Ohlhausen Marc, Nguyen Nam V, Kaftan Timothy, Song Helen, Kim Sean, Reiff Daniel, Yeh Steven
Department of Ophthalmology, Stanley M. Truhlsen Eye Institute, University of Nebraska Medical Center, Omaha, NE, USA.
College of Medicine, University of Nebraska Medical Center, Omaha, NE, USA.
Am J Ophthalmol Case Rep. 2024 Dec 12;37:102231. doi: 10.1016/j.ajoc.2024.102231. eCollection 2025 Mar.
To describe a rare case of presumed bilateral acute idiopathic maculopathy (AIM) in a pediatric patient.
An 11-year-old male was evaluated for a "fuzzy Dorito-shaped" spot in the central vision of his right eye (OD) that started 3 days before presenting to our clinic. On examination, best-corrected visual acuity (BCVA) was counting fingers at 5 feet OD, and 20/25 in the left eye (OS). Fundus examination demonstrated a central macular lesion with pigmentary changes OD and mild retinal pigment epithelium (RPE) changes OS. Optical coherence tomography (OCT) imaging showed elevated, irregular RPE with overlying subretinal fluid in the fovea OD and trace ellipsoid zone changes temporal to the fovea OS. Uveitis work-up was unremarkable for any infectious or inflammatory etiologies. Given the severe vision loss in the right eye and the negative infectious work-up, the decision was made to initiate oral corticosteroids in agreement with the patient's family. One week following the initial presentation, the patient developed visual symptoms OS without much improvement OD. The patient was admitted to the hospital, and further work-up revealed elevated coxsackie B3 antibody titers (1:20). Given the characteristic findings on multimodal imaging and elevated coxsackie B3 antibodies, the patient was presumptively diagnosed with bilateral AIM. Over the following 4 months, he would intermittently complain of nonspecific visual complaints such as blurry vision and floaters requiring an increase in corticosteroid dose and addition of immunomodulatory therapy, although the clinical examination and OCT findings remained unchanged. At the patient's follow-up visit 8 months after the initial visit, BCVA was stable at 20/40 OD and 20/30 OS, and OCT demonstrated stable foveal lesions in both eyes.
This case report describes a rare presentation of presumed bilateral AIM in a pediatric patient. This prompts further consideration of this condition in pediatric patients, especially after a prodromal flu-like illness.
描述一例儿科患者双侧疑似急性特发性黄斑病变(AIM)的罕见病例。
一名11岁男性因右眼(OD)中心视力出现“模糊的多力多滋形状”斑点前来就诊,该斑点在就诊前3天出现。检查时,右眼最佳矫正视力(BCVA)为5英尺数指,左眼(OS)为20/25。眼底检查显示右眼黄斑中心病变伴有色素改变,左眼有轻度视网膜色素上皮(RPE)改变。光学相干断层扫描(OCT)成像显示右眼黄斑中心凹RPE隆起、不规则,伴有视网膜下液,左眼黄斑中心凹颞侧椭圆体带轻度改变。葡萄膜炎相关检查未发现任何感染或炎症病因。鉴于右眼严重视力丧失且感染相关检查结果为阴性,经与患者家属协商后决定开始口服皮质类固醇治疗。初次就诊一周后,患者左眼出现视觉症状,右眼改善不明显。患者入院进一步检查发现柯萨奇B3抗体滴度升高(1:20)。鉴于多模态成像的特征性表现及柯萨奇B3抗体升高,该患者被疑似诊断为双侧AIM。在接下来的4个月里,他间歇性地抱怨视力模糊和飞蚊症等非特异性视觉症状,需要增加皮质类固醇剂量并加用免疫调节治疗,尽管临床检查和OCT结果未变。初次就诊8个月后的随访中,右眼BCVA稳定在20/40,左眼稳定在20/30,OCT显示双眼黄斑中心凹病变稳定。
本病例报告描述了儿科患者双侧疑似AIM的罕见表现。这提示在儿科患者中,尤其是在出现前驱性流感样疾病后,应进一步考虑这种情况。