Karuntu Jessica S, Almushattat Hind, Nguyen Xuan-Thanh-An, Plomp Astrid S, Wanders Ronald J A, Hoyng Carel B, van Schooneveld Mary J, Schalij-Delfos Nicoline E, Brands Marion M, Leroy Bart P, van Karnebeek Clara D M, Bergen Arthur A, van Genderen Maria M, Boon Camiel J F
Department of Ophthalmology, Leiden University Medical Center, Leiden, the Netherlands.
Department of Ophthalmology, Amsterdam University Medical Center, University of Amsterdam, Amsterdam, the Netherlands.
Prog Retin Eye Res. 2024 Dec 27;107:101324. doi: 10.1016/j.preteyeres.2024.101324.
Retinitis pigmentosa (RP) is a progressive inherited retinal dystrophy, characterized by the degeneration of photoreceptors, presenting as a rod-cone dystrophy. Approximately 20-30% of patients with RP also exhibit extra-ocular manifestations in the context of a syndrome. This manuscript discusses the broad spectrum of syndromes associated with RP, pathogenic mechanisms, clinical manifestations, differential diagnoses, clinical management approaches, and future perspectives. Given the diverse clinical and genetic landscape of syndromic RP, the diagnosis may be challenging. However, an accurate and timely diagnosis is essential for optimal clinical management, prognostication, and potential treatment. Broadly, the syndromes associated with RP can be categorized into ciliopathies, inherited metabolic disorders, mitochondrial disorders, and miscellaneous syndromes. Among the ciliopathies associated with RP, Usher syndrome and Bardet-Biedl syndrome are the most well-known. Less common ciliopathies include Cohen syndrome, Joubert syndrome, cranioectodermal dysplasia, asphyxiating thoracic dystrophy, Mainzer-Saldino syndrome, and RHYNS syndrome. Several inherited metabolic disorders can present with RP, including Zellweger spectrum disorders, adult Refsum disease, α-methylacyl-CoA racemase deficiency, certain mucopolysaccharidoses, ataxia with vitamin E deficiency, abetalipoproteinemia, several neuronal ceroid lipofuscinoses, mevalonic aciduria, PKAN/HARP syndrome, PHARC syndrome, and methylmalonic acidaemia with homocystinuria type cobalamin (cbl) C disease. Due to the mitochondria's essential role in supplying continuous energy to the retina, disruption of mitochondrial function can lead to RP, as seen in Kearns-Sayre syndrome, NARP syndrome, primary coenzyme Q10 deficiency, SSBP1-associated disease, and long chain 3-hydroxyacyl-CoA dehydrogenase deficiency. Lastly, Cockayne syndrome and PERCHING syndrome can present with RP, but they do not fit the abovementioned hierarchy and are thus categorized as miscellaneous. Several first-in-human clinical trials are underway or in preparation for some of these syndromic forms of RP.
视网膜色素变性(RP)是一种进行性遗传性视网膜营养不良,其特征是光感受器退化,表现为视杆 - 视锥营养不良。约20% - 30%的RP患者在综合征背景下还表现出眼外表现。本文讨论了与RP相关的广泛综合征、致病机制、临床表现、鉴别诊断、临床管理方法及未来展望。鉴于综合征性RP的临床和遗传情况多样,诊断可能具有挑战性。然而,准确及时的诊断对于最佳临床管理、预后评估和潜在治疗至关重要。广义而言,与RP相关的综合征可分为纤毛病、遗传性代谢紊乱、线粒体疾病和其他综合征。在与RP相关的纤毛病中,Usher综合征和Bardet - Biedl综合征最为知名。较不常见的纤毛病包括Cohen综合征、Joubert综合征、颅外胚层发育不良、窒息性胸廓发育不良、Mainzer - Saldino综合征和RHYNS综合征。几种遗传性代谢紊乱可伴有RP,包括Zellweger谱系障碍、成人Refsum病、α - 甲基酰基辅酶A消旋酶缺乏症、某些黏多糖贮积症、维生素E缺乏共济失调、无β脂蛋白血症、几种神经元蜡样脂褐质沉积症、甲羟戊酸尿症、PKAN/HARP综合征、PHARC综合征以及伴有同型胱氨酸尿症的甲基丙二酸血症(钴胺素(cbl)C病)。由于线粒体在为视网膜持续提供能量方面的重要作用,线粒体功能破坏可导致RP,如在Kearns - Sayre综合征、NARP综合征、原发性辅酶Q10缺乏症、SSBP1相关疾病和长链3 - 羟酰基辅酶A脱氢酶缺乏症中所见。最后,Cockayne综合征和PERCHING综合征可伴有RP,但它们不符合上述分类,因此归类为其他综合征。针对其中一些综合征性RP形式的多项首次人体临床试验正在进行或正在筹备中。