Cilial Entropion: Surgical Outcome with a New Modification of the Hotz Procedure

Cilial Entropion: Surgical Outcome with a New Modification of the Hotz Procedure

Cilial Entropion: Surgical Outcome with a New Modification of the Hotz Procedure Hirohiko Kakizaki, MD, PhD,1,2 Dinesh Selva, FRACS, FRANZCO,2 Igal Le...

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Cilial Entropion: Surgical Outcome with a New Modification of the Hotz Procedure Hirohiko Kakizaki, MD, PhD,1,2 Dinesh Selva, FRACS, FRANZCO,2 Igal Leibovitch, MD3 Purpose: To report the surgical outcome with a new modification of the Hotz procedure for cilial entropion repair. Design: Retrospective case series. Participants: Forty-nine lower eyelids of 29 patients (22 female, 7 male; average age 8.4 years, range 2–27 years). Methods: The study included all patients diagnosed with cilial entropion and significant ocular irritation causing epiphora, photophobia, and ocular discharge, with or without keratitis. Surgery is based on dividing the anterior layer of the lower eyelid retractors from the anterior tarsal plate surface, definite identification of the inferior tarsal plate border, and reinforcement of the skin to the tarsal plate and lower eyelid retractors. Surgical outcome was defined as “good” or “fair” when there was no contact between the eyelashes and the globe or when less than 5 asymptomatic eyelashes had direct contact with the globe, respectively. A “poor” outcome was defined as either the majority of eyelashes remained in contact with the globe or persistence of irritation/keratitis in the presence of residual in-turned eyelashes. A successful outcome (“good” or “fair”) required no additional surgical intervention. Main Outcome Measures: Surgical outcome (“good,” “fair,” or “poor”), postoperative complications, recurrence. Results: During a mean follow-up period of 27.4 months (range, 3–50 months), 40 eyelids (82%) were judged postoperatively as “good,” 7 eyelids (14%) were judged as “fair,” and 2 eyelids (4%) were judged as “poor.” The 2 eyelids with a poor outcome were reoperated successfully using the same technique. No postoperative complications were recorded. There were no cases of recurrence, except the 2 eyelids with a “poor” outcome, during the follow-up period. Conclusions: Our modification of the Hotz procedure is based on identifying and dividing the anterior layer of the lower eyelid retractors and reinforcing the skin to the lower border of the inferior tarsal plate and lower eyelid retractors. This is a useful and predictive technique for cilial entropion repair and results in a high success rate. Financial Disclosure(s): The author(s) have no proprietary or commercial interest in any materials discussed in this article. Ophthalmology 2009;116:2224 –2229 © 2009 by the American Academy of Ophthalmology.

Cilial entropion is a congenital eyelid malposition that is commonly seen in Asian children.1,2 In this entity, a lower eyelid skin fold and the underlying pretarsal orbicularis oculi muscle ride over the eyelid margin, thereby tilting the eyelashes vertically or even pushing them against the globe.3 The tarsal plate maintains its normal upright position.4 Cilial entropion generally involves the medial one third to one half of the lower eyelid and usually occurs bilaterally.1,2 Induced keratitis and astigmatism are indications for surgical intervention.1,2,5,6 In both cilial entropion and epiblepharon, the cilia are inverted, but only epiblepharon is associated with significant redundant skin. The lower eyelid retractors are divided into anterior and posterior layers.7 The anterior layer consists of the superficial part of the capsulopalpebral fascia (which originates from the Lockwood’s ligament), a suborbicularis fibrous tissue, and the orbital septum, which reaches the anterior surface of the tarsal plate and the subcutaneous tissue. The posterior layer consists of dense fibers from the capsulopalpebral fascia, with smooth muscle fibers that terminate on

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the inferior tapered end of the tarsal plate. The main tractional forces originate from the posterior layer, whereas the anterior layer contributes only to pulling the anterior lamella of the lower eyelid.7 Kakizaki et al7 have shown that both layers are easily divided surgically with sharp and blunt dissection. The exact cause of lower eyelid cilial entropion is not known.5 A possible cause is failure of the lower eyelid retractors to gain access to the skin.4,5 In this case, the lower eyelid retractors heading toward the tarsal plate (the posterior layer) have a normal insertion, whereas retractor fibers directed to the skin (anterior layer) fail to insert, allowing skin and orbicularis muscle to roll upward toward the globe.4,5 By reattaching the anterior layer of the retractors to the anterior lamella of the eyelid, this tendency to roll upward could be attenuated. Although there are various surgical techniques to repair cilial entropion,1,6,8,9 the different modifications of the Hotz procedure are perhaps the most commonly performed.1,8,10 Because the lower tarsal plate height is short, the fixation ISSN 0161-6420/09/$–see front matter doi:10.1016/j.ophtha.2009.04.018

Kakizaki et al 䡠 Modified Hotz Procedure in Cilial Entropion sutures must be placed in the inferior edge of the tarsal plate to be effective in everting the eyelid.9,11–13 However, because most tarsal plate borders taper inferiorly,12 it is sometimes difficult to identify the exact tarsal edge unless the overlying lower eyelid retractors are detached from the anterior surface of the tarsal plate. In this series, the surgical outcome with our modification of the Hotz procedure for cilial entropion is reported, which is based on the double-layer anatomy of the lower eyelid retractors and the possible pathogenesis of this eyelid malposition.

Materials and Methods This is a retrospective chart review of all patients who underwent operation for cilial entropion and significant ocular irritation resulting in epiphora, photophobia, and ocular discharge, with or without keratitis, between August 2004 and October 2008. All patients were operated by a single surgeon (HK) using the modified Hotz procedure. Statistical analysis was based on chi-square test. Statistical significance was defined as P⬍0.05. All statistical analysis was carried out using SPSS for Windows (SPSS Japan Inc., Tokyo, Japan).

Surgical Technique Figure 1A shows a preoperative photo of cilial entropion. After drawing a skin incision line from the lower lacrimal punctum to around the lateral corneal limbus line in a region 3 mm below the eyelashes (Fig 1B), the conjunctiva and the eyelid were infiltrated with 2 ml of 2% lidocaine with 1:100,000 epinephrine. Surgery was performed under local or general anesthesia, according to the patient’s age and cooperation. After incising the skin and fixing the eyelid with an entropion clamp, the layer under the orbicularis oculi muscle was detached toward the eyelashes, and the wound was retracted at 3 points with hooks with 4-0 silk sutures (Fig 1C). Inferior dissection of the pretarsal attachment of the anterior layer of the lower eyelid retractors enabled clear and definite visualization of the inferior tarsal plate border (Fig 1D, E). The entropion clamp and hooks were then removed. After that, the orbicularis oculi muscle in the eyelid margin was slightly debulked to enable easy marginal rotation (Fig 1F, G). The surgical site was carefully inspected for hemostasis, and cautery was applied as necessary; 6-0 nylon or polypropylene sutures were used to connect the detached anterior layer of the lower eyelid retractors, with the inferior tarsal border, dermis, and marginal orbicularis oculi muscle (Fig 1H–J). These sutures were placed at 3 points along the eyelid. After confirming improvement in the direction of eyelashes, the skin was closed with subcutaneous continuous 6-0 nylon or polypropylene sutures for uncooperative patients (Fig 1K) or interrupted 6-0 nylon or polypropylene sutures for cooperative patients. No redundant skin was removed in this series of patients because it tends to stretch later as facial bones mature.4 Postoperative outcome was judged by the treating surgeon (HK), according to a standard grading scale used in our clinical practice. A “good” outcome was defined as no contact between the eyelashes and the globe, a “fair” outcome was defined as fewer than 5 asymptomatic eyelashes having some contact with the globe and no irritative symptoms, and a “poor” outcome was defined as either the majority of eyelashes remaining in contact with the

globe or persistence of irritation/keratitis in the presence of residual in-turned eyelashes. A “good” or “fair” result was considered a successful outcome and required no additional surgical intervention.

Results The study included 49 lower eyelids of 29 patients (22 female, 7 male, average age 8.4 years, range 2–27 years) who underwent operation using the new modification of the Hotz procedure. Twenty cases were bilateral, and 9 cases were unilateral (6 right and 3 left). The mean follow-up period was 27.4 months (range, 3–50 months). There were no intraoperative complications, and it was easy to identify the inferior tarsal plate border in all cases. No changes were observed in the normal anatomic attachment of the lower eyelid retractors to the tarsal plate in any of our cases. On the basis of our defined criteria, 40 eyelids (82%) were judged to have a “good” outcome (Fig 1L), 7 eyelids (14%) were judged to have a “fair” outcome, and 2 eyelids (4%) were judged to have a “poor” outcome. The 2 eyelids with a “poor” outcome were reoperated shortly after the first operation, using the same technique, and achieved a “good” surgical result. There were no cases of postoperative corneal exposure. A very mild degree of superficial punctuate keratopathy was recorded in patients with a “fair” outcome and in none of the patients with a “good” outcome. There were 6 patients (11 eyelids) in our series who were 14 years of age or older at the time of surgery. Five of these patients (9 eyelids: 82%) had a “good” outcome, and 1 patient (2 eyelids: 18%) had a “fair” outcome. No statistically significant difference was found between the success rate of the younger group (⬍14 years) and the older group (ⱖ14 years) (P ⫽ 0.437).

Discussion Our modification of the Hotz procedure enables direct and definite visualization of the inferior tarsal plate border after separating the anterior layer of the lower eyelid retractors, thereby enhancing the effective repair of cilial entropion. The goal of surgery in cilial entropion is to create an adhesion between the anterior lamella of the lower eyelid and the lower eyelid retractors, thereby exerting an everting force on the eyelashes of the lower eyelid.9,14 On the basis of previous reports that showed a unique double (anterior and posterior) layer structure of the lower eyelid retractors, and that showed the anterior layer has no role in pulling the lower tarsal plate,7 the anterior layer could be safely detached and then reattached after identifying the inferior tarsal plate border. This allows tight fixation of the lower eyelid retractors to the inferior tarsal border and the overlying skin, and results in a good rolling-out effect of the lower eyelid margin. An outcome was judged as “fair” when fewer than 5 eyelashes gently touched the globe, but not causing any ocular irritation. Because eyelashes of young children are fine and soft, and the cornea is tolerant,15 most cases of cilial entropion produce no or only mild symptoms, even when the cornea is in direct contact with the inverted eyelashes.2 In addition, because the cilial entropion may resolve without treatment when facial bones mature,5 mild

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Kakizaki et al 䡠 Modified Hotz Procedure in Cilial Entropion

Figure 1. (Continued.) J, Eyelid margin is everted. K, The surgical procedure is completed. L, The postoperative 3-month photograph of the patient, judged as “good.”

postoperative inversion of a few eyelashes toward the globe may be managed conservatively. The postoperative touch between the eyelashes and the globe in the group of patients with a “fair” outcome was mainly at the most medial part of the lower eyelid. Hwang et al9 recently reported excellent results using an eyelid margin split technique to repair this kind of medial malposition in patients with cilial entropion. Because the lacrimal punctum is located in this area, it may be difficult to repair this malposition successfully without causing punctal eversion. By splitting the anterior and posterior lamellae of the lower eyelid, the eyelashes are everted without causing punctal eversion. The absence of an adhesion between the lower eyelid retractor fibers and the anterior lamella of the lower eyelid is a widely accepted cause of cilial entropion.1,4,5,9 This, however, is not supported by histologic studies. Microscopic anatomic studies of the lower eyelid in adults have demonstrated direct fibers from the lower eyelid retractors to the skin, without racial differences in fiber course.11–13 We speculate that the fibers in cilial entropion are not absent

but may be too loose to pull the anterior lamella of the lower eyelid effectively. Higher septal attachment to the lower tarsal plate has also been proposed as a possible cause of cilial entropion.1 Although the confluence point between the orbital septum and the lower eyelid retractors is higher in Asians than in Caucasians,11,12 the position is always lower than the inferior tarsal plate edge and not on the tarsal plate itself.12 In addition, the orbital septum is a part of the anterior layer of the lower eyelid retractors after the confluence and does not interrupt the connection between the skin and the lower eyelid retractors. Thus, the higher septum attachment theory is probably not a causative factor in cilial entropion. Debulking the pretarsal orbicularis oculi muscle enables easier rotation of the lower eyelid margin. Although hypertrophy of the marginal fibers of the orbicularis oculi muscle is also considered as a possible cause of cilial entropion,15 this finding has not been confirmed histopathologically. A study by Tse et al,16 on cases of congenital entropion (but not cilial entropion), found no histologic evidence of orbicularis oculi muscle hypertrophy. No histopathologic exam-

4™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™™ Figure 1. A, Preoperative photograph showing bilateral cilial entropion. B, Surgeon’s view: Drawing a skin incision line from the lower lacrimal punctum to around the line of the lateral corneal limbus in a region 3 mm below the eyelashes. C, Surgeon’s view: After incising the skin and fixing the eyelid with an entropion clamp, the layer under the orbicularis oculi muscle is detached toward the eyelashes, and the wound is pulled at 3 points with hooks with 4-0 silk sutures. At this stage, the inferior border of the tarsal plate is still obscure. D, Surgeon’s view: Inferior dissection of the pretarsal attachment of the anterior layer of the lower eyelid retractors enables clear and definite visualization of the inferior tarsal plate border. E, The diagram of Figure 1D. F, Surgeon’s view: The orbicularis oculi muscle in the eyelid margin is slightly debulked to enable easy marginal roll. G, The diagram of the Figure 1F. H, Surgeon’s view: The detached anterior layer of the lower eyelid retractors is fixed with 6-0 nylon or polypropylene sutures to the inferior tarsal end and the dermis with marginal orbicularis oculi muscle. I, The diagram of Figure 1H.

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Figure 2. A patient judged as having a “fair” postoperative outcome, observed for 50 months. A, Preoperative state (3 years and 3 months old) showing bilateral lower eyelid cilial entropion. B, Six months postoperatively (3 years and 9 months old), the medial part of the lower eyelid skin covers the lower eyelid margin, but only a few inverted eyelashes are in direct contact with the globe. C, Fifty months postoperatively (7 years and 4 months old); no skin covers the lower eyelid margin. Although the eyelashes grow upright, only a few inverted eyelashes are in direct contact with the globe, similar to the postoperative findings 4 years earlier.

inations were performed on debulked orbicularis oculi muscles in our patients, but our clinical impression is that these muscles were not significantly hypertrophied compared with other lower eyelid involutional entropion cases17 (Fig 1C, D). When considering the growth of children and facial bone maturation, the need to remove a small amount of subciliary skin in cases of cilial entropion may be disputed.1,5,9,18 In our series, no skin was removed, but the success rate remained high. On the basis of the findings of this study (and demonstrated in Fig 2), the amount of redundant skin gradually decreases with age, and there are strong indications against skin removal. Although after facial maturation it might be reasonable to remove the redundant skin, a definite recommendation cannot be established on the basis of the small number of older patients in our study. Future comparative studies are required to definitely answer these issues of skin removal. Some studies suggest attaching the lower eyelid retractors (i.e., posterior layer as well) to the tarsus and/or skin or dermis.4,8,16 However, because the definition of “cilial entropion” is based on a normal position of the tarsal plate,4 the pathology of cilial entropion is attributed only to the fibers directed to the skin (i.e., anterior layer). Therefore, we do not think that posterior layer advancement is required to improve cilial entropion. The posterior layer is the main tractional component of the lower eyelid retractors7 and should therefore be advanced in cases of involutional lower eyelid entropion17 and reverse ptosis.19 Advancing the posterior layer in cases of cilial entropion may result in significant lower eyelid retraction. Nonabsorbable sutures were used in our patients. Absorbable sutures may also be used and should have at least the same effect in creating the desired scar between the skin

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and orbicularis muscle and the inferior edge of the tarsal plate, as long as careful dissection and layer identification are carried out during surgery. There were no cases of recurrence, except the 2 eyelids with a “poor” outcome, during a mean follow-up period of 27 months. However, because the number of recurrences will be ever increasing with a longer follow-up period, the true recurrence rate might be higher once all our patients complete the usual 5-year postoperative follow-up period. In conclusion, our modification of the Hotz procedure, based on identifying and dividing the anterior layer of the lower eyelid retractors and reinforcing the skin to the lower border of the inferior tarsal plate and lower eyelid retractors, is a useful and predictive technique for cilial entropion repair and results in a high success rate and a low recurrence rate.

References 1. Woo KI, Yi K, Kim YD. Surgical correction for lower lid epiblepharon in Asians. Br J Ophthalmol 2000;84:1407–10. 2. Noda S, Hayasaka S, Setogawa T. Epiblepharon with inverted eyelashes in Japanese children. I. Incidence and symptoms. Br J Ophthalmol 1989;73:126 –7. 3. Levitt JM. Epiblepharon and congenital entropion. Am J Ophthalmol 1957;44:112–3. 4. Jordan R. The lower-lid retractors in congenital entropion and epiblepharon. Ophthalmic Surg 1993;24:494 – 6. 5. Preechawai P, Amrith S, Wong I, Sundar G. Refractive changes in epiblepharon. Am J Ophthalmol 2007;143:835–9. 6. Hayasaka S, Noda S, Setogawa T. Epiblepharon with inverted eyelashes in Japanese children. II. Surgical repairs. Br J Ophthalmol 1989;73:128 –30.

Kakizaki et al 䡠 Modified Hotz Procedure in Cilial Entropion 7. Kakizaki H, Zhao J, Nakano T, et al. The lower eyelid retractor consists of definite double layers. Ophthalmology 2006; 113:2346 –50. 8. Millman AL, Mannor GE, Putterman AM. Lid crease and capsulopalpebral fascia repair in congenital entropion and epiblepharon. Ophthalmic Surg 1994;25:162–5. 9. Hwang SW, Khwarg SI, Kim JH, et al. Lid margin split in the surgical correction of epiblepharon. Acta Ophthalmol 2008; 86:87–90. 10. Hotz FC. A new operation for entropion and trichiasis. Arch Ophthalmol 1879;8:249 – 63. 11. Hawes MJ, Dortzbach RK. The microscopic anatomy of the lower eyelid retractors. Arch Ophthalmol 1982;100:1313– 8. 12. Kakizaki H, Jinsong Z, Zako M, et al. Microscopic anatomy of Asian lower eyelids. Ophthal Plast Reconstr Surg 2006;22:430–3. 13. Lim WK, Rajendran K, Choo CT. Microscopic anatomy of the lower eyelid in Asians. Ophthal Plast Reconstr Surg 2004;20: 207–11.

14. Quickert MH, Wilkes TD, Dryden RM. Nonincisional correction of epiblepharon and congenital entropion. Arch Ophthalmol 1983;101:778 – 81. 15. Johnson CC. Epiblepharon. Am J Ophthalmol 1968;66: 1172–5. 16. Tse DT, Anderson RL, Fratkin JD. Aponeurosis disinsertion in congenital entropion. Arch Ophthalmol 1983;101: 436 – 40. 17. Kakizaki H, Zako M, Kinoshita S, Iwaki M. Posterior layer advancement of the lower eyelid retractor in involutional entropion repair. Ophthal Plast Reconstr Surg 2007;23: 292–5. 18. Tabuchi A, Yamamoto M. Surgery (skin resection) for childhood inferior epiblepharon [in Japanese]. Ganka Rinsho Iho (Jpn Rev Clin Ophthalmol) 1972;66:334 –7. 19. Kakizaki H, Zako M, Iwaki M. Reverse ptosis repair targeting the posterior layer of the lower eyelid retractor. Ophthal Plast Reconstr Surg 2007;23:288 –91.

Footnotes and Financial Disclosures Originally received: November 17, 2008. Final revision: April 6, 2009. Accepted: April 7, 2009. Available online: September 10, 2009.

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Manuscript no. 2008-1357.

1

Department of Ophthalmology, Aichi Medical University, Nagakute, Japan. 2

South Australian Institute of Ophthalmology and Discipline of Ophthalmology and Visual Sciences, University of Adelaide, Australia.

Division of Oculoplastic and Orbital Surgery, Department of Ophthalmology, Tel-Aviv Medical Center, Tel-Aviv University, Tel-Aviv, Israel. Financial Disclosure(s): The author(s) have no proprietary or commercial interest in any materials discussed in this article. Correspondence: Hirohiko Kakizaki, MD, PhD, Department of Ophthalmology, Aichi Medical University, Nagakute, Aichi 480-1195, Japan. E-mail: cosme@d1. dion.ne.jp.

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