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CASE REPORT
Year : 2014  |  Volume : 17  |  Issue : 2  |  Page : 248-250

Congenital upper lid eversion and severe chemosis in a new born


Department of Ophthalmology, University of Port Harcourt Teaching Hospital, Rivers State, Nigeria

Date of Acceptance28-Jun-2013
Date of Web Publication20-Feb-2014

Correspondence Address:
E A Awoyesuku
Department of Ophthalmology, University of Port Harcourt Teaching Hospital
Nigeria
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DOI: 10.4103/1119-3077.127569

PMID: 24553040

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   Abstract 

The purpose of this study is to report a case of bilateral congenital eversion of the upper eyelid in an 8 h- old male baby. JD is an 8 h-old male baby born by normal vaginal delivery after prolonged labor, to a 24-year-old primigravida. Pregnancy was said to be term but was complicated by pregnancy-induced hypertension. A midwife delivered the baby and it was by spontaneous vertex delivery. He was said to have cried immediately after birth, but shortly afterward parents noticed a reddish fleshy swelling over the eyes and this made it difficult for them to see the eyeballs. The reddish swelling steadily increased in size necessitating their prompt referral to our center. On examination, a full term baby was seen with complete eversion of both upper eyelids and marked conjunctival chemosis. Ocular examination revealed normal globes with healthy cornea and briskly reactive pupils. The child was admitted and started on 3-hourly chloramphenicol ointment, ciloxan eye drop (ciprofloxacin hydrochloride United States Pharmacopeia equivalent to ciprofloxacin 0.3% w/v) - 8 times daily and 5% hypertonic saline patch over the chemosed conjunctiva. The pediatrician was also invited to co-manage the patient. After 9-days of treatment, the conjunctival chemosis fully resolved and lids reverted back to normal position. At 4-week follow-up, the lids continued to maintain their normal position and child could now open eyelids spontaneously. Congenital upper eyelid eversion is a rare clinical entity even though it is said to be commoner in blacks. Knowledge of its complete resolution with conservative management will help in future management of such cases thereby preventing complications that may arise from poorly treated cases.

Keywords: Congenital, eversion, upper eyelids


How to cite this article:
Awoyesuku E A, Pedro-Egbe C N, Sibeudu O A. Congenital upper lid eversion and severe chemosis in a new born. Niger J Clin Pract 2014;17:248-50

How to cite this URL:
Awoyesuku E A, Pedro-Egbe C N, Sibeudu O A. Congenital upper lid eversion and severe chemosis in a new born. Niger J Clin Pract [serial online] 2014 [cited 2014 Oct 2];17:248-50. Available from: http://www.njcponline.com/text.asp?2014/17/2/248/127569


   Introduction Top


Congenital upper eyelid eversion is a rare disorder of unknown etiology. The condition is typically bilateral [1] but unilateral cases have also been reported [2] , with a higher incidence observed in black newborns, infants with trisomy 21, and children of multiparous mothers. [3] Congenital lid eversion can be due to infections, inflammation and birth trauma; or may be associated with systemic anomalies such as Down syndrome. [4] It has also been associated with prolonged and difficult labor [5] though in some case, no known cause has been found. [6] Congenital eversion of the upper eyelid was first described by Adams [7] in 1896, and he called it "double congenital ectropion". Since the first reported case, many more have appeared in the literature. In Nigeria, several authors have reported cases of congenital upper eyelid eversion. In South-South Nigeria, Osahon and Ibadin [8] in Benin City, and Dawodu [9] in the Delta State each reported cases of bilateral congenital upper eyelid eversion in male neonates. In the same vein, in Western Nigeria, Adeoti et al. [10] in Ibadan reported three cases of bilateral congenital lid eversion in males who presented at between 4 h and 4 days of birth. Similarly, Omolase et al. [11] in Owo reported a case in a male neonate who presented within 3 h of birth. However, no such case/cases have been reported in Rivers state or other neighboring states in the South-South.

We hereby report a case of bilateral congenital upper lid eversion with severe conjunctival chemosis in an 8- h old male baby.


   Case Report Top


JD is an 8- h old male baby born by normal vaginal delivery to a 24- year- old primigravida after a full term pregnancy complicated by pregnancy-induced hypertension. Mother was offered caesarean section but declined and eventually delivered in a private clinic after labor that lasted over 17-h. Baby was delivered by spontaneous vertex delivery and was said to have cried immediately after birth. Shortly after birth, parents noticed reddish swelling over the eyes, which had been steadily increasing in size thereby necessitating their prompt referral.

Examination revealed a healthy looking baby, not in any obvious distress but with complete eversion of both upper eyelids and marked conjunctival chemosis [Figure 1]. Attempts at repositioning the upper eyelids failed. The lids were then gently parted to examine the eyeball. Both globes appeared to be of normal size with clear corneas and pupils that were briskly reactive to light. The child was admitted into the eye ward and started on 3-hourly ointment chloramphenicol, ciproxin eye drop (ciprofloxacin hydrochloride United States Pharmacopoeia equivalent to ciprofloxacin 0.3% w/v) - 8 times daily and 5% hypertonic saline patching over the chemosed conjunctiva. The pediatrician was also invited to co-manage the patient. At 1- week follow- up, the condition improved and the child could open his eyes partially. The eyelids could be manually repositioned but everted back on releasing. At 9 days of admission the chemosis was completely resolved and baby could fully open both eyes [Figure 2] though they tended to evert on forceful opening while trying to examine the patient. The lids however reverted spontaneously. Baby was then discharged from the hospital to be followed-up on an outpatient basis.

At follow-up, 2 weeks after the baby was first seen, the condition was found to have improved greatly and mother reported that baby could now open his eyes spontaneously though on an intermittent basis. Examination revealed that the chemosis had completely resolved and the eyelids were now well- apposed. The mother was then advised to continue the application of topical lubricants. At 4 weeks follow-up, the eyelids continued to maintain their normal position and child could fixate and follow the light, and the conjunctivae and corneas were normal [Figure 3].
Figure 1 : Before treatment

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Figure 2 : 9 days after treatment

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Figure 3 : One month after treatment

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   Discussion Top


Congenital lid eversion is a rare clinical event but since the first description by Adams [7] several reports have appeared in the literature. The condition is typically bilateral, but unilateral cases have been described. The underlying reason for eversion remains obscure, several possible mechanisms have been proposed and associations have been recognized. The incidence appears higher in black infants [3] , trisomy 21 [8] and infants born with collodion skin disease. [12] Orbicularis hypotonia, birth trauma, vertical shortening of the anterior lamella or vertical elongation of the posterior lamella of the eyelid and failure of the orbital septum to fuse with the levator aponeurosis have all been proposed as possible mechanisms responsible for the eversion. [13]

Chemosis and prolapse of the conjunctiva may occur secondary to venous stasis during delivery, causing eversion of the eyelids. [14] This may be the case with our patient who was delivered following prolonged labor in a woman with pregnancy-induced hypertension. On the other hand, Ren and Liu [2] reported the presence of congenital upper eyelid eversion in baby born by caesarean section. This means that other factors, other than prolonged labor may be responsible for the lid eversion in Ren's case. Once everted, orbicularis spasm may act as sphincter leading to a vicious cycle of conjunctival strangulation and edema secondary to venous stasis. The prolapsed conjunctiva usually protects cornea thereby preventing or reducing corneal complications. [15] Our baby appeared normal phenotypically even though no deoxyribonucleic acid test was carried out to rule out Down's syndrome.

Several management modalities both conservative and surgical have been proposed for management of congenital eyelid eversion. Conservative treatment involves the use of ointments and lubricants, topical prophylactic antibiotics and patching with 5% hypertonic saline. [10],[16] An attempt to explain the mechanism by which the 5% hypertonic saline soaked gauze dressing worked was put forward by Adeoti et al. [10] as osmosis of fluid from edematous tissues through the semi permeable subconjunctival membrane, encouraged by the hyper tonicity of the patch. This may explain the resolution of the edema and subsequent lid reversion as proposed by Voet et al. [17] Surgical treatment involves lateral tarsorhaphy with excision of redundant conjunctiva, fornix sutures, full thickness skin graft of the upper lid, and subconjunctival injection with hyaluronic acid. [2],[9],[11],[18]

Our patient was managed conservatively and despite the marked conjunctival chemosis and lid eversion at presentation, this resolved fully at 9- days post-presentation without any surgical intervention.


   Conclusion Top


Congenital upper eyelid eversion is a rare clinical entity even though it is said to be commoner in blacks. A normal anatomical outcome with good cosmetic results can be effectively achieved using topical lubricants, antibiotics and application of 5% hypertonic saline patch. Careful ocular examination should however be carried out first to rule out other ocular or systemic complications, in the absence of which conservative management can be instituted. Knowledge of its complete resolution with conservative management will help in future management of such cases thereby preventing complications that may arise from poorly treated cases.

 
   References Top

1.Duke-Elder S. Congenital deformities. In: Duke-Elder S, editor. System of Ophthalmology. Vol. 3. St. Louis: CV Mosby; 1963. p. 866-7.  Back to cited text no. 1
    
2.Ren L, Liu J. Congenital total ectropion of upper lid. Vestn Oftalmol 1999;115:32-4.  Back to cited text no. 2
    
3.Kronish JW, Lingua R. Pressure patch treatment for congenital upper eyelid eversion. Arch Ophthalmol 1991;109:767-8.  Back to cited text no. 3
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4.American Academy of Ophthalmology. Orbit, eyelid and lacrimal system. Basic and Clinical Science Course. New York: The Academy; 1998. p. 99-132.  Back to cited text no. 4
    
5.Erb A. Ein fall von Doppelseitigen Congenital Ectropion der Oberlider. Korresp Bl Schweiz Arz 1909;39:733-4.  Back to cited text no. 5
    
6.Young RJ. Congenital ectropion of the upper lids. Arch Dis Child 1954;29:97-100.  Back to cited text no. 6
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7.Adams AL. A case of double congenital ectropion. Med Fortn 1896;9:137-8.  Back to cited text no. 7
    
8.Osahon AI, Ibadin MO. Case Report: Congenital bilateral eyelid eversion and microphthalmia in co-existence with trisomy 21. Sahel Med J 2005;8:23-4.  Back to cited text no. 8
    
9.Dawodu OA. Total eversion of the upper eyelids in a newborn. Niger Postgrad Med J 2001;8:145-7.  Back to cited text no. 9
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10.Adeoti CO, Ashaye AO, Isawumi MA, Raji RA. Non-surgical management of congenital eversion of the eyelids. J Ophthalmic and Vis Res 2010;5:188-92.  Back to cited text no. 10
    
11.Omolase CO, Aina AS, Omolade EO. Congenital eversion of the upper eyelids. Asian J Ophthalmol 2008;10:236-7.  Back to cited text no. 11
    
12.Shapiro RD, Soentgen ML. Colloidin skin disease and everted eyelids. Postgrad Med 1969;45:216-9.  Back to cited text no. 12
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13.Al Hussain H, Al-Rajhi AA, Al-Qahtani S, Meyer D. Congenital upper eyelid eversion complicated by corneal perforation. Br J Ophthalmol 2005;89:771.  Back to cited text no. 13
    
14.Conlon MR, Sutula FC. Congenital eyelid anomalies. In: Albert DM, Jakobiac FA, editors. Principles and Practice of Ophthalmology. 2 nd ed. Vol 4. Philadelphia, PA: WB Saunders Co.; 1999. p. 3336.  Back to cited text no. 14
    
15.Meheshwari R, Meheshwari S. Congenital eversion of upper eyelids: Case report and management. Indian J Ophthalmol 2006;54:203-4.  Back to cited text no. 15
    
16.Stern EN, Campbell CH, Faulkner HW. Conservative management of congenital eversion of the eyelids. Am J Ophthalmol 1973;75:319-20.  Back to cited text no. 16
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17.Voet D, Voet JG, Pratt CW. Fundamentals of Biochemistry. New York. John Wiley and Sons; 1999. p. 30.  Back to cited text no. 17
    
18.Ibechukwu BI, Iya CK. Early surgical management of two cases of bilateral congenital prolapse of the superior conjunctival fornices with lid eversion. West Afr J Med 1991;10:433-8.  Back to cited text no. 18
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    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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