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CASE: X is a 22-month-old White male infant with a complex medical history, including diagnoses of FBXO11 mutation, hypotonia, restrictive lung disease and mild intermittent asthma, laryngotracheomalacia, obstructive sleep apnea (OSA), feeding difficulties with a history of aspiration, gastroesophageal reflux disease (GERD), and developmental delays. X's medical presentation has resulted in multiple prior medical admissions for respiratory failure due to acute illnesses, procedures and treatments including gastrojejunostomy (GJ) tube dependence, supraglottoplasty to reshape tissues of the upper larynx, and the use of biphasic positive airway pressure (BiPAP) at night and room air during the day when he is at baseline. In addition, he has nocturnal events characterized by significant agitation, screaming, crying, body stiffening and limb movements with pauses in breathing, mouth breathing, restless sleep, and difficulty waking in the morning with concomitant daytime fatigue despite above treatments for OSA. There is no history of congenital heart disease or sudden unexplained death. Family history is noncontributory because parents are negative for the FBXO11 variant.X's sleep disruption has led to significant sleep deficits for both X and his caregivers, who spend much of the night strategizing on how to console him. X has undergone several sleep studies, starting when X was aged 4 months, at several children's hospitals across the nation to determine the cause of his chronic sleep disturbance, which yielded limited information and treatment success. As an infant, X received a medical workup and was subsequently treated with a proton pump inhibitor (PPI) for reflux. At 12 months, he was diagnosed with disordered sleep with myoclonic jerks and started on melatonin and gabapentin for involuntary movements. At 13 months, gabapentin was weaned back because of intolerance, and at 15 months, nortriptyline and clonidine were started because of worsening symptoms to target potential neuropathic pain. While most of his symptoms were at night, he had occasional daytime screaming episodes, particularly when experiencing illness. Gabapentin and clonidine were stopped because nortriptyline seemed most effective.At 17 months, the results from a sleep study led to a diagnosis of night terrors, and several clinicians agreed that X's sleep disruption was behavioral in nature. At this time, an infant mental health consultant met with a sleep psychologist on the family's behalf to support family in considering systematic desensitization therapy to increase tolerance to wearing his BiPAP mask, as well as other behavioral and sleep hygiene strategies, which were tried on several occasions and again, resulted in limited improvement in functioning.At 19 months, X's multidisciplinary team reconsidered a night terror diagnosis after a failed trial of clonazepam and pursued a differential diagnosis of periodic limb movement disorder (PLMD). X trialed gabapentin again, but this time only a nighttime dose, per sleep medicine and psychiatry recommendation. While this brought some temporary relief from nighttime distress, despite increasing to the highest dose for age and weight (15 mg/kg/dose), this became less effective, and he was weaned off at 22 months. He had been on iron supplementation since age 6 months and received an iron infusion at 22 months because of persistently low ferritin levels and PLMD in sleep.At 24 months, X was briefly trialed on levetiracetam. While no evidence for seizures on EEG was present, this medication was chosen for involuntary movements and genetic risk for seizures. However, this medication was not useful. At 25 months, an evaluation with a movement disorder physiatrist resulted in a diagnosis of nocturnal paroxysmal dystonia, and he was started on baclofen, which has provided some, but not complete relief to nighttime symptoms. Parents are reporting he has more "good nights" than "bad nights," but "bad nights" come in stretches of a few days in length with no known trigger or relief.Most recently, X was evaluated by general genetics. Whole exome sequencing (WES) was pursued which revealed a pathogenic de novo variant in FBXO11 and provides a likely cause for his neurodevelopmental phenotype. However, he has some features not explained by FBX011; thus, reanalysis of his WES was performed and revealed a de novo variant of uncertain significance in RAF1. Because pathogenic variants in RAF1 have been associated with dilated cardiomyopathy and Noonan spectrum disorder, it was recommended that X be followed periodically in a cardiac genetics clinic. Family is well connected into the FBXO11 community, including supportive Facebook groups. Parents have shared that they do not feel X's breathing issues and pain fit with the phenotype of other children with FBXO11 mutations.X is also enrolled in a medical child care program to facilitate development and social-emotional functioning and receives learning, speech, occupational, physical, and feeding therapy while in attendance. Despite periods of absence due to contracting numerous viral illnesses over the past several months, X continues to make progress across developmental therapies and happily engages when at the program.What additional diagnostic tests and treatment should be considered to better understand X's medical and behavioral presentation? What are the implications of chronic sleep deprivation and stress on the behavior and development of infant with X's profile? What are important psychosocial considerations because it relates to children with medical complexity (CMC), particularly for X and his family to support caregiver, family, and X's quality of life and overall well-being?

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Introducción: Las mioclonías son contracciones musculares paroxísticas de corta duración o pérdida abrupta del tono muscular, denominadas mioclonías positivas y negativas, respectivamente. Se presenta un caso clínico de mioclonías positivas y negativas generalizadas y se pretende describir los múltiples mecanismos fisiopatológicos y etiologías que lo desencadenan. Presentación del caso: Hombre de 35 años, con diabetes mellitus tipo 1 complicada con enfermedad renal diabética en hemodiálisis, desarrolló una bacteriemia asociada a catéter por Staphylococcus aureus y presentó mioclonías positivas y negativas. Se identificaron como posibles desencadenantes la uremia, la infección y los fármacos con potencial promioclónico; el hallazgo incidental de una lesión isquémica en núcleo caudado no explicaba la semiología encontrada en el paciente. Se hizo el control y retiro de todos los factores promioclónicos enunciados, junto a manejo farmacológico con levetiracetam, y con ello se logró el control de los síntomas. Discusión: Los pacientes con enfermedad renal crónica son susceptibles a la acumulación de productos tóxicos de tipo guanidinas, que tienen potencial para producir mioclonías. Además, las infecciones, el uso de fármacos con potencial promioclónico y lesiones estructurales como las isquemias corticales son etiologías que deben considerarse en el diagnóstico diferencial. El mayor impacto en los síntomas se observa con el control del factor desencadenante, y, en caso de persistir, la terapia farmacológica proporciona buenos resultados. Conclusión: Las mioclonías son trastornos del movimiento relativamente comunes en la enfermedad renal crónica. La identificación del desencadenante es crucial para su manejo junto al uso de fármacos con actividad antimioclónica.

Introduction: Myoclonus are paroxysmal muscle contractions of short duration or abrupt loss of muscle tone, called positive and negative myoclonus respectively. A clinical case of generalized positive and negative myoclonus is presented and the aim is to describe the multiple pathophysiological mechanisms and etiologies that trigger it. Case presentation: A 35-year-old man with type 1 diabetes mellitus complicated by diabetic kidney disease on hemodialysis developed catheter-associated bacteremia due to Staphylococcus aureus and presented positive and negative myoclonus. Uremia, infection, and drugs with pro-myoclonic potential were identified as possible triggers; The incidental finding of an ischemic lesion in the caudate nucleus did not explain the semiology found in the patient. The control and removal of all the pro-myoclonic factors mentioned was carried out, along with pharmacological management with levetiracetam, thus achieving control of the symptoms. Discussion: Patients with chronic kidney disease are susceptible to the accumulation of guanidine-type toxic products, which have the potential to produce myoclonus. Furthermore, infections, the use of drugs with pro-myoclonic potential and structural lesions such as cortical ischemia are etiologies that should be considered in the differential diagnosis. The greatest impact on symptoms is observed with the control of the triggering factor and if it persists, pharmacological therapy provides good results. Conclusion: Myoclonus are relatively common movement disorders in chronic kidney disease. Identification of the trigger is crucial for its management along with the use of drugs with anti-myoclonic activity.

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BACKGROUND: Chronic low back pain (CLBP) is a global health problem, and gabapentin and pregabalin are often used in the treatment of patients without associated radiculopathy or neuropathy. Therefore, determining their efficacy and safety is of enormous value. OBJECTIVE: To examine the efficacy and safety of using gabapentin and pregabalin for CLBP without radiculopathy or neuropathy. METHODS: We performed a search on the CENTRAL, MEDLINE, EMBASE, LILACS, and Web of Science data bases for clinical trials, cohorts, and case-control studies that evaluated patients with CLBP without radiculopathy or neuropathy for at least eight weeks. The data were extracted and inserted into a previously-prepared Microsoft Excel spreadsheet; the outcomes were evaluated using the Cochrane RoB 2 tool, and the quality of evidence, using the Grading of Recommendations Assessment, Development and Evaluation (GRADE) system. RESULTS: Of the 2,230 articles identified, only 5 were included, totaling 242 participants. In them, pregabalin was slightly less efficacious than amitriptyline, the combination of tramadol/acetaminophen, and celecoxib, and pregabalin added to celecoxib showed no benefit when compared to celecoxib alone (very low evidence for all). On the other hand, although one study with gabapentin did not support its use in a general sample of patients with low back pain, another found a reduction in the pain scale and improved mobility (moderate evidence). No serious adverse events were observed in any of the studies. CONCLUSION: Quality information to support the use of pregabalin or gabapentin in the treatment of CLBP without radiculopathy or neuropathy is lacking, although results may suggest gabapentin as a viable option. More data is needed to fill this current gap in knowledge.

ANTECEDENTES: Dor lombar crônica (DLC) é um problema de saúde global, e a gabapentina e a pregabalina são frequentemente utilizadas no tratamento de pacientes sem radiculopatia ou neuropatia associada. Por isso, determinar sua eficácia e segurança é de enorme valor. OBJETIVO: Examinar a eficácia e segurança do uso de gabapentina e pregabalina no tratamento da DLC sem radiculopatia ou neuropatia. MéTODOS: Realizamos uma pesquisa nas bases de dados CENTRAL, MEDLINE, EMBASE, LILACS e Web of Science por ensaios clínicos, coortes e estudos de caso e controle que avaliassem pacientes com DLC sem radiculopatia ou neuropatia por pelo menos oito semanas. Os dados foram extraídos e inseridos em uma planilha previamente elaborada no programa Microsoft Excel; os desfechos foram avaliados com a ferramenta RoB 2 tool da Cochrane, e a qualidade das evidências, pelo sistema Grading of Recommendations Assessment, Development and Evaluation (GRADE). RESULTADOS: Dos 2.230 artigos identificados, apenas 5 foram incluídos, com um total de 242 participantes. Neles, a pregabalina foi ligeiramente menos eficaz do que a amitriptilina, a combinação de tramadol/acetaminofeno, e o celecoxibe, assim como a pregabalina adicionada ao celecoxibe não mostrou benefício em comparação ao uso isolado de celecoxibe (evidência muito baixa para todos). Quanto à gabapentina, embora um estudo não respalde seu uso para uma amostra geral de pacientes com lombalgia, outro encontrou redução na escala de dor e melhora da mobilidade (evidência moderada). Nenhum evento adverso grave foi observado nos estudos. CONCLUSãO: Há carência de informações de qualidade que sustentem o uso de pregabalina ou gabapentina no tratamento da DLC sem radiculopatia ou neuropatia, embora resultados possam sugerir que a gabapentina é uma opção viável. Mais dados são necessários para preencher essa atual lacuna no conhecimento.

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BACKGROUND: Gabapentin is an effective therapeutic alternative for chronic low back pain, indicated in several guidelines for treating neuropathic pain as first-line medication. This study aimed to describe the pharmacodynamics of gabapentin in the central nervous system of patients with chronic low back pain (CLBP) by using single-photon emission CT (SPECT) with [99mTc]Tc-ECD. METHODS: We selected 13 patients with CLBP due to lumbar disc herniation. They underwent SPECT before and after using gabapentin, compared with a SPECT database of healthy volunteers. A second analysis compared regional cerebral blood flow (rCBF) changes between responders and non-responders to gabapentin and the healthy controls. RESULTS: The mean age of patients was 41 years, and the mean pain intensity was 5.92 points, measured by the Numeric Rating Scale. After using gabapentin, SPECT showed an increase of rCBF in the bilateral anterior cingulate gyrus and a decrease of rCBF in periaqueductal gray matter. Non-responder patients with gabapentin showed a post-treatment decrease of rCBF in the paracentral lobule of the brain. CONCLUSIONS: A lack of improvement in some patients with gabapentin may be associated with an activated affective circuit of pain, evidenced by the increase of rCBF of the anterior cingulate cortex. A maladaptive brain state in chronic pain can explain the decrease of rCBF in the default mode network structures. Gabapentin acts directly or indirectly on neurons of periaqueductal gray substance by increasing the pain threshold and decreasing the rCBF of this structure.

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OBJECTIVES: The aim of this study was to evaluate the influence of a preappointment oral dose of gabapentin on the neurological examination of cats. METHODS: A prospective, randomized and blinded clinical trial was conducted in 35 client-owned healthy cats. Cats were scheduled for two appointments and randomly assigned to receive either a placebo or a 100 mg gabapentin capsule prior to the second veterinary visit. A neurological examination was performed during each visit, and the results were compared between groups. Normal/abnormal response rates for each test were based on the number of cats that allowed the test to be performed. RESULTS: Gabapentin was administered to 17 cats. Gait and postural reactions were significantly affected in the gabapentin group. Comparing the gabapentin with the placebo groups, proprioceptive ataxia was identified in 4/17 (23.5%) vs 0/18 cats (P = 0.0288); paw placement deficits were seen in 10/11 (90.9%) vs 1/4 (25%) cats; table tactile placement deficits were identified in 13/17 (76.5%) vs 0/18 cats (P <0.0001); hopping deficits were seen in 5/17 (29.4%) vs 0/16 cats (P = 0.0185); and abnormalities on wheelbarrowing and extensor postural thrust were reported in 5/17 (29.4%) vs 0/18 cats (P = 0.0129). These results had no correlation with age or dose/kg received. No significant difference was noted in the assessment of level and content of consciousness, posture, cranial nerves and spinal nerves. No significant differences were noted in test compliance or examination duration. CONCLUSIONS AND RELEVANCE: Gabapentin significantly altered gait analyses and postural reactions in this group of healthy cats. The administration of gabapentin could lead to false-positive results and, possibly, an incorrect identification of neurological lesions. In contrast, gabapentin did not impair the assessment of cranial nerves and spinal reflexes, which can be assessed in patients receiving the drug.

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BACKGROUND: Studies have shown that the overall incidence rate of herpeszoster (HZ) in China is 6.64 cases per 1000 people, despite such harms brought by postherpetic neuralgia (PHN), the mechanism of the disease remains unclear in China. Currently, effective biomarkers to predict PHN remain unavailable, which makes it difficult to prevent and successfully treat PHN. OBJECTIVE: The aim of the study was to determine the serum interleukin-6 level in PHN. METHODS: The serum levels of interleukin 6 (IL-6) were measured by multi-antibody sandwich ELISA. The likert scale was used to represent the degree of neuralgia in the patients. Patients with PHN were divided into a mild PHN group and a severe PHN group according to the Likert scale. ROC curve was performed for evaluating the diagnostic efficiency of IL6 for PHN. The correlation between the IL6 level and the Likert scale before and after treatment with gabapentin and mecobalamin was analyzed. RESULTS: IL6 levels in PHN patients resulted higher compared to volunteers. Patients in the severe PHN group had a higher serum IL6 level than in the mild PHN group. The Likert scale score was related to the serum IL6 levels and the frequency of IL6 levels above the cutoff value (4.95 pg/mL) in PNH groups before and after treatment (p < 0.05). STUDY LIMITATIONS: Pain is subjective. Some mental states, such as anxiety and depression, greatly influence an individual's perception of pain, and pain tolerance can vary between people. Therefore, pain scores can be affected by different individual factors. CONCLUSIONS: The serum IL6 levels may be used as a biochemical indicator of the severity of PNH.

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OBJECTIVES: The aim of this study was to verify whether a single oral dose of gabapentin (100 mg) or melatonin (3 mg) given 60 mins before a cardiac evaluation would reduce anxiety without interfering with heart rate (HR), systemic blood pressure (SBP), electrocardiogram (ECG) and echocardiographic indexes. METHODS: Seventy-five client-owned healthy cats underwent two sets of cardiac assessments 60 mins apart, randomly divided into gabapentin, melatonin and placebo groups. The interval between treatment and the second ECG and SBP measurement was 60 mins, and 70 mins for echocardiography. A compliance score (CS) classified the behavior, focusing on the ease of handling. RESULTS: Most variables did not change between the examinations. The placebo group showed more significant changes (SBP, tricuspid annular plane systolic excursion, HR during echocardiography, aortic flow velocity, S' wave from lateral mitral annulus), but they were not considered to be hemodynamically relevant. Gabapentin and melatonin significantly increased the cats' compliance without interfering with cardiac assessment. Eight cats presented with mild sedation, seven after gabapentin and one after melatonin. No major side effects were observed. CONCLUSIONS AND RELEVANCE: Gabapentin tranquilized the cats when it was given 60 mins prior to ECG and SBP measurement, and 70 mins prior to echocardiography, without interfering with systolic echocardiographic indexes. Melatonin also decreased the CS, but without sedation in most cases. The waiting period may have relaxed the cats in the placebo group, resulting in lower SBP measurements. However, this tranquility did not last as some echocardiographic changes signaled a sympathetic predominance.

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OBJECTIVES: The aim of this study was to evaluate, using echocardiography, the effects of oral administration of a single dose of gabapentin on the physiologic variables (heart rate [HR], respiratory rate [RR] and systolic blood pressure [SBP]) and systolic and diastolic cardiac function of healthy cats. METHODS: This was a prospective, randomized and blinded study with 40 healthy cats aged between 6 months and 2 years. The cats' health status was assessed on the first appointment (T1) when they underwent a physical examination, complete blood count, biochemical profile, assessment of physiologic variables and echocardiogram. The echocardiogram was used to measure the left ventricle's (LV) internal diameter during systole and diastole, isovolumic relaxation time, transmitral flow, E-wave deceleration time and HR. The cats were randomly divided into two groups: (1) a treatment group with 20 cats that received a single oral dose of gabapentin (100 mg/cat); and (2) a control group with 20 cats that received a single oral dose of placebo. All variables of the physiologic and echocardiographic variables were re-evaluated 1-3 weeks after T1 (T2), 90 mins after medication or placebo administration. RESULTS: There was no difference in the physiologic variables evaluated in both groups. The proportion of cats in the treatment group that had their ventricular filling waves fused on T1 but did not have them fused on T2 was significantly higher (45%) compared with cats in the control group (15%; P = 0.0384). CONCLUSIONS AND RELEVANCE: There was no difference between the groups in regard to SBP, HR, RR and echocardiographic variables. Gabapentin improved evaluation of diastolic function on echocardiogram because it reduced the fusion of ventricular filling waves during the evaluation of the diastolic function of the LV. Gabapentin did not cause adverse effects on the cardiovascular hemodynamics of young healthy cats.

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Abstract Introduction Burns are a common trauma that cause acute severe pain in up to 80% of patients. The objective of this narrative review is to evaluate the efficacy of opioids, non-steroidal anti-inflammatory drugs, paracetamol, gabapentinoids, ketamine, and lidocaine in the treatment of acute pain in burn victims. Methodology The databases explored were PubMed, Embase, ClinicalTrials, and OpenGrey. The included randomized, controlled clinical trials assessed the analgesic efficacy of these drugs on hospitalized patients, had no age limit, patients were in the acute phase of the burn injury and were compared to placebo or other analgesic drugs. Studies describing deep sedation, chronic opioid use, chronic pain, and patients taken to reconstructive surgeries were excluded. The Jadad scale was used to evaluate quality. Results Six randomized controlled clinical trials (397 patients) that evaluated the analgesic efficacy of fentanyl (n = 2), nalbuphine (n = 1), ketamine (n = 1), gabapentin (n = 1), and lidocaine (n = 1) to treat post-procedural pain were included. Fentanyl, nalbuphine, and ketamine were effective, while lidocaine was associated with a slight increase in reported pain and gabapentin showed no significant differences. Two studies were of high quality, one was of medium high quality, and three were of low quality. No studies on the efficacy of NSAIDs or paracetamol were found. Conclusion Evidence of efficacy is very limited. Fentanyl, nalbuphine, and ketamine seem to be effective for controlling acute pain in burn patients, whereas gabapentin and lidocaine did not show any efficacy.

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Abstract Objective: To evaluate the effect of neuromodulatory drugs on the intensity of chronic pelvic pain (CPP) in women. Data sources: Searches were carried out in the PubMed, Cochrane Central, Embase, Lilacs, OpenGrey, and Clinical Trials databases. Selection of studies: The searches were carried out by two of the authors, not delimiting publication date or original language. The following descriptors were used: chronic pelvic pain in women OR endometriosis, associated with MESH/ENTREE/DeCS: gabapentinoids, gabapentin, amitriptyline, antidepressant, pregabalin, anticonvulsant, sertraline, duloxetine, nortriptyline, citalopram, imipramine, venlafaxine, neuromodulation drugs, acyclic pelvic pain, serotonin, noradrenaline reuptake inhibitors, and tricyclic antidepressants, with the Boolean operator OR. Case reports and systematic reviews were excluded. Data collection: The following data were extracted: author, year of publication, setting, type of study, sample size, intervention details, follow-up time, and results. Data synthesis: A total of 218 articles were found, with 79 being excluded because they were repeated, leaving 139 articles for analysis: 90 were excluded in the analysis of the titles, 37 after reading the abstract, and 4 after reading the articles in full, and 1 could not be found, therefore, leaving 7 articles that were included in the review. Conclusion: Most of the studies analyzed have shown pain improvement with the help of neuromodulators for chronic pain. However, no improvement was found in the study with the highest statistical power. There is still not enough evidence that neuromodulatory drugs reduce the intensity of pain in women with CPP.

Resumo Objetivo: Avaliar o efeito de drogas neuromoduladoras na intensidade da dor pélvica crônica em mulheres. Fontes de dados: As buscas foram realizadas nas bases de dados PubMed, Cochrane Central, Embase, Lilacs, OpenGrey e Clinical Trials. Seleção dos estudos: As buscas foram realizadas por dois dos autores, não delimitando data de publicação ou idioma de publicação. Foram usados os seguintes descritores: chronic pelvic pain in women OR endometriosis, associated with MESH/ENTREE/DeCS: gabapentinoids, gabapentin, amitriptyline, antidepressant, pregabalin, anticonvulsant, sertraline, duloxetine, nortriptyline, citalopram, imipramine, venlafaxine, neuromodulation drugs, acyclic pelvic pain, serotonin, noradrenaline reuptake inhibitors e tricyclic antidepressants, com o operador booleano OR. Relatos de caso e revisões sistemáticas foram excluídos. Coleta de dados: Foram extraídos os seguintes dados: autor, ano de publicação, local de origem, tipo de estudo, tamanho da amostra, detalhes da intervenção, tempo de seguimento e resultados. Síntese dos dados: Foram encontrados 218 artigos, sendo 79 deles excluídos por serem repetidos, restando 139 artigos para análise, dos quais 90 foram excluídos na análise dos títulos, 37 após a leitura do resumo e 4 após a leitura dos artigos na íntegra, e 1 não foi encontrado, restando, então, 7 artigos que foram incluídos na revisão. Conclusão: A maioria dos estudos analisados mostrou melhora da dor crônica com auxílio de neuromoduladores. No entanto, nenhuma melhora foi encontrada no artigo com maior poder estatístico. Ainda não há evidências suficientes de que drogas neuromoduladoras reduzam a intensidade da dor pélvica crônica em mulheres.

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