Spontaneous Bleb Formation in a Presumed Pellucid Marginal Corneal Degeneration With Acute Hydrops

Toriyama, Koji; Suzuki, Takashi; Higashiura, Ritsuko; Maeda, Naoyuki; Ohashi, Yuichi

doi:10.1097/ico.0b013e3182801685

Cited by 10 publications

(10 citation statements)

References 6 publications

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“…Spontaneous filtering bleb formation due to corneal pathologies such as Terrien and pellucid marginal degeneration have been reported by Soong et al .,[ 2 ] Munro et al .,[ 3 ] and Toriyama et al . [ 8 ] Mechanism proposed was the extreme distortion and stretching of the peripheral cornea resulting in rupture of the Descemet's membrane and bleb formation. Our patient had normal cornea without signs of thinning or degeneration.…”

Section: Discussionmentioning

confidence: 99%

Bilateral idiopathic spontaneous filtering bleb with ectopia lentis: A case report and review of literature

Chandran

Khairnar

Aboobacker

et al. 2018

Indian J Ophthalmol

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A 26-year-old male presented with superior filtering bleb with scleral thinning, dislocated lens, and hypotony in both the eyes. His cornea was normal without any sign of ectasia, and there was no history of recurrent redness, trauma, or surgery in either eye. Anterior segment optical coherence tomography did not reveal communicating fistula between the anterior chamber and subconjunctival space. Physical examination and blood investigations did not reveal any systemic association. He was diagnosed to have spontaneous filtering bleb, which is a rare condition observed with ocular or systemic abnormalities.

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Section: Discussionmentioning

confidence: 99%

Bilateral idiopathic spontaneous filtering bleb with ectopia lentis: A case report and review of literature

Chandran

Khairnar

Aboobacker

et al. 2018

Indian J Ophthalmol

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“…A similar mechanism was described for a case of pellucid marginal degeneration and spontaneous filtering bleb. 8 The common pathology in cases of Terrien's and pellucid marginal degeneration is the abnormal tissue of the corneal periphery. Our patient didn't have any sign of axenfeld syndrome, pellucid, or Terrien's marginal degeneration.…”

Section: Discussionmentioning

confidence: 99%

“…They are reported in association with ocular abnormalities like Axenfeld syndrome, Terrien's marginal degeneration, microspherophakia, ectopia lentis, scleritis, and pellucid marginal corneal degeneration. 2 , 3 , 4 , 5 , 6 , 7 , 8 There are reports of spontaneous filtering blebs in systemic conditions like scleroderma and Traboulsi syndrome. 9 , 10 , 11 , 12 In this study, we present a case with mild craniofacial dysmorphism, microspherophakia, and bilateral spontaneous filtering blebs.…”

Section: Introductionmentioning

confidence: 99%

Bilateral spontaneous filtering blebs

Mohammadi¹,

Tabatabaei²

2020

American Journal of Ophthalmology Case Reports

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Purpose To report a case of bilateral spontaneous filtering bleb, scleral thinning, microspherophakia, and mild craniofacial dysmorphism. Observations An 18-year-old girl was referred to our clinic for evaluation of bilateral spontaneous filtering blebs. Her corrected distance visual acuity (CDVA) was 20/400 in the right eye (RE) and 20/100 in the left eye (LE). She had superior scleral thinning, multicystic filtering bleb, and microspherophakia bilaterally. The IOP was 9 mmHg in the RE and 8 mmHg in the LE. A mild craniofacial dysmorphism including downward slanting of palpebral fissures and malar hypoplasia was present. There was no sign of corneal ectasia in tomography. Anterior segment optical coherence tomography revealed the filtering blebs as subconjunctival low reflective fluid-filled spaces. Due to severe scleral thinning un the RE we performed a tectonic scleral patch graft. 6 months after surgery the depth of the anterior chamber increased and CDVA improved. Conclusion and importance This case indicated that the formation of spontaneous filtering blebs may occur in a syndromic condition. In any case with this symptom, special attention should be paid to craniofacial features, sclera, crystalline lens, and IOP. Tectonic scleral patch graft could be a valuable option in selected patients.

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“…A previous report described spontaneous bleb formation due to a rupture in the stromal cleft, allowing a large edematous cavern to expand under the conjunctiva [ 5 ]. Where a break in Descemet’s membrane was close to the inferior limbus, and a corneal perforation resulted in aqueous humor to migrate into the subconjunctival space.…”

Section: Discussionmentioning

confidence: 99%

Acute hydrops with a 180-degree massive edematous cavern demonstrated by three dimensional view of anterior segment optical coherence tomography in a patient with pellucid marginal corneal degeneration, a case report

2018

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BackgroundPellucid marginal corneal degeneration is a non-inflammatory disorder complicated by severe inferior corneal thinning. The central portion of the cornea, consequently, appears to protrude outwards, decreasing vision by means of an irregular stigmatism. Additionally, acute hydrops can occur in case of Descemet’s membrane rupture.Case presentationA 41-year-old Japanese woman presenting with severe visual loss in the left eye was examined and observed to have had full thickness corneal perforation as well as a Descemet membrane rupture with massive edema in the corneal stroma. Anterior segment optical coherence tomography-based corneal topography revealed a distorted crab claw sign indicating pellucid marginal corneal degeneration. The Descemet membrane rupture allowed acute hydrops to occur which was especially noteworthy given the scale of edema present within the stroma, rotating 180 degrees along the limbus, causing a smiley-face like lesion. We visualized it via a gonioscopic three-dimensional optical coherence tomography to build a three-dimensional video. Patient history revealed a previous acute hydrops in the right eye as well, which was ultimately treated with anterior lamellar keratoplasty, suggesting the pellucid marginal corneal degeneration had a classic bilateral involvement, which was also characterized with bilateral acute hydrops.ConclusionThis appears to be a very rare and interesting presentation of bilateral pellucid marginal degeneration, wherein not only acute hydrops formed bilaterally, but the cavity within the cornea stroma was exceptionally large with an unusual shape. Using the gonioscopic three-dimensional optical coherence tomography imaging, we were able to easily visualize the massive intrastromal cavern, and appropriately planned the crescent-shaped anterior lamellar keratoplasty. The 3d video constructed using this data is particularly elucidative compared to 2d images. As such, we recommend utilizing 3d imaging in cases where more conventional topography is not as explanatory with respect to precise nature of deformation.Electronic supplementary materialThe online version of this article (10.1186/s12886-018-0757-7) contains supplementary material, which is available to authorized users.

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Spontaneous Bleb Formation in a Presumed Pellucid Marginal Corneal Degeneration With Acute Hydrops

Abstract: This case indicated that spontaneous bleb formation in a pellucid marginal corneal degeneration should be included in the differential diagnosis of a conjunctival mass of unknown origin.

Cited by 10 publications

References 6 publications

Bilateral idiopathic spontaneous filtering bleb with ectopia lentis: A case report and review of literature

Bilateral idiopathic spontaneous filtering bleb with ectopia lentis: A case report and review of literature

Bilateral spontaneous filtering blebs

Acute hydrops with a 180-degree massive edematous cavern demonstrated by three dimensional view of anterior segment optical coherence tomography in a patient with pellucid marginal corneal degeneration, a case report

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