Literature DB >> 28301413

OPHTHALMIC FINDINGS IN LATE STAGE SJOGREN-LARSSON SYNDROME.

Tavish Nanda1, Jaclyn L Kovach2.   

Abstract

PURPOSE: To report spectral domain optical coherence tomography and fundus autofluorescence documentation of late stage macular findings associated with Sjogren-Larsson Syndrome in three adult siblings.
METHODS: Three adult siblings with Sjogren-Larsson Syndrome underwent ophthalmic examination and imaging.
RESULTS: Crystalline maculopathy and subretinal deposits, presumably lipofuscin accumulation, with macular atrophy were present in varying degrees in all three adult siblings. DISCUSSION: In adults with Sjogren-Larsson Syndrome, crystalline retinopathy can progress to macular atrophy and the appearance of lipofuscin accumulation.

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Year:  2019        PMID: 28301413      PMCID: PMC5729048          DOI: 10.1097/ICB.0000000000000583

Source DB:  PubMed          Journal:  Retin Cases Brief Rep        ISSN: 1935-1089


  10 in total

1.  Oligophrenia in combination with congenital ichthyosis and spastic disorders; a clinical and genetic study.

Authors:  T SJOGREN; T LARSSON
Journal:  Acta Psychiatr Neurol Scand Suppl       Date:  1957

Review 2.  The Müller cell: a functional element of the retina.

Authors:  E Newman; A Reichenbach
Journal:  Trends Neurosci       Date:  1996-08       Impact factor: 13.837

Review 3.  New functions of Müller cells.

Authors:  Andreas Reichenbach; Andreas Bringmann
Journal:  Glia       Date:  2013-02-26       Impact factor: 7.452

4.  Studies of the skin in Sjögren-Larsson syndrome by electron microscopy.

Authors:  L Y Matsuoka; B G Kousseff; K Hashimoto
Journal:  Am J Dermatopathol       Date:  1982-08       Impact factor: 1.533

5.  Juvenile macular dystrophy associated with deficient activity of fatty aldehyde dehydrogenase in Sjögren-Larsson syndrome.

Authors:  M A Willemsen; J R Cruysberg; J J Rotteveel; A L Aandekerk; P H Van Domburg; A F Deutman
Journal:  Am J Ophthalmol       Date:  2000-12       Impact factor: 5.258

6.  Patients with Sjögren-Larsson syndrome lack macular pigment.

Authors:  Rob L P van der Veen; Joris Fuijkschot; Michèl A A P Willemsen; Johannes R M Cruysberg; Tos T J M Berendschot; Thomas Theelen
Journal:  Ophthalmology       Date:  2010-02-16       Impact factor: 12.079

7.  Subclinical changes in the juvenile crystalline macular dystrophy in Sjögren-Larsson syndrome detected by optical coherence tomography.

Authors:  Joris Fuijkschot; Johannes R M Cruysberg; Michèl A A P Willemsen; Jan E E Keunen; Thomas Theelen
Journal:  Ophthalmology       Date:  2007-09-12       Impact factor: 12.079

8.  Sjögren-Larsson syndrome is caused by mutations in the fatty aldehyde dehydrogenase gene.

Authors:  V De Laurenzi; G R Rogers; D J Hamrock; L N Marekov; P M Steinert; J G Compton; N Markova; W B Rizzo
Journal:  Nat Genet       Date:  1996-01       Impact factor: 38.330

9.  Lipofuscin and melanin content of the retinal pigment epithelium in a case of Sjögren-Larsson syndrome.

Authors:  S E Nilsson; S Jagell
Journal:  Br J Ophthalmol       Date:  1987-03       Impact factor: 4.638

10.  Phenotypic variability among adult siblings with Sjögren-Larsson syndrome.

Authors:  Alexander Lossos; Moona Khoury; William B Rizzo; John M Gomori; Eyal Banin; Abraham Zlotogorski; Saleh Jaber; Oded Abramsky; Zohar Argov; Hanna Rosenmann
Journal:  Arch Neurol       Date:  2006-02
  10 in total
  3 in total

1.  Topographic Distribution and Progression of Soft Drusen Volume in Age-Related Macular Degeneration Implicate Neurobiology of Fovea.

Authors:  Andreas Pollreisz; Gregor S Reiter; Hrvoje Bogunovic; Lukas Baumann; Astrid Jakob; Ferdinand G Schlanitz; Stefan Sacu; Cynthia Owsley; Kenneth R Sloan; Christine A Curcio; Ursula Schmidt-Erfurth
Journal:  Invest Ophthalmol Vis Sci       Date:  2021-02-01       Impact factor: 4.799

2.  Beyond retina in Sjogren-Larsson syndrome.

Authors:  Neelam Pawar; R Meenakshi; Devendra Maheshwari; A Fathima; Sabyasachi Chakrabarty; Mihir Paranjape
Journal:  Indian J Ophthalmol       Date:  2022-07       Impact factor: 2.969

Review 3.  Dysfunctional peroxisomal lipid metabolisms and their ocular manifestations.

Authors:  Chuck T Chen; Zhuo Shao; Zhongjie Fu
Journal:  Front Cell Dev Biol       Date:  2022-09-07
  3 in total

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