Abe Ricardo Yuji, Makarczyk Luciana de Sá Quirino, Ávila Marcos Pereira de, Sallum Juliana Maria Ferraz
Hospital Oftalmológico de Brasília, Brasília, DF, Brazil.
Department of Opthalmology, Universidade Federal de Goiás, Goiânia, GO, Brazil.
Arq Bras Oftalmol. 2023 Jan-Feb;86(1):74-78. doi: 10.5935/0004-2749.20220078.
We describe the case of a 15-year-old girl with decreased visual acuity associated with elevated intraocular pressure in both eyes and angle closure on gonioscopy. She also presented attenuation of retinal vessels and optic disc pallor with large excavation in the left eye. Ultrasound biomicroscopy revealed an anteriorly positioned ciliary body and absence of ciliary sulcus, confirming the plateau iris configuration. Spectral-domain optical coherence tomography revealed a bilateral cystoid macular edema. Genetic screening revealed heterozygous variants of the Crumbs homolog 1 (CRB1) gene (c.2843G>A and c.2506C>A). The patient underwent trabeculectomy for intraocular pressure control and topical treatment for macular edema. This case highlights the importance of performing gonioscopy and evaluating intraocular pressure in patients with a shallow anterior chamber despite young age. In addition, it also shows the importance of genetic screening, when available, in elucidating the diagnosis and providing patients and their families' information on the patient's prognosis and possible therapeutic options.
我们描述了一名15岁女孩的病例,她双眼视力下降,伴有眼压升高,前房角镜检查显示房角关闭。她还表现出视网膜血管变细以及左眼视盘苍白并伴有大的视盘凹陷。超声生物显微镜检查显示睫状体向前移位且睫状沟消失,证实为高褶虹膜构型。频域光学相干断层扫描显示双侧黄斑囊样水肿。基因筛查发现Crb1(Crumbs同源物1)基因存在杂合变异(c.2843G>A和c.2506C>A)。该患者接受了小梁切除术以控制眼压,并接受了黄斑水肿的局部治疗。该病例强调了尽管患者年龄较小,但对于前房浅的患者进行前房角镜检查和评估眼压的重要性。此外,它还显示了在可行时进行基因筛查对于明确诊断以及为患者及其家属提供有关患者预后和可能治疗选择信息的重要性。