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RS₃PE presenting in a unilateral pattern: Case report and review of the literature
Seminars in Arthritis and Rheumatism, 11/24/08

Keenan RT et al. – Although remitting seronegative symmetrical synovitis with pitting edema (RS₃PE) is almost always a symmetric disease of the upper extremities, it may rarely present in a unilateral fashion, suggesting a role for neural and possibly other local factors in the genesis/modulation of the onset or maintenance of the disease.

Methods

Study to review the clinical features and pathophysiologic implications of RS₃PE presenting in a unilateral manner
An index case of unilateral RS₃PE was identified and characterized
A systematic literature search to identify other reports of pts

Results

Index case: 76-yr-old male, cerebrovascular accident 25 yrs prior, developed a classic picture of RS₃PE accompanied by an elevated ESR, but only in the nonhemiparetic hand
Condition responded rapidly to low-dose prednisone
Literature search identified 5 other cases, including 2 presented only in the Italian or German literature
2/5 were in pts with preexisting neurologic disease, in which the neurologically affected side was spared
1 additional case initially presented as unilateral disease but rapidly progressed to bilaterality
2 cases presented in a fully unilateral manner despite no reported neurologic abnormalities on the unaffected sides

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