Orbit, 2014; 33(6): 444–448 ! Informa Healthcare USA, Inc. ISSN: 0167-6830 print / 1744-5108 online DOI: 10.3109/01676830.2014.950298

ORIGINAL ARTICLE

Congenital Entropion: Outcome of Posterior Layer Advancement of Lower Eyelid Retractors and Histological Study of Orbicularis Oculi Muscle Hypertrophy Yasuhiro Takahashi1, Hiroshi Ikeda2, Akihiro Ichinose3, and Hirohiko Kakizaki1

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1

Department of Ophthalmology and 2Department of Pathology, Aichi Medical University, Nagakute, Aichi, Japan, and 3Department of Plastic Surgery, Kobe University School of Medicine, Kobe, Hyogo, Japan

ABSTRACT Purpose: To examine surgical outcomes of the posterior layer advancement of the lower eyelid retractors (LER) in patients with congenital entropion and to ascertain whether the pretarsal orbicularis oculi muscle (OOM) was hypertrophic or not. Materials and Methods: This retrospective study included three Japanese patients with congenital entropion who underwent posterior layer advancement of the LER. We reviewed the surgical outcomes and postoperative complications, such as lower eyelid ectropion and retraction for each patient. We also measured the diameters of the pretarsal OOM fibers in these patients and compared them with those measured in the previously reported 67 eyelids of 41 Japanese patients with congenital epiblepharon. Results: Congenital entropion was successfully corrected in all three patients without recurrence during 12-months of follow-up. No patient exhibited lower eyelid ectropion or lower eyelid retraction. The mean diameter of the pretarsal OOM fibers was 21.9 mm (range, 20.5–23.7 mm), which was not significantly different from that of the congenital epiblepharon (25.3 mm; range, 18.1–34.7 mm; p = 0.272, Mann–Whitney U test). Conclusions: The surgical outcomes in the present study suggest that posterior layer advancement of the LER is a useful surgical option for treatment of congenital entropion. No histological evidence of pretarsal OOM hypertrophy was shown in congenital entropion, which demonstrated that debulking of the pretarsal OOM may not be significant for correction of this entity. Keywords: Congenital entropion, hypertrophy, lower eyelid retractors, posterior layer advancement, pretarsal orbicularis oculi muscle

INTRODUCTION

address the double-layer anatomy of the LER, in which the posterior layer acts as the main vertical tractional component.6,7 It is inferred from the etiology of congenital entropion that posterior layer advancement of the LER is an effective surgical option for these patients. The pretarsal orbicularis oculi muscle (OOM) has been assumed to be hypertrophic in patients with congenital entropion, which causes an inward push of the lower tarsal plate.8-10 Removal of the pretarsal OOM is therefore recommended to correct congenital entropion.8,9 However, this assumption is based

Congenital entropion is an uncommon anomaly characterized by an inward rolling of the entire lower eyelid toward the globe.1,2 The main etiology of congenital entropion is attenuation or dehiscence of the lower eyelid retractors (LER).1-4 As congenital entropion causes corneal abrasion and even visual impairment, surgical correction is required.1 Posterior layer advancement of the LER is a procedure to definitely advance and fix the posterior layer of the LER.5 This approach was designed to

Received 4 March 2014; Accepted 28 July 2014; Published online 10 September 2014 Correspondence: Hirohiko Kakizaki, Department of Ophthalmology, Aichi Medical University, Nagakute, Aichi 480-1195, Japan. Tel: +81-561-62-3311 (ext. 22181), Fax: +81-561-63-7255, E-mail: [email protected]

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Congenital Entropion 445 strictly on clinical observations.1 A previous histological study showed no evidence of the pretarsal OOM hypertrophy in two patients with congenital entropion, although no statistical comparison was performed.1 Here, we reviewed surgical outcomes of the posterior layer advancement of the LER for congenital entropion. We also examined the diameter of the pretarsal OOM in patients with congenital entropion to ascertain the presence of pretarsal OOM hypertrophy.

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MATERIALS AND METHODS The present study included three Japanese patients who underwent posterior layer advancement of the LER for congenital entropion. All patients exhibited unilateral congenital entropion (Table 1). All procedures were performed by a single oculoplastic surgeon (HK). Institutional review board approval was obtained from Aichi Medical University (No. 13–080), and informed consent was obtained from all patients or their guardians. The procedure was performed as follows. The 4-year-old boy was treated under general anesthesia with local anesthesia of 2 mL 1% lidocaine with 1:100,000 epinephrine, and the other two were treated

under the same local anesthesia. The skin was incised with length of 20 mm at 3 mm below the cilia. The layer under the OOM was dissected toward the cilia. The anterior layer LER on the tarsal plate was dissected inferiorly to the lower tarsal margin, where the insertion of the posterior layer LER was severed to expose the conjunctiva. Then, the posterior layer of the LER was detached from the conjunctiva to 5 mm below that point. The orbital septum was incised in the transverse direction, and the sheet-like LER was exposed (Figure 1A and B). The pretarsal OOM was minimally debulked to facilitate everting the lower eyelid margin. The site 2 mm below the posterior layer edge was fixed to the lower edge of the tarsal plate using 6-0 polyvinylidene fluoride (Asflex, Kono Seisakusho Co., Ltd., Tokyo, Japan) sutures, with simultaneous advancement of the anterior layer as reinforcement for the posterior layer (Figure 1C). We added two to five additional sutures and secured the pretarsal OOM and the lower tarsal edge at three to six points (Figure 1D). Finally, the skin was sutured with 6-0 polyvinylidene fluoride sutures. We retrospectively reviewed surgical outcomes and postoperative complications, such as lower eyelid ectropion and retraction at 12 months postoperatively. We measured the diameter of the pretarsal OOM fibers obtained during surgery. The measurement

FIGURE 1. Intraoperative photographs (top, cranial: right, temporal). (A, B) The sheet-like structure of the LER. The anterior and posterior layers are clearly defined. The posterior layer, which includes smooth muscle fibers, is contracted because of the epinephrine effect. (C, D) The posterior layer as well as the anterior layer are advanced and fixed to the lower edge of the tarsal plate. The pretarsal orbicularis oculi muscle and the lower edge of the tarsal plate are secured at four points. !

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446 Y. Takahashi et al. methods were described previously in detail.11 The specimens were dehydrated, embedded in paraffin, and cut into 7-mm slices, perpendicular to the direction of the muscle fibers. These sections were then stained with hematoxylin and eosin. Photographs of the sectioned areas were taken with a digital camera system attached to a microscope (Moticam 2000; Shimadzu Rika Kikai, Tokyo, Japan) at400 magnification. Only specimens with cross-sectional areas that included large muscle fibers were selected. In each section, 10 muscle fibers were measured across their smallest diameter with Adobe Photoshop CS5 Extended (Adobe Systems Inc., San Jose, CA, USA), thereby avoiding inaccurate measurements of muscle kinking occurring during the processing or by any obliquity of the plane of section. We used previously reported results as a reference.11 We selected the results for 67 eyelids of 41 Japanese patients with congenital epiblepharon with similar patient age (mean, 14.1 years) and showing no evidence of pretarsal OOM hypertrophy.11 The Mann– Whitney U-test was used for statistical comparison.

Statistical significance was defined as p50.05. Statistical analysis was carried out using the Dr. SPSS II for Windows (SPSS Japan Inc., Tokyo, Japan).

RESULTS Congenital entropion was successfully corrected in all three patients (Figures 2–4). Recurrence did not occur during the 12-month follow-up period. None of the patients exhibited postoperative lower eyelid retraction. The mean diameter of the pretarsal OOM fibers in the three patients was 21.9 mm (range, 20.5–23.7 mm; Figure 5A).This result was not statistically different from that obtained from controls (25.3 mm; range, 18.1–34.7 mm; p = 0.272; Figure 5B).

DISCUSSION Congenital entropion was successfully repaired using posterior advancement of the LER in all three patients

TABLE 1 Patient data. Patient 1 2 3

Age (years)

Gender

Side

Other eye diseases

Simultaneous operation

20 4 25

F M F

L R L

TED + R cilial entropion R congenital ptosis None

B orbital decompression + R modified Hotz procedure R suspension surgery None

F, female; M, male; L, left; R, right; TED, thyroid eye disease; B, bilateral.

FIGURE 2. (A) Preoperative photograph of a 20-year-old female patient with left congenital entropion. The right lower eyelid shows a congenital epiblepharon. Bilateral proptosis (Hertel exophthalmometric value, 21.0 mm OU) due to thyroid eye disease is also demonstrated. (B) Postoperative photograph. The right congenital epiblepharon and the bilateral proptosis (Hertel exophthalmometric value, 16.5 mm OU) were simultaneously repaired using the modified Hotz procedure and deep lateral orbital wall decompression, respectively.

FIGURE 3 (A) Preoperative photograph of a 4-year-old male patient with right congenital entropion. (B) Postoperative photograph. Symmetrical upper eyelid position was achieved through simultaneous suspension surgery on the right upper eyelid. Orbit

Congenital Entropion 447

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FIGURE 4 (A) Preoperative photograph of a 25-year-old female patient with left congenital entropion. (B) Postoperative face photograph.

FIGURE 5 Specimens of pretarsal orbicularis oculi muscle (hematoxylin & eosin stain; magnification,400; bar length = 20 mm). (A) Specimens from the 25-year-old female patient. (B) Specimens from a patient with congenital epiblepharon.

without recurrence. The mean diameter of the pretarsal OOM was not significantly different between the groups. Several reports have illustrated good surgical results with a LER re-insertion technique for congenital entropion.1,3,4,12 These procedures did not involve, however, dissection of the posterior layer of the LER from the conjunctiva. Exposing the posterior layer of the LER enables its definite advancement and firm fixation with a scar on the lower tarsal plate.5,13,14 We therefore believe that our procedure is more advantageous than the LER re-insertion technique to prevent recurrence in congenital entropion. The present study demonstrated no histological evidence of the pretarsal OOM hypertrophy in patients with congenital entropion. This finding did not correspond to the OOM hypertrophy hypothesis on clinical observations8-10 but was consistent with a previous histological analysis.1 Based on the histological findings in the present and previous studies,1 debulking of the pretarsal OOM may not be significant for correction of the congenital entropion. There are several limitations in our study. This study was retrospective in nature. The small sample size and relatively short follow-up period were other disadvantages. Although we included only Japanese patients, our technique may be also effective for congenital entropion repair in Caucasian patients as !

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the LER in Caucasians exhibit the double-layer structure as well.7 In conclusion, we successfully repaired congenital entropion in three patients using posterior layer advancement of the LER. This procedure is a useful surgical option for treatment of the congenital entropion. Hypertrophy of the pretarsal OOM was not shown in patients with congenital entropion, in which debulking of the pretarsal OOM may not be significant for correction of this entity.

DECLARATION OF INTEREST The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

REFERENCES 1. Tse DT, Anderson RL, Fratkin JD. Aponeurosis disinsertion in congenital entropion. Arch Ophthalmol 1983;101:436–440. 2. Quickert MH, Wilkes TDI, Dryden RM. Nonincisional correction of epiblepharon and congenital entropion. Arch Ophthalmol 1983;101:778–781. 3. Jordan R. The lower-lid retractors in congenital entropion and epiblepharon. Ophthalm Surg 1993;24:494–496. 4. Millman AL, Mannor GE, Putterman AM. Lid crease and capsulopalpebral fascia repair in congenital entropion and epiblepharon. Ophthalm Surg 1994;25:162–165.

448 Y. Takahashi et al. 10. Crawford JS. Congenital eyelid anomalies in children. J Pediatr Ophthalmol Strabis 1984;21:140–149. 11. Kakizaki H, Takahashi Y, Kang H, et al. No histological evidence of orbicularis oculi muscle hypertrophy in congenital entropion. Clin Experiment Ophthalmol 2013;41: 167–171. 12. Mandal AK, Honavar SG, Gothwal VK. The association of unilateral congenital glaucoma and congenital lower lid entropion: causal or casual? Ophthalm Surg Lasers 2001;32: 149–151 13. Anderson RL, Dixon RS. Aponeurotic ptosis surgery. Arch Ophthalmol 1979;97:1123–1128. 14. Kakizaki H, Zako M, Mito H, et al. Modified operation to correctly detect and fix the lower eyelid retractor in involutional entropion. Jpn J Ophthalmol 2005;49:330–332.

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5. 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: 292–295. 6. Kakizaki H, Zhao J, Nakano T, et al. The lower eyelid retractor consists of definite double layers. Ophthalmology 2006;113:2346–2350. 7. Kakizaki H, Chan W, Madge SN, et al. Lower eyelid retractors in Caucasians. Ophthalmology 2009;116: 1402–1404. 8. Pereira MGB, Rodrigues MA, Rodrigues SAC. Eyelid entropion. Semin Ophthalmol 2010;25:52–58. 9. Christiansen G, Mohney BG, Baratz KH, Bradley EA. Botulinum toxin for the treatment of congenital entropion. Am J Ophthalmol 2004;138:153–155.

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Congenital entropion: outcome of posterior layer advancement of lower eyelid retractors and histological study of orbicularis oculi muscle hypertrophy.

To examine surgical outcomes of the posterior layer advancement of the lower eyelid retractors (LER) in patients with congenital entropion and to asce...
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