10 February 2024: Articles
Reconstruction of an Eyelid Defect Associated with Congenital Coloboma in a 7-Month-Old Male Infant Using an Acellular Dermal Allograft
Challenging differential diagnosis, Unusual or unexpected effect of treatment, Diagnostic / therapeutic accidents, Unexpected drug reaction, Congenital defects / diseases, Educational Purpose (only if useful for a systematic review or synthesis)Wenhong Cao1ABCDEF, Li Li1A, Yunwei Fan1B, Yuan Wang1B, Qian Wu1ABD*
Am J Case Rep 2024; 25:e942645
BACKGROUND: Congenital eyelid coloboma in children often faces complications such as keratitis, symblepharon, and amblyopia. Repairing defects involving at least 50% of the eyelid margin can be challenging. Acellular dermal allograft (ADA) has achieved excellent results as a substitute in adult eye plastic surgery, with minimal morbidity. This report describes a case of reconstruction of an eyelid defect in a 7-month-old male infant using an ADA.
CASE REPORT: A 7-month-old male infant was referred due to congenital eyelid coloboma in the left eye, which affected nearly one-half of the upper and lower eyelids medially, with more than 9 mm of lagophthalmos and lacrimal duct malformation inducing dacryocystitis. Under general anesthesia, A U-shaped silicone drainage tube was inserted in the nasolacrimal duct to ensure an unobstructed lacrimal duct. The symblepharon release, pseudopterygium excision, and medial canthus reconstruction were performed sequentially. Then, the upper eyelid defect was repaired through the advancement of the lateral segment of the eyelid, following lateral cantholysis. A trimmed ADA was placed as a substitute for the tarsal plate in the lower eyelid defect area and sutured with the free edge of the retractor. Finally, the lower and lateral skin orbicular muscle flap was advanced to cover the acellular dermis composite graft. The postoperative eyelid morphology was satisfactory. At 6 months after surgery, lower eyelid retraction gradually appeared.
CONCLUSIONS: ADA is presented as an effective solution for reconstructing significant eyelid defects of infants. However, the potential of postoperative eyelid retraction still deserves future research and refinement in surgical techniques.
Keywords: Cleft Eyelid, Allografts, Surgery, Plastic
Congenital eyelid coloboma is a relatively uncommon condition, characterized by a partial- or full-thickness defect in the eyelid. It is estimated to be present in about 1 in 10 000 births. The condition arises due to the failure of fusion of the mesodermal lid folds. The accompanying anatomical defects include keratoconus, coloboma of the iris, micro-ophthalmia, symblepharon, and pseudopterygium . To protect the cornea, conservative therapies typically consist of artificial tears, ointment treatment, and moist chamber optical bandages. Should these measures fail to yield the desired results, surgical intervention can be required. The timing of the surgery depends upon the size of the defect, the presence of corneal exposure, and the general well-being of the infant . If the defect is so extensive, with obvious corneal exposure or obstruction of the visual axis, surgery should be performed as soon as possible. However, repairing defects involving at least 50% of the eyelid margin can be challenging. Gu et al  developed a composite graft of acellular human dermis to reconstruct injured posterior lamella due to burns or trauma. An allogenic acellular dermal graft was used by Chang et al  to rectify conditions such as lower lid retraction, superior sulcus deformity, and anophthalmic socket contraction. The long-term outcomes presented by Vahdani et al , observed over a period exceeding 80 months, demonstrated the effectiveness of acellular dermal allografts (ADA) as a replacement for the tarsal plate in repairing extensive full-thickness eyelid defects. The literature above indicated that the applications of ADA in adult ophthalmic plastic surgery brought excellent functional and cosmetic outcomes, with minimal morbidity. To the best of our knowledge, there is no specific research on the application of ADA in pediatric eye plastic surgery. This report describes a case of reconstruction of a large eyelid defect in a 7-month-old male infant using an ADA.
A 7-month-old male infant was referred due to congenital eyelid coloboma in the left eye, which affected nearly one-half of the upper and lower eyelids medially. The patient presented with more than 9 mm of lagophthalmos, accompanying with nasal exposure keratitis, micro-ophthalmia, iris coloboma, symblepharon, pseudopterygium, and lacrimal duct malformation, inducing dacryocystitis (Figure 1).
The study adhered to the principles outlined in the Declaration of Helsinki and was approved by the Beijing Children’s Hospital Ethics Committee (2020-Z-171). Informed consent was obtained from the infant’s parents.
To ensure the lowest risk of infection during subsequent treatment, the obstruction of the lacrimal passage was treated with probing before eyelid reconstruction surgery, leaving postoperative epiphora but no secretions. The subsequent blepharoplasty was performed under general anesthesia.
The upper and lower lacrimal puncta were observed to be patent and appeared normal. Therefore, a U-shaped silicone drainage tube was inserted, connecting the upper and lower lacrimal dots to the nasolacrimal duct, to ensure an unobstructed lacrimal duct. Under microscopic magnification, surgical release of the nasal symblepharon and excision of pseudopterygium were performed first, followed by freshening and conversion of the defect edges into raw surfaces. Then, the medial canthal tendon loss was addressed by creating a supranasal tarsal strip for direct, which was anchored to the orbital periosteum with non-absorbable sutures.
To further maintain the integrity of the eyelashes, the upper eyelid defect was repaired through the advancement of the lateral segment of the eyelid. Additionally, a semicircular skin flap was made below the lateral portion of the eyebrow and canthus to enable further mobilization of the eyelid. Tension was released with a lateral cantholysis.
After sufficient lysis of the symblepharon, the lower eyelid conjunctival advancement flap was obtained. A trimmed ADA (ReDerm, Jieya Company, Beijing, People’s Republic of China), measuring 12×4×0.6 mm, was placed as a substitute for the tarsal plate in the lower eyelid defect area and sutured with the free edge of the retractor (Figure 2). Subsequently, the lower and lateral skin orbicular muscle flap was advanced to cover the acellular dermis composite graft and carefully sutured in place to establish a blood supply. To prevent tissue adhesion, the upper and lower eyelid margins were temporarily sutured. Tobramycin and dexamethasone eye ointments were applied in the conjunctival sac, and a bandage dressing was placed for 48 h. The wound suture was removed after 1 week. At 4 weeks after surgery, the lacrimal drainage tube was then pulled out, leaving no epiphora.
During the postoperative follow-up period of 18 months, there were no reported instances of postoperative infections, allergic reactions, or immunologic rejections related to the use of the allograft in this patient. The eyelid morphology was found to be satisfactory, with no lagophthalmos (Figure 3). Six months after surgery, lower eyelid retraction gradually appeared (Figure 4).
In this section, we mainly discuss the feasibility and reliability of applying ADA in reconstructing infant eyelid defects, as well as potential issues during the postoperative period.
This was a case of severe congenital upper and lower eyelid defect, combined with lacrimal passage obstruction, secondary dacryocystitis, and severe eyelid malclosure. The patient was at potential risk of exposure to keratitis, corneal infection perforation, and even eyeball atrophy. In contrast to the adult cases referred to earlier [2–4], the patient in this study was younger, presented with a congenital condition, had a more rapid onset of symptoms, and had a greater number of comorbidities, as well as more profound visual impairment. Therefore, it was imperative to conduct eyelid defect reconstruction surgery combined with lacrimal passage reconstruction surgery at an early stage in the child’s life. This surgical intervention holds significant importance for safeguarding the child’s future eye protection and development.
Surgical methods are selected based on the size of the defect. For small (less than 25%) or moderate defects (25% to 50%), effective treatments include direct closure, Tenzel semicircular flap, Mustarde lid switch flap, Cutler-Beard reconstruction for the upper eyelid, and the Hughes tarsoconjunctival flap for the lower eyelid. These methods can all be effectively combined with lateral cantholysis. Severe defects (more than 50%) can require the procedure of substitute transplantation  if the methods above cannot completely close the defect. In our case, the surgical technique of a Tenzel semicircular flap with lateral cantholysis was used for reconstruction of the upper eyelid defects, to preserve the integrity of the eyelid eyelashes. However, given the limited residual tarsal plates in the lower eyelid, a surgical strategy was adopted to replace the tarsal plate with a graft and combine the lower and temporal skin orbicularis muscle transfer flap.
Autogenous tarsoconjunctiva is typically viewed as the best material for mending the posterior lamella. However, securing the right size for substantial defects with notable vertical range can be challenging. The process of procuring a tarsoconjunctival flap or graft gets more intricate when there is a lack of adequate donor tissue from the opposing contralateral eyelid, a situation that often arises from simultaneous diseases, injuries, or the sheer magnitude of the defect. For infants, the primary concern is the potential harm of using healthy eyelid tissue, leading to possible complications at the donor site. As an autologous graft, a mucoperiosteal graft from the hard palate is also appropriate. However, it comes with disadvantages, including donor site scarring, limited availability, and prolonged operating time . To avoid donor-site morbidity, several substitutes are available to replace autologous grafts. Given the particularity of infants, we decided to choose the acellular human dermis as a graft to minimize the risk of immune rejection and the surgical complications mentioned above.
ADA is an immunogenically inert acellular material derived from the human cadaveric dermis. Mechanical and chemical techniques are used to remove the epidermis and all dermal cellular structures before freeze-drying. Initially introduced in 1992 for surface grafting in burn victims, ADA has since found various applications, including serving as a spacer in the correction of lid retraction [6–8]. Over the past decade, it has become recognized as an excellent substitute for the tarsus and provides a favorable basement membrane surface that facilitates conjunctival migration. As a result, ADA has gained popularity in upper and lower eyelid defects, becoming a preferred choice for numerous oculoplastic and reconstructive procedures [9–12]. However, there is a lack of specific research on the application of ADA in eyelid plastic surgery in children. In our study, we used ADA as a posterior lamellar graft for the reconstruction of a congenital full-thickness lid defect in a 7-month-old infant, to achieve satisfactory functional and aesthetic outcomes. This indicates that ADA is safe and reliable for pediatric eye plastic surgery.
One possible limitation of using ADA is its tendency to undergo resorption and contraction over time. In response to this concern, Vahdani et al  suggested the intentional use of an oversized ADA that is approximately double the determined vertical size of the posterior lamella defect. This approach compensates for any postoperative contracture or resorption. In the present case, the lower eyelid showed significant retraction 6 months after surgery. We hypothesized that an insufficient estimation of graft size might have contributed to this eyelid regression. This suggests that we should appropriately increase the size of the graft in the future to counteract possible postoperative graft shrinkage. If there is a severe full-thickness defect of the lower eyelid, a combined surgical strategy for eyelid and cheek elevation may be required so that vertical traction on the lid can be avoided. This is worth further studying in a larger sample size.
To conclude, ADA offers a dependable, safe, and effective solution for reconstructing significant eyelid defects. The distant observations from this particular infant case demonstrate positive functional and aesthetic results. However, the potential of postoperative eyelid retraction suggests the need for a refined approach in graft sizing and surgical techniques, particularly when addressing severe full-thickness defects. Long-term postoperative eyelid retraction deserves further attention and research.
FiguresFigure 1.. The patient at 7 months of age showed left upper and lower lid colobomas with corneal erosions secondary to exposure and symblepharon and pseudopterygium. Figure 2.. A trimmed acellular dermal allograft (12×4×0.6 mm) as a substitute for the tarsal plate was placed in the lower eyelid defect area and sutured with the free edge of the retractor. Figure 3.. The lagophthalmos caused by the eyelid defect disappeared 1 month after surgery. Figure 4.. The lower eyelid showed significant retraction 18 months after surgery.
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6.. Wainwright DJ, Use of an acellular allograft dermal matrix (AlloDerm) in the management of full-thickness burns: Burns, 1995; 21(4); 243-48
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8.. Shorr N, Perry JD, Goldberg RA, The safety and applications of acellular human dermal allograft in ophthalmic plastic and reconstructive surgery: A preliminary report: Ophthalmic Plast Reconstr Surg, 2000; 16(3); 223-30
9.. Hayek B, Hatef E, Nguyen M, Acellular dermal graft (AlloDerm) for upper eyelid reconstruction after cancer removal: Ophthalmic Plast Reconstr Surg, 2009; 25(6); 426-29
10.. Levin F, Turbin RE, Langer PD, Acellular human dermal matrix as a skin substitute for reconstruction of large periocular cutaneous defects: Ophthalmic Plast Reconstr Surg, 2011; 27(1); 44-47
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