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Figure 1.  Asymmetric Epiblepharon
Asymmetric Epiblepharon

Asymmetric epiblepharon was diagnosed when the difference of the extent of epiblepharon was more than one-fourth of the eyelid length. In the right eye, epiblepharon was found to involve less than one-fourth of the medial eyelid; however, in the left eye, the medial half of the eyelid was involved.

Figure 2.  Asymmetric Unilateral Epiblepharon in a Patient With Head Tilt
Asymmetric Unilateral Epiblepharon in a Patient With Head Tilt

A, Head tilt to the right side in a girl. B, On slitlamp examination, epiblepharon was found to be asymmetric, with greater severity in the head-tilted right side.

Table.  Symmetry of Epiblepharon According to the Cause of Head Tilt
Symmetry of Epiblepharon According to the Cause of Head Tilt
1.
Johnson  CC.  Epiblepharon.  Am J Ophthalmol. 1968;66(6):1172-1175.PubMedGoogle ScholarCrossref
2.
McCord  CD, Tanenbaum  M, Nunery  WR.  Oculoplastic Surgery. 3rd ed. New York, NY: Raven Press; 1995:221-222.
3.
Lim  WK, Rajendran  K, Choo  CT.  Microscopic anatomy of the lower eyelid in Asians.  Ophthal Plast Reconstr Surg. 2004;20(3):207-211.PubMedGoogle ScholarCrossref
4.
Levitt  JM.  Epiblepharon and congenital entropion.  Am J Ophthalmol. 1957;44(1):112-113.PubMedGoogle ScholarCrossref
5.
Erickson  BP, Lee  WW.  Epiblepharon-induced head tilt masquerading as torticollis.  Ophthal Plast Reconstr Surg. 2015;31(1):e8-e9.PubMedGoogle ScholarCrossref
6.
World Medical Association.  World Medical Association Declaration of Helsinki: ethical principles for medical research involving human subjects.  JAMA. 2013;310(20):2191-2194.PubMedGoogle ScholarCrossref
7.
Sundar  G, Young  SM, Tara  S, Tan  AM, Amrith  S.  Epiblepharon in East Asian patients: the Singapore experience.  Ophthalmology. 2010;117(1):184-189.PubMedGoogle ScholarCrossref
8.
Kushner  BJ.  The influence of head tilt on ocular torsion in patients with superior oblique muscle palsy.  J AAPOS. 2009;13(2):132-135.PubMedGoogle ScholarCrossref
9.
Tomczak  KK, Rosman  NP.  Torticollis.  J Child Neurol. 2013;28(3):365-378.PubMedGoogle ScholarCrossref
10.
Clarren  SK, Smith  DW, Hanson  JW.  Helmet treatment for plagiocephaly and congenital muscular torticollis.  J Pediatr. 1979;94(1):43-46.PubMedGoogle ScholarCrossref
11.
Santiago  AP, Rosenbaum  AL.  Dissociated vertical deviation and head tilts.  J AAPOS. 1998;2(1):5-11.PubMedGoogle ScholarCrossref
12.
Jampolsky  A.  Management of vertical strabismus.  Trans New Orleans Acad Ophthalmol. 1986;34:141-171.PubMedGoogle Scholar
13.
Johnson  CC.  Epicanthus and epiblepharon.  Arch Ophthalmol. 1978;96(6):1030-1033.PubMedGoogle ScholarCrossref
14.
Park  JI.  Modified Z-epicanthoplasty in the Asian eyelid.  Arch Facial Plast Surg. 2000;2(1):43-47.PubMedGoogle ScholarCrossref
15.
Park  JI.  Z-epicanthoplasty in Asian eyelids.  Plast Reconstr Surg. 1996;98(4):602-609.PubMedGoogle ScholarCrossref
16.
Jones  MC.  Unilateral epicanthal fold: diagnostic significance.  J Pediatr. 1986;108(5, pt 1):702-704.PubMedGoogle ScholarCrossref
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Original Investigation
August 2016

Association Between Head Tilt and Asymmetric Epiblepharon

Author Affiliations
  • 1Department of Ophthalmology, Seoul National University Hospital, Seoul, Korea
  • 2Department of Ophthalmology, Seoul Metropolitan Government Seoul National University Boramae Medical Center, Seoul, Korea
  • 3Department of Ophthalmology, Seoul National University Bundang Hospital, Seongnam, Korea
  • 4Department of Ophthalmology, Hallym University Sacred Heart Hospital, Anyang, Korea
JAMA Ophthalmol. 2016;134(8):870-872. doi:10.1001/jamaophthalmol.2016.1375
Abstract

Importance  Epiblepharon is known to be bilateral and symmetric. However, asymmetric epiblepharon is frequently observed in patients with head tilt and, to date, this condition has not been investigated.

Objectives  To evaluate the clinical features of epiblepharon in patients with head tilt and to analyze the association between the direction of head tilt and epiblepharon asymmetry.

Design, Setting, and Participants  The medical records of 1074 Korean children who received a diagnosis of epiblepharon between January 1, 2006, and October 31, 2013, at Seoul National University Bundang Hospital were retrospectively reviewed. The data collected included sex, age, extent and asymmetric presentation of epiblepharon, and direction and cause of head tilt.

Main Outcomes and Measures  The presence of epiblepharon asymmetry in patients with head tilt as well as the association between the direction of head tilt and the side having more severe epiblepharon.

Results  Of 1074 patients (536 boys; mean [SD] age, 5.4 [2.6] years) with epiblepharon, 38 individuals (3.5%) showed head tilt. The causes of head tilt were superior oblique palsy (18 patients [47.4%]), congenital muscular torticollis (10 [26.3%]), dissociated vertical deviation (3 [7.9%]), and unknown (7 [18.4%]). Asymmetric epiblepharon was more common in children with vs without head tilt (34 [89.5%] vs 80 [7.7%]; P < .001, Fisher exact test). Of the 34 patients with head tilt and asymmetric epiblepharon, the direction of head tilt was consistent with the side having more severe epiblepharon in 29 individuals (85.3%) and inconsistent in 5 individuals (14.7%) (P = .009).

Conclusions and Relevance  These data suggest that, in patients with epiblepharon and head tilt, epiblepharon is most often asymmetric and is severe in the head-tilted side. These results suggest that patients with asymmetric epiblepharon should be evaluated for head tilt.

Introduction

Epiblepharon is a condition in which a fold of redundant skin and the underlying pretarsal orbicularis muscle overlaps the eyelid margin, pushing the eyelashes against the cornea. This condition can result in corneal erosion and increased astigmatism.1-3 Epiblepharon is most often bilateral and symmetrical.4 However, we have often observed asymmetric epiblepharon in our clinical practice, and it is more frequently found in patients with head tilt. To the best of our knowledge, other than a single case report,5 the presence and symmetry of epiblepharon in patients with head tilt has not been evaluated. In the present study, we investigated epiblepharon symmetry in patients with head tilt and analyzed the association between the 2 conditions.

Box Section Ref ID

Key Points

  • Question What is the prevalence of epiblepharon asymmetry in patients with head tilt?

  • Findings In this study of medical records of children, most patients with head tilt showed asymmetric epiblepharon, which was more severe in the head-tilted side.

  • Meaning Evaluation of head tilt should be considered when patients with asymmetric epiblepharon are encountered.

Methods

This study retrospectively reviewed the medical records of 1074 patients who had received a diagnosis of epiblepharon between January 1 2006, and October 31, 2013, at Seoul National University Bundang Hospital. Using electronic medical records and medical photographs, we collected data on sex, age, extent and asymmetric presentation of epiblepharon, direction of head tilt, and cause of head tilt. All of the surgical corrections of epiblepharon had been performed by 1 surgeon (N.K.).

The presence and direction of head tilt were identified by the same physician (N.K.) and the causes of head tilt, including superior oblique palsy (SOP), congenital muscular torticollis, and dissociated vertical deviation (DVD), were evaluated. Superior oblique palsy was diagnosed when inferior oblique overaction and superior oblique underaction were present. Congenital muscular torticollis was diagnosed by a pediatric orthopedic surgeon when the patient had unilateral sternocleidomastoid muscle hypertrophy or spasticity. Dissociated vertical deviation was characterized by slow vertical drifting of the nonfixating eye when the other eye was fixating on a target. If there was no definite cause of head tilt, it was classified as unknown origin.

On a thorough literature search, we found no definition of asymmetric epiblepharon. We defined asymmetric epiblepharon as a difference of the extent of epiblepharon between the 2 eyelids of more than one-fourth of the eyelid length (Figure 1). The degree of difference of epiblepharon or the degree of head tilt was not evaluated. The protocol for this study was approved by the institutional review board of Seoul National University Bundang Hospital, with waiver of the need for informed consent. The conduct of the study adhered to the tenets of the Declaration of Helsinki.6 Statistical analyses were performed using SPSS, version 19.0 (SPSS Inc).

Results

All 1074 children with epiblepharon (536 boys [49.9%], 538 girls [50.1%]) included in the study were Korean and their mean (SD) age was 5.4 (2.6) years. Among them, 38 patients (3.5%) showed head tilt. No significant difference was identified in sex (P = .76, χ2 test) or age (5.35 vs 5.40 years, difference 0.05; 95% CI, −0.43 to 0.33; P = .74; 2-tailed, unpaired t test) between patients with and those without head tilt. The most common cause of head tilt was SOP (18 patients [47.4%]), followed by congenital muscular torticollis (10 [26.3%]) and DVD (3 [7.9%]). Of the 38 patients with head tilt, 23 individuals (60.5%) underwent subsequent surgical correction of epiblepharon. Head tilt persisted after epiblepharon was surgically corrected.

Among the 38 patients with head tilt, asymmetric epiblepharon was present in 34 individuals (89.5%); among the 1036 patients without head tilt, 80 children (7.7%) showed asymmetric epiblepharon. The proportion of asymmetric epiblepharon in the patients with head tilt was significantly higher than that in those without head tilt (P < .001, Fisher exact test) Of the 34 patients with head tilt and asymmetric epiblepharon, 29 children (85.3%) showed more severe epiblepharon in the head-tilted side (13 [44.8%] on the right side, 16 [55.2%] on the left side). The direction of the tilt was consistent with the side having more severe epiblepharon (P = .009, Fisher exact test).

Characteristics of Epiblepharon

In the SOP group, 17 of 18 children (94.4%) had asymmetric epiblepharon; in 16 of those patients (94.1%), the direction of tilt was consistent with the side having more severe epiblepharon. In the congenital muscular torticollis group, 9 of 10 patients (90.0%) had asymmetric epiblepharon and, in 7 of those individuals (77.8%), the direction of tilt was consistent with the side having more severe epiblepharon. In the DVD group, all 3 patients had asymmetric epiblepharon; in 2 of those 3 patients (66.7%), the direction of tilt was consistent with the side having more severe epiblepharon. In the group of 7 children (18.4%) with unknown origin of epiblepharon, 5 children (71.4%) had asymmetric epiblepharon; in 4 of those 5 children (80.0%), the direction of the tilt was consistent with the side having more severe epiblepharon. No significant difference was identified in the prevalence or concordance of the direction of head tilt and the side with more severe epiblepharon between the groups (P = .44 and P = .29, respectively, Fisher exact test) (Table).

Case Report

One of the girls presented with photophobia and eye rubbing. On slitlamp examination, asymmetric lower eyelid epiblepharon was found, which was more severe in the right eye. Superior oblique palsy was identified in the child’s left eye, causing her head to tilt to the right side. (Figure 2).

Discussion

We found that epiblepharon was asymmetric in most patients with head tilt (89.5%) in contrast to bilateral and symmetric epiblepharon in the general population.7 In addition, the direction of head tilt and the side with more severe epiblepharon was consistent in most of the patients (85.3%). This finding suggests that head tilt is strongly associated with epiblepharon asymmetry. However, epiblepharon asymmetry did not differ according to the cause of head tilt. Therefore, it can be deduced that head tilt—not the cause of head tilt—likely leads to epiblepharon asymmetry.

Patients with SOP usually tilt their heads to the contralateral side of the affected eye, which serves to decrease hypertropia.8 Patients with congenital muscular torticollis present with head tilt to the side of the shortened sternocleidomastoid muscle.9 If SOP or torticollis is not treated, facial asymmetry frequently occurs. Therefore, for effective prevention of facial asymmetry, early diagnosis and treatment are important.10 Regarding DVD, Santiago and Rosenbaum11 reported that head tilt improved after strabismus surgery; Jampolsky12 found that anomalous head posture might be due to a superior rectus contracture occurring after prolonged DVD. Further research is needed to determine the underlying mechanism of this type of head tilt.

We are aware of only 1 case report5 describing a patient with head tilt and asymmetric epiblepharon; the head tilt resolved once epiblepharon was repaired, implying that asymmetric epiblepharon might cause head tilt. In contrast, our study found that head tilt can cause asymmetric epiblepharon; head tilt persisted even after epiblepharon was surgically corrected.

There are several possible mechanisms by which head tilt causes asymmetric epiblepharon. Patients with head tilt and epiblepharon generally have facial asymmetry, which leads to unilateral epicanthal folds and an overriding skin fold on the tilted side, which in turn contributes to more severe epiblepharon on the same side. This theory is supported by previous reports13-15 that patients with prominent epicanthal folds frequently experience severe ocular irritation due to contact of the eyelash with the cornea. Jones16 found that most cases of congenital torticollis are accompanied by unilateral epicanthal folds on the side of the head tilt and suggested that a unilateral epicanthal fold could be an early diagnostic indicator of congenital torticollis.

There are several limitations to our study. First, as a retrospective review, a selection bias may be present. Second, the severity and duration of head tilt were not evaluated. Mild head tilt of a short duration might not be sufficient to induce epiblepharon asymmetry. Prospective studies examining a larger number of patients with head tilt are warranted to investigate the effect of angle and duration of head tilt on epiblepharon asymmetry.

Conclusions

In this retrospective study of patients with epiblepharon and head tilt, most epiblepharon was asymmetric, with greater severity on the head-tilt side. These findings suggest that head tilt should be evaluated in patients with asymmetric epiblepharon.

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Article Information

Corresponding Author: Namju Kim, MD, Department of Ophthalmology, Seoul National University Bundang Hospital, 300 Gumi-dong, Bundang-gu, Seongnam, Gyeonggi-do 463-707, Korea (resourceful@hanmail.net).

Submitted for Publication: January 18, 2016; final revision received March 30, 2016; accepted April 3, 2016.

Published Online: May 26, 2016. doi:10.1001/jamaophthalmol.2016.1375.

Author Contributions: Drs Seol and Choung are considered equivalent first authors. Dr Kim had full access to all the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.

Study concept and design: Seol, Choung, Kim, Khwarg.

Acquisition, analysis, or interpretation of data: Seol, Choung, Kim, Lee.

Drafting of the manuscript: Seol, Kim, Khwarg.

Critical revision of the manuscript for important intellectual content: Choung, Kim, Lee.

Statistical analysis: Seol, Choung.

Administrative, technical, or material support: Choung.

Study supervision: Choung, Kim, Lee, Khwarg.

Conflict of Interest Disclosures: All authors have completed and submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest and none are reported.

Additional Contributions: We thank the patients for granting permission to publish this information.

References
1.
Johnson  CC.  Epiblepharon.  Am J Ophthalmol. 1968;66(6):1172-1175.PubMedGoogle ScholarCrossref
2.
McCord  CD, Tanenbaum  M, Nunery  WR.  Oculoplastic Surgery. 3rd ed. New York, NY: Raven Press; 1995:221-222.
3.
Lim  WK, Rajendran  K, Choo  CT.  Microscopic anatomy of the lower eyelid in Asians.  Ophthal Plast Reconstr Surg. 2004;20(3):207-211.PubMedGoogle ScholarCrossref
4.
Levitt  JM.  Epiblepharon and congenital entropion.  Am J Ophthalmol. 1957;44(1):112-113.PubMedGoogle ScholarCrossref
5.
Erickson  BP, Lee  WW.  Epiblepharon-induced head tilt masquerading as torticollis.  Ophthal Plast Reconstr Surg. 2015;31(1):e8-e9.PubMedGoogle ScholarCrossref
6.
World Medical Association.  World Medical Association Declaration of Helsinki: ethical principles for medical research involving human subjects.  JAMA. 2013;310(20):2191-2194.PubMedGoogle ScholarCrossref
7.
Sundar  G, Young  SM, Tara  S, Tan  AM, Amrith  S.  Epiblepharon in East Asian patients: the Singapore experience.  Ophthalmology. 2010;117(1):184-189.PubMedGoogle ScholarCrossref
8.
Kushner  BJ.  The influence of head tilt on ocular torsion in patients with superior oblique muscle palsy.  J AAPOS. 2009;13(2):132-135.PubMedGoogle ScholarCrossref
9.
Tomczak  KK, Rosman  NP.  Torticollis.  J Child Neurol. 2013;28(3):365-378.PubMedGoogle ScholarCrossref
10.
Clarren  SK, Smith  DW, Hanson  JW.  Helmet treatment for plagiocephaly and congenital muscular torticollis.  J Pediatr. 1979;94(1):43-46.PubMedGoogle ScholarCrossref
11.
Santiago  AP, Rosenbaum  AL.  Dissociated vertical deviation and head tilts.  J AAPOS. 1998;2(1):5-11.PubMedGoogle ScholarCrossref
12.
Jampolsky  A.  Management of vertical strabismus.  Trans New Orleans Acad Ophthalmol. 1986;34:141-171.PubMedGoogle Scholar
13.
Johnson  CC.  Epicanthus and epiblepharon.  Arch Ophthalmol. 1978;96(6):1030-1033.PubMedGoogle ScholarCrossref
14.
Park  JI.  Modified Z-epicanthoplasty in the Asian eyelid.  Arch Facial Plast Surg. 2000;2(1):43-47.PubMedGoogle ScholarCrossref
15.
Park  JI.  Z-epicanthoplasty in Asian eyelids.  Plast Reconstr Surg. 1996;98(4):602-609.PubMedGoogle ScholarCrossref
16.
Jones  MC.  Unilateral epicanthal fold: diagnostic significance.  J Pediatr. 1986;108(5, pt 1):702-704.PubMedGoogle ScholarCrossref
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