Management of Evaporative Dry Eye in Ectrodactyly-Ectodermal Dysplasia-Clefting Syndrome

Ota, Yu; Matsumoto, Yukihiro; Doğru, Murat; Goto, Eiki; Uchino, Yuichi; Endo, Koji; Tsubota, Kazuo

doi:10.1097/opx.0b013e31818527cd

Cited by 13 publications

(14 citation statements)

References 24 publications

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“…The absence of the meibomian glands leads to an instability of the tear film, with a normal Schirmer test but pathological BUT. Lemp [24] reported that patients with absent or deficient meibomian gland function should be categorized in the evaporative group of dry eye syndromes [15]. Matsumoto et al [13] measured the tear evaporation rate to clarify the changes in tear quality.…”

Section: Discussionmentioning

confidence: 99%

“…Although the Schirmer I test was 5/6 mm and BUT 6/6 s in one patient, he had no dry eye complaints. The authors attributed the corneal abnormalities to an unstable tear film due to a lack of lipid secondary to absence of meibomian glands and goblet cell abnormalities [15,[18][19][20].…”

Section: Discussionmentioning

confidence: 99%

“…Meibomian gland deformities have so far only been described in patients with EEC syndrome. There is evidence that, on the basis of the similar pathology, other ectodermal dysplasias should also be combined [6,14,15].…”

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confidence: 99%

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Ophthalmic Manifestations in Patients with Ectodermal Dysplasia Syndromes

Keklíkçí¹,

Yavuz²,

Tunik³

et al. 2014

Adv Clin Exp Med

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Background. Ectodermal dysplasia (ED) is a disorder that results from abnormal formation of at least two of the four major ectodermal derivatives in the developing embryo. The ectoderm of the embryo forms the skin, teeth, hair and nails, sweat glands and part of the eyes. Objectives. The aim of this article is to reveal ophthalmologic symptoms and signs as multidisciplinary, reliable criteria for ectodermal dysplasia. Material and Methods. In this retrospective study, 24 patients with ED were analyzed from the recorded data. Ophthalmological examination of the patients, who had previously received the diagnosis of ED in the dental department, was done. During the examination, ocular symptoms related to tear film, corneal changes, lacrimal duct, periorbital hyperpigmentation, alteration lashes and eyebrows were evaluated. Results. The age ranged between 3-45, and the mean and standard deviation (Mean ± SD) was 15.8 ± 7.4 years. The number of males was 13 (54.2%) and females, 11 (45.8%). Eighteen patients (75.0%) suffered from ocular complaints related to the ocular surface. In 11 of the patients with ED, there were dry eye symptoms. While the mean age of cases with eye involvement was 17.5, it was 23.1 in cases with dry eye symptoms. Conclusions. In the study, it was observed that, in patients with ED, ocular complaints, particularly dry eye symptoms, may increase as age advances (Adv Clin Exp Med 2014, 23, 4, 605-610).

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Ophthalmic Manifestations in Patients with Ectodermal Dysplasia Syndromes

Keklíkçí¹,

Yavuz²,

Tunik³

et al. 2014

Adv Clin Exp Med

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“…The main characteristics of this rare autosomal recessive hereditary disease include abnormalities of the epidermis and its appendages (hair, nails, eccrine and sebaceous glands) together with lack of sweat glands or more frequently decrease in sweat glands which leads to increase in body temperature (1). Ocular findings including strabismus, telecanthus, blepharophymosis, entropion, dacriocystitis, blepharitis, conjunctivitis, meibomian gland dysfunction, corneal opacity and cataract may be observed in individuals with ectodermal dysplasia (3)(4)(5)(6)(7)(8)(9). Recurring corneal erosions, opacifications, vascularization and perforations affect various ocular surface disorders.…”

Section: Introductionmentioning

confidence: 99%

“…Recurring corneal erosions, opacifications, vascularization and perforations affect various ocular surface disorders. However, the reason of ocular surface disorders in individuals with ectodermal dysplasia is not known exactly (6,7). To prevent corneal demage these patients should be followed up with artificial tear.…”

Section: Introductionmentioning

confidence: 99%

Ektodermal displazili olgularda konjonktiva bası sitolojisi

Söker¹,

Keklíkçí²,

Meşe³

et al. 2012

tpa

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Ectodermal dysplasia is a group of symptoms affecting tissues and organs of ectodermal origin. In this study, conjunctival impression cytology technique which is a non-invasive method was performed in patients with ectodermal dysplasia to evaluate the effects on the conjunctiva. Six eyes of three patients with ectodermal dysplasia who were followed up by the Ophthalmology Clinic of Dicle University Faculty of Medicine and Prosthetic Dentistry Departments were included in the study. Bulbar conjunctiva impression cytology method was applied to the upper surface of the bulbar conjunctiva and the cell groups taken to the surface of cellulose acetate paper were fixed by a solution containing 70% ethyl alcohol, 37% formaldehyde and glacial acetic acid with a ratio of 20:1:1 and later stained by Periodic acid-Schiff and hematoxylin-eosin. Using impression cytology method a decrease in integrity of the cells, picnotic changes in the nucleus, epithelial cell-like changes and loss of goblet cells were observed in patients with ectodermal dysplasia. The signs of eye dryness in patients with ectodermal dysplasia were supported by the impression cytology technique. Impression cytology can be used as a non-invasive method in ophthalmologic follow-up of patients with ectodermal dysplasia. (Turk Arch Ped 2012; 47: 72-5)

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Cleft Palate, with a Peculiar Limb Deformity

El-Darouti

2012

Challenging Cases in Dermatology

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Management of Evaporative Dry Eye in Ectrodactyly-Ectodermal Dysplasia-Clefting Syndrome

Abstract: Low dose lipid base ointment application may be a promising treatment modality for the ocular surface disease in ectrodactyly-ectodermal dysplasia (EEC)-clefting syndrome, which seems to help in alleviating the subjective complaints and in improving the objective clinical findings.

Cited by 13 publications

References 24 publications

Ophthalmic Manifestations in Patients with Ectodermal Dysplasia Syndromes

Ophthalmic Manifestations in Patients with Ectodermal Dysplasia Syndromes

Ektodermal displazili olgularda konjonktiva bası sitolojisi

Cleft Palate, with a Peculiar Limb Deformity

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