; 121: 280.
Bjorngren H, Holst R. Reticulate hyperpigmentation of Iijima, Naito and Uyeno. A European case. Acta Derm Venereol (Stockh) 1991; 71: 248–250.
Metta AK, Ramachandra S, Sadath N, Manupati S. Linear and whorled nevoid hypermelanosis in three successive generations. Indian J Dermatol Venereol Leprol. 2011; 77: 403.
Quecedo E, Febrer I, Aliaga A. Linear and whorled nevoid hypermelanosis. A spectrum of pigmentary disorders. Pediatr Dermatol. 1997; 14: 247-248.
Di Lernia V. Linear and whorled hypermelanosis. Pediatr Dermatol. 2007; 24: 205-210.
Ertam I, Turk BG, Urkmez A, Ozdemir F. Linear and whorled nevoid hypermelanosis: Dermatoscopic features. J Am Acad Dermatol 2009; 60: 328-331.
Harre J, Millikan LE. Linear and whorled pigmentation. Int J Dermatol 1994; 33: 529- 37.
Nehal KS, Pebenito R, Orlow SJ. Analysis of 54 cases of hypopigmentation and hyperpigmentation along the lines of Blaschko. Arch Dermatol 1996; 132: 1167-1170.
Schepis C, Siragusa M, Alberti A, Romano C. Linear and whorled nevoid hypermelanosis in a boy with mental retardation and congenital defects. Int J Dermatol 1996; 35: 654-655.
Megarbane A, Vabres P, Slaba S, Smahi A, Loeys B, Okais N. Linear and whorled nevoid hypermelanosis with bilateral giant cerebral aneuysms. Am J Med Genet 2002; 112: 95-98.
Yuksek J, Sezer E, Erbi AH. linear and whorled hypermelanosis. Dermatol Online J 2007; 13: 23.