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PubMedDate not supplied

Corneal opacity and congenital glaucoma associated with massive heparan sulfaturia: report of one case.

A four month-old male infant was noted to have had severe corneal opacity since birth. Buphthalmos, increased intraocular pressure and corneal opacity with neovascularization were noted during physical examination. There was neither dysmorphic face nor hirsutism and the liver and spleen were impalpable. In addition, hypotonia, poor head control, and absence of Moro and grasping reflexes were also noted. There was no evidence of congenital infection by TORCH study. Tests of both urine and plasma amino acids were within normal limits. However, excessive urinary excretion of heparan sulfate was detected by thin-layer chromatography. Corneal transplantation was performed at 6 months old. Histopathological examination of the corneal button showed homogeneous thickening of Bowmen's membrane and intracytoplasmic pinkish substances in corneal stroma. The Alcian blue stain was positive, which was consistent with mucopolysaccharidosis of cornea. The manifestation of this case may be a clinical variant of Sanfilippo's syndrome (Mucopolysaccharidosis type III).

Corneal Opacity↗
PubMed2000

Corneal opacities after cataract surgery with hypromellose.

26 cases of corneal opacity after cataract surgery occurred in a period of 2 weeks in one surgical unit. Cases occurred after a change in source of intraocular hypromellose solution and only among patients in whom the new product had been used.

Cataract Extraction↗
PubMed2006

[Increase in posterior segment depth in eyes with corneal opacities].

AIM: Studies in animals and in humans demonstrate an increase in axial length and induced myopia in eyes with early onset corneal scars. The purpose of this study was to compare the total ocular axial length and the length of the anterior and the posterior segments, in eyes with unilateral central corneal opacification with the unaffected fellow eyes. Elongation of the eye and progressive myopia, may lead to both anisometropia and amblyopia. METHODS: In six patients with unilateral corneal opacification that developed at an early age, complete eye examinations with refraction were performed. Ocular axial measurements were determined by A-mode ultrasound. RESULTS: The mean ocular length was greater in eyes with central corneal opacities compared with the fellow eyes (27.53 vs. 23.38 mm; p < 0.003), due to elongation of the posterior segment (19.75 mm vs. 15.87 mm; p<0.008). No significant change was found in the length of the anterior segments. CONCLUSIONS: Our study showed an ipsilateral increase in the posterior segment length in patients with unilateral corneal opacification which had occurred in childhood. The cause is unknown but may be due to altered visual input. Progressive myopia in corneal leukoma is a factor to be taken into account in the follow-up and treatment of patients with central corneal opacities that occurred in childhood.

Adult↗
PubMed1988

Evaluation of keratoplasty in cases of post-inflammatory corneal opacity with vascularization.

Penetrating keratoplasty was performed in 106 cases of post-inflammatory corneal opacity with vascularization. Donor material obtained post mortem was preserved in a moist chamber, MK-medium at 4 degrees C or in culture medium at room temperature. 55.7% of patients gained a useful vision after keratoplasty. The post-operative spherical error of refraction was found to be -1.4 +/- 3.6 D and the astigmatic error of refraction -3.3 +/- 2.0 D. The post-operative visual acuity was found to be 0.2 +/- 0.2. The ocular pathology associated with keratoplasty is discussed.

Adolescent↗
PubMed2004

Outcome of combined Ahmed glaucoma valve implant and penetrating keratoplasty in refractory congenital glaucoma with corneal opacity.

PURPOSE: To investigate surgical outcomes following simultaneous Ahmed glaucoma valve implant and penetrating keratoplasty (PKP) in the management of refractory congenital glaucoma with corneal opacity. METHODS: A retrospective review was undertaken of pediatric patients who underwent simultaneous Ahmed glaucoma valve implant and PKP at King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia, between January 1994 and September 1999. RESULTS: Twenty eyes of 17 patients were included in the study. Cumulative probabilities of success by Kaplan-Meier analysis showed 85%, 44%, and 33% IOP control and 85%, 43%, and 17% graft success at 2, 24, and 48 months. The most common cause of glaucoma failure that required subsequent surgery was subconjunctival scarring, which resulted in loss of long-term IOP control. Main graft-related complications included failure (13/20) and graft ulceration (6/20), and in 4/6 ulcerated grafts, Streptococcus pneumoniae was cultured positively. Subsequent surgery was the only significant clinical factor associated with poor outcome of glaucoma. However, low graft survival rate was significantly associated with delinquency of follow-ups, corneal ulcer, subsequent surgery, and postoperative complications. CONCLUSIONS: The long-term success of simultaneous Ahmed glaucoma valve implant and PKP in refractory congenital glaucoma associated with corneal opacity is low, and the complication rate is high.

Child, Preschool↗
PubMed1988

Encephalomyelitis, reproductive failure and corneal opacity (blue eye) in pigs, associated with a paramyxovirus infection.

A new clinicopathological disorder associated with a paramyxovirus infection in pigs is described. Central nervous system manifestations and corneal opacity are the main features in piglets two to 21 days old. Older pigs seem to be more resistant and only corneal opacity is commonly observed. In pregnant sows the virus appears to be responsible for reproductive disturbances such as stillbirth, mummification and a return to oestrus. The changes are mainly microscopic and characterised by a non-suppurative encephalomyelitis, interstitial pneumonia and anterior uveitis with corneal oedema. Experimental infection of one-day-old piglets with the virus reproduced clinical signs similar to those described in naturally infected piglets. The virus was recovered from the tonsils, lung and brain of the experimentally infected piglets between the fourth and 20th day after infection.

Animals↗
PubMed2000

Corneal opacity, hydration and endothelial morphology in the bovine cornea opacity and permeability assay using reduced treatment times.

The purpose of this study was to determine whether the standard bovine cornea opacity and permeability (BCOP) assay exposure time of 10 minutes overestimates the ocular irritancy of chemical substances. Corneas were subjected to BCOP protocol following 30-second and 1-minute exposures to irritants. Corneal opacity and hydration (mg H(2)O/mg cornea) were then measured and compared to data obtained after 10 minute irritant treatments. For most test substances corneal opacity and hydration were lower following reduced exposure times. It is suggested that using shorter exposure times in BCOP protocol may be more predictive of human response to ocular irritants, since irritants are usually in brief contact with the ocular surface during accidental exposure. A second purpose of this study was to examine effects of irritants on the corneal endothelium. Corneas were treated according to BCOP protocol following exposure to irritants for 1 or 10 minutes. The endothelium was stained with Alizarin Red and trypan blue, and examined using light microscopy. Severe irritants, such as NaOH and trichloroacetic acid, cause endothelial cell death. It was also determined that simply mounting the cornea in the BCOP assay holders caused damage to 20% of the endothelial cells. Because the endothelium is essential for normal corneal transparency and hydration, it is suggested that examination of the endothelium be added to the BCOP assay and that optimization of the assay will require modification of the cornea holders.

Animals↗
PubMed1992

Familial high-density-lipoprotein deficiency causing corneal opacities (fish eye disease) in a family of Dutch descent.

Fish eye disease (FED) is an extremely rare familial disorder characterized by severe HDL deficiency and extensive corneal opacities. This disorder appears to be a variant of familial lecithin: cholesterol acyltransferase (LCAT) deficiency in which the enzyme remains partly active yet the ability of the enzyme to esterify cholesterol in high-density lipoprotein (HDL) has been lost. The rarity of this disorder has limited advances in our understanding of the pathophysiology of the HDL deficiency. However, we here describe the clinical and biochemical presentation of a family with FED who are of Dutch descent. The proposition presented with HDL deficiency and corneal opacity. Subsequently, they were diagnosed as having FED by the absence of LCAT activity against a small proteoliposome substrate despite the presence of half-normal LCAT mass and a near-normal ratio of unesterified to total cholesterol in plasma. Heterozygotes presented with half-normal LCAT activity, but not with decreased HDL. With the identification of this three-generation family, renewed investigation of this intriguing disorder of HDL is now possible.

Adult↗
PubMedDate not supplied

Specular microscopy in the identification of deep corneal opacities.

Specular microscopy provides a level of magnification and differentiation of detail which approaches that of a histological examination, and has the advantage of being a noninvasive procedure. This makes specular microscopy useful not only in examining the corneal endothelium, but also in identifying deep corneal opacities, which fall into two broad groups: 1) exogenous materials deposited in the cornea; and 2) endogenous pathologic corneal changes. Exogenous materials in the cornea have a wide variety of appearances, including the typical appearance of caterpillar hairs, the multifaceted appearance of glass foreign bodies, the regular geometric appearance of cholesterol crystals and cigar-shaped myeloma crystals or the more irregular deposits of chlorpromazine. Endogenous changes include ghost vessels, larger striate corneal nerves, fasciculi of fine parallel lines in keratoconus, criss-cross irregularly running lines in resolved keratitis, and a shagreen or cellophane-like appearance suggesting thickening of Descemet's membrane. While some deep corneal opacities are easily diagnosed clinically, the nature of others may be difficult to determine and specular microscopy is a valuable aid in such cases.

Corneal Diseases↗
PubMed2006

Optical coherence tomography of the anterior segment in secondary glaucoma with corneal opacity after penetrating keratoplasty.

AIM: To evaluate secondary glaucoma after penetrating keratoplasty with anterior-segment optical coherence tomography (OCT). DESIGN: Case series. METHODS: Four eyes of four patients with corneal opacity and increased intraocular pressure (IOP) were evaluated using high-speed (2000 axial scans/s) OCT at 1.3 microm wavelength. Cross-sectional images of the anterior segment were analysed to assess the cause of increase in pressure. RESULTS: Slit-lamp evaluation of the anterior chamber in all cases was limited by corneal opacity. The OCT imaging allowed visualisation of anterior-segment structures behind the opaque corneas. Using OCT, iris-intraocular lens adhesion and pupillary block were identified as the probable reasons for the increased IOP in one case. Peripheral anterior synechiae and angle closure were identified in the three remaining cases. In two cases, we found that the tip of the aqueous drainage tube was blocked by peripheral anterior synechiae. CONCLUSIONS: OCT is similar to ultrasound in that it allows visualisation through opaque corneas. However, OCT has an advantage in that it requires neither contact nor immersion. It is a valuable tool for evaluating the depth of the anterior chamber angle and the causes of secondary angle closure.

Aged, 80 and over↗
PubMed1991

A 'Fish-eye disease' familial condition with massive corneal opacities and hypoalphalipoproteinaemia: clinical, biochemical and genetic features.

A Caucasian family of mediterranean origin comprising a patient whose parents were first cousins, his wife and their three children, and his two sisters have been studied. The patient and his two daughters were afflicted with the same corneal opacities and hypoalphalipoproteinaemia. The disease was shown to be transmitted as a non-sex-linked recessive trait. The corneal opacities develop at the end of the second decade of life and consist of numerous minute greyish dots in the entire corneal stroma that give the cornea a misty appearance. Vision slowly deteriorated from 40 years of age. At about 50 years of age, except in one of the two daughters who showed Marfanoid syndrome, the three patients had good general health and no symptoms of atherosclerosis. Biochemical investigations showed hypoalphalipoproteinaemia (with a faint fast-moving HDL band on polyacrylamide gel gradient electrophoresis and small arcs of HDL2 and HDL3 of low mobility determined by agarose gel immunoelectrophoresis), low total cholesterol (3.5-4.9 mmol l-1), slightly decreased cholesteryl ester/total cholesterol ratio (0.52-0.63), extremely low HDL cholesterol (0.20-0.21 mmol l-1), mild hypertriglyceridaemia (1.94-3.80 mmol l-1), and striking deficiency in apo A-I and apo A-II (0.45-0.72, 0.08-0.16 g l-1, respectively). The esterification of HDL cholesterol was low while that of LDL and VLDL was nearly normal. Other laboratory values were normal. The HDL subspecies and major apolipoprotein isoforms have been studied to differentiate FED from Tangier disease, LCAT deficiency, as Apo A-I, A-II, C-II, C-III deficiencies and variants.(ABSTRACT TRUNCATED AT 250 WORDS)

Adolescent↗
PubMed1984

A simple method to assess surfactant-induced bovine corneal opacity in vitro: preliminary findings.

The in vitro development of bovine corneal opacity induced by two anionic surfactants, sodium decyl sulphate (NaDS) and sodium lauryl sulphate (NaLS), was monitored using a simple, specially constructed instrument, the opacitometer. Results show clearly that NaDS was more potent, and acted more rapidly, than NaLS, an order of potency which has been shown by in vivo ocular irritancy tests with these agents but not in cytotoxicity tests. The measurement of irritant-induced opacity of bovine isolated cornea may therefore offer another approach to the search for viable in vitro alternatives to in vivo ocular irritancy testing.

Animals↗
PubMed2004

Topographically guided excimer laser photorefractive keratectomy to treat superficial corneal opacities.

OBJECTIVE: To demonstrate the efficacy and safety of topographically guided excimer laser photorefractive keratectomy (PRK) in treating superficial corneal opacities. DESIGN: Prospective, noncomparative interventional case series. METHODS: Twenty-six eyes of 24 patients with postinfectious (n = 6), post-traumatic (n = 18), and post-PRK (n = 2) scars were treated with an excimer laser linked to a computerized videokeratography unit with a topographically supported customized ablation workstation. MAIN OUTCOME MEASURES: Manifest spectacle refraction, best spectacle-corrected visual acuity (BSCVA), uncorrected visual acuity (UCVA), change of corneal clarity, and topographic indexes of surface regularity. RESULTS: The mean follow-up period was 7.7+/-2.1 months (+/- standard deviation) (range = 6-12). Corneal clarity and corneal topography patterns improved in all eyes. Uncorrected visual acuity improved from 0.7+/-0.8 logarithm of the minimum angle of resolution (logMAR) to 0.4+/-0.5 logMAR (P = 0.008), and BSCVA improved from 0.3+/-0.6 logMAR to 0.1+/-0.6 logMAR (P<0.001). The BSCVA increased in all eyes (>/=2 lines in 76.9% of eyes and 1 line in 23.1%). The mean spherical equivalent refraction changed from -0.125+/-2.77 diopters (D) to +1.33+/-1.28 D (P = 0.019). CONCLUSIONS: The use of topographically guided PRK with the topographically supported customized ablation method resulted in significant increases of UCVA and BSCVA and improved corneal clarity in all patients. The technique seems to be safe and effective for treating various superficial corneal diseases, including postinfectious, post-traumatic, and post-PRK scars.

Adult↗
PubMed1979

Fish-eye disease. A new familial condition with massive corneal opacities and dyslipoproteinaemia.

A man and his three daughters had massive corneal opacities called in their home village "fish-eye disease" because of the resemblance of the eyes to those of boiled fish. The two living daughters had the same dyslipoproteinaemia, characterised by normal serum cholesterol but raised serum triglycerides, raised very-low-density lipoproteins, strikingly high levels of low-density lipoprotein (LDL) triglycerides. LDL contained normal sized as well as abnormally large particles and a 90% reduction in the level of high-density lipoprotein (HDL) cholesterol. Lecithin:cholesterol acyltransferase (LCAT) activity and the percentage of plasma cholesterol esters were normal, with excluded LCAT-deficiency. Normal electrophoretic mobility of HDL as well as other lipoprotein findings excluded Tangier disease. The clinical and laboratory abnormalities in fish-eye disease are atherosclerosis at old age, visual impairment, and dense corneal opacification. Fish-eye disease thus differs both clinically and in its lipoprotein abnormalities from LCAT-deficiency and Tangier disease.

Adolescent↗
PubMed1982

Fish eye disease: a new familial condition with massive corneal opacities and dyslipoproteinaemia.

Fish eye disease (FED) is characterized by severe corneal opacities, causing impaired vision, and dyslipoproteinaemia: hypertriglyceridaemia, raised levels of very low density lipoproteins (VLDL), triglyceride enrichment of low density lipoproteins (LDL) and reduction of high density lipoproteins (HDL). The disease is described in two unrelated families. In both there was a high proportion of low HDL in relatives without eye disease. VLDL, LDL and HDL had normal electrophoretic mobilities. The concentrations of VLDL cholesterol and triglycerides were increased fivefold. LDL cholesterol levels were normal but LDL triglycerides markedly increased. HDL cholesterol was reduced by 90% as were the levels of HDL apolipoproteins. The major part of HDL cholesterol was in the HDL3 fraction. FED HDL were smaller than normal with molecular weights of 115,000 daltons. Lecithin: cholesterol acyltransferase activity and amount of cholesterol esters in serum were normal. Postheparin lipoprotein and hepatic lipases showed normal or subnormal values. Clinically FED differs from other familial conditions with deficiency of HDL such as Tangier disease, LCAT-deficiency and Milano-AI-apoprotein disease. In spite of the extremely low HDL cholesterol FED is not characterized by premature atherosclerosis. Mechanisms for the dyslipoproteinaemia are discussed.

Aged↗