Extrapyramidal signs occurring after sympathetic block for complex regional pain syndrome responding to diphenhydramine: Two case reports.

RATIONALE: To present an unusual extrapyramidal motor response occurring after a sympathetic block in CRPS and its successful treatment with diphenhydramine. PATIENT CONCERNS: Severe pain related to Complex Regional Pain Syndrome type 1 interfering with activities of daily living. DIAGNOSES: Complex Regional Pain Syndrome type 1. INTERVENTIONS: We report a video case reports of two patients with a diagnosis of CRPS type-I. Both patients exhibited similar presentation of unusual extrapyramidal motor response of the affected limb following lumbar sympathetic block. Both patients were treated with intravenous diphenhydramine to abort the extrapyramidal motor response. OUTCOMES: Both patients similarly responded to treatment with intravenous diphenhydramine with abrupt resolution of the motor response. LESSONS: Sympathetic blockade may interfere with the adaptive autonomic reflex circuits of the motor balance homeostasis in patients with complex regional pain syndrome. Disinhibition of extrapyramidal system may lead to immediate expression of extrapyramidal signs following the sympathetic block. Diphenhydramine, with its anti-histaminic and anticholinergic properties, may be effective in aborting such extrapyramidal signs, and should be considered as a treatment option in similar cases.

Spinal Cord Neuromodulation Therapy for Levofloxacin-Reinduced Complex Regional Pain Syndrome and Neurotoxicity: A Case Report.

Fluoroquinolones are a class of popular outpatient antimicrobial agents with a wide spectrum of therapeutic indications for respiratory and genitourinary infections. Though the most common side effects are gastrointestinal, fluoroquinolones have been increasingly associated with neurotoxicity including peripheral neuropathy and seizures. We present here a case of a 43-year-old woman with previously resolved type I complex regional pain syndrome (CRPS) who presented with symptoms of CRPS and neurotoxicity in the setting of levofloxacin administration. Our aim is to advocate for increased caution in prescribing to patients with a history of neuropathology including CRPS.

Role of peripheral endothelin receptors in an animal model of complex regional pain syndrome type 1 (CRPS-I).

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.

Cyclosporine-induced pain syndrome in a child undergoing hematopoietic stem cell transplant.

OBJECTIVE: To report a case of calcineurin-induced pain syndrome (CIPS) in a child undergoing his second hematopoietic stem cell transplant (HSCT). CASE SUMMARY: A 6.1-year-old child received cyclosporine and methotrexate for acute graft-versus-host disease (aGVHD) prophylaxis after his first HSCT for acute myeloblastic leukemia. Amlodipine was given for the treatment of hypertension. Symptoms of CIPS were not observed. After the second HSCT at the age of 6.7 years, the child received cyclosporine (target trough whole blood cyclosporine concentration range 150-200 microg/L), starting on day -3, and mycophenolate mofetil for aGVHD prophylaxis. With the first cyclosporine dose, the patient complained of leg pain that was most severe during the cyclosporine infusion. Analgesic agents and a change from intravenous to oral administration of cyclosporine were ineffective in controlling the pain. Magnetic resonance imaging findings on day 10 showed periosteal soft tissue changes and mild bone marrow edema of the femora and tibiae. Tacrolimus was substituted for cyclosporine on day 20; on day 21 amlodipine was initiated to manage hypertension. Trough whole blood tacrolimus concentrations ranged from 1.7 to 6.2 microg/L. Pain was reduced in severity by day 29 and completely resolved once tacrolimus was discontinued on day 42. In this case, CIPS was considered to be probably associated with cyclosporine according to the Naranjo probability scale. DISCUSSION: CIPS is hypothesized to result from calcineurin-induced vascular changes that disturb bone perfusion and permeability, leading to intraosseous vasoconstriction and bone marrow edema. In our patient, symptoms were most acute during the infusion, when whole blood cyclosporine concentrations were likely to be the highest. Our patient's symptoms were resolved when tacrolimus was substituted for cyclosporine and amlodipine was initiated. CONCLUSIONS: Interventions aimed at reducing pain associated with CIPS may include the initiation of calcium-channel blocker therapy and conversion to an alternative calcineurin inhibitor.

Sympathetic dystrophy associated with sirolimus therapy.

The reflex sympathetic dystrophy syndrome (RSDS) in organ transplant recipients has only previously been reported in patients treated with calcineurin inhibitors. We retrospectively analyzed 393 renal transplant patients treated with sirolimus, 9 of whom developed RSDS. All the patients reported varying degrees of pain in the legs, affecting the knees, ankles, and/or feet, plus cutaneous erythema. The onset of pain ranged from 1-6 months after transplantation. At the time of diagnosis of RSDS, the mean serum creatinine was 1.4 mg/dL (range 1.0-1.7) and bone scintigraphy with 99mTc pyrophosphate showed increased uptake in all cases. The symptoms remitted 3-10 months after treatment (mean, 4 months) with calcitriol, with or without nifedipine or calcitonin, and in one case with suppression of sirolimus. We conclude that sirolimus therapy may induce RSDS in renal transplant recipients.

[Complex regional pain syndrome type I induced by phenobarbital].

Complex regional pain syndrome type I (CRPS-I) requires the presence of regional pain and sensory changes associated with findings such as abnormal skin color, temperature change, sudomotor activity, or edema, following a noxious event. Complex regional pain syndrome type I induced by phenobarbital (PB) is not well known, although several reports have strengthened the association between PB and CRPS-I. I reviewed the charts of 99 patients treated with PB to assess the incidence, clinical characteristics, investigations, dosage and plasma concentration of PB, and risk factors in the development of CRPS-I. Six patients developed CRPS-I. Pain was severe and allodynia, swelling, discoloration, sweating were present in all patients. This syndrome manifested bilaterally in some patients. Affected patients included 5 men and 1 woman between the ages of 52 and 78 (average 64.2 years). A radiograph showed demineralization in one patient. Thermography showed temperature differences between affected and unaffected limbs, although in a few patients the differences were little because of bilateral affected limbs. 99Technetium methlyene diphosphonate bone scan showed increased periarticular changes in most of the patients. The patients developed CRPS-I at 9.7 weeks (average) after PB was begun. The average time was 7.5 months between CPRS-I and PB reduction. Neither sympathetic ganglion blockade nor physical therapy was effective. Treatment of CRPS-I consists of PB reduction and prednisone and/or Neurotropin. In all patients clinical symptoms and signs such as pain and edema, and range of motion of their shoulders were improved after PB discontinuation. One patient was followed longitudinally, documenting improvement following discontinuation, reexacerbation with PB rechallenge, and remission once more when PB were discontinued. The higher incidence should depend on the coexistence of separate risk factors such as age and PB dosage. Recognition of CRPS-I induced PB, early diagnosis, and withdrawal of PB are important for symptomatic relief and improvement of QOL.

Síndrome hombro-mano en una paciente tratada con anticonvulsivantes / Shoulder-hand syndrome in a patient treated with anticonvulsants

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Complex regional pain syndrome type-I after rubella vaccine.

Complex regional pain syndrome type I (CRPS-I) is a complex disorder characterised by pain, autonomic dysfunction, and decreased range of motion. The syndrome was believed as a well-recognized disorder in adults but, less commonly recognized in children. CRPS-I after vaccination has been rarely reported. We reported an 11-year-old young girl with CRPS-I due to rubella vaccine.

[Recurrent bilateral shoulder hand syndrome induced by phenobarbital]. / Syndrome epaule-main bilateral recidivant gardenalique: a propos d'un cas.

Algodystrophy syndrome is characterized by pain, vasomotor disorders and/or trophic skin changes, without laboratory signs of inflammation. Among the etiological factors of this syndrome (trauma, cerebrovascular accident, diabetes mellitus...), iatrogenic ones including phenobarbital can be observed. We report a case of phenobarbital induced recurrent bilateral shoulder-hand syndrome in a 67 years old women with hypertension, diabetes, and history of epileptic crisis following ischemic cerebrovascular accidents. The patient recovered after barbiturate withdrawal and treatment with calcitonin. It's necessary to diagnose promptly shoulder-hand syndrome of iatrogenic cause in order to withdraw definitively the responsible drug, to start an effective therapy, at this stage, on pain and bone demineralization and to prevent severe sequels.

[Anticonvulsant-induced rheumatism: does a possible role of carbamazepine exist? A clinical case with a 20-month follow-up]. / Reumatismo da anticonvulsivanti: esiste un possibile ruolo della carbamazepina? Caso clinico con follow-up di 20 mesi.

The rheumatism induced by anticonvulsants has been described in literature mostly because of phenobarbital therapy. The possible onset of this clinical picture due to other antiepileptic drugs is unusual and not well defined. We report the case of a 87-year-old female, affected by partial seizures treated with carbamazepine for 20 years, who came to our observation for the onset of disturbances that clearly resemble the classic syndrome of rheumatism induced by barbiturates: the diagnostic hypothesis of a drug side effect was confirmed by the marked clinical improvement of the patient after carbamazepine was stopped and substituted by gabapentin.

Three-month follow-up of shoulder-hand syndrome induced by phenobarbital and treated with gabapentin.

In the present study we evaluated the 3-month follow-up of 14 subjects with phenobarbital-induced shoulder-hand syndrome after discontinuation of their previous pharmacological treatment (group 1: gabapentin 100 mg/day; group 2: acetaminophen 3 g/day for 3 months). The aim of this study was to evaluate pain and joint function in each group after cessation of treatment and to compare the results in the two groups. The result for pain and joint function was better in the seven patients previously treated with gabapentin.

Heroin-induced rhabdomyolysis as a cause of reflex sympathetic dystrophy.

Reflex sympathetic dystrophy is an excessive or abnormal response of the sympathetic nervous system in an extremity to an injury or other condition. The authors describe a 37-year-old man who experienced constant pain and vasomotor instability in both feet after nontraumatic rhabdomyolysis secondary to smoking heroin. Three-phase bone scintigraphy was performed and revealed significantly increased blood-flow, blood-pool, and delayed-phase radioactivity. The follow-up three-phase bone scinitigram showed less radiotracer uptake that was consistent with a good response to calcitonin therapy. Heroin-induced rhabdomyolysis should be added to the list of precipitating conditions that can induce this syndrome.