|Year : 2016 | Volume
| Issue : 2 | Page : 137-139
Congenital bilateral eyelid imbrication in a neonate: A rare case
Manpreet Kaur1, Shakeen Singh1, Manpreet Singh2
1 Department of Ophthalmology, Sri Guru Ram Das Institute of Medical Sciences and Research, Amritsar, India
2 Department of Ophthalmic Plastic Surgery, Sohana Charitable Eye Hospital, Mohali, Punjab, India
|Date of Web Publication||8-Apr-2016|
Department of Ophthalmology, Sri Guru Ram Das Institute of Medical Sciences and Research, Amritsar, Punjab
Source of Support: None, Conflict of Interest: None
A newborn male child with polycythemia had watering and discharge from both eyes. In antenatal period, the mother had severe oligohydramnios. Clinically, both upper eyelids completely overlapped the lower and on forceful mechanical opening, spontaneous eversion was observed. The review of pathogenesis and management of eyelid imbrication syndrome is provided in this report.
Keywords: Eyelid imbrication syndrome, floppy eyelid syndrome, management, polycythemia
|How to cite this article:|
Kaur M, Singh S, Singh M. Congenital bilateral eyelid imbrication in a neonate: A rare case. J Clin Neonatol 2016;5:137-9
| Introduction|| |
Eyelid imbrication syndrome (EIS) is an abnormality of eyelid apposition in which the upper eyelid overrides the lower.  An acquired variant of EIS is found occasionally in adults who have floppy/lax eyelids or those who underwent lateral canthal strip tightening procedure for lower eyelid laxity. Floppy eyelids are often found in obese patients having slept disorders or sleep apnea syndrome. Surgical tightening of the upper eyelids is required in patients developing the secondary ocular surface disease. 
The congenital variety of EIS is extremely rare as only 4 cases are reported till date on PubMed search with keywords-congenital, eyelid imbrication. We hereby, report the 5 th case of literature (3 rd Indian origin) with bilateral congenital eyelid imbrication associated with polycythemia and respiratory distress.
| Case Report|| |
A 1-day-old male child was referred to us with an excessive discharge and watering from both eyes since birth. The child was born after an uneventful cesarean section at 37 weeks of pregnancy. An antenatal history of severe oligohydramnios was present, and the child was admitted to neonatal intensive care unit of our hospital due to respiratory distress. The local examination revealed bilateral upper eyelid boggy edema and erythema along with complete overlap over lower eyelids [Figure 1]a. Lower eyelids were not visible along the entire length. On measuring, the overlap of upper eyelids was around 4 mm each side [Figure 1]b. The horizontal and vertical (mid-point) dimensions of upper eyelids were 23 mm and 8 mm, respectively [Figure 2]. The eyelashes were matted with discharge.
|Figure 1: (a) Bilateral upper eyelid imbrication is noted clinically. The lower eyelid margin and eyelashes are not visible throughout the horizontal length. An attempt to lift left upper eyelid does not reveal the lower eyelid/eyelashes. (b) On mechanically lifting the upper eyelid, the extent of overlap is distinguished by soggy lower eyelid pretarsal skin (row of arrows). (c) A spontaneous eyelid eversion is noticed secondary to orbicularis oculi spasm (the soggy lower eyelid pretarsal skin is masked due to muscle contraction)|
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|Figure 2: (a) Measurement of horizontal length of the left upper eyelid -23 mm. (b) The mid-point vertical length measured is -8 mm|
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Both the upper eyelids were floppy (left >right) and on attempting a mechanical eyelid opening, the eyelids spontaneously everted showing hyperemic tarsal conjunctiva [Figure 1]c. The eversion was sustained until mechanically corrected secondary to forceful spasmodic contractions of orbicularis oculi. The pretarsal region skin of lower eyelids was soggy and blanched secondary to mechanical pressure and lacrimation. After retracting the eyelids with Desmarres retractor and torch light examination, the conjunctiva and cornea grossly appeared normal.
The systemic examination by pediatrician revealed polycythemia with hemoglobin (Hb) and packed cell volume values being 17.1 g% (normal, 13-16 g%) and 49.6% (normal, 40-54%), respectively. There was no cyanosis, icterus or pallor. The child underwent a partial exchange for the polycythemia.
The conjunctival swab showed no growth of pyogenic organisms. The child was given topical lubricating (carboxymethylcellulose 0.5%) and antibiotic (tobramycin 0.3%) eye drops 4 times/day. The upper eyelid overlap or imbrication resolved spontaneously after 4-5 days along with the resolution of eyelid edema and erythema [Figure 3]. At 7 th day of admission, the patient was discharged as Hb of child was 14 g% with normal respiratory rate. Topical antibiotic and lubricating eye-drops were discontinued after 1 week and 3 weeks, respectively.
|Figure 3: At 5th day of topical lubricating treatment, the upper and lower eyelid margins rests normally and the lower eyelashes are visible normally. The upper eyelid erythema has also reduced|
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| Discussion|| |
The spontaneous resolution of congenital EIS must have been a strong causative factor for its scarce literature. This self-limiting, bilateral rare condition is associated with lax eyelids in congenital variety while the acquired ones need a definitive surgical correction. An association with Down's syndrome is suggested but the child being reported here had no suggestive features of Down's syndrome. 
Previous literature on this benign and idiopathic condition has highlighted an early presentation after birth (mean 22.75 h; range 3-48 h), no gender predilection, lax and elongated canthi, spontaneous eversion of eyelids, and spontaneous recovery. The amount of upper eyelid override varied from >1 to 6 mm (mean 2.56 mm) which in our case was more than the average reported. The upper tarsal conjunctival hyperemia is secondary to the direct contact with eyelashes of the lower eyelid. ,,,, This might lead to the beginning of the vicious cycle of irritation, neuronal spasm of orbicularis, and more override of the upper eyelid. This makes the eyelids prone to spontaneous eversion as the orbicularis remains in constant subclinical spasm. This is a prominent feature describing the effect of vicious cycle which was reported by Chandravanshi et al., De Silva et al. and Odat and Hina. ,,
Treatment of ocular surface with a lubricant reduces the intensity of irritation caused by the lower eyelid cilia and breaks the vicious cycle. Along with the hypothesized growth of soft (eyelids) and bony (orbit) tissue, the reduced tone of orbicularis helps in attaining the normal anatomical position of the eyelids. , We think that the natural resolution of EIS could best possibly be explained by the thicker lipid layer and more mucin content of tear film in infants as compared to adults. , This readily provides the lubrication hence spontaneous recovery of the congenital EIS. As the condition resolves on its own without any sequela that might be the reason of its scarce reporting in the scientific literature.
We hereby, describe this rarely reported and possibly common condition in newly born infants and try to explain the pathomechanisms of congenital EIS. This is the first case of its kind associated with systemic polycythemia which could be explained by the presence of respiratory distress in the child.
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Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3]