Social interaction involves coordination between individuals across multiple domains, including neural activity, behaviour and physiology. An increasing number of studies shows that interpersonal coordination and synchrony can be detected in each of these modalities, but the functional significance of these different types of coordination remains unclear. This paper examines the challenge of understanding coordination in terms of the timescales of interaction. We provide a novel mapping of interaction types and measurement methods across timescales spanning from milliseconds to minutes. We review candidate mechanisms of synchrony and coordination in both neural systems and behaviour, and consider how these measures relate to real world outcomes such as learning and development. Overall, we provide an integrative approach that, by taking timescales into account, aims to offer a better understanding of the origins and interpretation of interpersonal coordination.
A growing body of literature has illustrated the importance of understanding the circadian clock due to its connection to a wide array of molecular and physiological processes. Numerous methods have been developed in order to monitor the status of the circadian clock in living tissue; however, most methods are either costly or labor intensive, and often require precise conditions that lowers throughput and limits flexibility in the size, age, or type of plant to be assayed. Here, we present an affordable and adaptable methodology for assaying circadian period using the well-characterized circadian output of leaf movement. Employing fully automated time-lapse photography using Raspberry Pi cameras and predominantly automated image post-processing, this methodology minimizes manual input to expedite circadian analysis, thus improving throughput. Additionally, the top-down setup used in this method is appropriate for a wide range of sizes and ages of plants, allowing for an expansion of the scientific questions that can be assayed by this methodology.
Circadian rhythms orchestrate metabolism and brain function, aligning internal physiological processes with the 24-hour day-night cycle. Growing evidence highlights a reciprocal relationship between circadian regulation, metabolism, and neurobiological processes. Circadian disruption impairs glucose and lipid homeostasis, alters neurotransmitter and endocrine signalling, and triggers stress response, forming a feedback loop that impacts metabolism and brain function. These disturbances are implicated in many conditions, such as obesity, diabetes, depression, and bipolar disorder. This review examines recent advances in the interplay between circadian regulation, metabolism, and mental health, emphasising shared molecular mechanisms and their role in disease progression. Understanding these connections may ultimately inform therapeutic strategies that integrate circadian-based approaches to improve treatments for metabolic and psychiatric disorders.
Surgery and postoperative complications disrupt circadian rhythms. While clinicians recognize the implicit value of circadian rhythms to evaluate postoperative recovery, the absence of a practical means to measure them has limited their exploration and integration into clinical practice in children. Consumer wearable device (Fitbit) was given to children 3-18 years old who underwent laparoscopic appendectomy for complicated appendicitis and data were collected during postoperative days (POD) 1 to 21. Three novel rest-activity rhythmicity (RAR) metrics were computed from minute-by-minute continuous Fitbit data: (1) Periodicity and (2) Amplitude of the 24-hour Activity Rhythm; and (3) Circadian quotient of the 24-hour heart rate (HR) Rhythm. RAR trajectories were aggregated for patients without postoperative complications and compared against trajectories for patients with complications. Ninety-four patients were included in the analysis (n=13 [14%] with complications). For patients without complications, three RAR metrics gradually increased in intensity until reaching plateaus between POD 11-20 (Periodicity of the 24-hour Activity Rhythm, 95% confidence interval [CI]: POD 9-14; Amplitude of the 24-hour Activity Rhythm, 95% CI: POD 16-24; Circadian quotient of the 24-hour HR Rhythm, 95% CI: POD 9-15). Although the RAR metrics for children with complications increased in intensity, none plateaued during the 21-day study period. Wearable-derived circadian rhythms demonstrated distinct patterns in RAR metrics for children with and without complications during the 21-day study period following appendectomy. This suggests that the RAR may be relevant digital biomarkers to track postoperative recovery and identify complications in pediatric populations.
Liver metabolism is under tight control of the circadian system. Disruption of key clock gene expression (desynchronosis) leads to the misalignment of metabolic pathways. However, the relationship between circadian dysregulation and hepatic protein-synthetic function, as well as its sexual dimorphism, remains poorly understood. To evaluate the effect of chronic photoperiod disruption on hepatic protein-synthetic function (total protein, albumin) and to establish its relationship with the expression of key circadian proteins (BMAL1, CLOCK, PER2) in male and female rats, as well as to assess the efficacy of exogenous melatonin in correcting the identified disturbances. The study was performed on 240 adult Wistar rats (120 males, 120 females). Animals were divided into 3 groups: control (light: dark 10:14 h), dark deprivation (LL, constant light for 21 days), and LL + melatonin (12 mg/L drinking water). Plasma levels of total protein and albumin were measured. Immunohistochemistry was used to assess the percentage of positively stained hepatocytes for BMAL1, CLOCK, and PER2. Statistical analysis included two-way ANOVA, Pearson correlation analysis, ANCOVA, and ROC analysis. Dark deprivation reduced albumin levels by 15.7% in males and by 15.9% in females compared to controls. Two-way ANOVA revealed significant effects of "lighting conditions" (F = 145.3, p < 0.0001), "sex" (F = 18.7, p < 0.01), and their interaction (F = 7.2, p < 0.05). BMAL1 and CLOCK expression decreased by more than 70% in both sexes, whereas PER2 expression paradoxically increased by 28.9-35.0%. Strong correlations were found between albumin levels and expression of BMAL1 (r = 0.79-0.81, p < 0.001), CLOCK (r = 0.69-0.74, p < 0.001), and PER2 (r= - 0.68 to - 0.71, p < 0.001). ANCOVA (R²=0.71, p < 0.0001) identified BMAL1 expression as the most significant independent predictor of albumin levels (β = 0.52, p < 0.0001), with sex retaining independent significance (p = 0.02). ROC analysis demonstrated high predictive performance of BMAL1 expression for hypoalbuminemia (AUC = 0.87-0.89, p < 0.0001). Melatonin treatment fully restored the expression of all examined circadian proteins and normalized protein synthetic parameters to control levels in both sexes. Chronic photoperiod disruption induces profound hepatic desynchronosis characterized by suppression of BMAL1/CLOCK and accumulation of PER2, which is associated with decreased protein synthetic function. A pronounced sexual dimorphism in susceptibility to desynchronosis was identified. BMAL1 expression is a highly informative predictor of hypoalbuminemia. Exogenous melatonin fully restores the impaired parameters, supporting its use as an effective chronobiotic. The liver works on a daily (circadian) rhythm. When this rhythm is disrupted—for example, by constant light exposure—it can harm liver function. However, scientists did not know exactly how rhythm disruption affects the liver’s ability to make proteins (like albumin), or whether males and females respond differently. This study tested whether constant light harms liver protein production, and whether the sleep hormone melatonin could fix it. They used 240 adult rats (120 males, 120 females). The rats were split into three groups:1. Normal light-dark cycle (control).2. Constant light for 21 days (to disrupt their body clock).3. Constant light plus melatonin in their drinking water.They measured blood levels of total protein and albumin (a key protein made by the liver). They also measured three key clock proteins (BMAL1, CLOCK, PER2) in the liver cells. Constant light lowered albumin levels by about 16% in both male and female rats. It also caused major changes in the liver’s clock proteins: BMAL1 and CLOCK dropped by over 70%, while PER2 increased. These changes were strongly linked to lower albumin levels. The study also found that sex mattered—males and females responded differently to rhythm disruption. Using a statistical model, BMAL1 was the best predictor of low albumin. Importantly, melatonin treatment fully restored all clock proteins and normalised protein production in both sexes. Disrupting the body’s daily rhythms harms the liver’s ability to make essential proteins. The clock protein BMAL1 may serve as a useful marker for liver problems. Melatonin—a natural hormone already used for sleep disorders—could potentially help protect liver function when circadian rhythms are disturbed (e.g., in shift workers, people with chronic jet lag, or those living under abnormal light cycles).
This paper examines the temporal organization of industrial dairy production in China. Based on ethnographic fieldwork on a small-scale farm in Hebei Province, it showcases how a seemingly seamless 24/7 milk production cycle is sustained through the coordination of multiple, divergent temporalities. These include reproductive rhythms shaped by both bovine biology and market dynamics, the processual temporality of care and repair, and the agricultural temporality rooted in the 24 solar terms that structure traditional farming cycles in China. Engaging with theoretical work on temporal multiplicity and coordination, this paper highlights the costs of sustaining industrial production time, borne disproportionately by cows, human workers, ecologies, and small farms. By making visible the embodied and more-than-human labor required to sustain industrial time, this paper contributes to current discussions to rethink modernity through time. It offers an empirically grounded account of how divergent rhythms are held together through friction, improvisation, and care in industrial agriculture, calling for a temporal ethic grounded in responsiveness, care, and relational coordination.
Postoperative complete heart block (CHB) constitutes a complication of congenital heart surgery, requiring a permanent pacemaker (PPM). This study aimed to determine the incidence of post-discharge recovery from CHB following PPM and the incidence of recurrent CHB after initial recovery. The cumulative incidences of recovery from CHB and post-discharge recurrent CHB after 9,892 congenital cardiac operations between 2001 and 2024 were analyzed. A Cox regression model was used to identify predictors of recovery from CHB and recurrent CHB. Of 327 patients with CHB, 193 subjects (59.0%) required a PPM within the same hospital stay, and 134 patients (41.0%) were discharged without a PPM due to complete recovery. Post-discharge recovery of atrioventricular conduction was observed in 32/193 (16.6%) patients with a PPM (median period of 3.8 (0.3-6.3) years). The type of recovery included 22 sinus rhythms, 8 first-degree, and 2 second-degree atrioventricular blocks. Post-discharge recurrent CHB requiring PPM was observed in 11/134 (8.2%) patients with a median period of 0.6 (0.2-4.7) years. The cumulative incidence of recovery and late PPM implantation at 10 years was 24.2% and 9.5%, respectively. Neonatal operation (hazard ratio (HR): 4.491, p=0.018), longer recovery time with an 8-day threshold (HR: 1.107, p=0.022), and catheter-intervention (HR: 4.587, p=0.025) were risk factors for post-discharge recurrent CHB. Our findings support the current class 1 guidelines for pacemaker implantation after 7 days of CHB. Post-discharge recurrent CHB is difficult to predict in individual patients and can present many years post-operatively, supporting life-long follow-up after congenital heart surgery.
Post-cardiac arrest (CA) prognostication primarily focuses on predicting poor outcome, whereas early markers of good prognosis remain less explored. We aimed to identify early predictors of favorable outcome at 3 months in comatose CA survivors. We prospectively enrolled adult patients with post-anoxic coma admitted to the Intensive Care Unit of a secondary-care hospital (2020-2025). Demographic, CA-related, clinical, electroencephalographic (EEG), neuron-specific enolase (NSE) and neuroimaging data were collected. Good outcome was defined as Cerebral Performance Category (CPC) 1-2 at 3 months. Logistic regression analyses identified independent predictors of good outcome. Model performance was internally validated with bootstrap resampling. Among 121 included patients (median age = 66; 72% male), outcome was available for 118, of whom 42.3% achieved good outcome. Patients with CPC 1-2 were younger, had predominant shockable initial rhythms and cardiac etiology of CA, higher Glasgow Coma Scale scores, lower NSE levels, and decreasing NSE trend. Peak NSE ≤ 36 ng/mL was the optimal cut-off for favorable prognosis. Early benign EEG (continuous/nearly continuous background with preserved reactivity ≤72 h) was associated with neurological recovery. In multivariable analysis, independent predictors of favorable outcome were younger age (OR 0.92, 95% CI 0.86-0.99), shockable initial rhythm (OR 28.0, 95% CI 3.87-202.1), early benign EEG (OR 10.76, 95% CI 1.95-59.4) and peak NSE ≤ 36 ng/mL (OR 43.17, 95% CI 6.01-309.9). The model showed excellent discrimination (apparent AUC 0.97, optimism-corrected AUC 0.96). In comatose post-CA patients, a model combining age, initial rhythm, early benign EEG and peak NSE independently predicted good outcome at 3 months.
Light is a major environmental factor regulating circadian rhythms, sleep- wake cycles, and mood-related behaviors. Patients with Myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) often experience circadian disruption and poor sleep quality, which severely compromise their quality of life; however, the relationship between light exposure and illness severity remains largely unknown. An observational cross-sectional cohort secondary study used collected data from 100 ME/CFS patients and 56 healthy controls to explore the impact of spontaneous light exposure on multidimensional health status and circulating biochemical parameters. Demographic and clinical features were assessed using validated patient-reported outcome measures. Light intensity, wrist temperature, and physical activity were continuously monitored at home over one week using wrist-worn actigraphy. Light intensity during predefined intervals and rhythmic variables of light cycle were calculated. Principal component analysis (PCA) was applied to reduce dimensionality of light variables. Multivariable analysis was performed adjusting for age, sex, body mass index, and physical activity. Following PCA of the light patterns, two components emerged across groups with high consistency: PC1 (explaining 61.7% of the total variance) reflected higher daytime light and rhythm stability, and PC2 (explaining 16.1%) represented nocturnal/early-morning light and rhythm instability. In ME/CFS patients, light variables were more extensively associated with clinical outcomes measures (FIS-40, PSQI and SF-36) than in healthy controls (all p < 0.05). Furthermore, PC2 was associated with higher levels of VCAM-1 and triglycerides, and lower serotonin concentrations (all p < 0.05). Four distinct light patterns were identified based on PCA scores: nocturnal light, healthy, adverse, and low diurnal light. ME/CFS patients exhibiting the healthy light pattern showed significantly lower fatigue, fewer sleep complaints, reduced autonomic dysfunction, and higher quality of life compared to those with the adverse light pattern (all p < 0.05). No significant differences were observed among healthy controls. Light exposure patterns show distinct associations with symptom variability in ME/CFS compared to healthy controls. More stable daytime light appears to relate to better symptom profiles, whereas irregular exposure and nocturnal light are linked to poorer health outcomes. Although causality cannot be inferred, these findings highlight light exposure as a potentially modifiable, non-invasive target for behavioral interventions aimed at improving the quality of life in ME/CFS, representing a promising emerging for future translational research.
As the scale of online learning expanded, students tended to exhibit declining learning behaviors and accumulating task backlogs during self-directed study. These issues further led to reduced learning motivation and increased psychological pressure. To address this, this study constructed an innovative model that integrated a knowledge graph with multi-agent reinforcement learning. The model enabled learning state risk identification and personalized intervention. The study conducted a systematic evaluation using comprehensive learning behavior data from seven selected courses. The results indicated that the model achieved a high level of risk identification at an early stage. The recall values for all courses ranged from 0.879 to 0.896. As the learning process progressed, accuracy steadily increased to above 0.889. The F1-score remained between 0.842 and 0.871 across all stages, which demonstrated strong stability. Furthermore, the intervention strategies significantly improved learning trajectories across two experimental semesters. Students' learning activities showed continuous improvement over time. Behavioral fluctuations and breakpoint frequency were both markedly reduced. These findings confirmed that the model consistently enhanced learning motivation, stabilized learning rhythms, and optimized patterns of resource utilization.
Melatonin (MT) is a natural indoleamine compound. In addition to regulating sleep and circadian rhythms, MT exhibits antioxidative, anti-inflammatory, anti-apoptotic, and ovarian protective properties. Recent studies have shown that MT alleviates polycystic ovary syndrome (PCOS), premature ovarian insufficiency (POI), ovarian injury, and age-related ovarian function decline. This review establishes an integrated mechanistic model in which oxidative stress, mitochondrial dysfunction, inflammatory responses, and autophagic imbalance collectively contribute to the pathogenesis of ovarian disorders, while melatonin acts as a pleiotropic regulator that counteracts these interconnected pathological pathways. Furthermore, this review also discusses recent advances regarding the inhibitory effects of melatonin on ovarian tumors. Although MT shows considerable clinical promise, further research is needed to determine its optimal dosage, administration regimens, and therapeutic targets to facilitate its clinical translation.
A 58-year-old man presented with out-of-hospital cardiac arrest. The initial rhythm was pulseless electrical activity, which deteriorated into ventricular fibrillation during resuscitation. After return of spontaneous circulation, electrocardiography demonstrated diffuse ST-segment depression with ST-segment elevation in lead aVR, strongly suggesting acute coronary syndrome. Emergent coronary angiography revealed simultaneous angiographic occlusion with TIMI 0 flow in the ostial left anterior descending artery and distal right coronary artery. During preparation for percutaneous coronary intervention, coronary flow spontaneously recovered to TIMI 3 without balloon dilation, stenting, or intracoronary vasodilator administration. Intravascular ultrasound demonstrated no plaque rupture or thrombus. Persistent coma prompted neuroimaging, which revealed diffuse subarachnoid hemorrhage with hydrocephalus. Although cardiac arrest associated with subarachnoid hemorrhage usually presents with non-shockable rhythms, this case demonstrated recurrent ventricular fibrillation and transient multivessel coronary occlusion closely mimicking acute myocardial infarction.
Animals in temperate zones have evolved sophisticated mechanisms to synchronize their physiology and behavior with the seasons. However, the molecular mechanisms underlying these seasonal adaptations have long remained poorly understood. Here, we highlight medaka (Oryzias latipes) as a powerful vertebrate model for studying seasonal adaptation, owing to its robust and experimentally tractable seasonal responses. Recent studies using medaka have uncovered the molecular mechanisms underlying several of these phenomena, such as seasonal reproduction, seasonal changes in feeding behavior, seasonal changes in color perception, stress-related defensive behavior, winter depression-like behavior, gut-length plasticity, and photoperiod-dependent metabolic reprogramming. Furthermore, medaka has enabled the first molecular-level investigation of the circannual clock, providing evidence for a tissue remodeling-based mechanism that drives endogenous annual rhythms. Together, these findings establish medaka as an invaluable model organism for deciphering the molecular architecture of seasonal adaptation in vertebrates.
Neurodivergent neurotypes refer to brains that function differently from what is considered the societal standard or "stereotypical" norm, and include autism and attention deficit hyperactivity disorder (ADHD). Currently, global estimates suggest over 15% to 20% of the global population is neurodivergent. Autistic people and those with ADHD often experience mental health problems and have a heightened likelihood of anxiety and depression. Epidemiological evidence shows that sleep and circadian rhythm disruption, such as low self-reported sleep quality, insomnia symptoms, and delayed sleep-wake phase disorder, are common in autistic people and in those with ADHD. Despite scientific advancements, a comprehensive framework integrating sleep and circadian factors associated with mental health problems in neurodivergent neurotypes remains currently undeveloped. This perspective, written by people with lived experience of neurodivergent neurotypes, examines mental health problems, particularly anxiety, depression, and suicide risk, in autistic people and those with ADHD; sleep and circadian rhythm disruption in these neurotypes; and the growing epidemiological evidence linking sleep, circadian, and mental health problems in these neurotypes compared to people with neurotypical development. Our perspective advocates for an integrated model that accounts for the interplay between sleep, circadian rhythms, and mental health in neurodivergent neurotypes. Moreover, it identifies gaps and future research avenues, and the need for personalized behavioural interventions to improve their sleep, circadian, and mental health problems. Co-production of research is advocated as a method to ensure that insights from neurodivergent neurotypes are included as equal partners in the entire research process, to best support their sleep, circadian, and mental health.
In this Perspective, we suggest combining physiological, neurobiological, behavioural, and social data from multiple marine mammal species to create evolutionary models of the emergence of social vocal coordination. Marine mammal sound production mechanisms have evolved to manage vocalizing and breathing in an aquatic environment, potentially releasing functional constraints on the control of vocal timing. Intervals in rhythmic cetacean vocalizations cover a remarkable temporal range, from less than a millisecond in porpoise burst pulses to 10 s in sperm whale slow clicks. Many cetaceans demonstrate temporally coordinated social behaviour, while pinnipeds express high variability in vocal plasticity and social behaviour. The systematic variability of vocal production mechanisms, vocal rhythms, and sociality can be compared phylogenetically to generate models for the evolution of social rhythm in the vocal domain.
When sampling odors, humans typically take a single long sniff, unlike most other mammals who sample odors through bouts of rapid repetitive sniffing. Decades of work has established that rapid sniffing rhythms underlie the organizational principles of odor coding, with sniff speed clocking odor responses in the olfactory bulb. In the absence of rapid sniffing, how are odor responses organized in the human olfactory system? Because most mammals sniff at rates centered around the theta frequency, we hypothesized that the olfactory bulb exploits a theta-range neural oscillation to set the pace of odor coding in the system. Here, we used high-precision neural recordings from the human olfactory bulb to show that initiation of a sniff elicits and temporally aligns theta oscillations in the bulb and that sniff-induced theta oscillations organize the timing and amplitude of odor responses. These findings suggest that, despite the lack of rapid sniffing bouts in humans, the system has preserved a similarly timed unit of olfactory processing.
Studies on the arrhythmia incidence in the Fontan population, especially comparing intracardiac (IC) and extracardiac (EC) Fontan, are limited, with inconsistent findings. This meta-analysis aims to compare arrhythmia risk in EC and IC Fontan patients. Eligible studies comparing arrhythmia outcomes following different Fontan procedures, including EC conduit (ECC), lateral tunnel (LT), and classic Fontan, were identified through a systematic search of the PubMed, Cochrane Library, EMBASE, and Google Scholar databases from 2016 to 2025. Random-effects meta-analysis was performed to pool odds ratios (ORs). A subgroup analysis based on follow-up duration and a restricted analysis comparing ECC versus LT were conducted to explore heterogeneity. Twelve publications with 6280 patients were included in the final analysis. In our systematic review and meta-analysis, the EC Fontan was associated with significantly lower odds of arrhythmia compared to the IC Fontan (OR = 0.38, 95% confidence interval [CI]: 0.27-0.52, P < 0.001), with substantial heterogeneity (I 2 = 74%). In a restricted analysis comparing only ECC with LT, the association remained significant (OR = 0.52, 95% CI: 0.39-0.69; P < 0.001). EC Fontan is linked to a lower observed arrhythmia burden compared to IC Fontan. However, this finding should be interpreted cautiously, as the observed effect may partly reflect the higher arrhythmia burden in classic Fontan, as well as differences in surgical era and follow-up duration. Given the persistent risk of arrhythmia in all Fontan patients, individualized management and long-term surveillance are essential.
Few studies have described physiological and cardiac rhythm responses to maximal voluntary breath-holding in elite freedivers. This case study presents minute ventilation, PETO2, PETCO2, involuntary breathing movements (IBMs; surface electromyography), SpO2, muscle and cerebral oxygenation (near-infrared spectroscopy), and cardiac rhythm changes (electrocardiogram) before, during, and after a maximal dry static voluntary breath-hold in a world champion freediver. Glossopharyngeal insufflation prior to the breath-hold increased forced vital capacity from 6.92L (138% predicted) to 9.04L (180% predicted). Compared to resting end-tidal gas pressures, the breathe-up in preparation of the breath-hold increased PETO2 (108mmHg to 135mmHg) and decreased PETCO2 (36mmHg to 21mmHg). The breath-hold was 06'07" in duration, of which 02'20" was spent in the easy-going phase and 03'47" in the struggle phase - the latter encompassing a total of 48 IBMs. Throughout the breath-hold, PETO2 decreased to 38mmHg and PETCO2 increased to 65mmHg. SpO2 decreased from 97% to 73%. Muscle oxygenation decreased from 50% to 22%, whereas cerebral oxygenation remained relatively stable until the final ∼20s of the breath-hold (∼68% to ∼62%). Heart rate variability-based markers of autonomic cardiac activity decreased during the easy-going phase, increased during the struggle phase, and normalized after. Asymptomatic bradycardia with competition between sinus bradycardia and junctional rhythm and (supra)ventricular extrasystoles manifested throughout the struggle phase and resolved after breath-hold cessation. This report of a world champion freediver shows that a long voluntary breath-hold induced transient asymptomatic cardiac arrhythmias, likely linked to the physiological stress of extreme voluntary breath-holding.
A 68-year-old woman presented with palpitations, though ECG monitoring showed sinus rhythm. Multimodal imaging (cardiac magnetic resonance and echocardiography) showed asymmetric septal hypertrophy with left ventricular outflow tract obstruction (resting gradient of up to 87 mmHg). Echocardiography showed severe mitral regurgitation with multiple regurgitant jets and systolic anterior motion of the mitral valve. Mitral valve anatomy was abnormal, with anterior leaflet restriction and a clefted posterior leaflet. Symptoms persisted despite medical therapy (beta-blockers), and following multidisciplinary discussion, she proceeded to surgery on symptomatic and prognostic grounds. The patient underwent successful septal myectomy and bioprosthetic mitral valve replacement with excellent valve function. Following a generally uneventful postoperative recovery, she was discharged on postoperative day ten with stable cardiac function. This case highlights a rare trileaflet configuration of the mitral valve contributing to severe mitral regurgitation in hypertrophic obstructive cardiomyopathy and underscores the importance of detailed anatomical assessment in guiding surgical decision-making.
This case report describes a 57-year-old woman with a history of transient loss of consciousness, initially treated as epilepsy, who presented with recurrent torsades de pointes requiring direct-current cardioversion. Her evaluation revealed a prolonged corrected QT (QTc) interval, and, after excluding acquired causes, she was managed with a dual-chamber implantable cardioverter-defibrillator, β-blocker and mexiletine therapy, and a base rate of 80 bpm. Genetic analysis identified a previously unreported, possibly pathogenic compound heterozygosity in the AKAP9 gene (c.9443C>T, p.Thr3148Met and c.10515_10520delAACCGG, p.Thr3506_Gly3507del). Upon diagnosis of congenital long QT syndrome, her antiepileptic drugs were discontinued. At 6-month follow-up, she remained free of arrhythmic events with noted improvement in her QTc interval, highlighting the critical importance of accurate diagnosis and genotype-guided therapy in such cases.