- Email: [email protected]
Large upper eyelid coloboma repair: a one-stage, one-site technique
Accepted Manuscript Large upper eyelid coloboma repair: a one-stage, one-site technique Lora R. Dagi Glass, MD, Alexandra T. Elliott, MD PII:
S1091-8531(16)30461-X
DOI:
10.1016/j.jaapos.2016.05.018
Reference:
YMPA 2469
To appear in:
Journal of AAPOS
Received Date: 1 December 2015 Revised Date:
27 April 2016
Accepted Date: 27 May 2016
Please cite this article as: Dagi Glass LR, Elliott AT, Large upper eyelid coloboma repair: a one-stage, one-site technique, Journal of AAPOS (2016), doi: 10.1016/j.jaapos.2016.05.018. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Large upper eyelid coloboma repair: a one-stage, one-site technique Lora R. Dagi Glass, MD,a Alexandra T. Elliott, MDb
RI PT
Author affiliations: aOphthalmic Plastic Surgery Service, Department of Ophthalmology, Massachusetts Eye and Ear Infirmary, Boston, Massachusetts; bDepartment of Ophthalmology, Boston Children’s Hospital and Harvard Medical School, Boston, Massachusetts Submitted December 1, 2015. Revision accepted May 27, 2016.
SC
Correspondence: Alexandra T. Elliott, MD, Department of Ophthalmology, Boston Children’s Hospital, 300 Longwood Avenue, Boston, MA 02115 (email: [email protected])
M AN U
Presented in part at the 41st Annual Meeting of the American Association for Pediatric Ophthalmology and Strabismus Annual Meeting, New Orleans, Louisiana, March 25-29, 2015, and at the European Society of Ophthalmology Congress, Vienna, Austria, June 6-9, 2015.
AC C
EP
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Word count: 960
ACCEPTED MANUSCRIPT
Current techniques for repairing large eyelid colobomas require preparation of other tissue sites and occasionally more than one procedure. We present a technique that requires only one procedure and is limited to the colobomatous eyelid; in addition, it is
RI PT
specifically designed to help avoid postoperative astigmatic and obstructive amblyopia. Outcomes are demonstrated in 3 cases of hemifacial microsomia. Large colobomas on the
incising only the congenitally abnormal eyelid.
SC
upper eyelid can be successfully and aesthetically repaired with only one procedure,
Large colobomas of the upper eyelid represent a management dilemma in children. Given
M AN U
findings of corneal exposure and concern for subsequent ulceration and perforation, repair is often urgent: there is no time to wait for maturation of potential skin graft sites or increased tissue laxity. While smaller eyelid colobomas can be closed directly or with a Tenzel semicircular rotational flap,1-5 larger ones require more extensive tissue rearrangement. The form
TE D
of rearrangement can have potentially lifelong amblyopic consequences, particularly with a Cutler-Beard technique. Other closure techniques include combinations of musculocutaneous, tarsoconjunctival, and mucous membrane tissue rearrangements, flaps, and/or grafts taken from
EP
the same lid, the ipsilateral lower lid, the contralateral lid, the brow area, or the penis.1,2,4-10 Postoperative skin color, texture, contour, or thickness mismatch can be disconcerting to parents
AC C
and may lead to social difficulties.
We introduce a one-stage, one-site technique for large upper eyelid coloboma closure that
has been successfully employed in coloboma defects exceeding 50%. A case series follows with discussion of outcomes. All 3 patients presented for treatment at Boston Children’s Hospital. Technique The following procedure is performed under general anesthesia (Figure 1). A pentagonal wedge
ACCEPTED MANUSCRIPT
excision of the coloboma is performed using Westcott scissors; the excised tissue is saved. Westcott scissors are used to perform a superior cantholysis, without a canthotomy. The skin of the most lateral aspect of the lid is then incised vertically with a 15c blade. The
RI PT
underlying orbicularis remains intact and is stretched as the coloboma edges are sutured close. Three 7-0 polyglactin 910 sutures are placed in a buried fashion at the margin of coloboma
closure. Two or three partial-tarsal-thickness sutures are placed along the coloboma bed, and the
SC
overlying orbicularis muscle is sutured together using interrupted 7-0 polyglactin 910 sutures. The skin is closed in an interrupted fashion using 6-0 fast absorbing gut suture.
M AN U
The excised pentagonal wedge is then removed from its saline bath. A full-thickness triangular skin graft is taken from the wedge and then sutured over the lateral, bare orbicularis using 8-0 polyglactin 910. A Telfa bolster soaked in erythromycin ophthalmic ointment is sewn securely over the graft site, to be removed after 1 week.
TE D
Case 1
A 3-month-old girl with Goldenhar syndrome presented with a large left upper eyelid coloboma (Figure 2). Associated syndromic abnormalities included a large left limbal dermoid, bilateral ear
EP
displacement, micrognathia, and vertebral and cardiac anomalies. Under general anesthesia with nasal intubation, the left upper lid coloboma was repaired using the technique described above.
AC C
Postoperative examination demonstrated excellent lid mobility and closure. A small residual notch remained at the colobomatous site. She was followed for 4 months thereafter, with patching for refractive amblyopia, afterward transferring care to a local ophthalmologist. Case 2
A 1-month-old boy with Goldenhar syndrome presented for evaluation of a large left upper eyelid coloboma (Figure 2). He had undergone left limbal dermoid excision at approximately 1
ACCEPTED MANUSCRIPT
week of life concurrent with a temporal canthoplasty; other associated abnormalities included a smaller limbal dermoid in the right eye, hemifacial microsomia and preauricular skin tags. Given adequate corneal lubrication, surgical repair of the coloboma was deferred until 9 months. This
RI PT
surgery was combined with revision of the limbal dermoid excision scar and symblepharon
release with placement of amniotic membrane. Despite significant improvement in his upper lid lagophthalmos in the postoperative period, symblepharon recurred between the lower lid and the
SC
dermoid revision site. This lower lid dystopia ultimately resulted in lower lid retraction and corneal abrasion and scarring ensued. The patient has continued to be followed for the past 2.5
M AN U
years, and the lower lid retraction has been managed with overnight lid taping and erythromycin ophthalmic ointment as needed. The patient is also undergoing prophylactic patching for refractive amblyopia. Case 3
TE D
A 6-day-old girl with Goldenhar syndrome presented with a large right upper eyelid coloboma (Figure 2). She also appeared to have brow involvement and a limbal dermoid in the same eye as well as preauricular tags. Given good compliance with corneal lubrication, surgery was deferred
EP
until 1 year of age. Postoperative follow-up demonstrated excellent healing, and by 7 months’ follow-up, the parents stated they were no longer asked about their daughter’s lid. Limbal
AC C
dermoid removal was deferred. No refractive error or amblyopia was present. Discussion
In each of the cases described, direct coloboma closure would have either proved impossible or caused significant astigmatism due to lack of eyelid laxity. A Tenzel flap would produce greater wound tension and require an extended incision beyond the eyelid. Previous attempts to repair large colobomas of the upper eyelid using a graft have relied upon a second site of tissue
ACCEPTED MANUSCRIPT
excision, whether ipsilateral brow or lower lid, contralateral lid, or penile.1,2,4-10 Some techniques also rely upon a second surgery for flap incision, with obvious concerns for occlusion amblyopia. Our technique allows for a single procedure at a single site. The skin graft itself is
RI PT
perfectly matched, having been harvested from the requisite same-lid pentagonal wedge
surrounding the coloboma. This technique also allows for relatively normal lash appearance. While amblyopia may still be present secondary to other ocular abnormalities, such astigmatism
SC
due to limbal dermoids, there is no concern for occlusional amblyopia. Astigmatic amblyopia
AC C
EP
TE D
superior lateral canthal tendon.
M AN U
from the lid repair is unlikely, given the lack of tension allowed by release of the skin and
ACCEPTED MANUSCRIPT
References 1.
Grover AK, Chaudhuri Z, Malik S, Bageja S, Menon V. Congenital eyelid colobomas in 51 patients. J Pediatr Ophthalmol Strabismus 2009;46:151-9. Seah LL, Choo CT, Fong KS. Congenital upper lid colobomas: management and visual outcome. Ophthal Plast Reconstr Surg 2002;18:190-95.
3.
RI PT
2.
Lawson A. Large Coloboma of the Upper Lid; Operation; Result. Proc R Soc Med
SC
1916;9(Sect Ophthalmol):27-9.
Collin JR. Congenital upper lid coloboma. Aust N Z J Ophthalmol 1986;14:313-17.
5.
Tawfik HA, Abdulhafez MH, Fouad YA. Congenital upper eyelid coloboma:
M AN U
4.
embryologic, nomenclatorial, nosologic, etiologic, pathogenetic, epidemiologic, clinical, and management perspectives. Ophthal Plast Reconstr Surg 2015;31:1-12. 6.
Lee H, Takahashi Y, Ichinose A, Kakizaki H. Reconstruction of a congenital upper eyelid
7.
TE D
coloboma using a lamellar-based technique. Ophthal Plast Reconstr Surg 2014;30:e95-6. Guerrissi JO. Surgical reconstruction of the palpebral border: upper lid eyebrow-musculocutaneous island flap. Plast Reconstr Surg 2005;115:1118-23. Hashish A, Awara AM. One-stage reconstruction technique for large congenital eyelid
EP
8.
coloboma. Orbit 2011;30:177-9. Hoyama E, Limawararut V, Malhotra R, Muecke J, Selva D. Tarsomarginal graft in upper
AC C
9.
eyelid coloboma repair. J AAPOS 2007;11:499-501.
10.
Hauben DJ, Tessler Z. One-stage reconstruction of a large upper lid defect in a newborn. Plast Reconstr Surg 1989;83:337-40.
ACCEPTED MANUSCRIPT
Legends FIG 1. Surgical steps. A, The pentagonal wedge markings surrounding the upper lid coloboma; of note, the upper triangle of skin was ultimately used as a graft. B, The upper lid after excision
RI PT
of the wedge; the dashed, green line indicates a superior cantholysis, specifically without a
canthotomy. C, Application of medial tension while a 15c blade is used to incise the skin of the most lateral aspect of the lid (red line). D, Cross-section of the lateral lid showing a stretched
SC
orbicularis muscle bridge sandwiched between released skin and posterior lamellae. E, The
lateral orbicularis on stretch revealing a triangular bridge of vascularized muscle. F, The skin
M AN U
graft in position overlying the stretched orbicularis muscle.
AC C
EP
TE D
FIG 2. Preoperative findings and postoperative results in cases 1 (A,B), 2 (C,D), and 3 (E,F).
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
S1091-8531(16)30461-X
DOI:
10.1016/j.jaapos.2016.05.018
Reference:
YMPA 2469
To appear in:
Journal of AAPOS
Received Date: 1 December 2015 Revised Date:
27 April 2016
Accepted Date: 27 May 2016
Please cite this article as: Dagi Glass LR, Elliott AT, Large upper eyelid coloboma repair: a one-stage, one-site technique, Journal of AAPOS (2016), doi: 10.1016/j.jaapos.2016.05.018. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Large upper eyelid coloboma repair: a one-stage, one-site technique Lora R. Dagi Glass, MD,a Alexandra T. Elliott, MDb
RI PT
Author affiliations: aOphthalmic Plastic Surgery Service, Department of Ophthalmology, Massachusetts Eye and Ear Infirmary, Boston, Massachusetts; bDepartment of Ophthalmology, Boston Children’s Hospital and Harvard Medical School, Boston, Massachusetts Submitted December 1, 2015. Revision accepted May 27, 2016.
SC
Correspondence: Alexandra T. Elliott, MD, Department of Ophthalmology, Boston Children’s Hospital, 300 Longwood Avenue, Boston, MA 02115 (email: [email protected])
M AN U
Presented in part at the 41st Annual Meeting of the American Association for Pediatric Ophthalmology and Strabismus Annual Meeting, New Orleans, Louisiana, March 25-29, 2015, and at the European Society of Ophthalmology Congress, Vienna, Austria, June 6-9, 2015.
AC C
EP
TE D
Word count: 960
ACCEPTED MANUSCRIPT
Current techniques for repairing large eyelid colobomas require preparation of other tissue sites and occasionally more than one procedure. We present a technique that requires only one procedure and is limited to the colobomatous eyelid; in addition, it is
RI PT
specifically designed to help avoid postoperative astigmatic and obstructive amblyopia. Outcomes are demonstrated in 3 cases of hemifacial microsomia. Large colobomas on the
incising only the congenitally abnormal eyelid.
SC
upper eyelid can be successfully and aesthetically repaired with only one procedure,
Large colobomas of the upper eyelid represent a management dilemma in children. Given
M AN U
findings of corneal exposure and concern for subsequent ulceration and perforation, repair is often urgent: there is no time to wait for maturation of potential skin graft sites or increased tissue laxity. While smaller eyelid colobomas can be closed directly or with a Tenzel semicircular rotational flap,1-5 larger ones require more extensive tissue rearrangement. The form
TE D
of rearrangement can have potentially lifelong amblyopic consequences, particularly with a Cutler-Beard technique. Other closure techniques include combinations of musculocutaneous, tarsoconjunctival, and mucous membrane tissue rearrangements, flaps, and/or grafts taken from
EP
the same lid, the ipsilateral lower lid, the contralateral lid, the brow area, or the penis.1,2,4-10 Postoperative skin color, texture, contour, or thickness mismatch can be disconcerting to parents
AC C
and may lead to social difficulties.
We introduce a one-stage, one-site technique for large upper eyelid coloboma closure that
has been successfully employed in coloboma defects exceeding 50%. A case series follows with discussion of outcomes. All 3 patients presented for treatment at Boston Children’s Hospital. Technique The following procedure is performed under general anesthesia (Figure 1). A pentagonal wedge
ACCEPTED MANUSCRIPT
excision of the coloboma is performed using Westcott scissors; the excised tissue is saved. Westcott scissors are used to perform a superior cantholysis, without a canthotomy. The skin of the most lateral aspect of the lid is then incised vertically with a 15c blade. The
RI PT
underlying orbicularis remains intact and is stretched as the coloboma edges are sutured close. Three 7-0 polyglactin 910 sutures are placed in a buried fashion at the margin of coloboma
closure. Two or three partial-tarsal-thickness sutures are placed along the coloboma bed, and the
SC
overlying orbicularis muscle is sutured together using interrupted 7-0 polyglactin 910 sutures. The skin is closed in an interrupted fashion using 6-0 fast absorbing gut suture.
M AN U
The excised pentagonal wedge is then removed from its saline bath. A full-thickness triangular skin graft is taken from the wedge and then sutured over the lateral, bare orbicularis using 8-0 polyglactin 910. A Telfa bolster soaked in erythromycin ophthalmic ointment is sewn securely over the graft site, to be removed after 1 week.
TE D
Case 1
A 3-month-old girl with Goldenhar syndrome presented with a large left upper eyelid coloboma (Figure 2). Associated syndromic abnormalities included a large left limbal dermoid, bilateral ear
EP
displacement, micrognathia, and vertebral and cardiac anomalies. Under general anesthesia with nasal intubation, the left upper lid coloboma was repaired using the technique described above.
AC C
Postoperative examination demonstrated excellent lid mobility and closure. A small residual notch remained at the colobomatous site. She was followed for 4 months thereafter, with patching for refractive amblyopia, afterward transferring care to a local ophthalmologist. Case 2
A 1-month-old boy with Goldenhar syndrome presented for evaluation of a large left upper eyelid coloboma (Figure 2). He had undergone left limbal dermoid excision at approximately 1
ACCEPTED MANUSCRIPT
week of life concurrent with a temporal canthoplasty; other associated abnormalities included a smaller limbal dermoid in the right eye, hemifacial microsomia and preauricular skin tags. Given adequate corneal lubrication, surgical repair of the coloboma was deferred until 9 months. This
RI PT
surgery was combined with revision of the limbal dermoid excision scar and symblepharon
release with placement of amniotic membrane. Despite significant improvement in his upper lid lagophthalmos in the postoperative period, symblepharon recurred between the lower lid and the
SC
dermoid revision site. This lower lid dystopia ultimately resulted in lower lid retraction and corneal abrasion and scarring ensued. The patient has continued to be followed for the past 2.5
M AN U
years, and the lower lid retraction has been managed with overnight lid taping and erythromycin ophthalmic ointment as needed. The patient is also undergoing prophylactic patching for refractive amblyopia. Case 3
TE D
A 6-day-old girl with Goldenhar syndrome presented with a large right upper eyelid coloboma (Figure 2). She also appeared to have brow involvement and a limbal dermoid in the same eye as well as preauricular tags. Given good compliance with corneal lubrication, surgery was deferred
EP
until 1 year of age. Postoperative follow-up demonstrated excellent healing, and by 7 months’ follow-up, the parents stated they were no longer asked about their daughter’s lid. Limbal
AC C
dermoid removal was deferred. No refractive error or amblyopia was present. Discussion
In each of the cases described, direct coloboma closure would have either proved impossible or caused significant astigmatism due to lack of eyelid laxity. A Tenzel flap would produce greater wound tension and require an extended incision beyond the eyelid. Previous attempts to repair large colobomas of the upper eyelid using a graft have relied upon a second site of tissue
ACCEPTED MANUSCRIPT
excision, whether ipsilateral brow or lower lid, contralateral lid, or penile.1,2,4-10 Some techniques also rely upon a second surgery for flap incision, with obvious concerns for occlusion amblyopia. Our technique allows for a single procedure at a single site. The skin graft itself is
RI PT
perfectly matched, having been harvested from the requisite same-lid pentagonal wedge
surrounding the coloboma. This technique also allows for relatively normal lash appearance. While amblyopia may still be present secondary to other ocular abnormalities, such astigmatism
SC
due to limbal dermoids, there is no concern for occlusional amblyopia. Astigmatic amblyopia
AC C
EP
TE D
superior lateral canthal tendon.
M AN U
from the lid repair is unlikely, given the lack of tension allowed by release of the skin and
ACCEPTED MANUSCRIPT
References 1.
Grover AK, Chaudhuri Z, Malik S, Bageja S, Menon V. Congenital eyelid colobomas in 51 patients. J Pediatr Ophthalmol Strabismus 2009;46:151-9. Seah LL, Choo CT, Fong KS. Congenital upper lid colobomas: management and visual outcome. Ophthal Plast Reconstr Surg 2002;18:190-95.
3.
RI PT
2.
Lawson A. Large Coloboma of the Upper Lid; Operation; Result. Proc R Soc Med
SC
1916;9(Sect Ophthalmol):27-9.
Collin JR. Congenital upper lid coloboma. Aust N Z J Ophthalmol 1986;14:313-17.
5.
Tawfik HA, Abdulhafez MH, Fouad YA. Congenital upper eyelid coloboma:
M AN U
4.
embryologic, nomenclatorial, nosologic, etiologic, pathogenetic, epidemiologic, clinical, and management perspectives. Ophthal Plast Reconstr Surg 2015;31:1-12. 6.
Lee H, Takahashi Y, Ichinose A, Kakizaki H. Reconstruction of a congenital upper eyelid
7.
TE D
coloboma using a lamellar-based technique. Ophthal Plast Reconstr Surg 2014;30:e95-6. Guerrissi JO. Surgical reconstruction of the palpebral border: upper lid eyebrow-musculocutaneous island flap. Plast Reconstr Surg 2005;115:1118-23. Hashish A, Awara AM. One-stage reconstruction technique for large congenital eyelid
EP
8.
coloboma. Orbit 2011;30:177-9. Hoyama E, Limawararut V, Malhotra R, Muecke J, Selva D. Tarsomarginal graft in upper
AC C
9.
eyelid coloboma repair. J AAPOS 2007;11:499-501.
10.
Hauben DJ, Tessler Z. One-stage reconstruction of a large upper lid defect in a newborn. Plast Reconstr Surg 1989;83:337-40.
ACCEPTED MANUSCRIPT
Legends FIG 1. Surgical steps. A, The pentagonal wedge markings surrounding the upper lid coloboma; of note, the upper triangle of skin was ultimately used as a graft. B, The upper lid after excision
RI PT
of the wedge; the dashed, green line indicates a superior cantholysis, specifically without a
canthotomy. C, Application of medial tension while a 15c blade is used to incise the skin of the most lateral aspect of the lid (red line). D, Cross-section of the lateral lid showing a stretched
SC
orbicularis muscle bridge sandwiched between released skin and posterior lamellae. E, The
lateral orbicularis on stretch revealing a triangular bridge of vascularized muscle. F, The skin
M AN U
graft in position overlying the stretched orbicularis muscle.
AC C
EP
TE D
FIG 2. Preoperative findings and postoperative results in cases 1 (A,B), 2 (C,D), and 3 (E,F).
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT