Taechajongjintana Marisa, Kasetsuwan Ngamjit, Reinprayoon Usanee, Sawanwattanakul Sirinuch, Pisuchpen Phattrawan
Department of Ophthalmology, Faculty of Medicine, Division of Cornea and Refractive Surgery, Chulalongkorn University, 1873 Rama 4 Road, Pathumwan, Bangkok, 10330, Thailand.
J Med Case Rep. 2018 Aug 19;12(1):225. doi: 10.1186/s13256-018-1765-1.
We report a rare case of Phialophora verrucosa fungal keratitis, which required various types of treatment according to the intractable natural history of the disease.
A 51-year-old Thai man with poorly controlled diabetes received a bamboo branch injury and developed a perforated corneal lesion on his left eye. A pathological study from therapeutic penetrating keratoplasty showed fungal hyphae. This was later identified as Phialophora verrucosa by polymerase chain reaction. This organism was aggressive and recalcitrant because it relapsed with two corneal grafts and was resistant to amphotericin B, natamycin, and itraconazole. However, we found that the efficacy of voriconazole was promising for treating Phialophora verrucosa. We also used corneal cross-linking to establish corneal integrity after the infection was under control.
Because of the chronic nature of Phialophora verrucosa, a patient's first visit may occur many years after trauma, and sometimes clinical presentation might not appear to indicate fungal infection. Therefore, a high index of suspicion is needed in this situation. Voriconazole showed good results in our case. Instead of using a more invasive keratoplasty, we used corneal cross-linking to strengthen the corneal biomechanics. To the best of our knowledge, this is the first case showing the benefit of corneal cross-linking to improve corneal biomechanics in resolved Phialophora verrucosa keratitis.
我们报告一例罕见的疣状瓶霉真菌性角膜炎病例,根据该疾病顽固的自然病程,需要进行多种类型的治疗。
一名51岁的泰国男性,糖尿病控制不佳,眼部被竹枝划伤,左眼出现角膜穿孔性病变。治疗性穿透性角膜移植的病理研究显示有真菌菌丝。后来通过聚合酶链反应确定为疣状瓶霉。这种病原体具有侵袭性且顽固,因为它导致两次角膜移植失败,并且对两性霉素B、那他霉素和伊曲康唑耐药。然而,我们发现伏立康唑治疗疣状瓶霉有良好前景。在感染得到控制后,我们还使用角膜交联来维持角膜完整性。
由于疣状瓶霉感染具有慢性特点,患者首次就诊可能在受伤多年后,有时临床表现可能并不提示真菌感染。因此,在这种情况下需要高度怀疑。伏立康唑在我们的病例中显示出良好效果。我们没有采用更具侵入性的角膜移植,而是使用角膜交联来增强角膜生物力学。据我们所知,这是第一例显示角膜交联对改善已治愈的疣状瓶霉性角膜炎角膜生物力学有益的病例。