This case report describes a man who self-diagnosed rapid-eye-movement (REM) sleep behavior disorder using a sleep-tracking wearable device, later confirmed by a sleep evaluation.
Abstract Introduction Dopamine agonists such as pramipexole/ropinirole are treatments for restless legs syndrome (RLS) and Parkinson’s disease. It is well established that these medications can cause impulse control disorders (ICDs), side effects with serious and potentially life-altering consequences. ICDs are recurrent difficult to resist behaviors that can be harmful to the patient or others around them. Common examples include pathologic gambling, sexual behaviors, eating, and shopping. We present a patient with RLS who developed pathologic nocturnal sleep related shopping, without recall of the events, which resolved after stopping pramipexole. Report of case(s) A 58-year-old woman presented with a 28-year history of RLS, for which she was initially prescribed pramipexole. She denied a prior history of parasomnias or compulsive behaviors. After starting pramipexole, she reported episodes of pathologic nocturnal shopping, without recollection of the events. For example, she would awaken each morning to discover orders placed online for various items, but did not remember doing so. Unnecessary items such as kitchen knives, T-shirts, and vacuum parts would later arrive at her home. These items were also ordered in exceptionally high quantities, such as 60 kitchen knives. The time-stamped purchases took place on average 1 hour after sleep onset. During the daytime she took protective and compensatory measures such as removing credit cards from common retail websites and returning items. However, during each event she navigated around the safeguards placed, such as purchasing personalized items that could not be returned. She reported feeling ashamed of what was happening but was too embarrassed to discuss this with her doctor. Eventually, the nocturnal shopping caused strain on her marriage and drove her into debt. After hearing stories of impulsive behavior from other patients on pramipexole, she disclosed her symptoms to her doctor. After switching from pramipexole to ropinirole, her nocturnal shopping behavior ended. Conclusion This is the first case of pramipexole induced sleep-related ICD, with severe negative impact. The symptomatology can be so unusual that patients might be reluctant to discuss with their prescribing doctor. When prescribing dopamine agonists, providers should counsel patients on the symptoms of ICD, including manifestations during wakefulness and sleep. Support (if any)
Abstract Introduction Rapid eye movement (REM) sleep behavior disorder (RBD) is a parasomnia strongly associated with α-synucleinopathies, most commonly Parkinson’s disease (PD). Among Veterans, posttraumatic stress disorder (PTSD) is common and may influence RBD presentation and outcomes. The extent to which PTSD modifies demographic, medical, and mortality profiles in Veterans with RBD, and how these differ from those with RBD and Parkinson’s disease (PD), remains poorly characterized. Methods We conducted a retrospective cohort study using the Veterans Health Administration Corporate Data Warehouse. Veterans with at least one ICD-9 or ICD-10 diagnostic code for RBD were identified between 1999 - 2020 and categorized into three, mutually-exclusive groups based on the presence or absence of co-occurring ICD codes: RBD-only, RBD+PTSD, or RBD+PD. Demographics, comorbidities (Charlson Comorbidity Index [CCI]), and mortality outcomes were compared using logistic regression models adjusted for age, sex, race/ethnicity, BMI, and CCI. Results The cohort included 11,283 Veterans with RBD only, 17,527 with RBD+PTSD, and 5,675 with RBD+PD. Veterans with RBD+PTSD were younger (mean 58.9 ± 14.9 y) and had higher psychiatric comorbidity (94.2%) compared to RBD only (54.2%) or RBD+PD (46.5%). Those with RBD+PD were older (73.6 ± 8.1 y), predominantly male (98.9%), and had the highest neurological comorbidity (92.6%). Unadjusted mortality was lowest in RBD+PTSD (15.3%) and highest in RBD+PD (53.6%). Adjusted models showed reduced mortality risk for RBD+PTSD (aOR 0.73, 95% CI 0.70–0.80, p < 0.001) and increased risk for RBD+PD (aOR 2.43, 95% CI 2.3–2.6, p < 0.001) vs. RBD alone. Higher age, male sex, and greater comorbidity independently predicted mortality. Conclusion In this large national VA cohort, comorbid PTSD and Parkinson’s disease were associated with distinct demographic and clinical profiles among Veterans with RBD. RBD+PTSD was linked to younger age, greater psychiatric burden, and lower mortality. These findings underscore the heterogeneity of RBD in Veterans and support stratified approaches to surveillance for synucleinopathy and trauma-related sleep disturbances. Support (if any) VA CDA # IK2CX002363-01A1 (MJ); NHLBI K25 #1K25HL152006-01 (JR)
Rapid eye movement sleep behavior disorder (RBD), a prodromal synucleinopathy that precedes Parkinson’s disease and related dementias, can present with subtle cognitive difficulties. However, limited data on longitudinal cognitive change in individuals with RBD has been presented. The present analysis aimed to quantify cognitive change across approximately one year in a cohort of individuals with polysomnogram-confirmed RBD (n=60 from the North American Prodromal Synucleinopathy Consortium), using previously validated standardized regression-based change scores developed in cognitively unimpaired participants from the National Alzheimer's Coordinating Center. In the entire sample, the mean change scores across the 13 cognitive scores ranged from -.37 to .04 compared to demographically matched peers. One-sample two-tailed t-tests revealed a significant decline on 1 of the 13 cognitive scores, category fluency for vegetables (p=.01), with small to medium effect size (d=.34). Although not statistically significant, subtle declines were also present on measures of attention (Number Backward, p= .06, d=.21), memory (Craft Story Delayed Recall, p=.18, d=.17), processing speed (Trail Making A, p=.22, d=.16), and visuospatial skills (Benson Copy, p=.23, d=.16). Overall, during this prodromal period, individuals with RBD appear to remain largely cognitively stable over approximately one year. Nonetheless, continued tracking of these individuals may provide important prognostic information and/or endophenotyping of patients. These results also have potential implications for clinical management and the design of clinical trials in RBD.
We developed a database of free-text descriptions of dream enactment behaviours (DEBs) from unstructured clinical notes of United States Veterans with computationally phenotyped diagnoses of rapid eye movement sleep behaviour disorder (RBD). We additionally examined characteristics of DEBs considered essential for RBD diagnosis according to expert consensus. Free-text DEB descriptions were manually extracted from the clinical records of 200 randomly sampled Veterans; RBD computational diagnoses were defined as ≥2 ICD codes for RBD (>30 days but <390 days apart) and procedural codes for in-lab polysomnography within 6 months. DEB descriptions were categorized by clinical features and labeled as essential or not through expert consensus. The proportions of essential versus non-essential DEB types were compared with Chi-Square of Fisher's exact tests. Out of 195 unique DEB descriptions, experts labeled 124 (63.6 %) as essential RBD symptomatology. These included variations of "dream enactment behaviour" (e.g., "acted out dreams"); hitting, punching, or kicking bedpartners, pets, and/or bedside objects; and other mentions of sleep-related violence. Non-essential descriptions mentioned sleep-talking, thrashing, vivid dreams, nightmares, and sleeping in separate beds. Descriptions of sleep-related movements (n = 119) were more likely labeled as essential (75.6 %) versus non-essential (24.4 %; p = 0.00). Potentially violent or injurious movements were more likely labeled as essential (91.4 %) versus non-essential (8.6 %; p = 0.00). Expert labeling emphasized the significance of sleep-related movements and potentially injurious behaviours for RBD diagnosis in free-text medical records. A natural language processing algorithm that detects DEBs from free-text could further characterize sleep-related violence and injury risks associated with DEBs according to demographics and comorbidities.
To elucidate whether awake handedness in sexsomnia is retained during sleep to uncover potential clues about the underlying neurophysiologic mechanisms. Participants’ and observers’ self-reported handedness during sexsomnia events. Case 1: A 22-year-old right-handed (RH) female with an 8-year history of nocturnal sleep-related masturbatory behavior (SMB) involving the left hand (LH) exclusively. Case 2: A 30-year-old ambidextrous male with SMB involving both hands, but most prominently the LH. Case 3: A 33-year-old RH female with exclusively LH SMB in the setting of acute multiple sclerosis exacerbation with cervical spinal cord and brainstem involvement. Case 4: A 44-year-old RH male with bilateral dream enactment behavior and LH-predominant SMB noted by the bed partner. Case 5: A 59-year-old RH female with a history of multiple system atrophy and rapid eye movement sleep behavior disorder developed a new SMB, noted by the bed partner involving exclusively the LH. Case 6: A 37-year-old RH male with inappropriate sexual behaviors involving the nondominant LH without retention of memory for the events. Case 7: A 17-year-old RH male with polysomnographic evidence of genital manipulation using both hands but predominantly involving the nondominant hand. We speculate that sexsomnias originate from central pattern generator in the brainstem and spinal cord, as opposed to the cerebral cortex. The lack of involvement of cerebral motor control is further substantiated by amnesia for sexsomnia events. The implications of these data provide fundamental new clues about the mechanism of sexsomnias with critical forensic implications. Avidan AY, Hasan R, Badami V, Schenck CH. Hand dominance shift during sleep in sexsomnia: a clue to pathophysiology? J Clin Sleep Med. 2025;21(5):937–941.
Rapid-eye-movement sleep behavior disorder (RBD) is a parasomnia causing motor behaviors and vocalizations during sleep, which can lead to injuries in patients and their bed partners. Adult-onset RBD generally precedes a neurodegenerative synucleinopathy, while other cases can be associated with antidepressant use, neurotrauma and narcolepsy. The management of RBD relies on the systematic identification of etiologic and contributing factors, implementation of safety measures, appropriate pharmacotherapy and counseling, which should be patient-centered. In this manuscript, we summarize the evidence on the management of RBD. We summarize the evidence supporting the use of clonazepam, melatonin, rivastigmine, and pramipexole, the four agents currently recommended by the American Academy of Sleep Medicine. For each agent and for alternative therapies, we discuss efficacy, dosing, adverse effects and indications. We integrate the current knowledge on therapies in RBD in treatment algorithms that can guide providers in choosing the most appropriate initial therapy, and alternative options based on the course of symptoms and comorbidities. There is a large need for additional, well tolerated therapies for reducing RBD symptoms. The last section of this manuscript discusses current challenges and unmet needs, as well as future directions in developing therapies and improving the care of patients with RBD.
Interictal epileptiform discharges (IEDs) increase during non-rapid eye movement (NREM) sleep, and decrease or disappear in REM sleep, especially during phasic REM sleep. REM sleep without atonia (RSWA), and its possible effects on IEDs, has not yet been studied. A retrospective review of 10-year data retrieved 205 adults with fronto-temporal epilepsy, with full clinical data, 18-channel EEG and polysomnography. Tonic and phasic REM sleep periods were analyzed, and REM atonia was scored with the latest criteria. EEG recordings and IEDs were also re-evaluated in NREM sleep from the first and second halves of the night, and during phasic/tonic REM, and RSWA periods. RSWA was detected in 31 patients (15.1
BackgroundREM sleep behavior disorder (RBD) is characterized by loss of normal muscle atonia during REM sleep, often associated with dream enactment behaviors, and is typically a prodromal neurodegenerative condition in middle-aged and older adults. However, emerging case reports and case series suggest that not all RBD presentations follow this trajectory, particularly in younger individuals.Case presentationA case of 7-year history of vivid, immersive dreaming perceived as continuous with waking life, accompanied by persistent dream-reality confusion, is described. The patient frequently engaged in reality-testing behaviors and reported significant cognitive fatigue. Video-polysomnography confirmed REM sleep without atonia and a concordant dream re-enactment episode. Neuropsychiatric evaluation ruled out dissociative or psychotic disorders, and no evidence of neurodegenerative disease was observed.ConclusionThis presented case illustrates a potentially distinct, non-neurodegenerative REM parasomnia phenotype that underscores the need to expand current parasomnia classifications to better capture the diverse cognitive and metacognitive dimensions of REM sleep disorders. Moreover, potential mechanisms underlying the main features of this case, including immersive dreaming and persistent dream-reality confusion, are discussed in relation to hypothesized dysfunction in melanin-concentrating hormone (MCH) signaling.
Few studies have surveyed the prevalence of sexsomnia and sexsomnic behaviors in general population samples. In the present study, 1002 respondents (508 males and 494 females), mean age 50.3 years (SD = 17.5), recruited from a Norwegian survey panel, participated in an online survey about sleep phenomena and sleep habits. The survey included questions about lifetime and current parasomnias, such as sleepwalking, sleep terrors, confusional arousals, and dream enactment, as well as sleep duration and sleep need. Questions about lifetime and current sexsomnia, various sexsomnic behaviors, and frequency of current sexsomnia episodes were included. Logistic regression analyses were conducted to identify predictors of lifetime and current sexsomnia. A total of 10.5
BACKGROUND AND OBJECTIVES:Idiopathic/isolated REM sleep behavior disorder (iRBD) has been strongly linked to neurodegenerative synucleinopathies such as Parkinson disease, dementia with Lewy bodies, and multiple system atrophy. However, there have been increasing reports of RBD as a presenting feature of serious and treatable autoimmune syndromes, particularly IGLON5. This study's objective was to investigate the frequency of autoantibodies in a large cohort of participants with iRBD. METHODS:Participants were enrolled in the North American Prodromal Synucleinopathy cohort with polysomnography-confirmed iRBD, free of parkinsonism and dementia. Plasma samples were systematically screened for the autoantibodies IGLON5, DPPX, LGI1, and CASPR2 using plasma IgG cell-based assay. Positive or equivocal results were confirmed by repeat testing, plus tissue-based indirect immunofluorescence assay for IGLON5. RESULTS:Of 339 samples analyzed, 3 participants (0.9%) had confirmed positive IGLON5 autoantibodies in the cell-based assay, which were confirmed by the tissue-based assay. An additional participant was positive for CASPR2 with low titer by cell-based assay only (of lower clinical certainty). These cases exhibited a variety of symptoms including dream enactment, cognitive decline, autonomic dysfunction, and motor symptoms. In 1 IGLON5 case and the CASPR2 case, evolution was suggestive of typical synucleinopathy, suggesting the possibility that findings were incidental. However, 2 participants with IGLON5 died before diagnosis was clinically suspected, with a final clinical picture highly suggestive of autoimmune disease. DISCUSSION:Our finding that nearly 1% of a large iRBD cohort may have a serious but potentially treatable autoantibody syndrome has important clinical implications. In particular, it raises the question of whether autoantibody testing for IGLON-5-IgG should be widely implemented for participants with iRBD, considering the difficulty in diagnosis of autoimmune diseases, their response to treatment, and the potential for rapid disease progression. However, any routine testing protocol will also have to consider costs and potential adverse effects of false-positive findings. TRIAL REGISTRATION INFORMATION:NCT03623672.
Sexual behavior during sleep, known as sexual parasomnias, has captured the interest of researchers and clinicians. These parasomnias involve various sexual activities that occur unconsciously during sleep. Although relatively rare, they can profoundly affect well-being and relationships and can carry legal consequences. Understanding their nature, prevalence, and causes is crucial for advancing knowledge in this field. This article revisits the topic of sexsomnia, presenting new data and discussing cases published from 2007 to 2023. By analyzing these cases, we aim to enhance recognition, diagnosis, and management of sexsomnia, reducing stigma and providing better support for affected individuals.
AIMS:Although clonazepam (CLO) and melatonin (MLT) are the most frequently used treatments for REM sleep behavior disorder, the polysomnographic features associated with their use are little known. The aim of this study was to evaluate polysomnographic and clinical parameters of patients with idiopathic/isolated REM sleep behavior disorder (iRBD) treated chronically with CLO, sustained-release MLT, alone or in combination, and in a group of drug-free iRBD patients. METHODS:A total of 96 patients were enrolled: 43 drug-free, 21 with CLO (0.5-2 mg), 20 with sustained-release MLT (1-4 mg), and 12 taking a combination of them (same doses). Clinical variables and polysomnography were collected. RESULTS:Although clinical improvement was reported in all groups, MLT impacted sleep architecture more than the other treatments, with significant and large increase in N3 stage, moderate reduction in N2 and REM sleep, and moderate increase in REM latency. CLO moderately increased the percentage of both REM sleep and especially N2, while reducing N1 and wakefulness. Patients treated with both CLO and MLT did not show major changes in sleep architecture. CONCLUSION:These results suggest that the administration of MLT or CLO impacts (positively) on sleep parameters of iRBD patients. However, there is a need to better stratify patients, in order to treat them in a targeted manner, depending on the patient's individual sleep architecture and expected differential effects of these agents.
We investigated the accuracy of International Classification of Diseases (ICD) codes for the identification of veterans with rapid eye movement sleep behavior disorder. The charts of 139 randomly sampled veterans with ≥ 1 ICD-9 and ICD-10 code(s) for rapid eye movement sleep behavior disorder were reviewed for documentation of a suspected, previous, or current diagnosis; clinical symptoms; and/or empiric treatments for this disorder. Notably, 71 (51.1
Abstract Introduction As a first step toward phenotyping large numbers of Veterans with rapid eye movement (REM) sleep behavior disorder (RBD), we investigated the ability of ICD-9 (327.42) and ICD-10 (G47.52) codes to detect Veterans with this clinical diagnosis. Methods Subjects were 150 randomly selected Veterans with relevant RBD ICD codes for at least 2 outpatient encounters or 1 inpatient discharge (+RBDcode). Notes from the date of the first ICD diagnosis were reviewed for any documentation of RBD, dream enactment behaviors (DEBs), and targeted pharmacotherapy. The nearest diagnostic, split night, or titration polysomnogram (PSG) report was also reviewed. To calculate sensitivity, specificity, positive predictive value (PPV), and negative predictive value (NPV) for probable RBD, a comparison group of 50 Veterans with at least 2 outpatient visits or 1 inpatient discharge summary with ICD-9/ICD-10 codes for OSA and no ICD codes for RBD (-RBDcode) were propensity-matched for age, sex, gender, race, ethnicity, and month-year of first diagnosis. Results The final sample consisted of 139 Veterans in +RBDcode (age 64.8±15.2, 94.6% male, 69.8% white, 10% Hispanic) and 47 Veterans in -RBDcode (age 69.6±2.7, 95.8% female, 77.1% white, 4.3% Hispanic) after excluding subjects with unavailable or restricted charts. In +RBDcode, a diagnosis of RBD, overt DEBs, and targeted pharmacotherapy were documented in 130 (93.5%), 103 (74.1%), and 71 (51.1%) of subjects, respectively. No PSGs were documented in 71 (51.5%), reports did not comment on REM sleep without atonia (RSWA) in 29 (20.9%), and REM did not occur in 8 (5.8%) subjects. Only 13 (9.3%) had documented DEBs and a PSG with RSWA. No subjects in -RBDcode had documentation of pRBD. The sensitivity, specificity, PPV, and NPV for pRBD was 100%, 83.9%, 93.5%, and 100%, respectively. Conclusion ICD-9 and ICD-10 codes accurately predicted the presence (or absence) of pRBD compared to manual chart review. Next steps are to phenotype Veterans with RBD diagnoses according to comorbidities and outcomes on a nation-wide scale. Support (if any) VA CSR&D CDA #IK2CX002363-01A1 [MJ]; NIH-NHLBI-K25 (#1K25HL152006-01) [JR]; seed funding by AHBPCE (#FY2024-002) [JR]; Center for Innovations in Quality, Effectiveness, and Safety (CIN 13-413), Michael E. DeBakey VA Medical Center, Houston, TX.
A novel form of injury associated with obstructive sleep apnea (OSA) that was comorbid with obesity hypoventilation syndrome and severe daytime somnolence is reported in a 55-year-old woman, manifesting as severe ocular and extraocular muscle injuries sustained from suddenly falling asleep and colliding with a sharp object, resulting in surgical enucleation of the right eye and orbital implant. The literature on injuries (falls, motor vehicle accidents) related to OSA and excessive day time sleepiness is reviewed, along with the literature on injuries from OSA-related parasomnias. The diverse health hazards, including physical injury, associated with OSA-excessive daytime sleepiness, are emphasized, further encouraging the need to educate primary care providers on early detection of OSA with prompt treatment intervention. Baker N, Schenck CH, Golden E, Varghese R. A case of accidental self-enucleation caused by obstructive sleep apnea. J Clin Sleep Med. 2024;20(8):1395–1397.