Kayukawa Kanae, Kitazawa Koji, Wakimasu Koichi, Sotozono Chie, Kinoshita Shigeru
Department of Ophthalmology, Kyoto Prefectural University of Medicine, Kyoto, Japan.
Department of Frontier Medical Science and Technology for Ophthalmology, Kyoto Prefectural University of Medicine, Kyoto, Japan.
Am J Ophthalmol Case Rep. 2023 Jan 12;29:101796. doi: 10.1016/j.ajoc.2023.101796. eCollection 2023 Mar.
To report a rare case of lattice corneal dystrophy type 1 (LCD1) with bilateral Mooren's ulcer.
This case involved a 62-year-old male patient with LCD1 who presented with the primary complaint of experiencing pain and photophobia in both eyes for 2 months prior to his initial visit. Upon examination, a peripheral corneal ulcer was observed in both eyes covering more than 3 of the 4 quadrants, accompanied with ciliary injection and severe corneal infiltration. He was diagnosed with Mooren's ulcer, and treatment with 0.1% betamethasone and 0.5% levofloxacin eye drops and systemic cyclosporine and betamethasone was initiated. At 1-month post treatment initiation, a remaining ulceration ridge was observed on the corneal surface in his left eye, which was subsequently resected. Complete epithelialization was achieved at 1-month postoperative in the left eye and after 6-months of conservative topical treatment in the right eye. At 8-9 years post onset of Mooren's ulcer, the patient underwent penetrating keratoplasty in both eyes while undergoing treatment with oral cyclosporine administration for severe corneal opacity due to progression of lattice dystrophy. Post treatment, there has been no recurrence of ulcerations, even though more that 10 years has passed since the onset of Mooren's ulcer.
To the best of our knowledge, this is the first reported case of LCD1 with bilateral Mooren's ulcer, and in this rare case, the patient was successfully treated with a combination of steroid, cyclosporine, and peripheral superficial keratectomy, and a good visual outcome was achieved after penetrating keratoplasty (PK) under the use of systemic cyclosporine.
报告1例罕见的伴有双侧蚕蚀性角膜溃疡的1型格子状角膜营养不良（LCD1）病例。
该病例为一名62岁男性LCD1患者，初次就诊前2个月主要表现为双眼疼痛和畏光。检查发现双眼周边角膜溃疡，累及4个象限中的3个以上，伴有睫状充血和严重角膜浸润。他被诊断为蚕蚀性角膜溃疡，并开始使用0.1%倍他米松和0.5%左氧氟沙星滴眼液以及全身应用环孢素和倍他米松进行治疗。治疗开始后1个月，左眼角膜表面观察到残留的溃疡嵴，随后进行了切除。左眼术后1个月实现完全上皮化，右眼经过6个月的保守局部治疗后也实现了上皮化。在蚕蚀性角膜溃疡发病8 - 9年后，由于格子状营养不良进展导致严重角膜混浊，患者双眼接受了穿透性角膜移植术，同时口服环孢素进行治疗。治疗后，即使自蚕蚀性角膜溃疡发病已过去10多年，溃疡也未复发。
据我们所知，这是首例报道的伴有双侧蚕蚀性角膜溃疡的LCD1病例，在这个罕见病例中，患者通过类固醇、环孢素和周边浅层角膜切除术联合成功治疗，并在全身应用环孢素的情况下进行穿透性角膜移植术（PK）后取得了良好的视觉效果。
