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Hypertrichosis of vellus hairs of the malar region after unilateral treatment with bimatoprost.

PURPOSE: To report hypertrichosis and increased pigmentation of vellus hairs of the malar region associated with topical bimatoprost usage. DESIGN: Interventional case report METHODS: In a female Hispanic patient with open-angle glaucoma, hypertrichosis, and increased pigmentation of the vellus hairs of the malar region developed after 8 weeks of unilateral treatment with bimatoprost. Increased growth of eyelashes was also noted. RESULTS: Bimatoprost was discontinued, with the intention of monitoring for resolution of hypertrichosis. The patient epilated the hair because of poor cosmetic appearance. Two months after epilating the hair, growth did not recur. CONCLUSIONS: Increased hair growth in the malar region can be an early side effect of topical use of ocular bimatoprost.

Amides↗

Keratosis lichenoides chronica in pediatric patients: a different disease?

Keratosis lichenoides chronica (KLC) is a rare acquired disease of adulthood, of unknown etiology, characterized by keratotic parallel linear lesions, retiform plaques, and keratotic, often follicular papules, chronicity and lichenoid histopathologic features. KLC of pediatric onset is considered extremely rare. Its features and relationship to adult onset KLC are unknown. We studied 8 cases of pediatric-onset KLC in the literature and 6 personal cases and compared them with 40 reported adult-onset KLC patients. The following features characterize pediatric-onset KLC: familial occurrence; probable autosomal recessive inheritance; early or congenital onset with facial erythemato-purpuric macules; forehead, eyebrow, and eyelash alopecia; pruritus; and a low frequency of other cutaneous and systemic abnormalities. Pediatric-onset KLC may represent a different disease or a subset of adult-onset KLC, with special genetic and clinical characteristics. Determining its precise nosology will have prognostic and therapeutic implications.

Adolescent↗

Retained anterior chamber cilium causing endophthalmitis after phacoemulsification.

An 81-year-old white man had uneventful cataract surgery by an experienced surgeon. Three days postoperatively, he presented with endophthalmitis and was treated in accordance with the standard departmental protocol. During the recovery, a curvilinear foreign body was identified and subsequently removed from the interior anterior chamber. Histological examination confirmed the foreign body as an eyelash. The patient improved to a final corrected visual acuity of 6/9. The relevant literature is reviewed, and ways to prevent this potentially blinding but avoidable complication of intraocular surgery are presented.

Aged↗

Abnormal electroretinogram and abnormal electrooculogram after lightning-induced ocular injury.

PURPOSE: To report a case of abnormal electrooculogram and abnormal electroretinogram after lightning-induced ocular injury. DESIGN: Interventional case report. METHODS: A 39-year-old man was struck on the left forehead by a bolt of lightning. After resuscitation, he regained consciousness. RESULTS: Ophthalmic examination disclosed a best-corrected visual acuity of right eye (RE): 20/25 and left eye (LE): 20/50, burned eyelashes, punctate keratitis, iridocyclitis, anterior subcapsular lens opacity, missing foveolar reflex, and macular pigment epithelial defect LE. Fluorescein angiography revealed only an area of punctate leaking in the left eye. Electrooculogram showed reduced amplitudes, in the left eye. Arden ratio was RE: 2.04 and LE: 1.52. The amplitudes of scotopic and photopic electroretinogram b-waves were reduced in the left eye. After uneventful cataract surgery in the left eye, the follow-up electrooculogram and electroretinogram still showed reduced amplitude in the left eye. CONCLUSION: Lightning is an uncommon cause of ocular injury. This is the first report of abnormal electro-oculogram and abnormal electroretinogram after lightning-induced ocular injury.

Adult↗

Prostaglandin-induced hair growth.

Latanoprost, used clinically in the treatment of glaucoma, induces growth of lashes and ancillary hairs around the eyelids. Manifestations include greater thickness and length of lashes, additional lash rows, conversion of vellus to terminal hairs in canthal areas as well as in regions adjacent to lash rows. In conjunction with increased growth, increased pigmentation occurs. Vellus hairs of the lower eyelids also undergo increased growth and pigmentation. Brief latanoprost therapy for 2-17 days (3-25.5 microg total dosage) induced findings comparable to chronic therapy in five patients. Latanoprost reversed alopecia of the eyelashes in one patient. Laboratory experiments with latanoprost have demonstrated stimulation of hair growth in mice and in the balding scalp of the stumptailed macaque, a primate that demonstrates androgenetic alopecia. The increased number of visible lashes is consistent with the ability of latanoprost to induce anagen (the growth phase) in telogen (resting) follicles while inducing hypertrophic changes in the involved follicles. The increased length of lashes is consistent with the ability of latanoprost to prolong the anagen phase of the hair cycle. Correlation with laboratory studies suggests that initiation and completion of latanoprost hair growth effects occur very early in anagen and the likely target is the dermal papilla.

Animals↗

Menkes' syndrome: ophthalmic findings.

PURPOSE: To report the prevalence and clinical significance of ocular findings in 20 patients with Menkes' syndrome recruited for a clinical trial at the National Institute of Child Health and Human Development (NICHHD). DESIGN: Retrospective observational case series. PARTICIPANTS: Twenty patients with Menkes' syndrome enrolled in a clinical trial at the NICHHD, who underwent ophthalmic evaluation at the National Eye Institute from 1990 through 1997. METHODS: Review of clinical ophthalmic examination records, photographs, and visual evoked potential recordings. MAIN OUTCOME MEASURES: Prevalence of ophthalmic signs of unusual frequency in patients with Menkes' syndrome. RESULTS: There was a high prevalence of very poor visual acuity (8 patients), myopia at 21 months old or younger (5 of 9 patients this age who underwent cycloplegic refraction), strabismus (11 of 18 patients whose motility was evaluated), blue irides (15 of 16 patients whose iris color was documented), iris stromal hypoplasia and bilateral peripheral transillumination (7 and 3 patients, respectively), peripheral retinal hypopigmentation (7 of 18 patients whose retinal periphery was noted at the initial examination), and aberrant eyelashes (5 patients). CONCLUSIONS: Patients with Menkes' syndrome exhibit a high prevalence of several ocular findings, including some (very poor visual acuity, myopia, strabismus) that may warrant special care. Early ocular examination is thus indicated for patients with Menkes' syndrome, particularly those with mild variants of the disease, whose neurologic status is better and lifespan is longer.

Abnormalities, Multiple↗

Trichotillomania.

PURPOSE: Trichotillomania is characterized by an irresistible urge to pull one's hair, and may involve the eyelashes or eyebrows. The authors present four cases of trichotillomania, and review the management of this unusual disorder. METHODS: The cases of four patients with trichotillomania were reviewed retrospectively. RESULTS: All four patients had characteristic areas of broken lashes along the lid in the absence of other signs of disease. Three of the four knew they were plucking the hair, yet could not control it. In the fourth, it was only after a lengthy observation period that she was discovered plucking. CONCLUSIONS: Trichotillomania has been infrequently reported in the ophthalmic literature. Management can be difficult. Many of these patients are aware of their behavior, but are unable to curtail it. Others may conceal or deny their habit. Psychiatric counseling may be of some benefit if patients are willing to undergo it.

Adult↗

Multiple follicular hamartomas with sweat gland and sebaceous differentiation, vermiculate atrophoderma, milia, hypotrichosis, and late development of multiple basal cell carcinomas.

We describe a 46-year-old woman with multiple symmetrical papulocystic lesions on the face, neck, chest, back, and upper arms since childhood. Vermiculate atrophoderma, entropion, and progressive loss of eyelashes was also noted. Since the age of 30, 12 basal cell carcinomas have developed on the face. Histopathologic examination revealed multiple anastomosing nests and strands that arose from the lower part of a hair follicle in early lesions. In more advanced lesions, multiple keratinizing microcysts within a moderately sclerotic stroma were noted in the upper and mid dermis. In some biopsy specimens, focal areas showing sweat gland (ductal) and sebaceous differentiation were also observed. Basal cell carcinomas developed from the basaloid component of the hamartomatous proliferation.

Atrophy↗

Investigating demodex in forensic autopsy cases.

Demodex is an ectoparasite living in the skin as a nonpathogen or a pathogen. It is also known that demodex acts as a vector of pathogenic microorganisms. In this study, we have investigated the rate of occurrence and vitality of demodex in forensic autopsies that have a high risk of contamination by infected organisms. The study, which was cross-sectional, conducted on 100 autopsy cases: 77 of the bodies were male and 23 female. The average age was 41. The samples were taken from the forehead, nose, cheeks, eyelashes and the temporal areas using the standardized skin surface biopsy and hair epilation techniques that were modified for this study. The data obtained were assessed statistically. Demodex was determined in 10% of all the cases. The cases revealed that demodex increased in old age and was more frequently seen in people with fair complexion. It was most frequently found in the forehead and the cheek. The longest postmortem interval in the positive cases was 55 h. The relationship between the postmortem interval and the presence of demodex was not statistically significant. Considering the fact that demodex, which is transmitted from human to human through skin contact, acts as a vector of pathogenic organisms and can stay alive in dead bodies for a long time, we think that the personnel performing autopsies without taking the necessary precautions are under risk.

Adult↗

Mutation of the FOXC2 gene in familial distichiasis.

OBJECTIVE: To examine the FOXC2 gene in a family with hereditary distichiasis. BACKGROUND: Distichiasis, ie, a second row of eyelashes arising from the meibomian glands of the eyelids, can be inherited either alone (Online Mendelian Inheritance in Man [OMIM] no. 126300) or, more commonly, as part of the lymphedema-distichiasis (LD) syndrome (OMIM no. 153400). More than 45 families with mutations in the FOXC2 gene and LD have been described. Both lymphedema and distichiasis are highly penetrant. Distichiasis without lymphedema is not commonly seen. METHODS: We examined three generations of a family (N = nine members) with hereditary distichiasis but without lymphedema or other features of LD syndrome. The FOXC2 gene was polymerase chain reaction--amplified from genomic DNA from all family members and examined for mutations. RESULTS: Clinical examination showed distichiasis of all four lids in two affected family members across two generations. There were no other consistent ophthalmologic abnormalities in the family. A cytosine-to-adenine transversion was identified in DNA from affected study participants at nucleotide position 1076, which would be predicted to cause truncation of the protein at codon 359. This change was not observed in any of the nine unaffected family members participating. CONCLUSIONS: This finding suggests that hereditary distichiasis and LD may not be separate genetic disorders but different phenotypic expressions of the same underlying disorder. Ophthalmologists should be aware that LD may present as distichiasis alone and counsel and refer their patients appropriately.

Adenine↗

FOXC2 truncating mutation in distichiasis, lymphedema, and cleft palate.

We report a family showing autosomal-dominant segregation of upper- and lower-eyelid distichiasis (double row of eyelashes) in seven affected relatives over three generations, in addition to below-knee lymphedema of pubertal onset (lymphoedema proecox) in three. Two children had cleft palate in addition to distichiasis, but without the previously reported association with the Pierre-Robin sequence. Other ophthalmologic anomalies included divergent strabismus and early-onset myopia. This family was found to be completely linked to markers mapped to 16q24.3 and thereby proposed to be allelic to the distichiasis-lymphedema syndrome (DL, MIM 153400), although pterygium colli, congenital heart disease, or facial dysmorphism were not features found here. As FOXC2/FKLH14 mutations were found to underlie DL and diverse hereditary lymphedema conditions, this gene was examined by sequence analysis. An out-of-frame deletion (914-921del) was identified and found to segregate with the disease, further highlighting the phenotypic heterogeneity of lymphedema conditions linked to FOXC2 truncating mutations. Whether such heterogeneity is related to genotype-phenotype correlation, a hypothesis not primarily supported by the apparent loss-of-function mechanism of the mutations, or governed by modifying genes, remains to be determined.

Abnormalities, Multiple↗

The effect of ketamine on clinical endpoints of hypnosis and EEG variables during propofol infusion.

BACKGROUND: We studied the effect of variable doses of ketamine on the endpoints of hypnosis, e.g., unresponsiveness to verbal commands (UVC), loss of eyelash reflex (LER), and inhibition of body movement response with or without sneezing to nasal membrane stimulation (INBMR), and processed EEG variables, e.g., bispectral index (BIS), 95% spectral edge frequency (SEF) and median frequency (MF) during propofol infusion. METHODS: Forty-eight patients received either propofol infusion, 30 mg.kg-1.h-1 (Group P; n = 12) or ketamine bolus, 0.25, 0.5 or 0.75 mg i.v., followed by propofol infusion, 30 mg.kg-1.h-1 + variable dose ketamine infusion, 0.25, 0.5 or 0.75 mg.kg-1.h-1 (Groups PK0.25, PK0.5 and PK0.75; n = 12 each) until UVC, LER and INBMR. BIS, 95% SEF and MF values were monitored and recorded at the endpoints of hypnosis. Propofol and ketamine concentrations were measured at INBMR. RESULTS: Propofol infusion, 30 mg.kg-1.h-1, induced UVC, LER and INBMR at BIS: 65 +/- 2, 63 +/- 9 and 33 +/- 7; 95% SEF: 17 +/- 3, 17 +/- 4 and 14 +/- 3; and MF values of 5 +/- 2, 5 +/- 3 and 3 +/- 2, respectively. With adjunctive ketamine (Groups PK0.5 and PK0.75), the hypnotic endpoints were achieved at higher BIS and 95% SEF values and lower propofol doses and concentrations as compared to Groups P and PK0.25 (9.9 +/- 5.8 and 9.4 +/- 3.4 vs. 13.4 +/- 4.5 and 14 +/- 5.8 micrograms.ml-1). CONCLUSIONS: Our results suggest additive interaction between propofol and ketamine (Groups PK0.5 and PK0.75) for achieving the hypnotic endpoints; however, ketamine did not depress the EEG variables in proportion to its hypnotic effect. The paradoxically higher BIS and 95% SEF values at the hypnotic endpoints may be due to lower propofol concentrations and/or no effect of ketamine on the EEG variables.

Adult↗

Trachoma: can trichiasis be treated with a sticking-plaster? A randomized clinical trial in China.

Trachoma is the most frequent cause of preventable blindness in the world. At the trichiasis/entropion stage, lid surgery is recommended, but many patients only use epilation, which does not prevent loss of vision. We developed a new treatment that should be more accessible than lid surgery and more effective than epilation: a sticking plaster that forces eyelashes back to their correct position. The first randomized controlled trial was conducted in Shanghai with 57 patients to compare the plaster method with epilation. After 3 months of follow-up, with no attrition, 67% of those treated by the new method presented a good clinical status, vs none of those treated by epilation (P < 0.001). The new treatment was well tolerated and lid function remained normal. Although our results show overwhelming benefit of this new, simple treatment for trachoma at the trichiasis stage, more research is needed at the primary health care level and in other settings to determine the potential use of the new method on a large scale and by nonspecialists.

Adult↗

Congenital lymphedema and distichiasis.

Distichiasis is defined as a double row of eyelashes. Inherited forms of distichiasis have been associated with early and late-onset congenital lymphedema. We report on a child with distichiasis and congenital lymphedema in which it is unknown whether the cause of lymphedema was due to a spontaneous mutation or acquired secondary to an intrauterine infection. Acquired forms of distichiasis are reviewed.

Abnormalities, Multiple↗

[Demodex folliculorum].

The author reports on Demodex folliculorum ("hair follicle mite"), a frequent and widespread parasite. These mites are found in hair follicles of eyelashes in particular. In ophthalmology they are responsible for chronic eczematous blepharitis ("blepharitis acarica") with trichiasis and madarosis. Therapy is unsatisfactory. The author has now discovered that isoptocarbachol 3% eyedrops successfully combat Demodex folliculorum. The solution was first tested experimentally on house flies (Musca domestica) and was later used to treat patients affected with the parasite: a cotton bud impregnated with this parasympathomimetic (anticholinesterase) drug was applied to the border of the eyelids several times.

Adult↗

Modified grading system for upper eyelid trachomatous trichiasis.

AIM: This study was conducted to determine the level of inter-observer agreement in the modified grading scheme for trichiasis of the upper eyelid. METHODS: A grading system that covers the entire spectrum of trachomatous trichiasis, extending from the simple to the severe forms, was initially developed by the first and second authors and field-tested. The grading scheme recognizes three levels of severity, based on deviation of the lid margin (TT1, TT2 and TT3); and four anatomical locations indicating where the eyelashes touch the globe in TT1 (nasal, central, temporal, or combined). The grading of trichiasis patients was then conducted on the basis of the modified grading scheme. RESULTS: The inter-observer agreement between the grades of simple trichiasis (TT1) and entropion (TT2) using Cronbach's alpha was 0.84, and the agreement within each sub-grade was 0.86. The weighted Kappa value was 0.68 (95% CI 0.57-0.79) for the main grades and 0.49 (95% CI 0.23-0.79) for the sub-grades. CONCLUSIONS: The inter-observer agreement was very reliable and could easily be attained by all levels of ophthalmic professionals. As the grading follows the natural progression of trichiasis from the simple to the severe forms in relation to the anatomical site involved, it helps to determine the degree of correction and how far to extend the surgical incision.

Eyelashes↗

Epidemiological, demographic, and economic analyses: measurement of the value of trichiasis surgery in The Gambia.

AIM: Untreated trichiasis can lead to corneal opacity. Surgery to prevent the eyelashes from rubbing against the cornea is available, but many individuals with trichiasis never undergo the operation. This study estimates the cost of illness of untreated trichiasis and the willingness to pay for surgery and compares them with the actual cost of providing surgery. MATERIALS AND METHODS: The cost of illness estimate is based on trichiasis patient demographics. Data on the implicit price of obtaining surgery and surgical utilization in a matched pair randomized trial are used to infer individual willingness to pay for trichiasis surgery. Patients in the study paid nothing out-of-pocket for surgery; the price of obtaining surgery is the value of the individual's time needed for travel and surgery plus the price of public transportation. The cost of producing surgery was calculated from project records. RESULTS: All monetary figures are reported in 1998 US dollars. The average cost of untreated trichiasis, or the net present value of life-time lost economic productivity, was $89. Individuals facing a lower cost were more likely to undergo an operation; the inferred average willingness to pay was $1.43 (SD 0.244). Surgery cost $6.13 to provide, including $0.86 for transportation to the village. DISCUSSION: Whether the value of trichiasis surgery exceeds the cost in The Gambia depends on how the value is measured. Individuals are willing to use only limited resources to obtain surgery even though lifetime economic productivity may increase substantially. All three economic measures can be used to inform policy.

Adult↗

Induction dose-response of propofol in unpremedicated children.

The induction dose requirements of propofol were compared in three age groups in 300 unpremedicated healthy Chinese children: group A, younger than 2 yr (n = 48); group B, 2-5 yr (n = 117); group C, 6-12 yr (n = 135). Patients in each group were allocated randomly to receive one of eight doses of propofol (1.2, 1.4, 1.6, 1.8, 2.0, 2.2, 2.4 and 2.6 mg kg-1). ED50 and ED95 for loss of eyelash reflex (LER) and acceptance of face mask (AFM) were determined using probit analysis. ED50 and ED95 for both LER and AFM were greatest in group A, less in B and smallest in C; ED95 (AFM) for groups A, B and C were 2.88 (2.55-3.36), 2.53 (2.31-2.86), and 2.20 (2.02-2.46) mg kg-1, respectively. This probably represented their effective induction dose. The incidence of apnoea was dose related, but not pain on injection.

Age Factors↗