Chen Yi-Hua, Huang Pei-Wei, Liu Yu-Jen, Tsai Yueh-Ju
Department of Ophthalmology, Linkou Chang Gung Memorial Hospital, Taoyuan, Taiwan.
College of Medicine, Chang Gung University, Taoyuan, Taiwan.
Mol Genet Metab Rep. 2023 Mar 17;35:100969. doi: 10.1016/j.ymgmr.2023.100969. eCollection 2023 Jun.
This retrospective observational case series is to evaluate the histopathological findings of drooping eyelids in patients with infantile-onset Pompe disease and assess the feasibility of levator muscle resection combined with conjoint fascial sheath suspension for ptosis correction. It included six patients from a single tertiary referral center with ptosis and infantile-onset Pompe disease between January 1, 2013, and December 31, 2021. They most suffered from recurrent ptosis after initial surgical correction (6/11 eyes, 54.55%). The recurrence rate was high in eyes with levator muscle resection alone (4/6 eyes, 66.67%). No recurrence of ptosis was observed in eyes with levator muscle resection combined with conjoint fascial sheath suspension. The follow-up period was approximately 16-94 months. Histopathological examination revealed that the levator muscle had the most abundant glycogen accumulation-related vacuolar changes, followed by Müller's muscle and extraocular muscles. No vacuolar changes were observed in the conjoint fascial sheath. For patients with infantile-onset Pompe disease-related ptosis, performing levator muscle resection alone is not sufficient, while utilizing conjoint fascial sheath suspension can achieve the desired long-term outcomes with minimal recurrence. These findings may have important implications for the management of ophthalmic complications in patients with infantile-onset Pompe disease.
本回顾性观察性病例系列旨在评估婴儿型庞贝病患者上睑下垂的组织病理学表现,并评估提上睑肌切除术联合联合筋膜鞘悬吊术矫正上睑下垂的可行性。该研究纳入了2013年1月1日至2021年12月31日期间来自单一三级转诊中心的6例患有上睑下垂和婴儿型庞贝病的患者。他们大多在初次手术矫正后出现复发性上睑下垂(6/11只眼,54.55%)。单纯提上睑肌切除术的患眼复发率较高(4/6只眼,66.67%)。提上睑肌切除术联合联合筋膜鞘悬吊术的患眼未观察到上睑下垂复发。随访期约为16 - 94个月。组织病理学检查显示,提上睑肌糖原积累相关的空泡样改变最为丰富,其次是米勒肌和眼外肌。联合筋膜鞘未观察到空泡样改变。对于婴儿型庞贝病相关上睑下垂患者,单纯行提上睑肌切除术是不够的,而采用联合筋膜鞘悬吊术可实现理想的长期效果,复发率最低。这些发现可能对婴儿型庞贝病患者眼科并发症的管理具有重要意义。