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Visual recovery in a man with the rare combination of mtDNA 11778 LHON mutation and a MS-like disease after mitoxantrone therapy.
- Source :
-
Acta neurologica Scandinavica [Acta Neurol Scand] 2002 Oct; Vol. 106 (4), pp. 236-9. - Publication Year :
- 2002
-
Abstract
- We describe a young man with prognostic unfavourable homoplasmatic mitochondrial DNA(mt DNA) 11778 Leber's hereditary optic neuropathy (LHON) point mutation and confirmed multiple sclerosis (MS). This combination of LHON and MS-like disease is rare in both sexes, and in men has been described in only a few case reports. In a 4-year follow-up during immunosuppressive therapy with mitoxantrone, we found a remarkable time delayed visual recovery 12 months after acute onset of rapid sequential bilateral subtotal visual loss followed by episodes of isolated acute demyelinative optic neuropathy. Visual recovery to such extent after this latency is uncommon in both mtDNA 11778 LHON mutation and optic neuritis (ON) in MS. Relapses in visual deterioration must be considered as extremely rare in LHON. This case might support the hypothesis of an immunological pathogenetic factor in combined LHON and MS, and possibly in LHON alone. We suggest a search for the LHON mutation in MS patients with predominant visual impairment, independent of patients' gender.
- Subjects :
- Adolescent
Blindness etiology
Blindness physiopathology
Humans
Male
Multiple Sclerosis complications
Optic Atrophy, Hereditary, Leber complications
Optic Atrophy, Hereditary, Leber genetics
Point Mutation
Treatment Outcome
Visual Acuity
Blindness drug therapy
DNA, Mitochondrial genetics
Immunosuppressive Agents therapeutic use
Mitoxantrone therapeutic use
Multiple Sclerosis drug therapy
Optic Atrophy, Hereditary, Leber drug therapy
Recovery of Function drug effects
Subjects
Details
- Language :
- English
- ISSN :
- 0001-6314
- Volume :
- 106
- Issue :
- 4
- Database :
- MEDLINE
- Journal :
- Acta neurologica Scandinavica
- Publication Type :
- Academic Journal
- Accession number :
- 12225323
- Full Text :
- https://doi.org/10.1034/j.1600-0404.2002.01323.x