Magalhães Augusto, Vilares-Morgado Rodrigo, Cunha Ana Maria, Leão-Teles Elisa, Falcão Manuel, Carneiro Ângela, Falcão-Reis Fernando
Department of Ophthalmology, Centro Hospitalar Universitário de São João, Porto, Portugal.
Department of Surgery and Physiology, Faculty of Medicine of University of Porto, Porto, Portugal.
Case Rep Ophthalmol. 2021 Sep 27;12(3):816-823. doi: 10.1159/000518443. eCollection 2021 Sep-Dec.
The purpose of this clinical case report is to describe a case of mucopolysaccharidosis type IVA (MPS IVA), or Morquio syndrome, with increased choroidal thickness in enhanced-depth imaging optical coherence tomography (EDI-OCT) which can represent choroidal deposition of glycosaminoglycans (GAGs). A 21-year-old male with genetically confirmed diagnosis of MPS IVA was examined at our Pediatric Ophthalmology clinic as part of our follow-up protocol for MPS patients. His best-corrected visual acuity was 4/10 in his right eye (OD) and 6/10 in the left eye (OS). Mild diffuse corneal opacification was evident. Intraocular pressure was within normal range. Fundus examination and color fundus photography revealed no abnormalities. EDI-OCT revealed significantly increased choroidal thickness in his right eye and in his left eye, suggesting the presence of choroidal deposition of GAGs, despite absence of retinal or optic disc GAG deposition or other chorioretinal involvement. To our knowledge, this is the first case of MPS IVA described in the literature with suspected choroidal deposition of GAGs. With improved control of systemic features of MPS IVA, life expectancy of these patients has increased, allowing for more ocular manifestations to develop. The parallel development of technology in ophthalmology, such as the EDI-OCT, further contributes to the detection of these unprecedented ocular features in MPSs.
本临床病例报告的目的是描述一例IVA型黏多糖贮积症(MPS IVA),即Morquio综合征,在增强深度成像光学相干断层扫描(EDI-OCT)中脉络膜厚度增加,这可能代表糖胺聚糖(GAGs)在脉络膜沉积。一名经基因确诊为MPS IVA的21岁男性,作为我们对MPS患者随访方案的一部分,在我们的儿科眼科诊所接受了检查。他的右眼最佳矫正视力为4/10,左眼为6/10。可见轻度弥漫性角膜混浊。眼压在正常范围内。眼底检查和彩色眼底照相未发现异常。EDI-OCT显示他的右眼和左眼脉络膜厚度显著增加,提示存在GAGs脉络膜沉积,尽管没有视网膜或视盘GAG沉积或其他脉络膜视网膜受累情况。据我们所知,这是文献中描述的首例疑似GAGs脉络膜沉积的MPS IVA病例。随着MPS IVA全身症状控制的改善,这些患者的预期寿命增加,使得更多眼部表现得以出现。眼科技术的同步发展,如EDI-OCT,进一步有助于检测这些MPS中前所未有的眼部特征。