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The evaluation of brain changes to a specific pain condition in pediatric and adult patients allows for insights into potential mechanisms of pain chronicity and possibly long-term brain changes. Here we focused on the primary somatosensory system (SS) involved in pain processing, namely the ventroposterolateral thalamus (VPL) and the primary somatosensory cortex (SI). We evaluated, using MRI, three specific processes: (a) somatotopy of changes in the SS for different pain origins (viz., foot vs. arm); (b) differences in acute (ankle sprain versus complex regional pain syndrome-CRPS); and (c) differences of the effects of CRPS on SS in pediatric versus adult patients. In all cases, age- and sex-matched individuals were used as controls. Our results suggest a shift in concurrent gray matter density (GMD) and resting functional connectivity strengths (rFC) across pediatric and adult CRPS with (a) differential patterns of GMD (VPL) and rFC (SI) on SS in pediatric vs. adult patterns that are consistent with upper and lower limb somatotopical organization; and (b) widespread GMD alterations in pediatric CRPS from sensory, emotional and descending modulatory processes to more confined sensory-emotional changes in adult CRPS and rFC patterns from sensory-sensory alterations in pediatric populations to a sensory-emotional change in adult populations. These results support the idea that pediatric and adult CRPS are differentially represented and may reflect underlying differences in pain chronification across age groups that may contribute to the well-known differences between child and adult pain vulnerability and resilience., (© 2019 Wiley Periodicals, Inc.)

Objective: Findings regarding small nerve fiber damage in complex regional pain syndrome type I (CRPS-I) are not uniform, and studies have not included a matched healthy control group. The aim was to assess intraepidermal nerve fiber density (IENFD) in relation to thermal sensitivity of the same skin areas in CRPS-I patients and a gender- and age-matched healthy control group., Methods: IENFD was investigated in skin biopsies from the CRPS-affected and contralateral limbs of eight CRPS-I patients and from an equivalent site in eight gender- and age-matched healthy controls (HCs). Thermal thresholds (cold/warm detection, cold- and heat-pain detection) were assessed on the affected limb, the matching contralateral limb, and on the equivalent limbs of HCs, and participants rated the intensity of cold/heat and pain to static thermal stimuli (5 °C and 40 °C)., Results: IENFD was significantly lower in both the affected and contralateral limbs of CRPS-I patients than HCs, but IENFD did not differ between the affected and contralateral limbs of patients. The heat pain threshold was lower in the affected CRPS-I limb than in HCs, but all other thermal thresholds were similar in both groups. CRPS-I patients rated the cold stimulus as colder and more painful in the affected limb, and the warm stimulus as hotter, bilaterally, than the HCs., Conclusions: CRPS-I may be associated with bilateral small fiber damage, and perhaps small fiber neuropathy and bilateral disturbances in thermo-sensory perception. These disturbances could stem from a systemic response to injury or might increase the risk of developing CRPS-I after physical trauma.

Complex regional pain syndrome type I (CRPS-I) is a chronic pain syndrome with no clinical evidence of nerve injury; however, recently, changes in muscle tissue have been found in case of CRPS-I. Our aim was to search for histological changes in peripheral nerves of amputated limbs from patients with therapy-resistant CRPS-I that could justify muscle tissue changes. Fifteen patients with CRPS-I (duration >1 year) were included. Multiple nerve samples were taken from upper (n=4) and lower (n=11) amputated limbs. Histological changes (signs of nerve fiber loss and regeneration), fiber diameters, fiber diameter distribution, and fiber density were studied through microscopy and morphometry. Samples from three healthy sural nerves were used as control data as well as data from the literature. All patients (93% of tissue samples) showed histological signs of nerve fiber loss and fiber regeneration, varying in severity. No specific preference was found for any nerve or the location within the nerve. Sural nerves showed loss of especially larger nerve fibers (>12 μm) in comparison with control data. Sympathectomy did not influence this finding. The morphometric results of the other nerves are more difficult to interpret because of the absence of good-quality control data from the literature. However, the percentages of nerve fibers greater than 12 μm seem to lie within the normal range. Besides the known pathology of thin nerve fibers innervating the skin or blood vessels in CRPS-I, this study also shows pathological changes more proximal in the nerves, especially in the sural nerve.

Complex regional pain syndrome (CRPS) is a chronic pain syndrome that causes intractable pain, disability, and poor quality of life for patients. The etiology and pathophysiology of CRPS are still poorly understood. Due to a lack of proper diagnostic tools, the prognosis of CRPS is primarily based on clinical observation. The objective of this work is to evaluate a new imaging modality, photoacoustic microscopy (PAM), for assisting diagnoses and monitoring the progress and treatment outcome of CRPS. Blood vasculature and oxygen saturation (sO₂) were imaged by PAM from eight adult patients with CRPS-1. Patients' hands and cuticles were imaged both before and after stellate ganglion block (SGB) for comparison. For all patients, both vascular structure and sO₂ could be assessed by PAM. In addition, more vessels and stronger signals were observed after SGB. The results show that PAM can help diagnose and monitor CRPS.

The complex regional pain syndrome (CRPS) is a rare but debilitating pain disorder that mostly occurs after injuries to the upper limb. A number of studies indicated altered brain function in CRPS, whereas possible influences on brain structure remain poorly investigated. We acquired structural magnetic resonance imaging data from CRPS type I patients and applied voxel-by-voxel statistics to compare white and gray matter brain segments of CRPS patients with matched controls. Patients and controls were statistically compared in two different ways: First, we applied a 2-sample ttest to compare whole brain white and gray matter structure between patients and controls. Second, we aimed to assess structural alterations specifically of the primary somatosensory (S1) and motor cortex (M1) contralateral to the CRPS affected side. To this end, MRI scans of patients with left-sided CRPS (and matched controls) were horizontally flipped before preprocessing and region-of-interest-based group comparison. The unpaired ttest of the "non-flipped" data revealed that CRPS patients presented increased gray matter density in the dorsomedial prefrontal cortex. The same test applied to the "flipped" data showed further increases in gray matter density, not in the S1, but in the M1 contralateral to the CRPS-affected limb which were inversely related to decreased white matter density of the internal capsule within the ipsilateral brain hemisphere. The gray-white matter interaction between motor cortex and internal capsule suggests compensatory mechanisms within the central motor system possibly due to motor dysfunction. Altered gray matter structure in dorsomedial prefrontal cortex may occur in response to emotional processes such as pain-related suffering or elevated analgesic top-down control.

Densitometry is considered to be the gold standard in bone quality assessment. However, since its introduction, the medical community has been aware that mineral density is only one of the factors that influence the bone risk of fracture, which also depends on the bone's trabecular arrangement and, in particular, on the trabecular architecture's load bearing capabilities. At the University of Trieste, in recent years, a test has been developed that simulates the application of compressive loads on trabecular architecture's reconstructions extracted from digital radiographs. In this work, the test is described, and the results obtained by applying the appraisal in a particular case of severe osteoporosis of the hand, complicating a Complex Regional Pain Syndrome (CRPS) type II, are presented. The test was able to quantify the pathological alterations of bone micro-architecture by means of a Structural Index (SI), which was absolutely significant and relevant to the clinical situation. Important research and clinical opportunities of application of the test include accurate evaluation of osteoporotic bone diseases, careful clinical follow-up and monitoring of responses to therapeutic approaches, and, prospectively, reliable quantification of biological damage (forensic field)., (Copyright © 2013 Elsevier Ltd. All rights reserved.)

Background: Although intima-media thickness (IMT) was increased in several inflammatory diseases, studies investigating whether the inflammatory processes lead to macrovascular alteration with increased IMT in complex regional pain syndrome (CRPS) lack., Methods: Using ultrasound (high-resolution B-mode), we compared bilaterally the IMT of the common carotid artery (CCA-IMT), the radial artery (RA-IMT), the brachial artery (BRA-IMT) and the quotient QRA/CCA, in CRPS type I (n=17), peripheral nerve injury (PNI, n=17) and pain-free controls (PFC, n=22, matched to CRPS by gender, age and traditional cardiovascular risk factors)., Statistics: Spearman's correlation, paired t-test, ANOVA (p<0.05)., Results: Compared to PFC, RA-IMT were significantly increased in both patient groups bilaterally (mean±standard deviation, CRPS affected side vs. PFC dominant side: 0.32±0.08 mm vs. 0.19±0.08 mm, p<0.001; PNI affected side vs. PFC dominant side: 0.27±0.09 mm vs. 0.19±0.08 mm, p< 0.05; CRPS non-affected side vs. PFC non-dominant side: 0.30±0.10 mm vs. 0.19±0.09 mm, p<0.001; PNI non-affected side vs. PFC non-dominant side: 0.25±0.10 mm vs. 0.19±0.09 mm, p<0.05) and QRA/CCA (CRPS affected-side vs. PFC dominant side: 0.49±0.12 vs. 0.30±0.11, p<0.001; PNI affected side vs. PFC dominant side: 0.41±0.10 vs. 0.30±0.11, p<0.05; CRPS non-affected side vs. PFC non-dominant side: 0.43±0.19 vs. 0.30±0.13, p<0.001; PNI non-affected side vs. PFC non-dominant side: 0.39±0.14 vs. 0.30±0.13, p<0.05), and BRA-IMT - only on the affected side in CRPS (CRPS: 0.42±0.06 mm vs. PFC: 0.35±0.08 mm; p<0.05). In CRPS, QRA/CCA was significantly higher on the affected side compared to PNI (p<0.05). However, only CRPS displayed within-group side-to-side differences with a significantly increased RA-IMT and QRA/CCA on the affected side (p<0.05). The CCA-IMT was comparable between all groups and sides., Conclusions: The increased IMT of peripheral arteries in CRPS suggests ongoing inflammatory process. Until now, only endothelial dysfunction has been reported. The presented morphological macrovascular alterations might explain the treatment resistance of some CRPS patients.

Objectives: This is an observational study looking at the epidemiology of cervical spondylotic myelopathy of patients presenting to our hospital., Materials and Methods: The notes and MRI scans of 41 patients presenting to the Leicester General Hospital with a clinical diagnosis of cervical myelopathy between January 2004 and December 2008 were reviewed retrospectively., Results: Cervical myelopathy was found to be more common in male patients to the ratio of approximately 2.7:1, with an average age at diagnosis of 63.8 years. Multi-level disease was seen in the majority of patients, with C5/6 being the most commonly affected level., Conclusions: Cervical myelopathy predominantly affects men in their 7th decade of life. It is often a multi-level disease with C5/6 being the most commonly affected. It has little in common with cervical radiculopathy and is more analogous to lumber spinal stenosis.

Complex regional pain syndrome (CRPS) is a pathologic condition in which the painful experience is disproportionate in time and intensity in comparison with the inciting event. At present, the pathophysiology of CRPS is not well understood. Several studies have indicated that cortical reorganization plays a role in the persistence of the symptoms. A new promising approach, graded motor imagery (GMI), seems to be effective, but there are limited data for the CRPS-1 upper extremity population. The aim of this study was to demonstrate the effectiveness of a modified GMI (mGMI) protocol based on the work of Moseley to reduce pain and enhance functional capacities for a population with nonchronic CRPS-1 of the upper extremity. The following outcome measures were used to assess the clinical effectiveness: pain (short form of the McGill Pain Questionnaire), grip force (Martin vigorimeter), perception of upper extremity function (Disabilities of the Arm, Shoulder and Hand Questionnaire), and patient's global impression of change. All outcomes at T4 were compared with the baseline data (T0) using the Mann-Whitney test and the χ test (nonparametric tests). Seven patients were recruited for the study. At the end of the mGMI (T4), we obtained significant results for the decrease in the pain experienced in the last 7 days (visual analog scale; P=0.046), improvement in the affected extremity grip force (P=0.042), and the patient's global impression of change (P=0.015). However, the data of the perception of upper extremity function (Disabilities of the Arm, Shoulder and Hand Questionnaire) were not clinically or statistically significant. Our results indicate that this mGMI protocol seems to be a promising therapeutic modality to reduce pain. However, more investigations are needed to determine whether mGMI has a significant impact on upper extremity function.

Background: Accumulating experimental and clinical evidence supports the hypothesis that complex regional pain syndrome type I (CRPS-I) may be a small fiber neuropathy., Objectives: To evaluate the use of commercially available standard biopsy methods to detect intradermal axon pathology in CRPS-I, and to ascertain if these structural changes can explain quantitative sensory testing (QST) findings in CRPS-I., Study Design: Retrospective review of charts and laboratory data., Setting: Outpatient clinic, Methods: Skin biopsies from 43 patients with CRPS-I were stained with PGP 9.5, and epidermal nerve fiber density, sweat gland nerve fiber density and morphological abnormalities were evaluated. Thirty-five patients had quantitative sensory testing., Results: Alterations in skin innervation were seen in approximately 20% of CRPS-I patients with commercial processing. There were no patient characteristics, including duration of disease, that predicted a decreased epidermal nerve fiber density (ENFD). There was no consistent relationship between QST changes and ENFD measured by standard commercial skin biopsy evaluation procedures., Limitations: Commercial processing of tissue does not utilize stereologic quantitative analysis of nerve fiber density. Biopsy material is utilized from a proximal and distal source only, and differences in denervation of a partial nerve territory may be missed. The functional attributes of small fibers cannot be assessed., Conclusions: The negative results indicate that CRPS-I may be associated with changes in the ultramicroscopic small fiber structure that cannot be visualized with commercially available techniques. Alternatively, functional rather than structural alterations of small fibers or pathological changes at a more proximal site such as the spinal cord or brain may be responsible for the syndrome.

Background: The purpose of this study was to evaluate oxygen extraction and utilization in end stage chronic complex regional pain syndrome type I (CRPS I) patients undergoing amputation and to relate these to muscle histology of the amputated limb., Materials and Methods: In 25 patients with severe CRPS I requiring amputation of the affected limb venous blood samples and in 11 patients skeletal muscle specimens were analyzed., Results: The mean venous oxygen saturation (S(v)O(2)) value (94.3% ± 4.0%) of the affected limb was significantly higher than S(v)O(2) values found in healthy subjects (77.5% ± 9.8%) pointing to a severely decreased oxygen diffusion or utilization within the affected limb. Histologic analysis showed a significant decrease of type I fibers and a significant increase of type IIB fibers. Ultrastructural investigations of soleus skeletal muscle capillaries revealed thickened endothelial cells and thickened basement membranes. Muscle capillary densities were decreased in comparison with literature data. High venous oxygen saturation levels were partially explained by impaired diffusion of oxygen due to thickened basement membrane and decreased capillary density., Conclusion: This study shows that venous oxygen saturation is significantly increased in chronic end stage CRPS I patients corresponding with impaired oxygen diffusion. The abnormal skeletal muscle findings points to severe disuse but only partially explain the impaired diffusion of oxygen; mitochondrial dysfunction seems a likely explanation in addition., (Copyright © 2012 Elsevier Inc. All rights reserved.)

The complex regional pain syndrome type 1 from the surgeon's point of view: description of the symptoms, imaging (nuclear medicine, MRI) and of the associated psychological context. Importance of the need for a multi-disciplinary organization from the diagnostic to the therapeutic care., (Copyright © 2011 Société française d’anesthésie et de réanimation (Sfar). Published by Elsevier SAS. All rights reserved.)

Introduction: The radiological diagnosis of cervical spondylotic myelopathy (CSM) has to be made as soon as possible, since surgery performed in earlier stages during the course of CSM was reported to be more successful when compared with later stages. We hypothesized that diffusion tensor imaging (DTI) may detect CSM in earlier stages, before the appearance of signal increase in T2-weighted sequences., Methods: A total of 16 patients with neurological signs and symptoms of CSM but without hyperintensity in spinal cord on T2-weighted sequences enrolled in the study. The magnetic resonance (MR) examinations were performed on a 3-T MR imaging system. Apparent diffusion coefficient (ADC) and fractional anisotropy (FA) maps were generated on axial plane. The ADC and FA measurements in each individual were made at the level of most severe cervical canal stenosis and at a nonstenotic level. Student's t test was used to compare FA and ADC values of the spinal cord in stenotic and nonstenotic segments. We also investigated if there was a correlation between DTI parametrics and duration of clinical symptoms by using Pearson correlation analysis., Results: All patients showed changes in DTI parametrics at stenotic segments. While FA values of the spinal cord at the stenotic level showed a statistically significant reduction, there was a statistically significant increase in the measured ADC values (p < 0.001). There was no statistical correlation between the duration of symptoms and DTI parametrics., Conclusion: Our preliminary findings indicate that DTI may show abnormalities in the spinal cord before the development of T2 hyperintensity on conventional sequences in patients with CSM.

"Pain exposure" physical therapy (PEPT) is a new treatment for patients with complex regional pain syndrome type 1 (CRPS-1) that consists of a progressive-loading exercise program and management of pain-avoidance behavior without the use of specific CRPS-1 medication or analgesics. The aim of this study was to investigate primarily whether PEPT could be applied safely in patients with CRPS-1. Twenty patients with CRPS-1 were consecutively enrolled in the study after giving informed consent. The diagnosis of CRPS-1 was defined using the Bruehl and Harden/IASP diagnostic criteria. CRPS-1 was diagnosed between 3 and 18 months after the inciting event (trauma). According to a multiple single-case design (baseline [A1], treatment [B], follow-up [A2]), multiple baseline and follow-up measurements were performed to evaluate changes in CRPS signs and symptoms and to assess functional parameters. When comparing the baseline with the follow-up phase, patients improved significantly with respect to pain on the visual analogue scale (57%), pain intensity (48%), muscle strength (52%), arm/shoulder/hand disability (36%), 10-meter walking speed (29%), pain disability index (60%), kinesiophobia (18%), and the domains of perceived health change in the SF-36 survey (269%). Three patients initially showed increased vegetative signs but improved in all other CRPS parameters and showed good functional recovery at follow-up. We conclude that PEPT is a safe and effective treatment for patients with CRPS-1. A progressive-loading exercise program and management of pain-avoidance behavior without the use of specific medication ("pain exposure" physical therapy) is safe and effective for patients with complex regional pain syndrome., (Copyright © 2011 International Association for the Study of Pain. Published by Elsevier B.V. All rights reserved.)

Background: Abnormal skin temperature in the shoulder is caused by various diseases. A thermography is unable to capture temperature changes over time. In contrast, a Thermocron is an effective measuring device to monitor temperature changes over time., Purposes: The purposes of this study employing a Thermocron were to measure shoulder skin temperature over time in healthy subjects and to detect shoulder skin temperature abnormalities in a patient with shoulder-hand syndrome., Subjects and Methods: Subjects comprised 10 healthy volunteers (20 shoulders; 4 men and 6 women, mean age 54 years). For measurements, a Thermocron was attached on both shoulders. Measurements were made from 21.00 to 07.00 the following morning at 15-minute intervals., Results: Gradual difference in right and left shoulder skin temperature was observed with the timing of measurements but no significant difference was apparent, i.e. dominant side 34.9 ± 0.8°C, non-dominant side 34.9 ± 0.9°C (P = 0.28). Presentation of a case with shoulder-hand syndrome. A 54-year-old woman with the diagnosis of rotator cuff tear underwent surgical treatment of rotator cuff repair, but the pain of the operated shoulder persisted due to phase 1 shoulder-hand syndrome. In postoperative week 3, skin temperature measurement using Thermocrons demonstrated a significant decrease in temperature on the operated side (affected side 34.3 ± 0.4°C, healthy side 35.2 ± 0.3°C; P < 0.05)., Conclusion: The changing of the skin temperature during night-time was successfully recorded both in the healthy subjects and a case with shoulder-hand syndrome using a Thermocron.

Chronic post-ischemic pain (CPIP) is an animal model of CRPS-I developed using a 3-h ischemia-reperfusion injury of the rodent hind paw. The contribution of local endothelin to nociception has been evaluated in CPIP mice by measuring sustained nociceptive behaviors (SNBs) following intraplantar injection of endothelin-1 or -2 (ET-1, ET-2). The effects of local BQ-123 (ETA-R antagonist), BQ-788 (ETB-R antagonist), IRL-1620 (ETB-R agonist) and naloxone (opioid antagonist) were assessed on ET-induced SNBs and/or mechanical and cold allodynia in CPIP mice. ETA-R and ETB-R expression was assessed using immunohistochemistry and Western blot analysis. Compared to shams, CPIP mice exhibited hypersensitivity to local ET-1 and ET-2. BQ-123 reduced ET-1- and ET-2-induced SNBs in both sham and CPIP animals, but not mechanical or cold allodynia. BQ-788 enhanced ET-1- and ET-2-induced SNBs in both sham and CPIP mice, and cold allodynia in CPIP mice. IRL-1620 displayed a non-opioid anti-nociceptive effect on ET-1- and ET-2-induced SNBs and mechanical allodynia in CPIP mice. The distribution of ETA-R and ETB-R was similar in plantar skin of sham and CPIP mice, but both receptors were over-expressed in plantar muscles of CPIP mice. This study shows that ETA-R and ETB-R have differing roles in nociception for sham and CPIP mice. CPIP mice exhibit more local endothelin-induced SNBs, develop a novel local ETB-R agonist-induced (non-opioid) analgesia, and exhibit over-expression of both receptors in plantar muscles, but not skin. The effectiveness of local ETB-R agonists as anti-allodynic treatments in CPIP mice holds promise for novel therapies in CRPS-I patients., (Copyright © 2010 International Association for the Study of Pain. Published by Elsevier B.V. All rights reserved.)

A combination therapy of morphine with an NMDA-receptor antagonist might be more effective than morphine without a NMDA-receptor antagonist for the relief of neuropathic pain in patients with complex regional pain syndrome (CRPS). In order to test the efficacy of this combination therapy we performed a double-blind randomized placebo-controlled study on patients suffering from CRPS of the upper extremity. We used functional magnetic resonance imaging during movement of the affected and unaffected upper hand before and after a treatment regimen of 49 days that contrasted morphine and an NMDA-receptor antagonist with morphine and placebo. We postulated superior pain relief for the combination therapy and concomitant changes in brain areas associated with nociceptive processing. Only the combination therapy reduced pain at rest and during movement, and disability. After treatment, activation in the contralateral primary somatosensory (cS1) and anterior cingulate cortex was significantly reduced when the affected hand was moved. Pain relief during therapy was related to decreased activation in cS1 and secondary somatosensory cortex (S2). Our data suggest that the combination of morphine with an NMDA-receptor antagonist significantly affects the cerebral processing of nociceptive information in CRPS. The correlation of pain relief and decrease in cortical activity in cS1 and S2 is in accordance with the expected impact of the NMDA-receptor antagonist on cerebral pain processing with emphasis on sensory-discriminative aspects of pain., (Copyright © 2010 International Association for the Study of Pain. Published by Elsevier B.V. All rights reserved.)

Objectives: Although the etiology of complex regional pain syndrome type 1 (CRPS 1) is still debated, many arguments favor central maladaptive changes in pain processing as an important causative factor., Methods: To look for the suspected alterations, 10 patients with CRPS affecting the left hand were explored with functional magnetic resonance imaging during graded electrical painful stimulation of both hands subsequently and compared with healthy participants., Results: Activation of the anterior insula, posterior cingulate cortex (PCC), and caudate nucleus was seen in patients during painful stimulation. Compared with controls, CRPS patients had stronger activation of the PCC during painful stimulation of the symptomatic hand. The comparison of insular/opercular activation between controls and patients with CRPS I during painful stimulation showed stronger (posterior) opercular activation in controls than in patients., Discussion: Stronger PCC activation during painful stimulation may be interpreted as a correlate of motor inhibition during painful stimuli different from controls. Also, the decreased opercular activation in CRPS patients shows less sensory-discriminative processing of painful stimuli.These results show that changed cerebral pain processing in CRPS patients is less sensory-discriminative but more motor inhibition during painful stimuli. These changes are not limited to the diseased side but show generalized alterations of cerebral pain processing in chronic pain patients.

Objective: Malfunction of the sympathetic nervous system (SNS) is common in early complex regional pain syndrome type I (CRPS I). This study was designed to evaluate the function of the SNS in patients with chronic CRPS I and to correlate the obtained data with hand function measurements., Materials and Methods: Thirty-two patients of both the sexes in whom the diagnosis of CRPS I of the upper extremity had been established for at least 3 years before they were included in the study. Besides a hand outcomes questionnaire [Michigan Hand Outcome Questionnaire, (MHQ)] the patients underwent a standardized testing of the peripheral sympathetic nervous system (pSNS) using laser Doppler flowmetry (LDF)., Results: Mean time since initial diagnosis was 4.6+/-2.1 years. The mean MHQ score of the affected limb was 57.3+/-17 compared with 69.4+/-13.7 for the unaffected hand (P=0.002). Twenty patients (62.5%) still demonstrated pathologic results regarding the pSNS function (mean sympathetic reflex 0.18+/-0.11, normal range>0.38). We found no statistically significant correlation between pSNS function either with the clinical outcome as measured by MHQ (r=0.246; P=0.175), or with the level of pain (r=0.132; P=0.473)., Conclusions: Even 5 years after the diagnosis of CRPS I of the upper extremity we detected significant impairments of the pSNS in nearly two thirds of our patients. Patients still have pain and present with a significant deterioration of their hand function in comparison with the not affected hand. In our study we could not identify any correlation between pSNS function and clinical outcome as measured by MHQ.

Complex regional pain syndrome is a condition that usually affects the upper or lower extremities. The cause is not clearly understood. We report a case of a severe form of a rapidly progressive complex regional pain syndrome type I developing after a right shoulder injury managed with spinal cord stimulation (SCS). After failed conservative treatments, a rechargeable SCS system was implanted in the cervical spine. Allodynia and dystonia improved but the patient subsequently developed similar symptoms in lower right extremity followed by her lower left extremity. The patient became wheelchair bound. A second rechargeable SCS with a paddle electrode was implanted for the lower extremity coverage. The patient's allodynia and skin lesions improved significantly. However, over time, her initial symptoms reappeared which included skin breakdown. Due to the need for frequent recharging, the system was removed. During explantation of the surgical paddle lead, it was noted by the neurosurgeon that the contacts of the paddle lead were detached from the lead. After successful implantation of another SCS system, the patient was able to reduce her medications and is now able to ambulate with the use of a left elbow crutch.

The diagnosis Complex Regional Pain Syndrome type I (CRPS-I) is based on clinical symptoms, including motor symptoms. Histological changes in muscle tissue may be present in the chronic phase of CRPS-I. Aim of this study was to analyze skeletal muscle tissue from amputated limbs of patients with CRPS-I, in order to gain more insight in factors that may play a role in changes in muscles in CRPS-I. These changes may be helpful in clarifying the pathophysiology of CRPS-I. Fourteen patients with therapy resistant and longstanding CRPS-I, underwent an amputation of the affected limb. In all patients histological analysis showed extensive changes in muscle tissue, such as fatty degeneration, fibre atrophy and nuclear clumping, which was not related to duration of CRPS-I prior to amputation. In all muscles affected, both type 1 and type 2 fibre atrophy was found, without selective type 2 fibre atrophy. In four patients, type grouping was observed, indicating a sequence of denervation and reinnervation of muscle tissue. In two patients even large group atrophy was present, suggesting new denervation after reinnervation. Comparison between subgroups in arms and legs showed no difference in the number of changes in muscle tissue. Intrinsic and extrinsic muscles were affected equally. Our findings show that in the chronic phase of CRPS-I extensive changes can be seen in muscle tissue, not related to duration of CRPS-I symptoms. Signs of neurogenic myopathy were present in five patients.

Objectives: Physiotherapy is considered an important treatment option in patients with upper limb complex regional pain syndrome type-1 (CRPS-1). In case of chronic CRPS-1, exercise therapy of the affected limb forms an important part of the physiotherapeutic program. We investigated whether muscle loading in chronic CRPS-1 patients is associated with impairments in muscle circulation of the forearm of the affected limb., Methods: Thirty patients with chronic CRPS-1 unilaterally affecting their upper limbs, and 30 age-matched and sex-matched control participants were included in this study. Local muscle blood flow and hemoglobin oxygenation were measured by near infrared spectroscopy within the muscles of the forearm at rest, after 1-minute isometric handgrip exercises, and after arterial occlusion. Main outcome parameters were: local muscle blood flow, O2 consumption (mVO2), and postischemic reoxygenation (ReOx)., Results: We found no differences in baseline muscle blood flow, mVO2, and ReOx between the affected CRPS-1, unaffected CRPS-1, and control arms. After exercise, mVO2 of the affected CRPS-1 arms was not different from the clinically unaffected CRPS-1 arms. Furthermore, in comparison with the control arms, unaffected CRPS-1 arms showed no difference in mVO2 or ReOx., Conclusions: Muscle loading does not seems to be related to impairments in muscle oxygen uptake in forearm muscles of upper limbs affected by chronic CRPS-1. Our results suggest that exercise therapy can be safely used in physiotherapeutic training programs for chronic CRPS-1 of the upper limb.

Bone marrow edema syndrome (BMES) refers to transient clinical conditions with unknown pathogenic mechanism, such as transient osteoporosis of the hip (TOH), regional migratory osteoporosis (RMO), and reflex sympathetic dystrophy (RSD). BMES is primarily characterized by bone marrow edema (BME) pattern. The disease mainly affects the hip, the knee, and the ankle of middle-aged males. Many hypotheses have been proposed to explain the pathogenesis of the disease. Unfortunately, the etiology of BMES remains obscure. The hallmark that separates BMES from other conditions presented with BME pattern is its self-limited nature. Laboratory tests usually do not contribute to the diagnosis. Histological examination of the lesion is unnecessary. Plain radiographs may reveal regional osseous demineralization. Magnetic resonance imaging is mainly used for the early diagnosis and monitoring the progression of the disease. Early differentiation from other aggressive conditions with long-term sequelae is essential in order to avoid unnecessary treatment. Clinical entities, such as TOH, RMO, and RSD are spontaneously resolving, and surgical treatment is not needed. On the other hand, early differential diagnosis and surgical treatment in case of osteonecrosis is of crucial importance.

Complex regional pain syndrome type I (CRPS-I) can affect an extremity after minor trauma or operation. The pathogenesis of this syndrome is unclear. It has clinical signs of severe local inflammation as a result of an exaggerated inflammatory response, but neurogenic dysregulation is also a contributor. Several studies investigated the role of inflammatory mediators and cytokines thus far; however, the results are heterogeneous and vary between different settings. This review summarizes recent study results that show a clear underlying inflammatory response at the local site, where systemic responses seem to be inconsistent. An induction of CRPS-like symptoms by application of neuroinflammatory mediators was shown recently. Local inflammation is involved in the pathophysiology of CRPS-I. We must expand our knowledge of pathophysiologic mechanisms, and we are still far away from using inflammatory markers in diagnosis and follow-up of CRPS-I.

Reflex sympathetic dystrophy (RSD) is a poorly understood neurovascular disorder characterized by pain, altered sensation, motor disturbance, soft tissue changes, vasomotor changes, and autonomic changes that occurs after trauma to an extremity. Unilateral leukonychia, Beau's lines, nailfold swelling, and nail clubbing have been an observed sequela of RSD. We present a case of a unilateral atypical trachyonychia occurring in the setting of RSD after traumatic fracture of a digit.

As CRPS I frequently develops after tissue trauma, we proposed that an exaggerated inflammatory response to tissue trauma may underlie CRPS I. Therefore, we studied the vascular inflammatory, nociceptive and apoptotic sequelae of (i) soft tissue trauma and (ii) exaggerated soft tissue trauma in comparison to those of (iii) sciatic nerve chronic constriction injury, modeling CRPS II. Standardized soft tissue trauma (TR) was induced by means of a controlled impact injury technique in the hind limb of pentobarbital-anesthetized rats. Additional animals received soft tissue trauma and femoral arterial infusion of mediator-enriched supernatant achieved by homogenization and centrifugation of traumatized muscle tissue in order to provoke an exaggerated trauma response (ETR). Infusion of supernatant of non-traumatized muscle served as control intervention (STR, sham trauma response). Neuropathy was induced by chronic constriction injury of the sciatic nerve (CCI). Untreated animals served as controls (CO). Detailed nociceptive testing showed temporarily decreased mechanical pain thresholds in ETR animals that resolved within 14 days, while TR and STR animals, i.e. those with singular limb trauma, and controls remained free of pain. Neither cold- nor heat-evoked allodynia developed in post-traumatic animals, whereas CCI animals presented the well-known pattern of ongoing neuropathic pain. Using high-resolution in vivo multifluorescence microscopy, muscle tissue of traumatized animals revealed an enhanced inflammatory response that was found most pronounced in ETR animals. CCI of the sciatic nerve was not accompanied by tissue inflammation; however, induced myocyte apoptosis. Collectively, these data indicate that exaggeration of trauma response induces signs and symptoms of acute CRPS I. Pain perception displays differences to that in CRPS II. Apoptosis turns out to be a distinctive marker for CRPS, warranting further evaluation in clinical studies.

Tibia fracture in rats evokes chronic hindpaw warmth, spontaneous extravasation, edema, allodynia, and periarticular bone loss, a syndrome resembling complex regional pain syndrome type I (CRPS I). Glucocorticoids such as methylprednisolone (MP) are probably effective analgesic and anti-edematous agents in patients suffering from CRPS and this study examined the effects of chronic MP treatment in the rat CRPS I model. Bilateral hindpaw thickness, temperature, and nociceptive thresholds were determined, and the hindlimb bone density was measured using dual-energy X-ray absorptiometry (DXA). Spontaneous cutaneous extravasation and substance P infusion evoked extravasation were determined using an Evans blue vascular permeability assay. After baseline testing, the distal tibia was fractured and the hindlimb casted for 4 weeks. At 2 weeks post-fracture MP infusion was started (1 mg/kg/day for 28 days). The rats were retested at 4, 6, and 8 weeks post-fracture. Hindpaw edema and warmth after fracture were reversed by MP infusion and these effects persisted after discontinuing treatment. Furthermore, there was an increase in spontaneous protein extravasation and an enhanced substance P evoked extravasation and edema response in the hindpaw at 4 weeks that was inhibited by MP infusion. Glucocorticoid treatment had no effect on the allodynia, hindpaw unweighting, or the periarticular bone loss observed after tibia fracture. We postulate that post-junctional facilitation of substance P signaling contributes to the hindpaw warmth, edema, and the enhanced spontaneous protein extravasation observed in this CRPS I model, and that the anti-edematous effects of glucocorticoid treatment are due to inhibition of post-junctional neuropeptide signaling.

CRPS-I consists of post-traumatic limb pain and autonomic abnormalities that continue despite apparent healing of inciting injuries. The cause of symptoms is unknown and objective findings are few, making diagnosis and treatment controversial, and research difficult. We tested the hypotheses that CRPS-I is caused by persistent minimal distal nerve injury (MDNI), specifically distal degeneration of small-diameter axons. These subserve pain and autonomic function. We studied 18 adults with IASP-defined CRPS-I affecting their arms or legs. We studied three sites on subjects' CRPS-affected and matching contralateral limb; the CRPS-affected site, and nearby unaffected ipsilateral and matching contralateral control sites. We performed quantitative mechanical and thermal sensory testing (QST) followed by quantitation of epidermal neurite densities within PGP9.5-immunolabeled skin biopsies. Seven adults with chronic leg pain, edema, disuse, and prior surgeries from trauma or osteoarthritis provided symptom-matched controls. CRPS-I subjects had representative histories and symptoms. Medical procedures were unexpectedly frequently associated with CRPS onset. QST revealed mechanical allodynia (P<0.03) and heat-pain hyperalgesia (P<0.04) at the CRPS-affected site. Axonal densities were highly correlated between subjects' ipsilateral and contralateral control sites (r=0.97), but were diminished at the CRPS-affected sites of 17/18 subjects, on average by 29% (P<0.001). Overall, control subjects had no painful-site neurite reductions (P=1.00), suggesting that pain, disuse, or prior surgeries alone do not explain CRPS-associated neurite losses. These results support the hypothesis that CRPS-I is specifically associated with post-traumatic focal MDNI affecting nociceptive small-fibers. This type of nerve injury will remain undetected in most clinical settings.

Complex regional pain syndromes (CRPS, type I and type II) are devastating conditions that can occur following soft tissue (CRPS type I) or nerve (CRPS type II) injury. CRPS type I, also known as reflex sympathetic dystrophy, presents in patients lacking a well-defined nerve lesion, and has been questioned as to whether or not it is a true neuropathic condition with an organic basis. As described here, glabrous and hairy skin samples from the amputated upper and lower extremity from two CRPS type I diagnosed patients were processed for double-label immunofluorescence using a battery of antibodies directed against neural-related proteins and mediators of nociceptive sensory function. In CRPS affected skin, several neuropathologic alterations were detected, including: (1) the presence of numerous abnormal thin caliber NF-positive/MBP-negative axons innervating hair follicles; (2) a decrease in epidermal, sweat gland, and vascular innervation; (3) a loss of CGRP expression on remaining innervation to vasculature and sweat glands; (4) an inappropriate expression of NPY on innervation to superficial arterioles and sweat glands; and (5) a loss of vascular endothelial integrity and extraordinary vascular hypertrophy. The results are evidence of widespread cutaneous neuropathologic changes. Importantly, in these CRPS type I patients, the myriad of clinical symptoms observed had detectable neuropathologic correlates.

Reflex sympathetic dystrophy (RSD) may be a misdiagnosis or at least not descriptive enough in patients with atypical hand posture and atypical edema. Seven patients with the previous diagnosis of RSD were investigated further because of inconsistent clinical picture with the underlying pathology and bizarre course of the disease. Four patients had clenched fist and three had factitious edema. These seven patients underwent psychological examination, and MMPI was applied to all. In two of these no psychological disorder was obtained according to DSM-IV. One patient could not adapt to MMPI. In two anxiety disorders, in one depression, and in one patient conversion disorder was diagnosed. We suggest that these patients are not motivated enough to improve their conditions and expectations of such patients may show some differences depending on the environment.

A neuropathic-like pain syndrome was produced in rats following prolonged hindpaw ischemia and reperfusion, creating an animal model of complex regional pain syndrome-Type I (CRPS-I; reflex sympathetic dystrophy) that we call chronic post-ischemia pain (CPIP). The method involves placing a tourniquet (a tight fitting O-ring) on one hindlimb of an anesthetized rat just proximal to the ankle joint for 3 h, and removing it to allow reperfusion prior to termination of the anesthesia. Rats exhibit hyperemia and edema/plasma extravasation of the ischemic hindpaw for a period of 2-4 h after reperfusion. Hyperalgesia to noxious mechanical stimulation (pin prick) and cold (acetone exposure), as well as mechanical allodynia to innocuous mechanical stimulation (von Frey hairs), are evident in the affected hindpaw as early as 8 h after reperfusion, and extend for at least 4 weeks in approximately 70% of the rats. The rats also exhibit spontaneous pain behaviors (hindpaw shaking, licking and favoring), and spread of hyperalgesia/allodynia to the uninjured contralateral hindpaw. Light-microscopic examination of the tibial nerve taken from the region just proximal to the tourniquet reveals no signs of nerve damage. Consistent with the hypothesis that the generation of free radicals may be partly responsible for CRPS-I and CPIP, two free radical scavengers, N-acetyl-L-cysteine (NAC) and 4-hydroxy-2,2,6,6-tetramethylpiperydine-1-oxyl (Tempol), were able to reduce signs of mechanical allodynia in this model.

Reflex sympathetic dystrophy (RSD) is a common rheumatic disorder manifesting painful swelling, discoloration, stiffening and atrophy of the skin. Radiographic alterations include small, spotty subperiosteal bone resorption (SBR) and diffuse porosis, and MR imaging shows bone and soft-tissue edema. The purposes of current investigation were to assess 99mTc HDP pinhole SPECT (pSPECT) findings of RSD, to correlate them with those of radiography and MRI and to speculate about causative mechanism of SBR which characterizes RSD. pSPECT was performed in five patients with RSD of the foot. pSPECT showed small, discrete, spotty hot areas in the subperiosteal zones of ankle bones in all five patients. Diffusely increased tracer uptake was seen in the retrocalcaneal surface where the calcaneal tendon inserts in two patients with atrophic RSD. pSPECT and radiographic correlation showed spotty hot areas, that reflect focally activated bone turnover, to closely match with SBR. Further correlation with MRI showed both spotty hot areas and SBR to coincide in location with the insertions of ligaments and tendons, onto which pulling strain is constantly exerted. In contrast, the disuse osteoporosis in unstrained bones did not show any more significantly increased tracer uptake than normal cancellous bones.

This study investigated how members of a hand team perceive clinical signs after a fracture of the distal radius. The risk of developing complex regional pain syndrome I (CRPS-I) was assessed on a 100-mm straight line based on clinical signs 5 weeks, 7 weeks and 10 weeks after the accident. Members of the hand team perceived clinical signs significantly differently.

Atypical presentations are common when spondyloarthropathy develops in older patients. We report two cases initially mistaken for reflex sympathetic dystrophy syndrome (RSDS). Both the patients were men, aged 62 and 75 years, respectively, with marked painful edema of a foot. One patient reported a moderate-energy trauma as the triggering event. Severe diffuse demineralization was noted on radiographs and diffuse hyperactivity on bone scans starting at the early vascular phase. These findings suggestive of RSDS led to treatment with calcitonin, griseofulvin, and pamidronate, all of which were ineffective. Laboratory tests showed severe inflammation, promoting investigations for other conditions. Spondyloarthropathy was diagnosed based on oligoarthritis with sacroiliitis, presence of HLA B27, and a favorable response to non-steroidal antiinflammatory therapy. In older patients, edema of the foot with severe demineralization and the laboratory evidence of inflammation should suggest a spondyloarthropathy.

To confirm the diagnostic usefulness of muscle sympathetic nerve activity (MSNA) in differentiation between cervical spondylotic amyotrophy (CSA) and amyotrophic lateral sclerosis (ALS) with cervical spondylosis (CS), MSNA, heart rate (HR) and blood pressure (BP) were recorded in 10 patients with CSA and ALS with CS, and age-matched healthy volunteers at rest and during head-up tilting. There were no differences in age, disability scores, pulmonary function, and HR or BP at rest between ALS and CSA groups. Resting MSNA was significantly greater in patients with ALS with CS than in comparison groups (P<0.001) with virtually no overlap between ALS and the CSA groups. During head-up tilting, changes in BP and MSNA were significantly less in patients with ALS than in patients with other subjects. MSNA at rest clearly differentiated CSA from ALS with CS, suggesting diagnostic utility.

Introduction: Reflex sympathetic dystrophy (Complex Regional Pain Syndrome type 1) is little known by dermatologists. We report a pediatric case of reflex sympathetic dystrophy with predominant cutaneous involvement., Case Report: A 10 year-old girl presented a warm, painful and relapsing right hand edema for seven months (three outbreaks). The hand was cyanotic, pigmented and painful. Routine blood tests were normal. Radiography and radionuclide bone scan were consistent with stage 1 reflex sympathetic dystrophy. Physiotherapy led to dramatic improvement., Discussion: Reflex sympathetic dystrophy is known since the XVIIIth century. In the last decade, progress in radiology and bone scan have provided elements for understanding the physiopathology of the disease. Microvascular abnormalities under the control of sympathetic nervous system are characteristic of different stages of reflex sympathetic dystrophy. Recently, neurovascular system experiments showed that sympathetic reflex tonus changes may be controlled by the central nervous system. Dermatologic changes of reflex sympathetic dystrophy are well known: edema and erythema in first stage, cyanosis in second stage, sclerosis and atrophia in third stage, but pediatric cases are rarely reported., Conclusion: Reflex sympathetic dystrophy is a complex disease, however its physiopathology is now understood. The clinical presentation can be atypical and the dermatologist may be the first to be consulted.

Objective: Ketamine hydrochloride (KET), an agent used for general anesthesia, has local anesthetic effects and N-methyl-D-aspartate (NMDA) receptor antagonist action. Because recent studies emphasized the role of peripherally distributed NMDA receptors in processing the nociceptive information, we investigated whether peripheral application of the ointment containing KET is able to attenuate the symptoms of local neuropathic pain., Case Reports: We applied ointment containing KET (0.25%-1.5%) to the affected area on limbs in 5 patients with complex regional pain syndrome type I (CRPS I) and in 2 patients with type II (CRPS II). One to 2 weeks later, we observed improvement of the report of pain intensity, measured by the visual analog scale, in 4 patients with acute early dystrophic stage of CRPS I. Swelling of the affected limbs subsided as well. No apparent changes were noticed in 1 patient with chronic atrophic stage of CRPS I and in both patients with CRPS II., Conclusion: Topical application of KET appears to be beneficial for the patients with acute early dystrophic stage of CRPS I because of either its local anesthetic effect or NMDA receptor antagonist action. Patients with chronic atrophic stage of CRPS I and CRPS II patients do not appear to respond to this treatment.

Painful stiffness of the shoulder is an ill-defined clinical entity that is difficult to assess and delicate to treat. The nomenclature used is broad and includes terms such as frozen shoulder, adhesive capsulitis, focal algodystrophy, stiff shoulder, contracted shoulder, and others. Apart from its idiopathic form, the disease can be initiated by trauma, infection, tumour, radiation, systemic and local metabolic disturbances. Pathoanatomically, the common denominator is an inflammatory vascular proliferation followed by thickening, scarring, and retraction of the joint capsule. The inflammatory process often starts at the rotator interval and may extend to the subacromial space. Clinical diagnosis is based on history and physical examination. Generally the onset of pain precedes the perception of a reduced range of motion by weeks or months. In early stages of the disease, the inflammatory type of pain dominates, i.e., the patient's main complaint ist pain at night. In the later stage, range of motion gradually decreases. Patients do not often complain about reduced motion, probably because of its slow onset. Treatment options are a combination of mobilisation exercises with intra-articular steroids, hydraulic distension of the joint capsule, manipulation under anaesthesia, arthroscopic and/or open arthrolysis. The appropriate choice of protocol is just as important as its correct timing. In the inflammatory phase, aggressive treatment protocols are probably contraindicated. Complications of invasive protocols are rare but deleterious and therefore have to be taken into consideration. New anti-anglogenetic agents may enhance functional results and shorten the rehabilitation phase.

Introduction of the term complex regional pain syndromes (CRPS) as a replacement of the older terminology, reflex sympathetic dystrophy (RSD) and causalgia, has achieved two goals: it has focused attention on the diagnosis and treatment, and sent basic scientists back to their laboratories. The relation of sympathetically maintained pain and sympatholysis is examined, particularly as a neuropathic process that is found in many conditions, including CRPS. This review also focuses on recent observations proposing a pathologic basis in support of diagnosis and treatment of these disorders.

There is still controversy whether transient osteoporosis of the hip joint represents a distinct self-limiting disease, or reflects only an early, reversible subtype of non-traumatic osteonecrosis (ON). Transient osteoporosis has several synonyms: algodystrophy of the hip; transient marrow oedema; or bone marrow oedema syndrome--BMOES. Clinical presentation of BMOES shows mechanical hip joint pain, ON risk factors, and a diffuse bone marrow oedema in MR imaging. Histomorphological changes resemble early ON, but with diffuse sufficient repair in BMOES and focal and insufficient repair only at the border of the necrotic lesion in ON. Therefore the clinical course and outcome are significant different, with restitution occurring in BMOES, while progressive destruction of the joint takes place in ON. So far, the preferred treatment strategies are protected weight bearing for BMOES, but operative treatment for ON. In a prospective study of patients with BMOES, the clinical, radiographic, and MRI course of 43 hip joints after core decompression treatment were investigated. All patients showed immediate relief of pain after surgery and the average duration of symptoms with conservative treatment could be dramatically reduced by core decompression from 6 months down to 2 months. There were no perioperative complications. Based on our experience with over 100 BMOES patients, we are convinced that this syndrome represents not a distinct disease but an early reversible subtype of non-traumatic ON. Due to the excellent clinical results of core decompression, we recommend this operative therapeutical concept in patients with painful BMOES.

Because of the controversy concerning the manner in which the sympathetic nervous system is involved in reflex sympathetic dystrophy (RSD), its name was changed to one having no mechanistic connotations. This article reviews the relevant literature in support of not only the taxonomical changes to complex regional pain syndrome (CRPS) but also provides evidence of sympathetic dysfunction demonstrated in animal models of neuropathic pain.

Objective: A difference in temperature between the affected and normal extremity is one of the signs or symptoms of reflex sympathetic dystrophy (RSD). These temperature differences are scored anamnestically, by palpation, or by measurements. We investigated the relationship between objective and subjective differences in skin temperature between the hands., Subjects and Design: Fifty-one patients with RSD of one upper extremity participated (mean age 55 years, range 18-80). In the first 16 we investigated the reliability of the visual analogue scale (VAS) for recording perceived differences in skin temperature. In the remaining 35 patients the relationship between the VAS and objective infrared radiometry of the hands was investigated., Setting: Outpatients clinics of two university hospitals., Results: The VAS was found to be reliable in terms of response stability. There was no significant correlation between the objective and subjective outcomes., Conclusions: There is lack of correspondence between objective and subjective temperature measurements, which may be due to a normal discrepancy between objective and perceived temperature, and/or to alterations in perception due to RSD.

Few series on reflex sympathetic dystrophy syndrome (RSDS) have included children. The present series reviewed 10 affected children. The group consisted of 9 girls and one boy with an average age at onset of 11 years (5 years to 16 years). The diagnosis was based on the clinical findings of pain, dysesthesia and autonomic system dysfunction. All patients underwent x rays and bone scans. Their results showed great variation. Minor trauma was the most common trigger factor. The lower extremities were more often involved. The treatment consisted of pain relief and progressive mobilization. Less conventional treatments in children, such as calcitonin and bisphosphonate were also used. The severity and duration of the disease varied greatly among these children. Moderate pain and sympathetic dysfunction persisted often up to two years after onset. Reflex sympathetic dystrophy is more common in children than previously thought. There are differences with the adult form in presentation and clinical course: the diagnosis is often delayed, the lower extremities are more often involved, girls are affected more often and idiopathic forms are frequent. Significant emotional dysfunction is found in a majority of patients and they are best treated as inpatients by a multidisciplinary team.

Over a period of 12 years, 104 cases of posttraumatic reflex sympathetic dystrophy have been diagnosed followed up and treated according to a standard protocol, including, among other clinical items, a psychiatric examination. Apart from the traditional clinical recordings, the following has been noted: 1. All patients were over 30 years of age; 2. No relationship was found between the significance of the trauma and the severity of the dystrophy; 3. The dystrophy nearly always emerged at the time of the primary healing of the injury; 4. On psychiatric examination, 96% of the patients showed signs of chronic depression; 5. Forty-nine percent had elevated gamma GT suggestive of alcohol abuse; 6. The socioprofessional context always revealed: either a state of inactivity (jobless persons, disabled persons, childless housewives, pensioners); or an opportunity for inactivity (work injury suffered by workers, lower-rank employees, bankrupt self-employed people). There were no tradesmen, executives, lawyers, physicians, consultants, artists, sportsmen or musicians in the series, and no housewives with young children. 7. In the second phase of the survey, we decided to complement the drug therapy by systematically adding antidepressant agents. This led to a significant improvement in the course of the disease. These elements have led us to consider whether traumatic algodystrophy could be a psychosomatic disease.

We present a case of refractory reflex sympathetic dystrophy (RSD) (complex regional pain syndrome, type I) whose symptoms (ongoing pain, allodynia, hyperhydrosis and temperature abnormalities) were resolved after the patient suffered a traumatic cerebral contusion in the left temporal lobe, which caused no neurological deficit. This case suggests that symptoms of some RSD patients may largely sustained by a complex network involving the brain.