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Sökning: onr:"swepub:oai:DiVA.org:uu-493156" > Severe Raynaud's ph...

Severe Raynaud's phenomenon from ethosuximide raised concern over possible onset of systemic vasculitis : a case report

Berntson, Lillemor, 1957- (författare)
Uppsala universitet,Pediatrisk inflammations- och metabolismforskning samt barnhälsa
Liminga, Gunnar (författare)
Uppsala universitet,Barnonkologisk och neurologisk forskning
 (creator_code:org_t)
2022-12-22
2022
Engelska.
Ingår i: Pediatric Rheumatology. - : BioMed Central (BMC). - 1546-0096. ; 20
  • Tidskriftsartikel (refereegranskat)
Abstract Ämnesord
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  • Background: Ethosuximide and other anti-epileptic drugs have been reported to cause idiosyncratic reactions such as lupus-like syndromes, with elevated antinuclear antibody (ANA) levels. Herein, we present a case of a girl who developed a very severe Raynaud's phenomenon reaction and anti-Scl-70 antibodies related to treatment with ethosuximide, due to juvenile absence epilepsy (JAE).Case presentation: A 12-year-old girl was diagnosed with JAE and treatment with ethosuximide was initiated. Two and a half months later her fingers, digits II-V bilaterally, began to ache and were discolored, alternatingly white, blue, or normal-colored. Two weeks later, her fingers were bluish-black, aching severely, almost continuously. The family sought medical advice. Ethosuximide was halted and due to the severe symptoms, treatment with both prednisolone and intravenous iloprost was commenced. Laboratory tests revealed high ANA levels with anti-Scl-70 pattern and confirmed anti-Scl-70 antibodies. After a few weeks, she started to improve and the symptoms slowly decreased over five months. Anti-Scl-70 was still detectable four months after onset of symptoms, though she was much improved. After eleven months, repeated ANA analyses were completely negative.Conclusion: Although extremely rare, it is important to recognize that severe Raynaud's phenomenon, threatening peripheral digital circulation, may occur as an idiosyncratic reaction to ethosuximide, raising concern over possible onset of vasculitis.

Ämnesord

MEDICIN OCH HÄLSOVETENSKAP  -- Klinisk medicin -- Reumatologi och inflammation (hsv//swe)
MEDICAL AND HEALTH SCIENCES  -- Clinical Medicine -- Rheumatology and Autoimmunity (hsv//eng)

Nyckelord

Ethosuximide
Anti-epileptic drugs
Absence epilepsy
Adverse effect
Drug-induced
Vasculitis
Raynaud's syndrome
Anti-scl-70

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Liminga, Gunnar
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