Floppy eyelid syndrome: Clinical features and the association with obstructive sleep apnea

doi:10.1016/j.sleep.2005.07.001

Sleep Medicine

Volume 7, Issue 2, March 2006, Pages 117-122

https://doi.org/10.1016/j.sleep.2005.07.001 Get rights and content

Abstract

Floppy eyelid syndrome (FES) is a recently recognized entity, originally described in obese men with easily everted upper eyelids and chronic ocular irritation. Although the eyelids are primarily involved, other ocular structures such as the conjunctiva and cornea are frequently affected and cause much of the morbidity. Recent studies have found an interesting association with obstructive sleep apnea (OSA) and with chronic diseases such as hypertension and diabetes. The association of FES with OSA has both diagnostic and therapeutic implications; FES may be a presenting symptom in patients with undiagnosed OSA, and, in addition, treatment of obesity and OSA may have a favorable effect on the course of FES.

Introduction

In 1981, Culberston and Ostler [1] were the first to describe a unique syndrome characterized by easily everted floppy upper eyelids and papillary conjunctivitis in obese middle-aged men, which they named as the floppy eyelid syndrome (FES). They found that the floppy eyelids everted spontaneously during sleep, and as a consequence of nocturnal exposure, these patients developed chronic conjunctivitis of the upper lid [1]. The affected side seemed to correspond to the side that the patient usually slept on, and in bilateral cases, there was either no preferred side or the patient was sleeping face down.

Since that first report, a growing number of publications have reported a high prevalence of other ophthalmic pathologies in patients with FES; these include corneal abnormalities (superficial punctate keratopathy, corneal vascularization, keratoconus, corneal scars and ulcers) [2], [3], eyelid abnormalities (ptosis, lash ptosis, upper lid entropion, lower lid ectropion) [4], [5], [6], and glaucoma [7].

FES is also associated with systemic conditions, most frequently obesity, but also hypertension, ischemic heart disease, diabetes, and skin pathologies [8]. It is now well accepted that obstructive sleep apnea (OSA) is an important systemic condition that is closely associated with FES. Recent reports found a high prevalence of OSA in patients with FES and a high prevalence of floppy eyelids in patients with OSA [9], [10], [11], [12].

The aim of this report is to review the clinical manifestations and management of FES and to discuss its association with OSA and its role in management and prognosis for patients.

Section snippets

Eyelid anatomy

The skin of the eyelids is the thinnest of the body and has no subcutaneous fat. The orbicularis oculi muscle, which is one of the superficial muscles of facial expression, lies beneath the skin, and serves as the main protractor of the eyelids. The skin and the orbicularis muscle constitute the anterior lamella of the eyelid (Fig. 1). The tarsal plates, which are composed of dense fibrous tissue, are responsible for the structural integrity of the eyelids [13]. Each tarsal plate measures

Ophthalmic manifestations of FES

The spectrum of ophthalmic involvement by FES is wide. It may affect the eyelids, conjunctiva, cornea, and possibly result in blindness. Patients usually present with prolonged symptoms of non-specific ocular irritation, and a foreign body sensation. In addition, there may be significant mucous discharge, photosensitivity and redness [1], [2], [8].

The eyelids, which are designed to protect the ocular surface with their rigidity, are the main organ affected by the FES. The upper eyelids are

Pathogenesis of FES

The pathogenic mechanism responsible for the pathologic changes in FES is not fully elucidated. Culberston and Ostler [1] postulated that the loss of tarsal elasticity and difficulties in nocturnal complete eye closure, resulting from spontaneous eversion of the eyelids during the night, are the main pathogenic factors leading to a series of events, resulting in mechanical irritation of the conjunctiva, conjunctival keratinization, and secondary superficial corneal lesions. The mechanical

Systemic findings in patients with FES

Although FES denotes an ophthalmic condition, it is associated with systemic diseases, some of which can be life-threatening.

Obesity is the major systemic pathology described in a high percentage of patients with FES. In the original description by Culberston and Ostler [1], all 11 patients were overweight and had excessive facial fat. The authors postulated that the facial configuration of these patients increased the contact of the eyelids with the sheet or pillow, resulting in enhanced

FES and obstructive sleep apnea

Gonnering and Sonneland [17] were the first to describe a patient with FES and symptoms suggestive of OSA, but the actual report on a possible association between these two disorders was published three years later by Woog et al. [9] They described three patients with FES who had OSA and speculated that it could be related to an abnormality of the facial connective tissue. McNab [8] was the first to publish a large series of patients with FES who were diagnosed with OSA. In his first series in

Making the diagnosis of FES

Although the diagnosis of FES is usually made by ophthalmologists, there are important anamnestic details and findings in the clinical examination which may suggest this diagnosis when the patient is evaluated by a non-ophthalmologist. The patients are usually obese middle-aged males, who will complain of prolonged ophthalmic symptoms such as unilateral or bilateral eye irritation, a foreign body sensation and tearing. In most cases there is an associated eye redness and mucoid discharge, which

Management of patients with FES

Treatment of patients with FES is based on medical measures for symptom relief and on surgical procedures when conservative treatment proves insufficient.

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Citation Excerpt :
FES was first described in association with OSA in 1981 [1]. There have been many reports of this association in case reports and case series since then [2–8]. There are also reports of improvement of FES after PAP therapy [4,8].
Serious corneal complications and undiagnosed floppy eyelid syndrome; A case series and a 10-year retrospective review
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To describe three individuals with severe keratitis and a substantial delay before floppy associated eyelid syndrome (FES) was identified, and to estimate the prevalence of severe corneal disease in individuals with FES.
We defined severe keratitis as corneal ulceration, vascularization or scar that affected vision. We recorded the clinical characteristics, the duration of symptoms before the diagnosis of FES, subsequent management and outcome. Then, to determine the proportion of individuals with FES who had severe corneal disease, we interrogated the Moorfields Eye Hospital electronic patient record (EPR) for the diagnosis of FES made in the ten-year interval from 2008.
Three individuals presented with severe progressive keratitis (median duration of symptoms 19 months, range 2–48 months). All were male and with a high body mass index (BMI, range 38.9–41.2). In each the etiology of the keratitis was unclear before FES was identified. All had very lax lids and were aware they had periods of lid malposition during sleep. None mentioned symptoms of obstructive sleep apnoea (OSA) until they or their partner were directly questioned. The management of keratitis included both medical and surgical corneal treatments, with tarsorrhaphy and lid shortening surgery. We identified an additional 104 cases of FES from the EPR, of which 4 (3.8%) had severe keratitis.
FES can be missed unless signs of lid laxity are directly elicited. A delay in diagnosis can result in clinical deterioration, with unnecessary investigations and treatments. An assessment for FES should be included as part of the evaluation of individuals with severe or chronic keratitis.
Floppy eyelid syndrome in stickler syndrome
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Histologically, collagen appears normal in FES eyelids, while elastin is decreased and disrupted.9 It seems possible that the collagen mutation contributed to the development of FES in our patient, though there are data that suggests that OSA might cause FES by its effect on metalloproteinases and development of a proinflammatory response.10 Even if OSA is not the cause of FES, there is certainly an association between the two.
To report the possible connection between Stickler syndrome and floppy eyelid syndrome.
A 36-year-old man with genetically confirmed Stickler syndrome presented with prominent bilateral eyelid laxity consistent with floppy eyelid syndrome. He had a high-arched palate and reported years of apneic episodes during sleep consistent with obstructive sleep apnea (OSA).
To our knowledge, there have been no prior reported cases of floppy eyelid syndrome in Stickler syndrome patients. However, many patients with Stickler syndrome have palate abnormalities, which are associated with a higher risk of OSA. Given the known association between floppy eyelid syndrome and OSA, this case highlights the potential role for the ophthalmologist in identifying these patients and referring them for a sleep study if floppy eyelid syndrome is identified.
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Review articleFloppy eyelid syndrome: Clinical features and the association with obstructive sleep apnea

Abstract

Introduction

Section snippets

Eyelid anatomy

Ophthalmic manifestations of FES

Pathogenesis of FES

Systemic findings in patients with FES

FES and obstructive sleep apnea

Making the diagnosis of FES

Management of patients with FES

Am J Ophthalmol

Ophthalmology

Ophthalmology

Am J Ophthalmol

Am J Ophthalmol

Am J Ophthalmol

Ophthalmology

Ophthalmology

Ophthalmology

Am J Ophthalmol

Ophthalmology

Review article
Floppy eyelid syndrome: Clinical features and the association with obstructive sleep apnea