Ocular Surface Stem Cell Transplantation for Treatment of Keratitis-Ichthyosis-Deafness Syndrome
Ocular Surface Stem Cell Transplantation for Treatment of Keratitis-Ichthyosis-Deafness Syndrome
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DOI：
10.1097/ico.0000000000001802
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发表时间：
2019-01-01
期刊：
影响因子：
2.8
通讯作者：
Holland, Edward J.
中科院分区：
文献类型：
--
作者：
Cheung, Albert Y.;Patel, Sunny;Holland, Edward J.
目的：报告应用眼表干细胞移植(OSST)治疗角膜炎-鱼鳞病-耳聋(KID)综合征的角膜缘干细胞缺乏症(LSCD)的手术经验。OSST术后平均随访8.3+/-4.3年(3.4~11.4年)。2只眼接受活体结膜缘移植(LR-CLAL)，3只眼接受同种异体角膜缘移植(KLAL)。5只眼中有4只眼接受了随后的角膜移植。两只LR-CLAL眼在最后一次随访时都保持了稳定的眼表。相反，所有的KLAL眼都出现了失败的表面，需要重复KLAL手术。由于多个KLAL失败，1只眼接受了人工角膜植入术。尽管OSST可以稳定表面，但儿童综合征的长期治疗可能是具有挑战性的。LR-CLAL在这些眼睛中可能比KLAL有更多的好处，因为它是HLA和ABO匹配的组织；它还有助于治疗干燥性角结膜炎，这通常是KID综合征的一个显著特征。
Purpose: To report our surgical experience with ocular surface stem cell transplantation (OSST) for limbal stem cell deficiency (LSCD) in the setting of keratitis-ichthyosis-deafness (KID) syndrome.Methods: Retrospective interventional case series.Results: We present 5 eyes of 3 patients with KID syndrome that developed LSCD and underwent OSST. Mean follow-up after OSST was 8.3 +/- 4.3 years (range 3.4-11.4 years). Two eyes underwent living-related conjunctival limbal allograft (lr-CLAL), and 3 eyes were treated with keratolimbal allograft (KLAL). Four of the 5 eyes underwent subsequent keratoplasty. Both lr-CLAL eyes maintained a stable ocular surface at final follow-up. Conversely, all KLAL eyes developed a failed surface requiring repeat KLAL surgery. Because of multiple failed KLALs, 1 eye underwent placement of a keratoprosthesis.Conclusions: KID syndrome is a rare cause of LSCD. Although OSST can stabilize the surface, long-term treatment of KID syndrome can be challenging. An lr-CLAL may offer further benefit over a KLAL in these eyes because it is HLA-and ABO-matched tissue; it also helps to treat keratoconjunctivitis sicca, often a prominent feature of KID syndrome.