RSD/CRPS Studies 2003

RESEARCH 2003


INTRODUCTION:

Some of these studies are abstracts and others are review articles of research studies on RSD/CRPS. We have featured the prominent researchers first and then listed more studies alphabetically according to author. Important studies opening new ground or discoveries are listed with *** at beginning.

Charles Berde MD PhD

Robert Schwartzman MD

Studies A-M

Studies N-Z


INTRODUCTION

Charles Berde MD PhD, presenter at the Tampa conference, is a well known expert in the treatment of CRPS in children and teens. The Pain Treatment Service has treated over 600 children since 1986 and continues to do so.There is also ongoing research at the Boston Hospital where he works.

 

PHYSICAL THERAPY AND COGNITIVE BEHAVIOURAL TREATMENT FOR CRPS

Berde CB,Lee BH, Scharff L, Sethna NF, McCarthy CF, Scott-Sutherland J, Shea AM, Sullivan P, Meier P, Zurakowski D,Masek BJ. J Pediatr 2002 Jul;141(1):135-40

Pain Treatment Service and the Departments of Physical Therapy, Orthopaedic Surgery, and Psychiatry,Children's Hospital, Boston, Massachusetts.

Complex regional pain syndromes (CRPS; type 1, reflex sympathetic dystrophy, and type 2, causalgia) involve persistent pain, allodynia, and vasomotor signs. We conducted a prospective, randomized, single-blind trial of physical therapy (PT) and cognitive-behavioral treatment for children and adolescents with CRPS. Children 8 to 17 years of age (n = 28) were randomly assigned to either group A (PT once per week for 6 weeks) or group B (PT 3 times per week for 6 weeks). Both groups received 6 sessions of cognitive-behavioral treatment. Assessments of pain and function were repeated at two follow-up time periods. Outcomes were compared at the three time points through the use of parametric or nonparametric analysis of variance and post hoc tests. All five measures of pain and function improved significantly in both groups after treatment, with sustained benefit evident in the majority of patients at long-term follow-up. Recurrent episodes were reported in 50% of patients, and 10 patients eventually received sympathetic blockade. Most children with CRPS showed reduced pain and improved function with a noninvasive rehabilitative treatment approach. Long-term functional outcomes were also very good.
PMID: 12091866


For a copy of this or any CRPS article, visit McMaster Health Sciences Library. Search Pub Med using the PMID number listed.


GENDER DIFFERENCES IN CRPS I/ RSD IN CHILDREN AND ADOLESCENTS
Charles Berde, M.D., Ph.D., Harvard Medical School, Director, Pain Treatment Service, Boston Children's Hospital, Boston, Massachusetts

Complex regional pain syndrome, type 1 (CRPS1) or reflex sympathetic dystrophy (RSD) is being diagnosed more frequently in children and adolescents. Over the last 12 years, our pain center has examined the demographics, associated medical and lifestyle factors, and responses to treatment in over 450 patients. Some demographic features in children appear different from those described for adults:

(1) Children and adolescents have lower extremity involvement 6 times more often than upper extremity involvement.

(2) Girls are affected roughly 5 times as often as boys.

(3) RSD/CRPS1 is rare below age 8; the incidence increases markedly just before puberty.

(4) Female dancers, gymnasts and competitive athletes comprise a high percentage of the patients.

A high percentage of patients present either confined to crutches or in a wheelchair. School absenteeism is more common in these patients than in a matched population of rheumatology patients. Coexisting psychiatric diagnoses commonly include depression, anxiety disorders, post-traumatic stress disorders, distorted body image and eating disorders, and stressful family dynamics.

Treatments advocated for RSD have varied largely according to subspecialty and training of health providers. A program for prospective evaluation of these patients was established recently with funding from NICHD. Preliminary studies by Dr. Sethna and others on quantitative sensory and autonomic evaluation will be summarized. Treatments under prospective controlled evaluation include physical therapy, cognitive-behavioral interventions, tricyclics and anticonvulsants, and continuous infusion sympathetic blocks.


RSDS IN CHILDREN AND ADOLESCENTS
(summary of Dr. Berde’s presentation at RSDSA's Tampa conference)

Treatment varies according to the individual child. It is essential to maintain a close rapport with the patient and family so that they can understand and cope with the treatment.
Active PT and CBT (cogntive behaviour therapy) are at the top of the list for treatment and a large percentage of kids improve with these two methods. It is a stepwise return to weight bearing and understanding the role of pain. Desensitization and aqua therapy are also used. CBT includes relaxation training, biofeedback and other coping pain skills. Many children recount that CBT helps with daily stress and solving problems. If the pain and limb problem becomes chronic, then treatment for depression or anxiety is considered. Tricyclic antidepressants help with sleep and sometimes anticonvulsants (Neurontin) are used. Finding the right dose of the right drug and assessing side effects are all very important.Those who do not improve with PT and CBT, can receive sympathetic blockade.

Various methods can be used from lumbar paravertebral, lumbar epidural or IV regional with single shot or continuous methods. It depends on the individual case. Invasive procedures are saved for those who have tried all other therapies and did not respond well. These treatments include spinal cord or peripheral stimulation, implantable pumps and sympathectomy. No form of neurodestructive sympathectomy is done due to possible long term complications.

Note: This review article appears in the Winter Issue of the PARC PEARL.


FURTHER READING

Berde, C.B., & Sethna, N.F. (in press).

Sethna, N.F. (1999). Pharmacotherapy in long-term pain: Current experience and future direction. In P. McGrath, & G.A. Finley (Eds.), Chronic and recurrent pain in children and adolescents. Progress in pain research and management (pp. 243-266). Seattle: IASP Press.

Sherry DD et al Short and long term outcomes of children with CRPS 1 treated with exercise therapy. Clinical Journal of Pain 1999; 15:218-33

Stanton, R.P., Malcolm, J.R., Wesdock, K.A., & Singsen, B.H(1993). Reflex sympathetic dystrophy in children: An orthopedic perspective. Orthopedics, 16, 773-779.

Wilder, R.T., Berde, C.B., Wolohan, M., Vieyra, M.A., Masek, B.J., & Micheli, L.J. (1992). Reflex sympathetic dystrophy in children. Clinical characteristics and follow-up of seventy patients. Journal of Bone & Joint Surgery American Volume, 74, 910-919.


INTRODUCTION

Dr Robert Jay Schwartzman, presenter at the Tampa conference, is a well known expert in the treatment of CRPS in children and adults.He has been treating and researching CRPS for many years and continues to break new ground. He was the first to document the spread of CRPS (see Studies file) and discover the movement disorder component of CRPS. What follows are some of his most recent studies.


KETAMINE-MIDAZOLAM ANAESTHESIA FOR INTRACTABLE CRPS-1

INTRODUCTION

CRPS-1 is a very hard to treat syndrome characterized by neuropathic pain. CRPS can spread and become resistant to therapy. More treatments are urgently needed. NMDA receptors are thought play an active role in central pain and NMDA agonists as therapy for CRPS (and neuropathic pain) are being studied.

METHODS
Ten patients with intractable CRPS-1 were given anesthesia which consisted of ketamine and midazolam for 5 days. Some patients were intubated and some had spontaneus breathing. On day 6 they were slowly tapered from infusions.

OBSERVATIONS

INITIAL
All TEN responded by having no pain, no hyperalgesia, no allodynia and an absence of CRPS-1 signs.
LONG TERM
Five out of ten had full pain relief for 2 months up to 3.5 years. In 8/10 patients, after 6-8 weeks the original nociceptive pain returned. In 7/8 the pain was at the original injury site. Hyperalgesia and allodynia recurred in 4/10 who then received another ketamine treatment. Success happened in 2/3. Patients were all able to use less pain medication.

CONCLUSIONS
The authors are quick to say that this treatment shows potential and could be an effective treatment option for severe CRPS-1. They do raise the following issues: which patients would benefit most, what selection criteria would be used to select patients, when to treat the CRPS, and maintenance schedules for re-treatment.

This is the first attempt at using ketamine anesthesia for intractable CRPS-1 and it does not come without its risks. Patients need to be asleep and monitored carefully during the five day treatment. Perhaps in future trials, a less risky form of treatment can be found.

Source: Schwartzman, RJ et al "Ketamine-Midazolam Anesthesia for Intractable CRPS-1" 2002 presented to IASP 2002 Conference

SKIN BLOOD FLOW CHANGES DURING KETAMINE/MIDAZOLAM ANESTHESIA FOR INTRACTABLE CRPS-1

REVIEW

Skin temperature and color changes are an integral part of CRPS. The skin can turn blue, reddish, mottled, or purple. Temperatures vary from hot to warm or cold. Monitoring skin blood flow changes help clinicians decide on the effectiveness of CRPS treatments e.g. when using sympathetic blocks. One such new promising treatment is ketamine/midazolam anesthesia for chronic CRPS-1. This study is the first of its kind to investigate this treatment.

AIMS
Data on skin blood flow (SBF) at rest and during vasomotoric challenge (with heating probe) was collected. Patient's status and responses before, during and after ketamine anesthesia were observed. Could this method be used to asses the ability of the blood vessels to regulate themselves or determine vasomotor impairment in CRPS? Could this be used as a tool to assess treatment success ?

PATIENTS
Eight patients with intractable CRPS-1 were analyzed for blood flow with laser doppler flowmetry (PF 4001) and a heating probe was used to raise skin temperature. Patients were measured at rest and during the vasomotor challenge.

RESULTS
Under ketamine anesthesia, a significant increase in SBF was observed in the first 72 hours. Interestingly, the areas of the most pain showed the strongest increase, up to 10 fold. Then SBF normailzed and vasomotor reagibility occured e.g. decreased, edema, hyperemia and temperature changes. When the limb was locally heated, the highest increase of SBF was found on day 2 and 3 of therapy.

CONCLUSIONS
The authors are the first to admit that more studies must be done before LDF can be recommended as a diagnostic tool or for therapy. Suggestions include monitoring SBF changes in CRPS, possibly predicting therapeutic success, to quantify vasomotor reactive capacity (rest/vasomotor challenge), to help assess sympathetic activity and to contribute to a piece of the diagnostic puzzle of CRPS. Using LDF as a diagnostic tool would be another first since there is none available at this time.

Source: Schwartzman, RJ et al "Skin Blood Flow Changes During Ketamine/Midazolam Anesthesia for Intractable CRPS-1 2002

Note: Low dose ketamine treatments are now being developed in Canada. Contact us for the name of Canadian doctors offering ketamine treatments. These review articles can be found in the PARC PEARL March issue.


STUDIES A-M

***Argoff CE. A focused review on the use of botulinum toxins for neuropathic pain. Clin J Pain. 2002 Nov-Dec;18(6 Suppl):S177-81.
Cohn Pain Management Center, North Shore University Hospital, New York University School of Medicine, Bethpage, New York 11714, USA. pargoff@optonline.net

Understanding the pathophysiology of a pain syndrome is helpful in selecting appropriate treatment strategies. Nociceptive pain is related to damage to tissues due to thermal, chemical, mechanical, or other types of irritants. Neuropathic pain results from injury to the peripheral or central nervous system. Common examples of neuropathic pain include postherpetic neuralgia, diabetic neuropathy, complex regional pain syndrome, and pain associated with spinal cord injuries. Nociceptive pain may have similar clinical characteristics to neuropathic pain. It is also possible for acute nociceptive pain to become neuropathic in nature, as with myofascial pain syndrome. A clear benefit of botulinum toxin therapy for treatment of neuropathic pain disorders is that it often relieves pain symptoms. Although the precise mechanism of pain relief is not completely understood, the injection of botulinum toxin may reduce various substances that sensitize nociceptors. As a result, botulinum toxin types A and B are now being actively studied in nociceptive and neuropathic pain disorders to better define their roles as analgesics.

PMID: 12569966

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Basford JR, Sandroni P, Low PA, Hines SM, Gehrking JA, Gehrking TL.Effects of linearly polarized 0.6-1.6 &mgr;M irradiation on stellate ganglion function in normal subjects and people with complex regional pain (CRPS I). Lasers Surg Med. 2003 Jun;32(5):417-423.
Department of Physical Medicine and Rehabilitation, Autonomic Disorder Center, Mayo Clinic and Foundation, 200 Southwest Second Street, Rochester, Minnesota 55905.

BACKGROUND AND OBJECTIVES: Stellate ganglion blocks are an effective but invasive treatment of upper extremity pain. Linearly polarized red and near-infrared (IR) light is promoted as a safe alternative to this procedure, but its effects are poorly established. This study was designed to assess the physiological effects of this latter approach and to quantitate its benefits in people with upper extremity pain due to Complex Regional Pain Syndrome I (CRPS I, RSD). STUDY DESIGN/MATERIALS AND METHODS: This was a two-part study. In the first phase, six adults (ages 18-60) with normal neurological examinations underwent transcutaneous irradiation of their right stellate ganglion with linearly polarized 0.6-1.6 &mgr;m light (0.92 W, 88.3 J). Phase two consisted of a double-blinded evaluation of active and placebo radiation in 12 subjects (ages 18-72) of which 6 had upper extremity CRPS I and 6 served as "normal" controls. Skin temperature, heart rate (HR), sudomotor function, and vasomotor tone were monitored before, during, and for 30 minutes following irradiation. Analgesic and sensory effects were assessed over the same period as well as 1 and 2 weeks later. RESULTS: Three of six subjects with CRPS I and no control subjects experienced a sensation of warmth following active irradiation (P = 0.025). Two of the CRPS I subjects reported a >50% pain reduction. However, four noted minimal or no change and improvement did not reach statistical significance for the group as a whole. No statistically significant changes in autonomic function were noted. There were no adverse consequences. CONCLUSIONS: Irradiation is well tolerated. There is a suggestion in this small study that treatment is beneficial and that its benefits are not dependent on changes in sympathetic tone. Further evaluation is warranted. Lasers Surg. Med. 32:417-423, 2003. Copyright 2003 Wiley-Liss, Inc.

PMID: 12766967

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Bennett GJ. Are the complex regional pain syndromes due to neurogenic inflammation?
Neurology. 2001 Dec 26;57(12):2161-2.
Comment on:
Neurology. 2001 Dec 26;57(12):2179-84.
Letter
PMID: 11756591


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***Cordivari, MD 1 2, V. Peter Misra, MD, FRCP 1, Santiago Catania, MRCP 1, Andrew J. Lees, MD, FRCP 2 *
Treatment of dystonic clenched fist with botulinum toxin.
Mov Disord. 2001 Sep;16(5):907-13.
Department of Clinical Neurophysiology, The National Hospital for Neurology and Neurosurgery, Queen Square, London, United Kingdom
2Reta Lila Weston Institute of Neurological Studies, University College of London, United Kingdom
email: Andrew J. Lees (alees@ion.ucl.ac.uk)

* Correspondence to Andrew J. Lees, Reta Lila Weston Institute of Neurological Studies, Windeyer Building, 46 Cleveland Street, University College of London W1T 4JF, UK
A videotape accompanies this article.

Abstract
Fourteen patients with dystonic clenched fist (three with Corticobasal Ganglionic Degeneration, seven with Parkinson's disease, and four with Dystonic-Complex Regional Pain Syndrome) were treated with botulinum toxin A (BTXA, Dysport®). The muscles involved were identified by the hand posture and EMG activity recorded at rest and during active and passive flexion/extension movements of the finger and wrist. EMG was useful in distinguishing between muscle contraction and underlying contractures and to determine the dosage of BTX. All patients had some degree of flexion at the proximal metacarpophalangeal joints and required injections into the lumbricals. The response in patients depended on the severity of the deformity and the degree of contracture. All patients had significant benefit to pain, with accompanying muscle relaxation, and palmar infection, when present, was eradicated. Four patients with Parkinson's disease and one patient with Dystonia-Complex Regional Pain Syndrome obtained functional benefit. © 2001 Movement Disorder Society.
Received: 18 December 2000; Revised: 4 April 2001; Accepted: 19 April 2001
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***Kobana,M. S. Leisa, S. Schultze-Mosgaub and F. Birklein
Tissue hypoxia in complex regional pain syndrome
c/a Neurologische Klinik, Friedrich-Alexander-Universität Erlangen-Nürnberg, Universitätsstraße 17, D-91054, Erlangen, Germany
b Klinik für Mund-, Kiefer- und Gesichtschirurgie Friedrich-Alexander-Universität Erlangen-Nürnberg, Universitätsstraße 17, D-91054, Erlangen, Germany c Neurologische Klinik, Johannes Gutenberg-Universität Mainz, Langenbeckstraße 1, D-51101, Mainz, Germany
Received 26 February 2002; accepted 6 December 2002. ; Available online 14 May 2003.

Untreated complex regional pain syndrome (CRPS) may progress from acute stages with increased hair and nail growth in the affected limb to chronic stages with atrophy of the skin, muscles and bones. The aim of this study was to investigate whether tissue hypoxia could be one mechanism responsible for this late CRPS symptoms.

Nineteen patients with CRPS and two control groups (healthy control subjects, surgery patients with edema) participated in this study. Skin capillary hemoglobin oxygenation (HbO2 ) was measured non-invasively employing micro-lightguide spectrophotometry (EMPHO). The EMPHO probe was mounted force-controlled onto the skin of the affected and unaffected hand. HbO2 was measured at rest and during postischemic reactive hyperemia.

HbO2 did not differ between the right (58.20%±1.12) and left (57.79%±1.31, ns) hand in control subjects. However, in patients, HbO2 of the affected side (36.63%±2.16) was significantly decreased as compared to the clinically unaffected side (46.35%±2.97, P<0.01). As compared to controls, HbO2 in CRPS was reduced on both sides (P <0.001). Postischemic hyperoxygenation was impaired on the affected side in CRPS (60.81%±2.90) – as compared to the unaffected side (67.73%±1.50, P<0.04) and to controls (68.63%±0.87, P<0.005). The unaffected limb in CRPS did not differ from controls. Despite skin edema, pre- (49.06%±2.02) and postsurgery HbO2 (53.15%±4.44, ns) were not different in the second control group.

Our results indicate skin hypoxia in CRPS. Impairment of nutritive blood flow in the affected limb may be one factor contributing to atrophy and ulceration in chronic CRPS. The investigation of patients after surgery revealed that edema could not be the only reason for hypoxia.

[Corresponding Author Contact Information] Corresponding author. Tel.: +49-6131-173270; fax: +49-6131-175625

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Kuczkowski, KM. Bretylium in the treatment of complex regional pain syndrome: uncommon side-effect of a common drug. Anaesthesia 2003 Feb;58(2):201-2 Letter.
PMID: 12562438

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STUDIES N-Z

 

McCabe CS, Haigh RC, Ring EF, Halligan PW, Wall PD, Blake DR. A controlled pilot study of the utility of mirror visual feedback in the treatment of complex regional pain syndrome (type 1).
Rheumatology (Oxford) 2003 Jan;42(1):97-101
The Royal National Hospital for Rheumatic Diseases, Upper Borough Walls, Bath BA1 1RL, UK. candy.mccabe@rnhrd-tr.swest.nhs.uk

BACKGROUND: We assessed mirror visual feedback (MVF) to test the hypothesis that incongruence between motor output and sensory input produces complex regional pain syndrome (CRPS) (type 1) pain. METHODS: Eight subjects (disease duration > or =3 weeks to < or =3 yr) were studied over 6 weeks with assessments including two controls (no device and viewing a non-reflective surface) and the intervention (MVF). Pain severity and vasomotor changes were recorded. RESULTS: The control stages had no analgesic effect. MVF in early CRPS (< or =8 weeks) had an immediate analgesic effect and in intermediate disease (< or =1 yr) led to a reduction in stiffness. At 6 weeks, normalization of function and thermal differences had occurred (early and intermediate disease). No change was found in chronic CRPS. CONCLUSIONS: In early CRPS (type 1), visual input from a moving, unaffected limb re-establishes the pain-free relationship between sensory feedback and motor execution. Trophic changes and a less plastic neural pathway preclude this in chronic disease.

PMID: 12509620

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Parisod E, Murray RF, Cousins MJ.
Conversion disorder after implant of a spinal cord stimulator in a patient with a complex regional pain syndrome. Anesth Analg 2003 Jan;96(1):201-6, University of Sydney, Pain Management & Research Centre, Royal North Shore Hospital, St. Leonards, Australia.

IMPLICATIONS: This case history describes the treatment of a patient suffering with persistent pain. He was treated surgically with implantation of a spinal cord stimulator. After surgery, a partial paralysis that could not be explained medically and that was probably related to emotional factors occurred, and cognitive behavioral treatment was begun. This paper discusses the importance of considering social and psychological factors when medical treatment options are considered.

PMID: 12505953
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***Sandroni,P., Lisa M. Benrud-Larson, Robyn L. McClelland and Phillip A. Low Complex regional pain syndrome type I: incidence and prevalence in Olmsted county, a population-based study
Mayo Clinic, 200 First Stret SW, Rochester, MN 55905, USA
Received 5 September 2002; accepted 9 December 2002. ; Available online 8 May 2003.


Abstract

The objective of this study is to undertake a population based study on the incidence, prevalence, natural history, and response to treatment of complex regional pain syndrome (CRPS). All Mayo Clinic and Olmsted Medical Group medical records with codes for reflexsympathetic dystrophy (RSD), CRPS, and compatible diagnoses in the period 1989–1999 were reviewed as part of the Rochester Epidemiology Project. We used IASP criteria for CRPS. The study population was in the Olmsted County, Minnesota (1990 population, 106,470). The main outcome measures were CRPS I incidence, prevalence, and outcome. Seventy-four cases of CRPS I were identified, resulting in an incidence rate of 5.46 per 100,000 person years at risk, and a period prevalence of 20.57 per 100,000. Female:male ratio was 4:1, with a median age of 46 years at onset. Upper limb was affected twice as commonly as lower limb. All cases reported an antecedent event and fracture was the most common trigger (46%). Excellent concordance was found between symptoms and signs and vasomotor symptoms were the most commonly present. Three phase bone scan and autonomic testing diagnosed the condition in >80% of cases. Seventy-four percent of patients underwent resolution, often spontaneously. CRPS I is of low prevalence, more commonly affects women than men, the upper more than the lower extremity, and three out of four cases undergo resolution. These results suggest that invasive treatment of CRPS may not be warranted in the majority of cases.

[Corresponding Author Contact Information] Corresponding author. Tel.: +1-507-284-2090; fax: +1-507-266-6754

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Schasfoort FC, Bussmann JB, Zandbergen AM, Stam HJ.
Impact of upper limb complex regional pain syndrome type 1 on everyday life measured with a novel upper limb-activity monitor.
Pain 2003 Jan;101(1-2):79-88

Department of Rehabilitation Medicine, Erasmus MC, University Medical Center Rotterdam, PO Box 1738, 300 DR Rotterdam, Rotterdam, The Netherlands. schasfoort@rev.fgg.eur.nl

Complex regional pain syndrome type 1 (CRPS1) often leads to serious activity limitations in everyday life. To date, however, limitations in patients with CRPS1 of an upper limb have not been objectively measured.Therefore, the aim of this study was to determine the long-term impact of upper limb CRPS1 on general mobility and upper limb usage during everyday life, as measured with a novel upper limb-activity monitor (ULAM). In ten female chronic CRPS1 patients and ten healthy control subjects, 24-h activity patterns were measured with the ULAM. This ULAM consists of body-fixed acceleration sensors, connected to a recorder worn around the waist. The ULAM automatically detects upper limb activity during mobility-related activities. Several outcome measures related to general mobility and upper limb usage were compared between patients and controls. The results showed that CRPSI in the dominant upper limb had modest impact on general mobility; i.e. on the percentages spent in body positions and body motions and on mean intensity of body activity. For upper limb usage outcome measures during sitting, there was a marked difference between CRPS1 patients and controls. Especially patients with dominant side involvement clearly showed less activity of their involved limb during sitting, indicated by significant differences for the mean intensity (P=0.014), percentage (P=0.004), and proportion (P=0.032) of upper limb activity. It is concluded that these ten chronic CRPS1 patients still had limitations in upper limb usage during everyday life, 3.7 years (average) after the causative event.

PMID: 12507702
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Schouten AC, Van De Beek WJ, Van Hilten JJ, Van Der Helm FC.
Proprioceptive reflexes in patients with reflex sympathetic dystrophy.
Delft University of Technology, Department of Mechanical Engineering, Man Machine Systems and Control, Mekelweg 2, 2628 CD Delft, Leiden, The Netherlands.

Reflex sympathetic dystrophy (RSD) is a syndrome that frequently follows an injury and is characterized by sensory, autonomic and motor features of the affected extremities. One of the more common motor features of RSD is tonic dystonia, which is caused by impairment of inhibitory interneuronal spinal circuits. In this study the circuits that modulate the gain of proprioceptive reflexes of the shoulder musculature are quantitatively assessed in 19 RSD patients, 9 of whom presented with dystonia. The proprioceptive reflexes are quantified by applying two types of force disturbances: (1) disturbances with a fixed low frequency and a variable bandwidth and (2) disturbances with a small bandwidth around a prescribed centre frequency. Compared to controls, patients have lower reflex gains for velocity feedback in response to the disturbances around a prescribed centre frequency. Additionally, patients with dystonia lack the ability to generate negative reflex gains for position feedback, for these same disturbances. Proprioceptive reflexes to the disturbances with a fixed low frequency and variable bandwidth present no difference between patients and controls. Although dystonia in the RSD patients was limited to the distal musculature, the results suggest involvement of interneuronal circuits that mediate postsynaptic inhibition of the motoneurons of the proximal musculature.

PMID: 12743675

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Singh B, Moodley J, Shaik AS, Robbs JV.
Sympathectomy for complex regional pain syndrome.
J Vasc Surg 2003 Mar;37(3):508-11
Department of Surgery, Nelson R. Mandela School of Medicine, Faculty of Health Sciences, University of Natal, 4013 Congella, South Africa.

BACKGROUND: With the easier and earlier recognition of complex regional pain syndrome (CRPS), a reappraisal of its therapy, particularly the role and timing of sympathectomy, is warranted. PATIENTS AND METHODS: Over a 9-year period, 42 patients with CRPS type II of the upper extremity were referred for sympathectomy. Patients were categorized according to the duration of the symptoms (group I, <3 months; group II, >3 months). All patients underwent initial medical treatment; stellate ganglion blocks were performed when symptoms persisted beyond 6 weeks. Patients were referred for thoracoscopic sympathectomy on persistence of the pain syndrome. A visual linear analogue scale was used to evaluate outcome of sympathectomy. RESULTS: Thoracoscopic dorsal sympathectomy was successfully undertaken in 32 patients. In the remaining 10 patients, thoracoscopy was not technically feasible and open sympathectomy was performed. There was an overall improvement in all 42 patients undergoing sympathectomy (P <.001, Wilcoxon signed rank test). The outcome in group I was significantly better than in group II (P <.003, Mann-Whitney U test). The diagnosis of sympathetically mediated pain with stellate blockade did not correlate with clinical outcome. Patients undergoing thoracoscopic sympathectomy had a better outcome than those undergoing open sympathectomy. There were no complications, and the hospital stay was shorter in the thoracoscopic group. CONCLUSION: Early recognition of CRPS and prompt recourse to surgical sympathectomy is a useful option in the management of CRPS.

PMID: 12618683

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Son UC, Kim MC, Moon DE, Kang JK.
Motor cortex stimulation in a patient with intractable complex regional pain syndrome type II with hemibody involvement. Case report.
J Neurosurg 2003 Jan;98(1):175-9
Department of Neurosurgery, Kangnam St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seoul, Korea. sbc@cmc.cuk.ac.kr

The authors describe the effectiveness of motor cortex stimulation (MCS) in a patient with complex regional pain syndrome (CRPS) Type II, formerly known as causalgia, with hemibody allodynia. During MCS, a subjective sensation of warm paresthesia developed in the painful hand and forearm and spread toward the trunk. Pain and allodynia in the areas associated with this sensation were alleviated significantly. The analgesic effect of stimulation proved to be long lasting and was still present at the 12-month follow up. The authors speculate that MCS might exert its effect through the modulation of thalamic activity in this particular case of CRPS with hemisensory deficit. A central mechanism associated with functional disturbance in noxious-event processing in the thalamus might have an important role in the pathogenesis of the condition.

PMID: 12546368

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van de Beek WJ, Schwartzman RJ, van Nes SI, Delhaas EM, van Hilten JJ. Diagnostic criteria used in studies of reflex sympathetic dystrophy.
Neurology. 2002 Feb 26;58(4):522-6.
Department of Neurology, Leiden University Medical Center, the Netherlands.

OBJECTIVE: Assessment of the diagnostic criteria of reflex sympathetic dystrophy (RSD) and evaluation of the impact of the introduction of the diagnostic criteria of complex regional pain syndrome (CRPS) on the international application of diagnostic criteria of RSD. METHODS: Randomized controlled trials and clinical investigations, published between January 1980 and June 2000, were evaluated with regard to the applied diagnostic criteria of RSD. RESULTS: One hundred seven studies were identified. Thirty-four of these studies were excluded because of inadequate reporting of diagnostic criteria. The 73 included studies were not homogeneous with regard to the diagnostic criteria because they applied many different aspects of sensory and autonomic features. Only 12% of the studies considered the presence of motor features, mostly vaguely described, as mandatory for the diagnosis RSD. Although 10 of the 23 studies published since the introduction of CRPS have applied this term, only 3 used the exact criteria without additions or other modifications. CONCLUSION: Diagnostic criteria sets of RSD focus on many different aspects of sensory and autonomic features that generally are described vaguely. This has not changed since the introduction of the CPRS criteria. These findings question whether the current criteria adequately define RSD.

PMID: 11865127

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***van de Beek WJ, Vein A, Hilgevoord AA, van Dijk JG, van Hilten BJ.
Neurophysiologic aspects of patients with generalized or multifocal tonic dystonia of reflex sympathetic dystrophy.
J Clin Neurophysiol. 2002 Jan;19(1):77-83.
Department of Neurology, Leiden University Medical Center, 2300 RC Leiden, The Netherlands.

Reflex sympathetic dystrophy (RSD) is a syndrome dominated by sensory, autonomic, and motor features of the extremities. In this study, 10 severely affected RSD patients who progressed to multifocal or generalized tonic dystonia underwent H-reflex evaluation, needle electromyography (EMG), polysomnography, somatosensory evoked potentials, and transcranial magnetic stimulation. H-reflex evaluation revealed an impaired vibratory inhibition of the H-reflex and a higher facilitation peak in the recovery curve between 200 to 350 msec. Needle EMG revealed an impaired reciprocal inhibition, and many patients were unable to alter the amount of muscle activity voluntarily. Evaluations of the stretch reflex showed a markedly decreased threshold and abnormal responses to tonic and phasic changes. Polysomnography performed in five patients revealed no abnormal EMG activity during nonrapid eye movement and rapid eye movement sleep, but EEG arousal phenomena provoked abnormally high and brief bursts of surface EMG activity in all registered muscle groups. Somatosensory evoked potentials and transcranial magnetic stimulation were normal. Taken together, the findings in these patients with tonic dystonia of RSD are in accordance with an impairment of inhibitory interneuronal circuits at the level of the brainstem or spinal cord.

PMID: 11896357

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***van de Beeka, Willem-Johan T., Bart O. Roepb, Arno R. van der Slikb, Marius J. Giphartb and Bob J. van Hilten,
Susceptibility loci for complex regional pain syndrome
a) Department of Neurology, Leiden University Medical Center, P.O. Box 9600, 2300 RC, Leiden, The Netherlands b) Department Immunohematology and Blood Transfusion, Leiden University Medical Center, Leiden, The Netherlands
Received 11 April 2002; accepted 22 October 2002. ; Available online 27 March 2003.

An association between HLA-DR13 and patients with complex regional pain syndrome (CRPS) who progressed towards multifocal or generalized tonic dystonia was recently reported. We now report on a new locus, centromeric in HLA-class I, which was significantly associated with a spontaneous development of CRPS, suggesting an interaction between trauma severity and genetic factors conferring CRPS susceptibility. Additionally, an association with the D6S1014 locus was found, supporting the previous finding of an association with HLA-DR13.
[Corresponding Author Contact Information] Corresponding author. Tel.: +31-71-526-2895; fax: +31-71-524-8253


 

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