Pediatric Continuous Brachial Plexus Catheter for a Case of Complex Regional Pain Syndrome (CRPS)

Introduction: Complex regional pain syndrome (CRPS) is a disorder characterized by pain as well as a myriad of sensory, autonomic, and motor disturbances. We are reporting a case of child diagnosed with CRPS and successfully treated with supraclavicular brachial plexus catheter infusion of local anesthetic. Case Report: An eight-year-old male underwent a left thoracotomy, repair of esophageal vascular ring, and translocation of the left subclavian to the left carotid artery. Post-operative course was relevant for severe intractable left shoulder and left arm pain associated with allodynia and hyperalgesia. A supraclavicular catheter was inserted, and an infusion of ropivacaine 0.2% was started. The child was sent for physical therapy as he gradually regained all functions of his left arm and resumed his regular activities. Conclusion: Continuous supraclavicular brachial plexus catheter infusion of local anesthetic is a valuable method of reducing pain in severe cases of upper extremity pediatric CRPS and may be safer and more effective than other invasive measures such as sympathetic blocks and epidural catheterization. Further research surrounding the diagnosis and treatment of pediatric CRPS is needed to allow early diagnosis and treatment and to improve outcome.

Impact of adolescent complex regional pain syndrome on the psychopathology of young men ahead of military service: A retrospective cohort analysis of Korean conscription data

Background: The relationship between physical and psychopathological features in complex regional pain syndrome(CRPS) has been a subject of constant interest, but no data are available in adolescents. Therefore, we aimed to identify the factors associated with psychopathology in adolescents with CRPS ahead of military service.Methods: We retrospectively reviewed all conscription examinees who had completed a Military Personality Inventory(MPI) during a period between February 2013 and December 2016. A total of 63 persons with a history of CRPS(19-years of age for all) were enrolled. Basic demographic and pain-related data were analyzed to examine their association with MPI results. The mean FGR score as well as the 8 subdomain scores were compared between those with pain duration at <15 months(n=30) vs. ≥15 months(n=33). Binary MPI results(normal-abnormal) were also compared between the two groups.Results: In multivariate analysis, abnormal MPI was associated with pain duration, with an odds ratio(OR) at 1.05 for every 1-month increase [95% confidence interval(CI) 1.02–1.08;P=0.002]. Subjects with pain duration at ≥15 months have lower faking good response score(P<0.001 vs. those with pain duration at <15 months), and higher abnormal MPI result rate, faking bad response, inconsistency, anxiety, depression, somatization, paranoid, personality disorder cluster A, and personality disorder cluster B scores(P<0.05). Pain duration was significantly associated with the MPI variables.Conclusions: Pain duration is associated with psychopathology in adolescents with CRPS. Psychopathologic features increased as the disease duration increased. A comprehensive understanding of time-dependent psychopathological factors could support the planning of multimodal approaches for managing adolescent CRPS.

Resolution of Crohn's disease and complex regional pain syndrome following treatment of paratuberculosis

A cohort of family members with various chronic diseases including Crohn's disease, asthma, complex regional pain syndrome, hypothyroidism, type 1 diabetes mellitus, and lymphangiomatosis and/or evidence of infection by Mycobacterium avium subsp. paratuberculosis(MAP) are described in this series of case reports. MAP was cultured from the blood of three members affected by the first five diseases and there was accompanying elevated anti-MAP Ig G in two members. The patient affected by the sixth disease has a markedly elevated anti-MAP titer. The two patients affected by the first four diseases have been treated with a combination of anti-MAP antibiotics and ultraviolet blood irradiation therapy with resolution of the disease symptomatology and inability to culture MAP in post treatment blood samples. These case reports of patients with MAP infections provide supportive evidence of a pathogenic role of MAP in humans.

Failure to fire after an electrical injury——A complex syndrome in a soldier: complex regional pain syndrome(CRPS)

Complex regional pain syndrome(CRPS) is a disorder characterized by an intractable, disabling pain of the affected limb. It is triggered by various injuries and is often resistant to standard therapy. We report a young soldier with CRPS of the right hand sustained from an electrical injury, who had improvement in resting pain with Zoledronic acid. In this report, we discuss the therapeutic options and the role of bisphosphonates in CRPS.

Two Cases of Double Crush Syndrome Progressing to Complex Regional Pain Syndrome

The double crush syndrome is described as an increased risk of distal nerve injury after a more proximal injury. This was a case series of two patients who developed Complex Regional Pain Syndrome under circumstances when a double crush phenomenon could have occurred. Both initially had spinal stenosis and subsequent spinal surgery. Both later had crush injuries to a unilateral lower extremity, which progressed to CRPS. There is no documented correlation between double crush syndrome and CRPS;however, these cases raise awareness about a heightened propensity for CRPS in such patients, which will lead to earlier, accurate diagnosis and treatment.

Frequency and Risk Factors of Complex Regional Pain Syndrome in Cotonou (Benin)

Aim: To determine the frequency and the risk factors of Complex Regional Pain syndrome (CRPS) in Cotonou (Benin). Patients and Méthod: This has been a transversal study carried out over 22 years on files of CRPS infected patients and submitted to rheumatologic consultation in the National Hospital University of Cotonou. Results: 73 out of 17,342 patients examined (0.42%) were suffering from CRPS. Those 73 patients (40 women, 54.8% and 33 men, 45.2%) were in average 54.66 years old and enjoyed an average duration of evolution of 5.79 months. The trauma (41 cases, 56.1%), the stroke (19 cases, 25%), the diabetes (8 cases, 11.3), were the main risk factors that were observed. CRPS was preferably located at the shoulder-hand (34.2%), shoulder (28.8%), wrist-hand (16.4%) and knee (11%). Inflammatory pain was observed in 55 cases. The treatment was dominated by griseofulvina (41 cases, 56.1%), antiinflammatory drugs (38 cases, 52%), analgesic (20 cases, 27, 3%), joint injection by betamethasone (17 cases, 27%). Conclusion: CRPS is not rare in our country. The first risk factor remains the trauma in rheumatologic consultation in Cotonou.

Beliefs and Clinical Practice for Complex Regional Pain Syndrome (CRPS) Managed by Physiotherapists on the South Island of New Zealand

On the South Island of New Zealand, Anaesthetists and other Medical Professionals, frequently refer their patients with Complex Regional Pain Syndrome (CRPS) for physiotherapy management. Beliefs about what is important for the management of patients with CRPS are lacking across all medical and allied health disciplines. Difficulties are no gold standard for diagnosis and evidence for intervention methods is moderate or can be conflicting. This paper explores what Physiotherapists believe to be important in a clinical setting for their management of CRPS, as well as documenting and evaluating their interventional methods used in everyday clinical practice across the region of the South Island of New Zealand. This has not been recorded before. Eighty-one Physiotherapists replied to questions on their usual treatment interventions for the management of CRPS, their frequency of use of these treatment interventions, and what they believed to be important in the management of pain and improvement of function. The results demonstrated that CRPS is not a common condition seen regularly by Physiotherapists;that there is a high level of variation between the physiotherapy interventions used and that Physiotherapists’ beliefs regarding interventions used for pain management and functional restoration differ. Education was reported as the most frequently used intervention method. Those physiotherapists seeing CRPS patients more frequently are more likely to use evidence based intervention methods like graded motor imagery or sensory motor training.

Septic Arthritis of the Hand during Treatment of Complex Regional Pain Syndrome

Complex regional pain syndrome (CRPS) complicated with septic arthritis of the hand has not previously been described. We treated a patient in whom swelling of the wrist recurred during treatment of CRPS and septic arthritis of the hand was induced by group G hemolytic streptococcus. There are case reports of tuberculous arthritis of the hand during treatment of CRPS;however, the causal relationship of wrist joint inflammation with CRPS is unclear. Careful follow-up is necessary for wrist joint inflammation if a patient with CRPS relapses with swelling of the wrist.

Loss of Surround Inhibition and After Sensation as Diagnostic Parameters of Complex Regional Pain Syndrome

Complex Regional Pain Syndrome (CRPS) is a severe chronic pain condition. Patients with CRPS experience allodynia, hyperalgesia, autonomic dysfunction, movement difficulties and dystrophic changes. However other characteristics that may be unique to the pain in CRPS require further study. This study evaluated pain parameters in ninety five subjects composed of three groups: healthy pain free controls, patients with radiculopathy and CRPS patients. Healthy subjects were tested in all four extremities, whereas radiculopathy and CRPS patients were tested only on the most affected extremity. All subjects were tested for the following pain parameters: thermal allodynia, mechanical static and dynamic allodynia, windup, and a hyperalgesic mechanical stimulus. All subjects were also evaluated for autonomic dysfunction, movement disorder and dystrophic skin changes. Statistically significant differences were found between both pain groups and the healthy control subjects as well as between the two pain groups. The finding that statistically differentiated CRPS from radiculopathy and normal controls was pain spread following an algesic mechanical and cold stimulus as well as after sensations to these stimuli. The study demonstrated a simple bedside test that discriminated between CRPS, radiculopathy and healthy control subjects.

Systemic Complications of Complex Regional Pain Syndrome

Complex Regional Pain Syndrome (CRPS) is a neuropathic pain disorder that is characterized by: 1) Severe pain beyond the area of injury;2) Autonomic dysregulation;3) Neuropathic edema;4) A movement disorder, atrophy and dystrophy. It is most often caused by a fracture, soft-tissue injury or surgical procedure and is divided into Type I, in which no nerve lesion is identified (classic reflex sympathetic dystrophy), and Type II where a specific nerve has been damaged (causalgia). In addition to the peripheral manifestations, there are many internal medical complications whose etiology is often not appreciated. This article will examine how CRPS affects the systems of: cognition;constitutional, cardiac, and respiratory complications;systemic autonomic dysregulation;neurogenic edema;musculoskeletal, endocrine and dermatological manifestations;as well as urological and gastrointestinal function.

Complex regional pain syndrome: From diagnosis to rehabilitation

Complex regional pain syndrome(CRPS) is a debilitating pathology characterised by intense chronic pain associated with vasomotor, sensory and motor dysfunction of the affected limb. Although the pathophysiology of CRPS is not fully understood, it is recognised that inflammatory processes and autonomic dysfunction are involved. These processes are associated with peripheral and central sensitisation as well as changes in brain structure and function, and are reflected in the clinical presentation of CRPS. CRPS management requires an interdisciplinary team and requires the therapeutic approach to be individualised. With regard to pharmacological treatment, bisphosphonates, corticosteroids, ketamine and anticonvulsants have been demonstrated to be effective for CRPS management. Psychotherapy, including cognitive-behavioural therapy, has produced promising results but more studies are needed to confirm its efficacy. Among rehabilitation interventions, there is evidence of the efficacy of physiotherapy and occupational therapy in diminishing CRPS symptoms and achieving a higher level of functioning. In this regard, the rehabilitation modality that seems the most promising according to the actual literature is graded motor imagery, which can help to reverse the maladaptive neuroplasticity occurring in CRPS.

Traditional Chinese medicine for foot pain in a patient with complex regional pain syndrome: A case report

BACKGROUND Complex regional pain syndrome(CRPS)is characterized by pain as well as sensory,motor,and sudomotor disorders.Generally,it is classified into two types CRPS-I and CRPS-II.There is no single diagnostic test or treatment approach for CRPS,and a multidisciplinary approach is gaining attention to improve patients’symptoms and their quality of life.CASE SUMMARY A 35-year-old woman with an unremarkable medical history sought treatment for CRPS at a hospital of Korean medicine.During her first visit,she was wheelchairbound due to severe pain in her left lower extremity.She had edema and discoloration of the left foot.She was treated with a combination of traditional Chinese medicine(TCM)approaches,including acupuncture,moxibustion,pharmacopuncture,and herbal decoction,for approximately 20 sessions.The foot and ankle outcome score(FAOS)and visual analog scale(VAS)score for pain were evaluated,along with general signs and functions.Her symptoms,signs,FAOS,and VAS scores improved after treatment,with a significant 7-degree decrease in the VAS score and a 62-point increase in the FAOS score.Additionally,the foot swelling and discoloration gradually resolved.During the phone follow-up,5 mo after the last visit,additional improvements in outcomes were observed.CONCLUSION Combined TCM treatment may be a reasonable and safe option for alleviating symptoms and improving function in patients with CRPS.

International study of the Complex Stress Reaction Syndrome:Implications for transdiagnostic clinical practice

BACKGROUND The debate regarding diagnostic classification systems in psychiatry(categorial vs dimensional systems)has essential implications for the diagnosis,prevention and treatment of stress reactions.We previously found a unique pattern of stress reaction in a study executed during the coronavirus disease 2019 pandemic using large representative samples in two countries,and termed it the Complex Stress Reaction Syndrome(CSRS).AIM To investigate CSRS,Type A(psychiatric symptoms,spanning anxiety,depression,stress symptoms,and posttraumatic stress disorder(PTSD)),with or without long-coronavirus disease(COVID)residuals(CSRS,Type B,neuropsychiatric symptoms spanning cognitive deficits and fatigue,excluding systemic symptoms).Our two-tailed hypothesis was that CSRS is a condition related to an unrecognized type of stress reaction in daily life in the general population(Type A)or that it is related to the severe acute respiratory syndrome coronavirus 2 infection and its long-COVID residuals(Type B).METHODS 977 individuals in four continents(North America,Europe,Australia and the Middle East)completed the online study questionnaire in six languages using the Qualtrics platform.The study was managed by six teams in six countries that promoted the study on social media.The questionnaire assessed anxiety,depression,stress symptoms and PTSD(CSRS,Type A),cognitive deficits and fatigue(CSRS,Type B).The data were analyzed using Proportion Analyses,Multivariate Analysis of Co-Variance(MANCOVA),linear regression analyses and validated clinical cutoff points.RESULTS The results of the Proportion Analyses showed that the prevalence of 4 symptoms spanning anxiety,depression,stress symptoms,and PTSD was significantly higher than the most prevalent combinations of fewer symptoms across 4 continents,age groups,and gender.This supports the transdiagnostic argument embedded in the CSRS(Type A).The same pattern of results was found in infected/recovered individuals.The prevalence of the 4 psychiatric symptoms combination was significantly greater than that of 5 and 6 symptoms,when adding cognitive deficits and fatigue,respectively.MANCOVA showed a significant three-way interaction(age×gender×continent).Further analyses showed that the sources of this three-way interaction were threefold relating to two sub-populations at-risk:(1)Individuals that self-identified as non-binary gender scored significantly higher on all 4 psychiatric symptoms of the CSRS,Type A at young age groups(<50 years old)in North America compared to(self-identified)women and men located in the 4 continents studied,and to other ages across the adult life span;and(2)This pattern of results(CSRS,Type A)was found also in women at young ages(<40 years old)in North America who scored higher compared to men and women in other continents and other ages.Linear regression analyses confirmed the MANCOVA results.CONCLUSION These results show a combined mental health risk factor related to stress reactivity,suggesting that the CSRS is sensitive to populations at risk and may be applied to future identification of other vulnerable sub-populations.It also supports the transdiagnostic approach for more accurate prevention and treatment.Time will tell if such transdiagnostic syndromes will be part of the discussions on the next revisions of the traditional classification systems or whether the crisis in psychiatry further evolves.

Hyoid-complex elevation and stimulation technique restores swallowing function in patients with lateral medullary syndrome:Two case reports

BACKGROUND A swallowing disorder may occur following a brainstem stroke,especially one that occurs in the swallowing centers.Lateral medullary syndrome(referred to as LMS),a rare condition in which a vascular event occurs in the territory of the posterior inferior cerebellar artery or the vertebral artery,has been reported to lead to more severe and longer lasting dysphagia.CASE SUMMARY We report two patients with dysphagia due to LMS and propose a novel technique named hyoid-complex elevation and stimulation technique(known as HEST).The two patients had no other functional incapacity back into life,but nasogastric feeding was the only possible way for nutrition because of severe aspirations.Swallowing function was evaluated by functional oral intake scale,modified water swallow test,surface electromyographic signal associated with video fluorography swallowing study to assess the situation of aspiration,pharyngeal residue,pharyngeal peristalsis,upper esophageal opening and the ability of deglutition.Both patients were treated with the HEST method for dysphagia and recovered quickly.CONCLUSION HEST is effective for shortening the in-hospital time and improving the quality of life for patients with dysphagia who suffer from LMS and likely other strokes.

Comparison of intravitreal anti-vascular endothelial growth factor agents and treatment results in Irvine-Gass syndrome

AIM:To compare the efficacy of bevacizumab,ranibizumab,and aflibercept in pseudophakic cystoid macular edema(CME)patients with Irvine-Gass syndrome(IGS).METHODS:This study is designed as retrospective consecutive case series.Those who developed postoperative pseudophakic CME that refractory to topical treatment and were treated with anti-vascular endothelial growth factor(VEGF)agents included in the study.Optical coherence tomography(OCT)examination including central macular thickness(CMT),total macular volume(TMV),retinal nerve fiber layer(RNFL),ganglion cell layer(GCL)and choroidal thickness(ChT)measurements at the baseline,1 st,3 rd and 6 th month controls were performed.RESULTS:Fifty-nine eyes of 59 patients with CME and other healthy eyes of the patients(Control group)were evaluated.There were 22 eyes of 22 patients in the bevacizumab group(group 1),19 eyes of 19 patients in the ranibizumab group(group 2),and 18 eyes of 18 patients in the aflibercept group(group 3).There was no difference in terms of age,gender,axial length,IOP,and spherical equivalent values.The baseline subfoveal and mean ChT were higher in the IGS group.The difference between the baseline and sixth month values of subfoveal and mean ChT were compared in the CME groups,thinning was observed in all three groups.GCL was thinner in the patient group at the 6 th month of treatment.The resolution time of CME was observed faster in group 1.CONCLUSION:All three anti-VEGF agents seem to be effective in CME but bevacizumab appears to be slightly more cost-effective than the other two alternatives.

Mystery behind labial and oral melanotic macules: Clinical, dermoscopic and pathological aspects of Laugier-Hunziker syndrome

Labial and oral melanotic macules are commonly encountered in a broad range of conditions ranging from physiologic pigmentation to a sign of an underlying life-threatening disease. Although Laugier-Hunziker syndrome(LHS) shares some features of labial and oral pigmentation with a variety of conditions, it is a benign and acquired condition, frequently associated with longitudinal melanonychia. Herein, the demographic, clinical, dermoscopic, and pathological aspects of LHS were reviewed comprehensively. The important differential diagnoses of mucocutaneous and nail pigmentation are provided. An accurate diagnosis is crucial to design a reasonable medical strategy, including management options, malignant transformation surveillance, and psychological support. It is important that clinicians conduct long-term follow-up and surveillance due to the potential risks of malignant transformation and local severe complications in some conditions.

The Concept Study of Recombinant Human Soluble Thrombomodulin in Patients with Acute Respiratory Distress Syndrome

Background: Recombinant human soluble thrombomodulin (rhTM) was approved for the treatment of disseminated intravascular coagulation in Japan, and rhTM has anti-inflammatory effects. Disordered coagulation is a part of the acute respiratory distress syndrome (ARDS) pathophysiology and thus we hypothesize that anticoagulant therapy may help. This preliminary study was to observe the safety of rhTM administration and the improvement on biomarker levels after the therapy for ARDS-patients. Objectives: Case series of ARDS-patients. Methods: Seventeen ARDS-patients that required ventilatory management were treated with rhTM and clinical and laboratory data were collected including platelets, thrombin-antithrombin complex (TAT), fibrinogen degradation products, oxygen saturation/the fraction of inspired oxygen (SpO2/FIO2), and high-mobility group-1 (HMG-1). The administration of rhTM was started during 6 days at a bolus dose of 0.06 mg/kg/day immediately after the diagnosis of ARDS. Results: Eleven of the 17 ARDS-patients were alive at 28 days after the beginning of the administration of rhTM. The serial pattern of the SpO2/FIO2 showed remarkable differences between the survivors and nonsurvivors from day 5 to day 7. The TAT in the survivors significantly decreased after treatment, and there were significantly lower levels in the TAT on day 7 in comparison to that of the nonsurvivors. The serial changes of HMG-1 showed increased levels in the nonsurvivors until day 5 after the administration of rhTM. Conclusions: Additional rhTM administration can safely improve the parameters in survival ARDS-patients, as demonstrated by significant improvements in the SpO2/FIO2, HMG-1 and TAT.

A Case with XXXX Syndrome Who Was Incidentally Diagnosed during Examination for Suspected Post-Human Papillomavirus Vaccination Syndrome

A 19-year-old Japanese woman was referred to us with the complaints of arthralgia and meralgia following human papillomavirus (HPV) vaccination. She received HPV vaccination at the age of 15 years and three years later, she developed intermittent arthralgia, meralgia, and numbness in limbs. There were no orthostatic dysregulation symptoms. She had hypertelorism, and brachydactyly in both the hands, and revealed mild cubitus varus deformity with lateral instability. X-ray examination disclosed hypoplasia of the humeral capitellum and trochlea in elbow joints. G-banded chromosomes were shown to be composed of 48, XXXX. She was, therefore, diagnosed with XXXX syndrome, which explained the reason for her limb symptoms. Although some girls with HPV vaccination complain of various symptoms including limb pain and numbness, exact underlying cause of these symptoms needs to be ascertained carefully for reaching a final diagnosis.

Lady Windermere Syndrome: An Interesting Clinical and Radiological Presentation

Lady Windermere Syndrome is an uncommon pulmonary disease characteristically observed in elderly white females with chronic cough suppression and dyspnea. It is classically associated with Mycobacterium avium complex (MAC) species. An 84-year-old female was evaluated for longstanding non-productive cough and intermittent dyspnea. A chest computed tomography scan revealed densities in the right upper lobe and scattered nodular-cystic bronchiectasis. Biopsy was negative for malignancy;however, bronchioalveolar lavage studies showed presence of MAC. Patient was treated with a 12-month course of azithromycin, rifampin and ethambutol resulting in resolution of her symptoms.

Familial primary pigmented nodular adrenocortical disease without Carney complex (CNC): A case report and review of literature

Primary pigmented nodular adrenocortical disease (PPNAD) is a rare cause of familial Cushing's syndrome. It is characterized by bilateral adrenocortical hyperplasia with small to normal-sized adrenal glands containing multiple small adrenal cortical pigmented nodules [1,2]. PPNAD may occur in an isolated form or as familial PPNAD. Familial cases of PPNAD are usually associated with Carney complex in which Cushing’s syndrome is the most common endocrine manifestation [3]. Familial cases of PPNAD without associated Carney complex are very rare. Only a few cases of familial isolated PPNAD have been reported in the literature, mostly in females [4]. Isolated familial PPNAD has got a better prognosis than familial PPNAD associated with Carney Complex. This observation has important consequences for clinical management, follow-up and genetic counselling of such patients. Familial cases of PPNAD are rare and mostly present in females with associated Carney complex. We herein report a case of familial Cushing’s syndrome in male siblings due to PPNAD without associated Carney complex.