Incontinentia pigmenti

Incontinentia pigmenti (IP) is a rare X-linked dominant multi-systemic ectodermal dysplasia usually lethal in males and presenting neonatally in females with a bullous rash along Blashko's lines (BL) followed by verrucous plaques evolving over time to hyperpigmented swirling patterns. It is further characterized by teeth abnormalities, alopecia, nail dystrophy and affects occasionally the retina and the central nervous system (CNS).

Retinopathy

Any noninflammatory disease of the retina. This nonspecific term is retained here because of its wide use in the literature, but if possible new annotations should indicate the precise type of retinal abnormality.


Total: 17

                       


(per page)
PMID (PMCID)
30768227
 FEMALE Infant
Revascularization After Intravitreal Bevacizumab and Laser Therapy of Bilateral Retinal Vascular Occlusions in Incontinentia Pigmenti (Bloch-Sulzberger Syndrome).
Cernichiaro-Espinosa LA, Patel NA, Bauer MS, Negron CI, Fallas B, Pogrebniak A, Berrocal AM.
Ophthalmic Surg Lasers Imaging Retina. 2019;50(2):e33-e37.
No consensus exists for the treatment of retinopathy in incontinentia pigmenti (IP).
27472514
 FEMALE
SUCCESSFUL TREATMENT OF REFRACTORY PROLIFERATIVE RETINOPATHY OF INCONTINENTIA PIGMENTI BY INTRAVITREAL RANIBIZUMAB AS ADJUNCT THERAPY IN A 4-YEAR-OLD CHILD.
Ho M, Yip WWK, Chan VCK, Young AL.
Retin Cases Brief Rep. 2017;11(4):352-355.
Intravitreal ranibizumab can be considered as an adjunct therapy in proliferative retinopathy secondary to incontinentia pigmenti when monotherapy with panretinal photocoagulation fails.
25695859
 FEMALE Infant
Extended Follow-up of Treated and Untreated Retinopathy in Incontinentia Pigmenti: Analysis of Peripheral Vascular Changes and Incidence of Retinal Detachment.
Chen CJ, Han IC, Tian J, Munoz B, Goldberg MF.
JAMA Ophthalmol. 2015;133(5):542-8.
Extended Follow-up of Treated and Untreated Retinopathy in Incontinentia Pigmenti: Analysis of Peripheral Vascular Changes and Incidence of Retinal Detachment.
25313552
 FEMALE Infant, Newborn
Intravitreal bevacizumab for incontinentia pigmenti.
Shah PK, Bachu S, Narendran V, Kalpana N, David J, Srinivas CR.
J Pediatr Ophthalmol Strabismus. 2013;50 Online:e52-4.
Two eyes of two siblings affected with incontinentia pigmenti having severe proliferative retinopathy are reported.
19373689
 MALE Infant, Newborn
Prospects for treatment of pediatric vitreoretinal diseases with vascular endothelial growth factor inhibition.
Lin KL, Hirose T, Kroll AJ, Lou PL, Ryan EA.
Semin Ophthalmol. 2009;24(2):70-6.
This paper will examine the current evidence for use of anti-VEGF therapy in a number of pediatric vitreoretinal disorders and describe a case of anti-VEGF therapy in retinopathy of incontinentia pigmenti.
19373689
 MALE Infant, Newborn
Prospects for treatment of pediatric vitreoretinal diseases with vascular endothelial growth factor inhibition.
Lin KL, Hirose T, Kroll AJ, Lou PL, Ryan EA.
Semin Ophthalmol. 2009;24(2):70-6.
While angiogenesis inhibitors are already widely used to treat retinal disease in adults, only limited reports are currently available for the use of anti-VEGF in pediatric vitreoretinal diseases such as retinopathy of prematurity, Coats' disease, familial exudative vitreoretinopathy and retinopathy of incontinentia pigmenti.
17976725
 FEMALE Middle Aged
Histopathologic and trypsin digestion studies of the retina in incontinentia pigmenti.
Bell WR, Green WR, Goldberg MF.
Ophthalmology. 2008;115(5):893-7.
To report the ocular histopathologic features of a 55-year-old patient with incontinentia pigmenti retinopathy.
17976725
 FEMALE Middle Aged
Histopathologic and trypsin digestion studies of the retina in incontinentia pigmenti.
Bell WR, Green WR, Goldberg MF.
Ophthalmology. 2008;115(5):893-7.
A 55-year-old patient with incontinentia pigmenti retinopathy.
15165204
 MALE Child
The importance of screening for sight-threatening retinopathy in incontinentia pigmenti.
Wong GA, Willoughby CE, Parslew R, Kaye SB.
Pediatr Dermatol. 2004;21(3):242-5.
The importance of screening for sight-threatening retinopathy in incontinentia pigmenti.
14566654
 FEMALE
Retinopathy of incontinentia pigmenti: a case report with thirteen years follow-up.
Cates CA, Dandekar SS, Flanagan DW, Moore AT.
Ophthalmic Genet. 2003;24(4):247-52.
Retinopathy of incontinentia pigmenti: a case report with thirteen years follow-up.
14566654
 FEMALE
Retinopathy of incontinentia pigmenti: a case report with thirteen years follow-up.
Cates CA, Dandekar SS, Flanagan DW, Moore AT.
Ophthalmic Genet. 2003;24(4):247-52.
We describe a young girl with incontinentia pigmenti in whom the retinopathy has shown a fluctuating but stable course over 13 years without treatment.
11507587
 FEMALE Infant, Newborn
Laser photocoagulation in preproliferative retinopathy of incontinentia pigmenti.
Nguyen JK, Brady-Mccreery KM.
J AAPOS. 2001;5(4):258-9.
Laser photocoagulation in preproliferative retinopathy of incontinentia pigmenti.
7618473
 FEMALE Infant, Newborn
Retinopathy in incontinentia pigmenti: a neonatal case report.
Fu LW, Soong WJ, Tsai SC, Hwang B.
Zhonghua Min Guo Xiao Er Ke Yi Xue Hui Za Zhi. 1995;36(3):210-3.
Retinopathy in incontinentia pigmenti: a neonatal case report.
1942603
 FEMALE Infant, Newborn
Cryotherapy for retinopathy of incontinentia pigmenti.
Yu YS, Cho BJ.
Korean J Ophthalmol. 1991;5(1):47-50.
Cryotherapy for retinopathy of incontinentia pigmenti.
2378846
 FEMALE Infant
Early diagnosis of the retinopathy of incontinentia pigmenti: successful treatment by cryotherapy.
Rahi J, Hungerford J.
Br J Ophthalmol. 1990;74(6):377-9.
Early diagnosis of the retinopathy of incontinentia pigmenti: successful treatment by cryotherapy.
2378846
 FEMALE Infant
Early diagnosis of the retinopathy of incontinentia pigmenti: successful treatment by cryotherapy.
Rahi J, Hungerford J.
Br J Ophthalmol. 1990;74(6):377-9.
Prospective examination of the ocular fundi was carried out in the newborn female sibling of a girl with incontinentia pigmenti and bilateral retinopathy.
6184762
 FEMALE Child
Proliferative retinopathy associated with incontinentia pigmenti.
Nix RR, Apple DJ.
Retina. 1981;1(3):156-61.
Proliferative retinopathy associated with incontinentia pigmenti.