Penetrating Keratoplasty as a Globe-Saving Procedure in Fragile Cornea

Natarajan, Radhika; Shah, Gaurav Y; Rao, Srinivas K; Padamanabhan, Prema

doi:10.1097/00003226-200303000-00017

Cited by 16 publications

(21 citation statements)

References 6 publications

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“…Primary penetrating keratoplasty is a good option to restore ocular integrity in patients with corneal perforations due to BCS, especially when tissue loss precludes simple repair as reported by Natarajan et al 14. In our patient too, suturing the corneal tear as a primary procedure was not possible due to pre-existing corneal pathology (brittle cornea).…”

Section: Discussionmentioning

confidence: 62%

Unusual case of globe perforation: the brittle cornea without systemic manifestations

Joshi

Uppapalli

et al. 2016

BMJ Case Reports

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Brittle cornea syndrome is a rare generalised connective tissue disorder with ocular features like keratoglobus or keratoconus, severe corneal thinning and a high risk of perforation. Various authors in different case reports and case series have brought out the fact that brittle cornea is a disorder with characteristic systemic manifestations such as deafness, joint hypermobility, hyperelasticity of skin, kyphoscoliosis and dental abnormalities alongwith ophthalmic features. We report a case of globe perforation following trivial trauma, in an individual with brittle cornea without any extraocular manifestations, posing a challenge in the diagnosis and dilemma in surgical repair of cornea, restoration of globe integrity and visual rehabilitation. The absence of systemic manifestations decreased the index of suspicion and led to a surprise in the theatre-a point this case emphasised.

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Section: Discussionmentioning

confidence: 62%

Unusual case of globe perforation: the brittle cornea without systemic manifestations

Joshi

Uppapalli

et al. 2016

BMJ Case Reports

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“…On inspection under anesthesia, there was severe host circumferential limbal and scleral thinning. The concern with utilizing conventional limbal wounds was the possible inability to reapproximate the compromised tissues [5]. The approach taken was to place the trocars through the donor keratoplasty tissue, 0.5 mm anterior to the graft-host junction (Supplementary video).…”

Section: Case Reports In Ophthalmological Medicinementioning

confidence: 99%

“…2 Case Reports in Ophthalmological Medicine loss. Additionally, sutures that are implanted during a primary keratoplasty have an increased risk of cheese-wiring due to the thin cornea, and there is a risk of overriding dueto mismatch between graft and host thickness, both of which could result in increased risk of postoperative infection and leakage[5].…”

mentioning

confidence: 99%

Case Series of Brittle Cornea Syndrome

Eleiwa

Raheem

Patel

et al. 2020

Case Reports in Ophthalmological Medicine

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Purpose. This case series demonstrate diagnostic features, treatment options, and challenges for Brittle Cornea Syndrome. Observations. Three cases presented with bluish sclera and extremely thin cornea. Genetic workup was performed and confirmed the diagnosis of Brittle Cornea Syndrome, a rare autosomal recessive disorder characterized by corneal thinning and blue sclera. Case 1 was a 4-year-old boy who developed cataract and glaucoma after undergoing right tectonic penetrating keratoplasty (PK) secondary to a spontaneous corneal rupture. Glaucoma was controlled medically. Later, the kid underwent right transcorneal lensectomy and vitrectomy with synechiolysis. After 6 weeks, he sustained graft dehiscence that was repaired using onlay patch graft. Case 2 was a 7-year-old boy who underwent PK in the right eye, then a pericardial patch graft in the left eye following spontaneous corneal rupture. Glaucoma in both eyes was controlled medically. Case 3 was the 2-year-old sister of the 2nd case. She had a pachymetry of 238 μm OD and 254 μm OS. In the 3 cases, parents were instructed to take protective measures for both eyes and to continue with follow-up visits. Also, they were instructed to have regular screenings for late-onset hearing loss, dental abnormalities, and bone deformities. Conclusions. Long-term follow-up of children diagnosed with Brittle Cornea Syndrome is paramount to minimize the morbidity of corneal rupture and late-onset extraocular conditions.

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“… 1 Some CTDs such as Ehlers-Danlos syndrome (EDS) confer an increased risk of globe rupture, 2 but there are very few reported instances of open globe injury in patients with OI. 3 , 4 , 5 We report a case of sequential traumatic open globes in a patient ultimately diagnosed with OI type I.…”

Section: Introductionmentioning

confidence: 97%

Sequential traumatic corneal open globe rupture in a patient with osteogenesis imperfecta type I

Campagna

Al-Mohtaseb

Khandelwal

et al. 2018

American Journal of Ophthalmology Case Reports

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PurposeTo report a case of sequential open globe rupture in a young patient with osteogenesis imperfecta type I following minor accidental blunt injury. This represented the patient's sole clinical manifestation of connective tissue disease, leading to a diagnosis of osteogenesis imperfecta type I at the age of 12 years old.ObservationsA 12-year-old male presented with right eye pain following accidental blunt trauma at school while wearing protective lenses. One year ago, he required surgical repair of a left open globe following blunt trauma during a middle school basketball game. His exam was significant for a full-thickness corneal laceration, necessitating open globe repair of his right eye, which was remarkably difficult given the poor tissue constitution of the cornea and sclera. He was referred to a genetics specialist, where he was found to have a pathogenic heterozygous splice site variant in the COL1A1 gene, consistent with osteogenesis imperfecta type I.Conclusions and importanceConnective tissue disease should be considered in any case of open globe rupture following minor trauma, even in the absence of other clinical manifestations of the disease. The surgical management of these patients is particularly challenging due to the fragility of the connective tissue. Early diagnosis of connective tissue disease is important to preserve visual acuity and prevent further damage to the eyes.

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Penetrating Keratoplasty as a Globe-Saving Procedure in Fragile Cornea

Abstract: Primary penetrating keratoplasty is a viable option to restore ocular integrity in fragile corneas following trauma when tissue loss precludes simple repair.

Cited by 16 publications

References 6 publications

Unusual case of globe perforation: the brittle cornea without systemic manifestations

Unusual case of globe perforation: the brittle cornea without systemic manifestations

Case Series of Brittle Cornea Syndrome

Sequential traumatic corneal open globe rupture in a patient with osteogenesis imperfecta type I

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