Thalidomide Has Activity in Treating Complex Regional Pain Syndrome

Schwartzman, Robert J.; Chevlen, Eric; Bengtson, Keith A.

doi:10.1001/archinte.163.12.1487

Cited by 61 publications

(31 citation statements)

References 11 publications

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“…Adalimumab, a recombinant human monoclonal antibody that blocks tumor necrosis factor (TNF-α) binding was tested in an uncontrolled study of 10 patients with CRPS-I, with 6 responders [60]. Case reports and a study of 42 patients reported efficacy of thalidomide, a tumor necrosis factor-α inhibitor [61], but a phase II, blinded RCT of lenalidomide, a safer thalidomide derivative, in 184 patients with CRPS-I did not find efficacy [62]. Perhaps it would have been more effective if given earlier in the course.…”

Section: Complex Regional Pain Syndromementioning

confidence: 99%

Immunotherapy Prospects for Painful Small-fiber Sensory Neuropathies and Ganglionopathies

Oaklander

2016

Neurotherapeutics

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The best-known peripheral neuropathies are those affecting the large, myelinated motor and sensory fibers. These have well-established immunological causes and therapies. Far less is known about the somatic and autonomic "small fibers"; the unmyelinated C-fibers, thinly myelinated A-deltas, and postganglionic sympathetics. The small fibers sense pain and itch, innervate internal organs and tissues, and modulate the inflammatory and immune responses. Symptoms of small-fiber neuropathy include chronic pain and itch, sensory impairment, edema, and skin color, temperature, and sweating changes. Small-fiber polyneuropathy (SFPN) also causes cardiovascular, gastrointestinal, and urological symptoms, the neurologic origin of which often remains unrecognized. Routine electrodiagnostic study does not detect SFPN, so skin biopsies immunolabeled to reveal axons are recommended for diagnostic confirmation. Preliminary evidence suggests that dysimmunity causes some cases of small-fiber neuropathy. Several autoimmune diseases, including Sjögren and celiac, are associated with painful small-fiber ganglionopathy and distal axonopathy, and some patients with "idiopathic" SFPN have evidence of organ-specific dysimmunity, including serological markers. Dysimmune SFPN first came into focus in children and teenagers as they lack other risk factors, for example diabetes or toxic exposures. In them, the rudimentary evidence suggests humoral rather than cellular mechanisms and complement consumption. Preliminary evidence supports efficacy of corticosteroids and immunoglobulins in carefully selected children and adult patients. This paper reviews the evidence of immune causality and the limited data regarding immunotherapy for small-fiberpredominant ganglionitis, regional neuropathy (complex regional pain syndrome), and distal SFPN. These demonstrate the need to develop case definitions and outcome metrics to improve diagnosis, enable prospective trials, and dissect the mechanisms of small-fiber neuropathy.

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Section: Complex Regional Pain Syndromementioning

confidence: 99%

Immunotherapy Prospects for Painful Small-fiber Sensory Neuropathies and Ganglionopathies

Oaklander

2016

Neurotherapeutics

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“…18 Both thalidomide and lenalidomide have also shown some benefit in the treatment of CRPS. 19,20 Converging evidence supports the development of a sterile inflammatory response at the site of injury in CRPS patients. This is precipitated and maintained by a pro-inflammatory cytokine profile (IL-1, IL-6, TNF-α, IL-1β) and depressed anti-inflammatory cytokines (IL-4 and IL-10) derived from extravasated leukocytes and recruited immune cells.…”

Section: Case Reportmentioning

confidence: 99%

Gardner-Diamond syndrome associated with complex regional pain syndrome

Edinger¹,

Schwartzman²

2013

J Dermatol Case Rep

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Background: Gardner-Diamond syndrome (GDS) is also known as psychogenic purpura, autoerythrocyte sensitization syndrome and painful bruising syndrome.Main observation: This is a case report of 27-year-old woman who presented with unexplained bruising and intramuscular hematomas after a seven year history of complex regional pain syndrome. Her evaluation consisted of hematological studies, skin and muscle biopsy; it failed to reveal an underlying coagulopathy, vasculitis or other demonstrable cause. In the absence of any other etiology, she was diagnosed as Gardner-Diamond syndrome. Conclusion:This patient is unique because of intramuscular hematomas and the association of Complex regional pain syndrome with Gardner-Diamond syndro-

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“…Thus, CRPS is alleviated with free radical scavengers, 31,67,104 and antitumor necrosis factor-a antibodies, 40,56,80 which would mitigate the inflammatory and damaging effects of oxygen-free radicals and proinflammatory cytokines. CRPS has also been shown to be alleviated by nitric-oxide donors and other vaosodilators, 34,55,63 or a-adrenergic antagonists, 63,68 and topical or regional clonidine, 15,28,43,69 which act by inhibition of the effects of sympathetic vasoconstrictors on vasculature smooth-muscle cells.…”

Section: Novel Therapeutics and Conclusionmentioning

confidence: 99%

Complex Regional Pain Syndrome: What's in a Name?

Coderre

2011

The Journal of Pain

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Within a two year period in the 1940's, two Boston physicians published dramatically opposing views on the underlying nature of a syndrome now known as complex regional pain syndrome (CRPS). Evans suggested, in several papers in [1946][1947], that sympathetic reflexes maintain pain and dystrophy in affected limbs. Foisie, in 1947, suggested arterial vasospasms were key in the etiology of this pain syndrome. Evans' hypothesis established the nomenclature for this syndrome for 60 years, and his term "reflex sympathetic dystrophy" guided clinical treatment and research activities over the same period. Foisie's proposed nomenclature was unrecognized, and had virtually no impact on the field. Recent evidence suggests that Evans' contribution to the field may have in fact lead clinicians and researchers astray all those years. This focus article on CRPS compares recent observations with these 2 earlier theories and asks the question --what if we had adopted Foisie's nomenclature from the beginning?

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Thalidomide Has Activity in Treating Complex Regional Pain Syndrome

Cited by 61 publications

References 11 publications

Immunotherapy Prospects for Painful Small-fiber Sensory Neuropathies and Ganglionopathies

Immunotherapy Prospects for Painful Small-fiber Sensory Neuropathies and Ganglionopathies

Gardner-Diamond syndrome associated with complex regional pain syndrome

Complex Regional Pain Syndrome: What's in a Name?

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