Lens-sparing vitrectomy for pediatric tractional retinal detachment in homozygous protein C deficiency

Purpose: To describe the ophthalmic presentation and management of a male infant with homozygous protein C deficiency (PCD). Methods: Case report. Observation: An 8-week-old male infant presented with bilateral vitreous hemorrhage and tractional retinal detachment in the right eye. Work up revealed...

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Main Authors: Moath A. Alowairdhi, Mohammed I. Alkhodair, Alwaleed M. Alsulaiman, Sulaiman M. Alsulaiman
Format: Article
Language:English
Published: Elsevier 2024-12-01
Series:American Journal of Ophthalmology Case Reports
Subjects:
Online Access:http://www.sciencedirect.com/science/article/pii/S2451993624002160
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author Moath A. Alowairdhi
Mohammed I. Alkhodair
Alwaleed M. Alsulaiman
Sulaiman M. Alsulaiman
author_facet Moath A. Alowairdhi
Mohammed I. Alkhodair
Alwaleed M. Alsulaiman
Sulaiman M. Alsulaiman
author_sort Moath A. Alowairdhi
collection DOAJ
description Purpose: To describe the ophthalmic presentation and management of a male infant with homozygous protein C deficiency (PCD). Methods: Case report. Observation: An 8-week-old male infant presented with bilateral vitreous hemorrhage and tractional retinal detachment in the right eye. Work up revealed low protein C levels. Whole exome sequencing showed a homozygous likely pathogenic variant in PROC gene. The management included laser photocoagulation to both eyes followed by lens-sparing vitrectomy with membrane peeling to the right eye. Successful outcome was achieved in both eyes. Conclusion and Importance: Congenital PCD is a rare life-threatening disease that usually presents in infancy with purpura fulminans or disseminated intravascular coagulation. However, ophthalmologists should be aware of this condition, as its ocular signs may appear first. Early recognition, laser therapy of the attached ischemic retina and early vitrectomy may be successful in achieving good anatomical and functional outcomes.
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institution Kabale University
issn 2451-9936
language English
publishDate 2024-12-01
publisher Elsevier
record_format Article
series American Journal of Ophthalmology Case Reports
spelling doaj-art-d95903302970427eb64309be60ad1c952024-12-18T08:50:47ZengElsevierAmerican Journal of Ophthalmology Case Reports2451-99362024-12-0136102206Lens-sparing vitrectomy for pediatric tractional retinal detachment in homozygous protein C deficiencyMoath A. Alowairdhi0Mohammed I. Alkhodair1Alwaleed M. Alsulaiman2Sulaiman M. Alsulaiman3King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia; College of Medicine, King Saud bin Abdulaziz University for Health Sciences, Riyadh, Saudi ArabiaKing Khaled Eye Specialist Hospital, Riyadh, Saudi ArabiaKing Khaled Eye Specialist Hospital, Riyadh, Saudi ArabiaKing Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia; Corresponding author. Vitreoretinal Division, King Khaled Eye Specialist Hospital, P.O. Box 7191, Riyadh, 11462, Saudi Arabia.Purpose: To describe the ophthalmic presentation and management of a male infant with homozygous protein C deficiency (PCD). Methods: Case report. Observation: An 8-week-old male infant presented with bilateral vitreous hemorrhage and tractional retinal detachment in the right eye. Work up revealed low protein C levels. Whole exome sequencing showed a homozygous likely pathogenic variant in PROC gene. The management included laser photocoagulation to both eyes followed by lens-sparing vitrectomy with membrane peeling to the right eye. Successful outcome was achieved in both eyes. Conclusion and Importance: Congenital PCD is a rare life-threatening disease that usually presents in infancy with purpura fulminans or disseminated intravascular coagulation. However, ophthalmologists should be aware of this condition, as its ocular signs may appear first. Early recognition, laser therapy of the attached ischemic retina and early vitrectomy may be successful in achieving good anatomical and functional outcomes.http://www.sciencedirect.com/science/article/pii/S2451993624002160Protein C deficiencyRetinal detachmentPediatricVitreous hemorrhage
spellingShingle Moath A. Alowairdhi
Mohammed I. Alkhodair
Alwaleed M. Alsulaiman
Sulaiman M. Alsulaiman
Lens-sparing vitrectomy for pediatric tractional retinal detachment in homozygous protein C deficiency
American Journal of Ophthalmology Case Reports
Protein C deficiency
Retinal detachment
Pediatric
Vitreous hemorrhage
title Lens-sparing vitrectomy for pediatric tractional retinal detachment in homozygous protein C deficiency
title_full Lens-sparing vitrectomy for pediatric tractional retinal detachment in homozygous protein C deficiency
title_fullStr Lens-sparing vitrectomy for pediatric tractional retinal detachment in homozygous protein C deficiency
title_full_unstemmed Lens-sparing vitrectomy for pediatric tractional retinal detachment in homozygous protein C deficiency
title_short Lens-sparing vitrectomy for pediatric tractional retinal detachment in homozygous protein C deficiency
title_sort lens sparing vitrectomy for pediatric tractional retinal detachment in homozygous protein c deficiency
topic Protein C deficiency
Retinal detachment
Pediatric
Vitreous hemorrhage
url http://www.sciencedirect.com/science/article/pii/S2451993624002160
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AT alwaleedmalsulaiman lenssparingvitrectomyforpediatrictractionalretinaldetachmentinhomozygousproteincdeficiency
AT sulaimanmalsulaiman lenssparingvitrectomyforpediatrictractionalretinaldetachmentinhomozygousproteincdeficiency