BackgroundInteroception refers to the sensing, interpretation, integration, and regulation of signals about the body’s internal physiological state. Interoceptive sensibility is the subjective evaluation of interoceptive experience, as assessed by self-report measures, and is abnormal in numerous neuropsychiatric disorders. Research examining interoceptive sensibility in individuals with chronic tic disorders (CTDs), however, has yielded conflicting results, likely due to methodologic differences between studies and small sample sizes.ObjectiveWe sought to compare interoceptive sensibility between adults with CTD and healthy controls, adjusting for co-occurring psychiatric symptoms, and to examine the relationship of interoceptive sensibility with other CTD clinical features, in particular, premonitory urge.MethodsWe recruited adults with CTDs and sex- and age-matched healthy controls to complete the Multidimensional Assessment of Interoceptive Awareness, Version 2 (MAIA-2), as well as a battery of measures assessing psychiatric symptoms prevalent in CTD populations. CTD participants additionally completed scales quantifying tic severity, premonitory urge severity, and health-related quality of life. We conducted between-group contrasts (Wilcoxon rank-sum test) for each MAIA-2 subscale, analyzed the effect of psychiatric symptoms on identified between-group differences (multivariable linear regression), and examined within-group relationships between MAIA-2 subscales and other clinical measures (Spearman rank correlations, multivariable linear regression).ResultsBetween adults with CTD (n = 48) and healthy controls (n = 48), MAIA-2 Noticing and Not-Worrying subscale scores significantly differed. After adjusting for covariates, lower MAIA-2 Not-Worrying subscale scores were significantly associated with female sex (β = 0.42, p < 0.05) and greater severity of obsessive-compulsive symptoms (β = –0.028, p < 0.01), but not with CTD diagnosis. After adjusting for severity of tics and obsessive-compulsive symptoms, a composite of MAIA-2 Noticing, Attention Regulation, Emotional Awareness, Self-Regulation, Body Listening, and Trusting subscales (β = 2.52, p < 0.01) was significantly associated with premonitory urge.ConclusionStudy results revealed three novel findings: adults with CTD experience increased anxiety-associated somatization and increased general body awareness relative to healthy controls; anxiety-associated somatization is more closely associated with sex and obsessive-compulsive symptoms than with CTD diagnosis; and increased general body awareness is associated with greater severity of premonitory urges.
OBJECTIVE Down syndrome (DS) affects 1 in 700 live births and approximately one-third of patients develop craniovertebral junction (CVJ) instability, diagnosed by clinical examination and radiological measures such as the atlantodens interval (ADI) and space available for the cord (SAC). Patients with symptomatic CVJ instability are at increased risk for spinal cord injury. There are no guidelines for surgical management of CVJ instability in DS, the existing literature is sparse, and there is a lack of consistent pediatric data. This systematic review aimed to synthesize practice patterns of the surgical management of CVJ stability in pediatric DS patients to facilitate future standardization of care. METHODS Peer-reviewed studies reporting surgical management of CVJ instability in pediatric DS patients were systematically reviewed. Inclusion criteria were studies reporting primary data on patients younger than 18 years with DS, who had CVJ instability evaluation and underwent surgical treatment. Bias risk was assessed. Descriptive statistics of the independent patient data were presented. Interval variables were analyzed using the Wilcoxon rank-sum test. RESULTS Of 1056 records, 38 studies were included. Of the included patients, 169 (6%) underwent surgery. The surgical indication was symptomatic, radiologically confirmed CVJ instability in 81% of the patients, presenting with myelopathy (30%), weakness (25%), abnormal gait (24%), torticollis (15%), and neck pain (14%). A cutoff of ADI ≥ 4 mm or SAC ≤ 14 mm, cord compression, cord signal change, and anomalous bony anatomy were used in diagnosing CVJ instability. Surgical approaches focused on internal fixation with posterior occipitocervical or atlantoaxial instrumented fusion in 57% and 44% of patients, respectively. Autograft, wiring, and allograft constructs were used in 48%, 45%, and 9% of patients. Anterior cervical approaches were performed in 6% of patients. Preoperative and postoperative external orthoses were used in approximately 50% of patients. The surgical mortality rate was 3%, and the complication rate was 36%. CONCLUSIONS Assessment of CVJ instability in DS is based on radiographic and clinical factors. Surgery is recommended if symptoms are present, and the procedure type depends on patient factors, degree of instability, anomalous bony anatomy, and reduction results to relieve cord compression. Most commonly, posterior instrumented fusion is used. However, further research is required to determine the strength of evaluation methods, create standardized guidelines for evaluation and surgical treatment, and investigate the long-term results of different surgical techniques.
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