{"title":"一名戈登哈尔综合征患儿的眼睑皮样囊肿切除术后出现假性翼状胬肉","authors":"Dea Prita Caesarita, Julie D. Barliana","doi":"10.35749/0e622333","DOIUrl":null,"url":null,"abstract":"Introduction : Goldenhar syndrome is a rare congenital disorder also known as oculoauricularvertebral dysplasia. Limbal dermoid associated with this syndrome. This case report aims to present an reccurent pseudopterygium after three times limbal dermoid cyst excision in patient with Goldenhar syndrome. \nCase Illustration : Patient 13 years old male with pseudopterygium in right eye. Visual acuity both eyes 6/6 snellen chart. Fibrovascular tissue from tarsal conjunctival inferotemporal to 2 mm limbal cornea in right eye. Patient had limbal dermoid cyst in right eye since birth and underwent limbal dermoid cyst excision for three times. First time surgery used amniotic membrane transplantation, then second and third surgery used conjunctival limbal graft. Goldenhar syndrome have been diagnosed since the age of 2 years old. External examination noticed asymmetrical hemifacial microsmia, grade III microtia right ear, conduction deafness, and laryngomalacia. \nDiscussion : Limbal dermoid cyst is one of characteristic of Goldenhar syndrome. In this case pseudopterygium grade II in right eye developed after limbal dermoid cyst excision for three times. Limbal dermoid simple excision may lead to corneal vascularization persistent epithelial defect, scar, and pseudopterygium. Various surgical include superficial keratectomy, amniotic membrane transplantation and lamellar or penetrating keratoplasty. Pseudopterygium still occurs in this case, even though recurrence has been prevented by administration of amniotic membrane transplantation and conjunctival limbal graft. \nConclusion : We should be concerned about management of limbal dermoid cyst with Goldenhar syndrome to prevent recurrent pseudopterygium.","PeriodicalId":165753,"journal":{"name":"Ophthalmologica Indonesiana","volume":"133 12","pages":""},"PeriodicalIF":0.0000,"publicationDate":"2024-02-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Pseudopterygium After Limbal Dermoid Cyst Excision in a Child with Goldenhar Syndrome\",\"authors\":\"Dea Prita Caesarita, Julie D. Barliana\",\"doi\":\"10.35749/0e622333\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"Introduction : Goldenhar syndrome is a rare congenital disorder also known as oculoauricularvertebral dysplasia. Limbal dermoid associated with this syndrome. This case report aims to present an reccurent pseudopterygium after three times limbal dermoid cyst excision in patient with Goldenhar syndrome. \\nCase Illustration : Patient 13 years old male with pseudopterygium in right eye. Visual acuity both eyes 6/6 snellen chart. Fibrovascular tissue from tarsal conjunctival inferotemporal to 2 mm limbal cornea in right eye. Patient had limbal dermoid cyst in right eye since birth and underwent limbal dermoid cyst excision for three times. First time surgery used amniotic membrane transplantation, then second and third surgery used conjunctival limbal graft. Goldenhar syndrome have been diagnosed since the age of 2 years old. External examination noticed asymmetrical hemifacial microsmia, grade III microtia right ear, conduction deafness, and laryngomalacia. \\nDiscussion : Limbal dermoid cyst is one of characteristic of Goldenhar syndrome. In this case pseudopterygium grade II in right eye developed after limbal dermoid cyst excision for three times. Limbal dermoid simple excision may lead to corneal vascularization persistent epithelial defect, scar, and pseudopterygium. Various surgical include superficial keratectomy, amniotic membrane transplantation and lamellar or penetrating keratoplasty. Pseudopterygium still occurs in this case, even though recurrence has been prevented by administration of amniotic membrane transplantation and conjunctival limbal graft. \\nConclusion : We should be concerned about management of limbal dermoid cyst with Goldenhar syndrome to prevent recurrent pseudopterygium.\",\"PeriodicalId\":165753,\"journal\":{\"name\":\"Ophthalmologica Indonesiana\",\"volume\":\"133 12\",\"pages\":\"\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2024-02-12\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Ophthalmologica Indonesiana\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.35749/0e622333\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Ophthalmologica Indonesiana","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.35749/0e622333","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0
摘要
导读:戈登哈尔综合征是一种罕见的先天性疾病,又称眼耳椎体发育不良。眼睑皮样瘤与该综合征有关。本病例报告旨在介绍一名患有戈登哈尔综合征的患者,在接受了三次肢端皮样囊肿切除术后,再次出现了假性翼状胬肉。病例说明:患者男性,13 岁,右眼假性翼状胬肉。双眼视力均为 6/6 斯奈伦视力表。右眼跗骨结膜下颞部至 2 mm 角膜缘有纤维血管组织。患者自出生起右眼就患有角膜缘样皮样囊肿,曾三次接受角膜缘样皮样囊肿切除术。第一次手术采用羊膜移植,第二次和第三次手术采用结膜角膜移植。两岁时就被诊断出患有戈登哈尔综合症。外部检查发现他有不对称的半侧颜面小肌症、右耳三级小耳症、传导性耳聋和喉头畸形。讨论肢端皮样囊肿是戈登哈尔综合征的特征之一。在本病例中,右眼假性翼状胬肉在进行了三次睑缘皮样囊肿切除术后形成。角膜缘蝶窦囊肿单纯切除术可能会导致角膜血管化、上皮持续缺损、瘢痕和假性翼状胬肉。各种手术包括浅层角膜切除术、羊膜移植术、板层或穿透性角膜移植术。尽管通过羊膜移植和结膜缘移植防止了复发,但该病例仍出现了假性翼状胬肉。结论:我们应该关注伴有戈登哈尔综合征的睑缘皮样囊肿的治疗,以防止假性翼状胬肉复发。
Pseudopterygium After Limbal Dermoid Cyst Excision in a Child with Goldenhar Syndrome
Introduction : Goldenhar syndrome is a rare congenital disorder also known as oculoauricularvertebral dysplasia. Limbal dermoid associated with this syndrome. This case report aims to present an reccurent pseudopterygium after three times limbal dermoid cyst excision in patient with Goldenhar syndrome.
Case Illustration : Patient 13 years old male with pseudopterygium in right eye. Visual acuity both eyes 6/6 snellen chart. Fibrovascular tissue from tarsal conjunctival inferotemporal to 2 mm limbal cornea in right eye. Patient had limbal dermoid cyst in right eye since birth and underwent limbal dermoid cyst excision for three times. First time surgery used amniotic membrane transplantation, then second and third surgery used conjunctival limbal graft. Goldenhar syndrome have been diagnosed since the age of 2 years old. External examination noticed asymmetrical hemifacial microsmia, grade III microtia right ear, conduction deafness, and laryngomalacia.
Discussion : Limbal dermoid cyst is one of characteristic of Goldenhar syndrome. In this case pseudopterygium grade II in right eye developed after limbal dermoid cyst excision for three times. Limbal dermoid simple excision may lead to corneal vascularization persistent epithelial defect, scar, and pseudopterygium. Various surgical include superficial keratectomy, amniotic membrane transplantation and lamellar or penetrating keratoplasty. Pseudopterygium still occurs in this case, even though recurrence has been prevented by administration of amniotic membrane transplantation and conjunctival limbal graft.
Conclusion : We should be concerned about management of limbal dermoid cyst with Goldenhar syndrome to prevent recurrent pseudopterygium.