Incontinentia pigmenti: the development of pseudoglioma.

Brown, C A

doi:10.1136/bjo.72.6.452

Cited by 18 publications

(6 citation statements)

References 10 publications

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“…). Besides, abnormal vascular changes may start during the early postnatal weeks and progress throughout the following months (Brown ). Because of the large number of lesions and very early onset of retinal changes, we highly suggest that fundus screening should be performed as soon as the diagnosis of IP is made.…”

Section: Discussionmentioning

confidence: 99%

Incontinentia pigmenti‐associated ocular anomalies of paediatric incontinentia pigmenti patients in China

Peng

Zhang

Long³

et al. 2018

Acta Ophthalmologica

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Section: Discussionmentioning

confidence: 99%

Incontinentia pigmenti‐associated ocular anomalies of paediatric incontinentia pigmenti patients in China

Peng

Zhang

Long³

et al. 2018

Acta Ophthalmologica

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“…Mensheha-Manhart et al believed the abnormality begins at the RPE leading to retinal changes with dysplasia and resulting in retinal detachment. 5 On the other hand, primary arterial insufficiency 13 and retinal ischemia leading to the synthesis of angiogenic factor 14 was also hypothesized. Retinal changes have also been attributed to mutations in a gene on the X-chromosome leading to impairment of proteins required for cell differentiation.…”

Section: Discussionmentioning

confidence: 99%

Assessment of the Retinal Structure in Children With Incontinentia Pigmenti

Mangalesh

Chen

Tran-Viet

et al. 2017

Retina

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Purpose This report aims at expanding the current knowledge of retinal microanatomy in children with incontinentia pigmenti using hand-held spectral domain optical coherence tomography (SDOCT). Methods We reviewed OCT scans from seven children (4 weeks–13 years) obtained either in the clinic or during an examination under anesthesia. The scans were analyzed for anatomical changes in the outer and inner retina, by certified graders. Medical records were assessed for systemic findings. Results We observed abnormal retinal findings unilaterally in three children. We found inner and outer retinal thinning temporally in two participants. This thinning was present prior to and persisted after treatment. One child showed a distorted foveal contour and significant retinal thickening secondary to dense epiretinal membrane and vitreomacular traction. All other children had normal retinae. Conclusion Hand-held SDOCT imaging of the retina has brought to light additional retinal structural defects that were not previously reported or visualized via routine clinical ophthalmic examination including retinal photography. Despite a normal foveal structure and visual acuity, we identified inner and outer retinal thinning on SDOCT which may benefit from future functional assessment such as visual field testing.

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“…Nishimara et al (1980) speculated that a primary arterial insufficiency might lead to obstruction, ischaemia and proliferation of vessels. On the other hand, Brown (1988) thought that the retinal ischaemia seen in IP could be the source of an angiogenic factor. Retinal changes in IP have also been ascribed to mutations in a gene on the X-chromosome, which impairs the production of an enzyme or other protein needed for cell differentiation (Catalano et al 1990).…”

Section: Discussionmentioning

confidence: 99%

“…The vascular changes seem to start during the first postnatal weeks and progress during the following weeks or even months (Brown 1988;Krey & Laux 1974;Nishimura et al 1980). In the final stage, the retina becomes detached and included in a retrolental fibrotic mass.…”

Section: Discussionmentioning

confidence: 99%

Ocular manifestations of incontinentia pigmenti

Holmström

Thorén

2000

Acta Ophthalmologica Scandinavica

107

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ABSTRACT.Purpose: The study aimed to evaluate the ocular manifestations in patients with incontinentia pigmenti (IP). Methods: Thirty patients from different parts of Sweden participated. Orthoptic and ocular examinations were performed as well as evaluation of refraction and visual acuity. Results: Ocular manifestations, probably associated with IP, were found in 77% (23/30) of the patients. Thirteen had serious or vision-threatening eye manifestations in one eye, of whom 7 were totally blind in that eye from retinal detachments. Ten patients had minor retinal and/or corneal changes. Conclusion: Ocular lesions in patients with IP may be serious and lead to blindness because of retinal disease. Ophthalmological follow-up is essential in the neonatal period and such a programme is recommended.

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Incontinentia pigmenti: the development of pseudoglioma.

Abstract: A case is described of incontinentia pigmenti in an infant with relatively normal retinae at seven days after birth who went on to total blindness by three months. This was due to excessive neovascularisation of retinae and vitreous, leading to bilateral pseudoglioma.

Cited by 18 publications

References 10 publications

Incontinentia pigmenti‐associated ocular anomalies of paediatric incontinentia pigmenti patients in China

Incontinentia pigmenti‐associated ocular anomalies of paediatric incontinentia pigmenti patients in China

Assessment of the Retinal Structure in Children With Incontinentia Pigmenti

Ocular manifestations of incontinentia pigmenti

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