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Acquired Lower Eyelid Epiblepharon
The patients included a 39-year-old man (Patient 1), a 15-year-old girl (Patient 2), and her 41-year-old uncle (Patient 3). We analyzed the eye movements of all patients with tangent screen and electro-oculography. Additionally, the lateral and superior rectus muscles were examined electromyographically in Patient 1. The three patients had double vision in upgaze or lateral gaze, or both. The ophthalmologic examina tion and eye movement analysis with tangent screen showed V-incomitance and an abduction deficit in all patients. Patient 2 showed a retraction of the globe in upgaze. Figure 1 shows the horizontal and vertical positions of the left and right eyes of Patient 2 during vertical saccades. The electro-oculographic pattern disclosed a twitch abduction of the occluded eye on upward saccades, followed by a postsaccadic back drift and a slight, slower abduction on downward saccades. This phenomenon was found in the nonfixing eye regardless of which eye was fixing. The other two patients had similar electro-oculographic findings in both eyes. Electromyographic findings of the lateral and superior rectus muscles were recorded simultane ously in Patient 1. Figure 2 shows that lateral rectus muscle firing activity was elicited during upgaze and downgaze. This finding indicates the presence of synergistic innervation between the lateral rectus muscle and elevating and depressing muscles. Among the few cited cases of vertical Duane's retraction syndrome in the literature, our patients shared great similarities with one described by Kommerell and Bach.3 The origin of Duane's retraction syndrome is still controversial in the literature. Most investigators suggest that a teratogenic event during the second month of gestation is the most likely cause of Duane's retraction syndrome. Two of our patients were close relatives, uncle and niece; therefore, a genetic compo nent is possible in these patients. We conclude that V-incomitance, limitation of abduction, retraction of the globe on upgaze, and twitch abduction on vertical saccades belong to a variant of Duane's retraction syndrome with synergis tic innervation between the abducens nerve and the upper and lower divisions of the oculomotor nerve. Our report contributes three additional cases of this rare variant and documents electromyographic evi dence of synergistic innervation between the lateral rectus muscle and ipsilateral vertical-acting muscles. VOL.122, N o . 3
REFERENCES 1. Hotchkiss M, Miller N, Clark A, Green W. Bilateral Duane's retraction syndrome. Arch Ophthalmol 1980;98:870-4. 2. Huber A. Electrophysiology of retraction syndromes. Br J Ophthalmol 1974;58:293-300. 3. Kommerell G, Bach M. A new type of Duane's syndrome. Neuro-ophthalmology 1986;6:159-644. Pruksacholawit K, Ishikawa S. Atypical vertical retraction syndrome. J Pediatr Ophthalmol 1976;13:215-20. 5. Khodadoust A, von Noorden G. Bilateral vertical retraction syndrome. Arch Ophthalmol 1967;78:606-12.
Acquired Lower Eyelid Epiblepharon Robert I. Park, M.D., and Dale R. Meyer, M.D. PURPOSE: Although congenital epiblepharon is a relatively common condition, particularly in Asians, acquired lower eyelid epiblepharon is rare. METHOD: We examined an 8-year-old boy who developed unilateral lower eyelid epiblepharon af ter a subciliary incisional approach for repair of a zygomatico-orbital fracture. RESULTS: The patient demonstrated an overrid ing fold of right lower eyelid skin and loss of the lower eyelid crease. At the time of surgical repair, disruption and scarring of the lower eyelid retrac tors, suborbicularis fascia, and orbital septum were noted. The epiblepharon was successfully corrected by plication of the retractors and skin to the tarsus. CONCLUSION: Disruption of the anterior subcu taneous fibers of the lower eyelid retractors (that is, capsulopalpebral fascia) contributes to epi blepharon.
C
ONGENITAL EPIBLEPHARON IS A COMMON CONDItion, particularly in Asian populations. The affected eyelids demonstrate a horizontal fold of skin that overrides the eyelid margin and compresses the eyelashes into the cornea, producing irritation. Lower eyelid epiblepharon is more severe in downgaze and is
Accepted for publication March 29, 1996. Division of Orbital and Ophthalmic Plastic Surgery, Lions Eye Institute. Inquiries to Dale R. Meyer, M.D., Division of Orbital and Ophthal mic Plastic Surgery, Lions Eye Institute, 35 Hackett Blvd., Albany, NY 12208.
BRIEF REPORTS
449
Fig. 1 (Park and Meyer). Left, An 8-year-old boy with acquired right lower eyelid epiblepharon after subciliary approach for repair of right zygomatico-orbital fracture. Right, Right lower eyelid shows overriding horizontal fold of skin with loss of lower eyelid crease and compression of eyelashes into cornea. Note that the eyelid margin is in normal position.
Fig. 2 (Park and Meyer). Left, Intraoperative photograph; scissors point to the central band of scar tissue involving suborbicularis fascia, orbital septum, and lower eyelid retractors. Right, Postoperative photograph shows correction of epiblepharon.
typically accompanied by loss of the normal lower eyelid crease. With epiblepharon, unlike entropion, the eyelid margin is unaffected and retains its normal position.1'3 In contrast to congenital epiblepharon, acquired lower eyelid epiblepharon is quite rare.3 We present a case of acquired unilateral lower eyelid epiblepharon after surgical repair of a zygomaticoorbital (tripod) fracture via a subciliary incision. A n 8-year-old boy sustained a displaced right zygomatico-orbital fracture after a sledding accident in January 1995. Open reduction with internal fixa tion of the tripod fracture was performed by another surgeon. The surgical approach included a subciliary 450
skin incision 1 to 2 mm below the eyelash line, with dissection then continued in the preseptal plane to the inferior orbital rim. A miniplate was used for fixation of the fracture involving the inferior orbital rim. The inferior periosteal incision and the lower eyelid orbicularis muscle incision were closed with 4-0 chromic suture. The skin incision was not closed directly. The patient did well initially; however, during a 2la-month postoperative period, discomfort developed in his right eye. He was referred for examination. O n examination, visual acuity was 20/20 in both eyes; no extraocular muscle limitation was noted and there was no evidence of enophthal-
AMERICAN JOURNAL OF OPHTHALMOLOGY
SEPTEMBER
1996
mos. Examination of the right lower eyelid demon strated loss of distinction of the right lower eyelid crease with a roll of skin overriding the tarsus, compressing the eyelashes into the cornea (Fig. 1). The eyelid margin and tarsus remained in normal position in primary gaze and downgaze. Slit-lamp biomicroscopy disclosed superficial punctate keratopathy. Surgical exploration and repair of the lower eyelid epiblepharon was performed. An incision was made 1 to 2 mm below the eyelash line. Dissection disclosed a broad band of scar tissue in the central and medial portion of the eyelid, involving the suborbicularis fascia, the orbital septum, and the lower eyelid retractors and surrounding orbital fat (Fig. 2). There was no involvement of the lateral and more extreme medial aspect of the eyelid. The scar tissue was lysed and excised. The lower eyelid retractors were identi fied, freed from the surrounding scar tissue, and then advanced and plicated to the inferior tarsus with 5-0 polyglactin sutures. The skin and orbicularis were closed with interrupted 6-0 plain gut sutures, taking deep bites of the underlying tarsus and lower eyelid retractors to enhance the eyelid crease. Postoperatively, the epiblepharon was corrected with complete relief of symptoms and no recurrence for 12 months. Acquired lower eyelid epiblepharon is a rare condi tion that, to our knowledge, has only been reported once previously.5 The clinical findings in this case were identical to congenital epiblepharon. Although the cause of lower eyelid epiblepharon is debated, most evidence suggests a defect in the capsulopal-
VOL.122, No. 3
pebral fascia (that is, lower eyelid retractors).1'3 Ante rior extensions of the capsulopalpebral fascia to the overlying skin are thought to form the lower eyelid crease and to contribute to a normally functioning eyelid.14 The clinical and intraoperative findings in our case of acquired epiblepharon support the theory that disruption of anterior subcutaneous extensions of the lower eyelid retractors contributes to epiblepharon formation. Repair of the epiblepharon in this case by plication of the lower eyelid retractors and skin to the tarsal plate successfully corrected the epiblepharon and further supports this theory. Other types of eyelid malposition, predominantly lower eyelid retraction and ectropion, have been reported with transcutaneous subciliary eyelid approaches for repair of orbital fractures. Fewer such complications are seen with a transconjunctival approach.5 REFERENCES 1. Quickert MH, Wilkes DI, Dryden RM. Nonincisional correc tion of epiblepharon and congenital entropion. Arch Ophthalmol 1983;101:778-81. 2. Millman AL, Mannor GE, Putterman AM. Eyelid crease and capsulopalpebral fascia repair in congenital entropion and epiblepharon. Ophthalmic Surg 1994;25:162-5. 3. Jordan R. The lower eyelid retractors in congenital entropion and epiblepharon. Ophthalmic Surg 1993;24:494-6. 4- Hawes M], Dortzbach RK. The microscopic anatomy of the lower eyelid retractors. Arch Ophthalmol 1982;100:1313-8. 5. Appling WD, Patrinely JR, Salzer TA. Transconjunctival approach vs subciliary skin-muscle flap approach for orbital fracture repair. Arch Otolaryngol Head Neck Surg 1993;119:1000-7.
BRIEF REPORTS
451
REFERENCES 1. Hotchkiss M, Miller N, Clark A, Green W. Bilateral Duane's retraction syndrome. Arch Ophthalmol 1980;98:870-4. 2. Huber A. Electrophysiology of retraction syndromes. Br J Ophthalmol 1974;58:293-300. 3. Kommerell G, Bach M. A new type of Duane's syndrome. Neuro-ophthalmology 1986;6:159-644. Pruksacholawit K, Ishikawa S. Atypical vertical retraction syndrome. J Pediatr Ophthalmol 1976;13:215-20. 5. Khodadoust A, von Noorden G. Bilateral vertical retraction syndrome. Arch Ophthalmol 1967;78:606-12.
Acquired Lower Eyelid Epiblepharon Robert I. Park, M.D., and Dale R. Meyer, M.D. PURPOSE: Although congenital epiblepharon is a relatively common condition, particularly in Asians, acquired lower eyelid epiblepharon is rare. METHOD: We examined an 8-year-old boy who developed unilateral lower eyelid epiblepharon af ter a subciliary incisional approach for repair of a zygomatico-orbital fracture. RESULTS: The patient demonstrated an overrid ing fold of right lower eyelid skin and loss of the lower eyelid crease. At the time of surgical repair, disruption and scarring of the lower eyelid retrac tors, suborbicularis fascia, and orbital septum were noted. The epiblepharon was successfully corrected by plication of the retractors and skin to the tarsus. CONCLUSION: Disruption of the anterior subcu taneous fibers of the lower eyelid retractors (that is, capsulopalpebral fascia) contributes to epi blepharon.
C
ONGENITAL EPIBLEPHARON IS A COMMON CONDItion, particularly in Asian populations. The affected eyelids demonstrate a horizontal fold of skin that overrides the eyelid margin and compresses the eyelashes into the cornea, producing irritation. Lower eyelid epiblepharon is more severe in downgaze and is
Accepted for publication March 29, 1996. Division of Orbital and Ophthalmic Plastic Surgery, Lions Eye Institute. Inquiries to Dale R. Meyer, M.D., Division of Orbital and Ophthal mic Plastic Surgery, Lions Eye Institute, 35 Hackett Blvd., Albany, NY 12208.
BRIEF REPORTS
449
Fig. 1 (Park and Meyer). Left, An 8-year-old boy with acquired right lower eyelid epiblepharon after subciliary approach for repair of right zygomatico-orbital fracture. Right, Right lower eyelid shows overriding horizontal fold of skin with loss of lower eyelid crease and compression of eyelashes into cornea. Note that the eyelid margin is in normal position.
Fig. 2 (Park and Meyer). Left, Intraoperative photograph; scissors point to the central band of scar tissue involving suborbicularis fascia, orbital septum, and lower eyelid retractors. Right, Postoperative photograph shows correction of epiblepharon.
typically accompanied by loss of the normal lower eyelid crease. With epiblepharon, unlike entropion, the eyelid margin is unaffected and retains its normal position.1'3 In contrast to congenital epiblepharon, acquired lower eyelid epiblepharon is quite rare.3 We present a case of acquired unilateral lower eyelid epiblepharon after surgical repair of a zygomaticoorbital (tripod) fracture via a subciliary incision. A n 8-year-old boy sustained a displaced right zygomatico-orbital fracture after a sledding accident in January 1995. Open reduction with internal fixa tion of the tripod fracture was performed by another surgeon. The surgical approach included a subciliary 450
skin incision 1 to 2 mm below the eyelash line, with dissection then continued in the preseptal plane to the inferior orbital rim. A miniplate was used for fixation of the fracture involving the inferior orbital rim. The inferior periosteal incision and the lower eyelid orbicularis muscle incision were closed with 4-0 chromic suture. The skin incision was not closed directly. The patient did well initially; however, during a 2la-month postoperative period, discomfort developed in his right eye. He was referred for examination. O n examination, visual acuity was 20/20 in both eyes; no extraocular muscle limitation was noted and there was no evidence of enophthal-
AMERICAN JOURNAL OF OPHTHALMOLOGY
SEPTEMBER
1996
mos. Examination of the right lower eyelid demon strated loss of distinction of the right lower eyelid crease with a roll of skin overriding the tarsus, compressing the eyelashes into the cornea (Fig. 1). The eyelid margin and tarsus remained in normal position in primary gaze and downgaze. Slit-lamp biomicroscopy disclosed superficial punctate keratopathy. Surgical exploration and repair of the lower eyelid epiblepharon was performed. An incision was made 1 to 2 mm below the eyelash line. Dissection disclosed a broad band of scar tissue in the central and medial portion of the eyelid, involving the suborbicularis fascia, the orbital septum, and the lower eyelid retractors and surrounding orbital fat (Fig. 2). There was no involvement of the lateral and more extreme medial aspect of the eyelid. The scar tissue was lysed and excised. The lower eyelid retractors were identi fied, freed from the surrounding scar tissue, and then advanced and plicated to the inferior tarsus with 5-0 polyglactin sutures. The skin and orbicularis were closed with interrupted 6-0 plain gut sutures, taking deep bites of the underlying tarsus and lower eyelid retractors to enhance the eyelid crease. Postoperatively, the epiblepharon was corrected with complete relief of symptoms and no recurrence for 12 months. Acquired lower eyelid epiblepharon is a rare condi tion that, to our knowledge, has only been reported once previously.5 The clinical findings in this case were identical to congenital epiblepharon. Although the cause of lower eyelid epiblepharon is debated, most evidence suggests a defect in the capsulopal-
VOL.122, No. 3
pebral fascia (that is, lower eyelid retractors).1'3 Ante rior extensions of the capsulopalpebral fascia to the overlying skin are thought to form the lower eyelid crease and to contribute to a normally functioning eyelid.14 The clinical and intraoperative findings in our case of acquired epiblepharon support the theory that disruption of anterior subcutaneous extensions of the lower eyelid retractors contributes to epiblepharon formation. Repair of the epiblepharon in this case by plication of the lower eyelid retractors and skin to the tarsal plate successfully corrected the epiblepharon and further supports this theory. Other types of eyelid malposition, predominantly lower eyelid retraction and ectropion, have been reported with transcutaneous subciliary eyelid approaches for repair of orbital fractures. Fewer such complications are seen with a transconjunctival approach.5 REFERENCES 1. Quickert MH, Wilkes DI, Dryden RM. Nonincisional correc tion of epiblepharon and congenital entropion. Arch Ophthalmol 1983;101:778-81. 2. Millman AL, Mannor GE, Putterman AM. Eyelid crease and capsulopalpebral fascia repair in congenital entropion and epiblepharon. Ophthalmic Surg 1994;25:162-5. 3. Jordan R. The lower eyelid retractors in congenital entropion and epiblepharon. Ophthalmic Surg 1993;24:494-6. 4- Hawes M], Dortzbach RK. The microscopic anatomy of the lower eyelid retractors. Arch Ophthalmol 1982;100:1313-8. 5. Appling WD, Patrinely JR, Salzer TA. Transconjunctival approach vs subciliary skin-muscle flap approach for orbital fracture repair. Arch Otolaryngol Head Neck Surg 1993;119:1000-7.
BRIEF REPORTS
451