Abstract
Moyamoya disease is a cerebrovascular disease characterized by stenosis and occlusion of the arteries of the circle of Willis, with abnormal telangiectatic collateral circulation at the base of the brain. An association between moyamoya disease and neurofibromatosis 1, a neurocutanoeus disorder, is well established in the literature. However its association with other neurocutaneous syndromes is infrequently reported. Hypomelanosis of Ito, another neurocutaneous syndrome, is characterized by macular hypopigmented skin whorls and variable neurologic involvement. Only one case study of an association between hypomelanosis of Ito and moyamoya disease has been reported in the English literature. We report a 17-year-old girl with both hypomelanosis of Ito and angiographic moyamoya disease. She presented with intractable seizures, progressive left hemiparesis, and skin manifestations of hypomelanosis of Ito. Although one might consider a coincidental association, this second case points to an association between the two disorders. Detailed neuroimaging, in particular angiography, should be considered in children with hypomelanosis of Ito and abnormal neurologic findings.
| Original language | English |
|---|---|
| Pages (from-to) | 924-926 |
| Number of pages | 3 |
| Journal | Journal of Child Neurology |
| Volume | 20 |
| Issue number | 11 |
| DOIs | |
| Publication status | Published - Nov 2005 |
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ASJC Scopus subject areas
- Clinical Neurology
- Pediatrics, Perinatology, and Child Health
Cite this
Hypomelanosis of Ito and Moyamoya disease. / Rafay, Mubeen F.; Al-Futaisi, Amna; Weiss, Shelly; Armstrong, Derek.
In: Journal of Child Neurology, Vol. 20, No. 11, 11.2005, p. 924-926.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - Hypomelanosis of Ito and Moyamoya disease
AU - Rafay, Mubeen F.
AU - Al-Futaisi, Amna
AU - Weiss, Shelly
AU - Armstrong, Derek
PY - 2005/11
Y1 - 2005/11
N2 - Moyamoya disease is a cerebrovascular disease characterized by stenosis and occlusion of the arteries of the circle of Willis, with abnormal telangiectatic collateral circulation at the base of the brain. An association between moyamoya disease and neurofibromatosis 1, a neurocutanoeus disorder, is well established in the literature. However its association with other neurocutaneous syndromes is infrequently reported. Hypomelanosis of Ito, another neurocutaneous syndrome, is characterized by macular hypopigmented skin whorls and variable neurologic involvement. Only one case study of an association between hypomelanosis of Ito and moyamoya disease has been reported in the English literature. We report a 17-year-old girl with both hypomelanosis of Ito and angiographic moyamoya disease. She presented with intractable seizures, progressive left hemiparesis, and skin manifestations of hypomelanosis of Ito. Although one might consider a coincidental association, this second case points to an association between the two disorders. Detailed neuroimaging, in particular angiography, should be considered in children with hypomelanosis of Ito and abnormal neurologic findings.
AB - Moyamoya disease is a cerebrovascular disease characterized by stenosis and occlusion of the arteries of the circle of Willis, with abnormal telangiectatic collateral circulation at the base of the brain. An association between moyamoya disease and neurofibromatosis 1, a neurocutanoeus disorder, is well established in the literature. However its association with other neurocutaneous syndromes is infrequently reported. Hypomelanosis of Ito, another neurocutaneous syndrome, is characterized by macular hypopigmented skin whorls and variable neurologic involvement. Only one case study of an association between hypomelanosis of Ito and moyamoya disease has been reported in the English literature. We report a 17-year-old girl with both hypomelanosis of Ito and angiographic moyamoya disease. She presented with intractable seizures, progressive left hemiparesis, and skin manifestations of hypomelanosis of Ito. Although one might consider a coincidental association, this second case points to an association between the two disorders. Detailed neuroimaging, in particular angiography, should be considered in children with hypomelanosis of Ito and abnormal neurologic findings.
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UR - http://www.scopus.com/inward/citedby.url?scp=31144471453&partnerID=8YFLogxK
U2 - 10.1177/08830738050200111301
DO - 10.1177/08830738050200111301
M3 - Article
C2 - 16417866
AN - SCOPUS:31144471453
VL - 20
SP - 924
EP - 926
JO - Journal of Child Neurology
JF - Journal of Child Neurology
SN - 0883-0738
IS - 11
ER -