Hypomelanosis of Ito with neurological involvement
Keywords:
Hypomelanosis of Ito, Incontinentia Pigmenti Achromicans, neurocutaneous syndrome, white matter atrophyAbstract
Hypomelanosis of Ito is also known as Incontinentia pigmenti achromians because the distribution of depigmented lesions is the negative image of hyperpigmented streaks of Incontinentia pigmenti. We report an infant of 9 months with hypopigmented macules along blaschkoid lines, developmental delay, macrocephaly and seizures. MRI brain revealed white matter atrophy and prominent temporal horns of lateral ventricles. This case report highlights the coexistence of Hypomelanosis of Ito with neurological involvement and thus the need to differentiate it from other similar conditions presenting with Blaschkoid dyspigmentation.References
Pavone P, Praticò AD, Ruggieri M, Falsaperla R. Hypomelanosis of Ito: a round on the frequency and type of epileptic complications. Neurol Sci 2015; 36: 1173.
Geetha K, Priya G L, Rameshwari T. Hypomelanosis of Ito associated with cafe-au-lait spot and angiomatous nevi. Indian J Paediatr Dermatol 2015; 16: 221-3.
Pascual-Castroviejo I, Lopez-Rodrigucz L, de la Cruz Medina M, Salamanca-Maesso C, Roche Herrero C; Hypomelanosis of Ito, Neurological complications in 34 cases; Can J Neurol Sci. 1998; 5: 124-9.
Nehal KS, PeBenito R, Orlow SJ. Analysis of 54 cases of hypopigmentation and hyperpigmentation along the lines of Blaschko. Arch Dermatol. 1996; 132(10):1167-70.
Ross DL, Liwnicz BH, Chun RW, Gilbert E. Hypomelanosis of Ito (incontinentia pigmenti achromians)--a clinicopathologic study: macrocephaly and gray matter heterotopias. Neurology 1982; 32: 1013-6.
Saha A, Seth J, Mukherjee S, Basu S. Hypomelanosis of Ito with brain abscess: An incidental association or consequence of underlying structural defect. Indian J Paediatr Dermatol 2015; 16: 186-8.
Trägårdh M, Thomsen CR, Thorninger R, Møller-Madsen B. Hypomelanosis of Ito presenting with pediatric orthopedic issues: a case report. J Med Case Rep 2014; 8:156.
El-Sawy T, He L, Chiang MF, Anyane-Yeboa K, Morel KD, Folberg R, et al. Retinoblastoma presenting in a child with hypomelanosis of Ito. Open Ophthalmol J. 2011; 5: 55-8.
Ruiz-Maldonado R, Toussaint S, Tamayo L, Laterza A, del Castillo V. Hypomelanosis of Ito: diagnostic criteria and report of 41 cases. Pediatr Dermatol 1992; 9: 1-10.
Cohen J 3rd, Shahrokh K, Cohen B. Analysis of 36 cases of Blaschkoid dyspigmentation: reading between the lines of Blaschko. Pediatr Dermatol 2014; 31: 471-6.
