Late-onset Kjellin syndrome: Diagnosis of SPG11 on fundus examination

Eur J Ophthalmol. 2024 Jul;34(4):NP44-NP46. doi: 10.1177/11206721241247418. Epub 2024 Apr 13.

Abstract

Introduction: Spastic paraplegia (SPG) is a heterogenous group of neurodegenerative disorders, that may include ocular involvement. Here we report the clinical data of a patient with late-onset Kjellin syndrome, a peculiar form of hereditary SPG with macular dystrophy.

Materials and methods: Clinical, functional and multimodal retinal imaging data were collected. Genetic testing was performed by Whole Exome Sequencing (WES).

Results: A 52-year-old female patient with SPG of unknown origin was referred for a progressive visual acuity loss. Multimodal fundus imaging revealed a peculiar macular dystrophy. Given the specific association of macular dystrophy and SPG, a Kjellin syndrome was suspected and genetic testing performed. WES revealed biallelic pathogenic variants in SPG11, co-segregating with disease in the family.

Conclusion: Careful ophthalmological examination prompted the diagnosis and guided molecular testing. This case underlines the importance of a neuro-ophthalmologic assessment in patients with SPG.

Keywords: Kjellin syndrome; SPG11; hereditary spastic paraplegia; maculopathy; pattern dystrophy.

Publication types

  • Case Reports

MeSH terms

  • Exome Sequencing
  • Female
  • Fluorescein Angiography / methods
  • Fundus Oculi*
  • Humans
  • Macular Degeneration / diagnosis
  • Macular Degeneration / genetics
  • Middle Aged
  • Multimodal Imaging
  • Mutation
  • Pedigree
  • Proteins / genetics
  • Spastic Paraplegia, Hereditary* / diagnosis
  • Spastic Paraplegia, Hereditary* / genetics
  • Tomography, Optical Coherence*
  • Visual Acuity / physiology

Substances

  • SPG11 protein, human
  • Proteins