Hyperkinetic movement disorder following Covid-19 Temporal correlation or Coincidental

Objective: Hypokinetic movement disorder and parkinsonian picture has been well described in literature following covid-19 but hyperkinetic MDS are very in global literatures. To investigate the epidemiology,clinical picture,the diagnostic and therapeutic challenges in patients hyperkinetic MDS in this context and to know the time schedule of the onset of the MDS with exploring the possible pathogenesis Background: Infections are up to 20% of movement disorders.The most frequent agents are beta-hemolytic streptococcus,and flavivirus causing Japanese encephalitisThe role of the viral stimulation of microglial activation in neuroinflammation has regained attention in the context of covid19 Methods: Patients of MDS attended the clinic from 31st march 2020 to March 2022,with recent onset of hyperkinetic movements were screened. Subjects had medical history either prior to the study or medical history reviewed by physicians suggestive of covid.PCR +VE or Presence of covid antibody in blood or csf in patients with recent onset hyperkinetic MDS within 6-12 weeks of onset of symptoms except.Ventilatory cases Other markers were used to rule out other viral infections causing MDS.MRI brain and EEG as a routine in all Immune markers in very selected cases in suspected immuomediated MDSThe attempted treatment were symptomatic and immunotherapy Results: In last 2 years 50 cases of new onset Hyperkinetic MDS are recorded, out of which only 9 cases were directly or indirectly linked to Covid,Nystagmus, orofacial dyskinesia and segmental or generalized myoclonus and ataxic gait associated delirium,tremors and ocular movement disorders along with epileptic seizures are also seen.Positive EEG findings are in the form of diffused bihemispheric slowing or periodic complexes with polyspikes at irregular interval and delta brush in few cases .MRI findings varied between non-specific changes to bitemporoparietal hyperintensities in flair and T2 both cortical and subcortical or bilateral basal ganglia. Treatment response in all the cases are statisfactory Conclusion(s): observational study revealed MDS in covid do happen Myoclonus is the most Frequent movement disorder associated with COVID-19 followed by dystonia and tremors .pathophysiology included neuro inflammation, autoimmune mechanisms and small vessels thrombosis hence not be co-incidental , response to steroid also s/o immune mediated.

Assessing the Effectiveness of a Standardized and Concise End-of-Life (EOL) Care Toolkit for Medical Residents in a Teaching Hospital: A Mixed Methods Exploratory Study (Sch409)

Outcomes: 1. Assess baseline knowledge, attitudes, and practices on EOL non-pain symptom management among internal medicine residents in a teaching hospital using a cross-sectional survey. 2. Develop a standardized inpatient EOL non-pain symptom management educational toolkit for internal medicine residents. Introduction: With palliative care gaining traction as a vital specialty to help patients living with serious illnesses comes the need for further training of healthcare professionals. Frontline providers such as medical residents can benefit from end-of-life (EOL) care training in symptom management. Method(s): There are three phases (over a period of 4 years) to this study: (1) administration of a needs assessment survey of baseline knowledge, attitudes, and practices on EOL non-pain symptom management;(2) development and implementation of a standardized inpatient EOL symptom management toolkit;and (3) a comparison of pre-and postassessment after the educational intervention. Result(s): The baseline survey had 66 participants. There were six non-pain symptoms that were elicited as important for further education and training. These were anorexia, nausea/vomiting, dyspnea, oral secretions, myoclonus, and delirium. Competency-based comfort and confidence levels were assessed using a Likert scale (1-5), with the highest number as the most comfortable. The residents were noted to be more comfortable with EOL communication compared to symptom management. Furthermore, residents who had had previous EOL care experiences with patients were more comfortable in symptom management. The educational intervention implemented at a later time revealed that there was an improvement in posttest scores for EOL symptom management. Discussion(s): This study highlights the needs and gaps in EOL symptom management training for medical residents. The implementation of a standardized inpatient EOL symptom management toolkit might serve as a potential intervention to address the needs and narrow gaps in medical training. This can serve as a possible template for other institutions to integrate an EOL care curriculum in medical residency. Limitations of the study include a small sample size, implementation during the COVID-19 pandemic, variable participant response rate, and interrupted timelines. The next steps include ongoing training for all residents, long-term follow-up postintervention, and institutional buy-in.Copyright © 2023

Neurological and Neuropsychiatric Impacts of COVID-19 Pandemic.

BACKGROUND: Albeit primarily a disease of respiratory tract, the 2019 coronavirus infectious disease (COVID-19) has been found to have causal association with a plethora of neurological, neuropsychiatric and psychological effects. This review aims to analyze them with a discussion of evolving therapeutic recommendations. METHODS: PubMed and Google Scholar were searched from 1 January 2020 to 30 May 2020 with the following key terms: "COVID-19", "SARS-CoV-2", "pandemic", "neuro-COVID", "stroke-COVID", "epilepsy-COVID", "COVID-encephalopathy", "SARS-CoV-2-encephalitis", "SARS-CoV-2-rhabdomyolysis", "COVID-demyelinating disease", "neurological manifestations", "psychosocial manifestations", "treatment recommendations", "COVID-19 and therapeutic changes", "psychiatry", "marginalised", "telemedicine", "mental health", "quarantine", "infodemic" and "social media". A few newspaper reports related to COVID-19 and psychosocial impacts have also been added as per context. RESULTS: Neurological and neuropsychiatric manifestations of COVID-19 are abundant. Clinical features of both central and peripheral nervous system involvement are evident. These have been categorically analyzed briefly with literature support. Most of the psychological effects are secondary to pandemic-associated regulatory, socioeconomic and psychosocial changes. CONCLUSION: Neurological and neuropsychiatric manifestations of this disease are only beginning to unravel. This demands a wide index of suspicion for prompt diagnosis of SARS-CoV-2 to prevent further complications and mortality.

Les impacts neurologiques et neuropsychiatriques d'une infection à la COVID-19. CONTEXTE: Bien qu'il s'agisse principalement d'une maladie des voies respiratoires, la maladie infectieuse à coronavirus apparue en 2019 (COVID-19) s'est avérée avoir un lien de causalité avec une pléthore d'impacts d'ordre neurologique, neuropsychiatrique et psychologique. Cette étude entend donc analyser ces impacts tout en discutant l'évolution des recommandations thérapeutiques se rapportant à cette maladie. MÉTHODES: Les bases de données PubMed et Google Scholar ont été interrogées entre les 1er janvier et 30 mai 2020. Les termes clés suivants ont été utilisés : « COVID-19 ¼, « SRAS ­ CoV-2 ¼, « Pandémie ¼, « Neuro ­ COVID ¼, « AVC ­ COVID ¼, « Épilepsie ­ COVID ¼, « COVID ­ encéphalopathie ¼, « SRAS ­ CoV-2 ­ encéphalite ¼, « SRAS ­ CoV-2 ­ rhabdomyolyse ¼, « COVID ­ maladie démyélinisante ¼, « Manifestations neurologiques ¼, « Manifestations psychosociales ¼, « Recommandations thérapeutiques ¼, « COVID-19 et changement thérapeutiques ¼, « Psychiatrie ¼, « Marginalisés ¼, « Télémédecine ¼, « Santé mentale ¼, « Quarantaine ¼, « Infodémique ¼ et « Médias sociaux ¼. De plus, quelques articles de journaux relatifs à la pandémie de COVID-19 et à ses impacts psychosociaux ont également été ajoutés en fonction du contexte. RÉSULTATS: Il appert que les manifestations neurologiques et neuropsychiatriques des infections à la COVID-19 sont nombreuses. Les caractéristiques cliniques d'une implication des systèmes nerveux central et périphérique sautent désormais aux yeux. Ces caractéristiques ont fait l'objet d'une brève analyse systématique à l'aide de publications scientifiques. En outre, la plupart des impacts d'ordre psychologique de cette pandémie se sont révélés moins apparents que les changements réglementaires, socioéconomiques et psychosociaux. CONCLUSION: Les manifestations neurologiques et neuropsychiatriques de cette maladie ne font que commencer à être élucidées. Cela exige donc une capacité accrue de vigilance en vue d'un diagnostic rapide, et ce, afin de prévenir des complications additionnelles et une mortalité accrue.

Hyperkinetic movement disorders following SARS-CoV-2 infection and vaccination - an update.

The aim of this review was to summarise current knowledge regarding hyperkinetic movement disorders related to SARS-CoV-2 infection and vaccination in terms of phenomenology, epidemiology, pathogenesis and treatment. After a thorough review of the PubMed and Google Scholar databases (2020-2022), we identified myoclonus and ataxia sometimes accompanied by opsoclonus (AMS) as the two most frequent COVID-19 sequelae, with chorea, tremor and dystonia being very rare. The pathogenesis seems to be variable, but in the majority of AMS cases it was autoimmunological, with good response and recovery after corticosteroids or intravenous immunoglobulins infusions. Vaccination may be complicated by hyperkinetic movement disorders (e.g. tremor, dystonia), but this is very rare. Patients with Deep Brain Simulation depletion should not be postponed due to lockdowns as this may result in fatal outcomes.

COVID19-associated new-onset movement disorders: a follow-up study.

BACKGROUND: Neurological symptoms are common manifestation in acute COVID-19. This includes hyper- and hypokinetic movement disorders. Data on their outcome, however, is limited. METHODS: Cases with new-onset COVID-19-associated movement disorders were identified by searching the literature. Authors were contacted for outcome data which were reviewed and analyzed. RESULTS: Movement disorders began 12.6 days on average after the initial onset of COVID-19. 92% of patients required hospital admission (mean duration 23 days). In a fraction of patients (6 of 27; 22%; 4 males/2 females, mean age 66.8 years) the movement disorder (ataxia, myoclonus, tremor, parkinsonism) was still present after a follow-up period of 7.5 ± 3 weeks. Severe COVID-19 in general and development of encephalopathy were risk factors, albeit not strong predictors, for the persistence. CONCLUSIONS: The prognosis of new-onset COVID-19-associated movement disorder appears to be generally good. The majority recovered without residual symptoms within several weeks or months. Permanent cases may be due to unmasking of a previous subclinical movement disorder or due to vascular/demyelinating damage. Given the relatively low response rate of one third only and the heterogeneity of mechanisms firm conclusions on the (long-term) outome cannot, however, be drawn.

Opsoclonus Myoclonus Ataxia Syndrome Due to SARS-CoV-2.

Opsoclonus myoclonus syndrome (OMS)/opsoclonus myoclonus ataxia syndrome (OMAS), also known as Kinsbourne's syndrome or 'dancing eyes-dancing feet' syndrome, is a rare central nervous system manifestation of COVID-19 but an increasing number of articles have reported patients in whom COVID-19 was complicated by OMS/OMAS. This narrative review aims at summarising and discussing current knowledge about the clinical presentation, diagnosis, treatment and outcome of SARS-CoV-2 associated OMS/OMAS. Altogether, 29 articles reporting 45 patients with SARS-CoV-2 associated OMS/OMAS were retrieved. Their ages ranged from 2 to 88 years. Three patients were children and the remainder adults. Gender was male in 32 patients and female in 13 patients. Opsoclonus was described in 29 patients, which was associated with myoclonus in 28 cases. Myoclonus was described in 43 patients, which was associated with opsoclonus and ataxia in 18 patients. Cerebral magnetic resonance imaging and cerebrospinal fluid investigations were not informative in the majority of the cases. OMS/OMAS was treated with steroids in 28 patients and with intravenous immunoglobulin (IVIG) in 15 patients. Clonazepam was given to 18 patients, levetiracetam to 13 patients, and sodium valproate to eight patients. Complete recovery was achieved in 12 cases and incomplete recovery in 22 cases. Diagnosing SARS-CoV-2 associated OMS/OMAS requires extensive neurological work up and exclusion of various differentials. SARS-CoV-2 associated OMS/OMAS may not always present with the full spectrum of manifestations but as an abortive syndrome. OMS/OMAS should not be missed as it usually responds favourably to steroids or IVIG.

Myoclonus status revealing COVID 19 infection.

INTRODUCTION: At the beginning of the coronavirus virus (COVID-19) pandemic, the Severe Acute Respiratory Syndrome Coronavirus 2 (SARS-CoV2) was thought to cause mainly respiratory symptoms, largely sparing the brain and the rest of the nervous system. However, as the knowledge about COVID-19 infection progresses and the number of COVID19-related neurological manifestations reports increases, neurotropism and neuroinvasion were finally recognized as major features of the SARS-CoV-2. Neurological manifestations involving the central nervous system are sparse, ranging from headaches, drowsiness, and neurovascular attacks to seizures and encephalitis [1]. Thus far, several cases of non-epileptic myoclonus were reported in critical patients [2,3]. Here, we report the first case of myoclonus status as the inaugural and sole symptom of COVID-19 in a conscious patient. OBSERVATION: A 60-year-old man with unknown family history and no medical issues other than smoking one cigarette packet a day over the span of 25 years. The patient presented with 5 days of abnormal movements in bilateral arms following the COVID vaccination. They were described as brief, involuntary jerking, like in sleep starts, in the proximal part of their upper members, and his face with a regular tremor in his arms exacerbated by movements and emotion. His movement disorder worsened the second day, and he developed an abnormal gait with slurred speech, concomitantly with diarrhea. Seven days following the symptoms onset, the patient was alert. His neurological exam revealed multifocal myoclonic jerks affecting four limbs predominantly proximal, the face, and the trunk (video 1). The myoclonic jerks were sensitive to tactile and auditory stimuli, without enhanced startle response or hyperekplexia. His gait was unsteady due to severe myoclonus, without cerebellar ataxia (video 2) and he had mild dysarthria. No dysmetria at the finger-to-nose and heel-to-shin tests were found. Examination of eye movements revealed paralysis of Down-Gaze and no opsoclonus was detected. Physical exam was unremarkable, including lack of fever and meningitis signs. The electroencephalogram (EEG) did not show any abnormalities concomitant with myoclonic jerks (Fig.1). The cerebral Magnetic Resonance Imaging (MRI) was normal (Fig. 2). An extensive biological work-up including a complete blood count, a comprehensive metabolic panel, an arterial blood gas analysis, a urine drug screen, a thyroid function test, a vitamin B12, folate, and ammonia level, and HIV and syphilis serologies were inconclusive. Testing for autoimmune and paraneoplastic antineuronal antibodies including anti-NMDA-R was negative. The cerebrospinal fluid (CSF) study was unremarkable (0.3 g/l of proteinorachia, 1 white blood cell). Polymerase chain reaction (PCR) for herpes simplex virus, varicella-zoster virus, and SARS-CoV-2 in CSF was negative. However, the patient tested positive for COVID-19 through PCR for viral RNA from the nasopharyngeal swab. After the administration of 12mg/day of Dexamethasone for 3 days, along with clonazepam and levetiracetam, the patient's symptoms started improving on day 3 and he displayed a very slow but progressive recovery. DISCUSSION: Our patient presented with acute isolated multifocal myoclonus status without cognitive impairment. These movements were prominent, spontaneous, worsened by action, and sensitive to touch and sound. The anatomical source of this myoclonus could be cortical or subcortical despite the absence of evident EEG discharges. Several diseases can cause acute myoclonus such as severe hypoxia, metabolic disturbances, and paraneoplastic syndromes. these diagnoses were ruled out in our patient. Post-vaccinal origin was also suggested, but its accountability was not proven. Thus, the two hypothetic etiologies raised were either para-infectious or infectious mechanisms in relation to SARS-Cov 2 infection. HIV, VZV, HSV, and syphilis infections were eliminated and the patient tested positive for SARS-Cov2 infection. In the literature, COVID-19-related myoclonus was reported as a complication of an already-known SARS-CoV-2 infection in about 50 patients so far. It generally occurs between 6 days and 26 days following the SARS-CoV-2 infection [2-5], and affects critical illness patients with cognitive decline, mainly from the intensive care unit [3,4]. Yet, our patient did not display any symptoms of COVID-19 infection before the occurrence of these abnormal movements. Furthermore, he had a relatively good general condition and no cognitive impairment. Several pathophysiological mechanisms were suggested regarding the COVID-19-related myoclonus. Either central nervous invasion by SARS-Cov 2 after transneuronal spread and/or auto-immune cross-reactivity reaction, are likely incriminated in the pathophysiology of most of the cases [6]. We believe that there is an inflammatory process involved with increased levels of proinflammatory cytokines and systemic inflammation, including cytokine storm or cytokine release syndrome targeting the brain and more specifically the cortex and basal ganglia [6]. Data collection in clinical registries is needed to increase our knowledge of the prevalence of neurological symptoms in patients with COVID-19 and will hopefully clarify the causal relationship between SARS-CoV-2 infection and post-COVID-19 myoclonic syndrome.

Myoclonus associated with infections: A narrative review

Different movement disorders are reported in association with infectious diseases. In addition, myoclonus can be associated with different types of viral and bacterial infections. We screened three electronic databases for cases of myoclonus as a feature of different infections and collected cases and series describing myoclonus associated with infections. Data regarding study design, sample size, neurological assessment, and diagnostic workup including brain imaging and cerebrospinal fluid analysis were extracted from the identified studies. In this narrative review, we review different infections associated with myoclonus and discuss their salient features. The infections presenting with myoclonus include predominantly subacute sclerosing panencephalitis due to measles. In addition, we describe other viral infections that are reported to associated with myoclonus. Recently, coronavirus disease 2019 infections have been reported to be increasingly associated with myoclonus. The hypothesized mechanisms of infection-related myoclonus are vasculopathy, autoimmune reactions, and inflammation. Although myoclonus is considered to be a result of heredodegenerative, metabolic, or autoimmune disorders, infections may present with myoclonus, especially in tropical and developing countries. In this review, we describe the infections that are associated with myoclonus. © 2022 Annals of Movement Disorders ;Published by Wolters Kluwer - Medknow.

The onset of functional movement disorders after COVID-19: A case series.

Patients with post-acute sequelae after coronavirus disease (COVID-19) report a variety of non-specific neurological complications (e.g., myoclonus, limb weakness). In particular, they manifest scenarios as medically unexplained symptoms and are known as functional movement disorders (FMDs). We present three cases of FMDs in patients of the Institute of Clinical Medicine named after N. V. Sklifosovsky (Sechenov University). All patients had a history of COVID-19 infection and reported fatigue, weakness, and jerks of upper and lower limbs. In conclusion, there might be a major possibility of the virus negatively affecting the central nervous system, including such rare neuropsychiatric complications.

Possible Autoimmune Encephalitis Associated with the Severe Acute Respiratory Syndrome Coronavirus 2 Omicron Variant Successfully Treated with Steroids: A Case Report.

We encountered a 55-year-old woman with possible autoimmune encephalitis associated with the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) Omicron variant. She was not vaccinated against coronavirus disease 2019 (COVID-19). Consciousness disturbance, myoclonic-like movements and gait disturbance occurred 10 days after the COVID-19 symptom onset. Her neurological symptoms improved two days after methylprednisolone pulse therapy. Cerebrospinal fluid (CSF) was negative for SARS-CoV-2 reverse transcription-polymerase chain reaction, the CSF-to-serum albumin quotient was mildly elevated, and interleukin 6 and 8 levels were normal in serum but mildly elevated in CSF. Omicron variant infection may increase blood-brain barrier permeability and intrathecal inflammation, causing autoimmune encephalitis.

A CLASSIC CASE OF CREUTZFELDT-JAKOB DISEASE (CJD)

CASE: A 64-year-old woman was brought in by husband for inability to care for patient. Previously active, she developed gait instability, slurred speech, and memory lapse to the point of selective mutism and being bed-bound within three months. Her medical history was notable for hypertension and Covid four months prior. She had had mild upper respiratory symptoms and recovered in ten days. Examination revealed general encephalopathy, dysarthria, limited ability to follow commands. She had decreased strength but increased tone and rigidity in all extremities. She had rhythmic jaw movement and bradykinesia with scatter myoclonic movements. Cerebellar exam was notable for ataxia, but she had normal cranial nerve and sensory exams and normal reflexes. MRI of the brain revealed restricted diffusion and T2/Flair signal abnormality involving bilateral basal ganglia, ventral medial thalami, hippocampi, and cerebral cortices. Toxic metabolic workup was unrevealing. CSF was positive for 14-3-3 protein and elevated total tau protein, confirming Creutzfeldt-Jakob disease. IMPACT/DISCUSSION: Creutzfeldt-Jakob Disease (CJD) is a prion disease with one in a million prevalence. Patients present with rapidly progressing dementia, myoclonus, and signs of cerebellar, corticospinal and extrapyramidal involvement including nystagmus, ataxia, hyperreflexia, spasticity, hypokinesia, bradykinesia, dystonia, and rigidity. CJD is fatal within months to two years. Patients with end stage disease may have akinetic mutism. Magnetic resonance imaging (MRI), electroencephalogram (EEG), and cerebrospinal fluid (CSF) analysis are important for evaluation of CJD. Most sensitive in early stages, MRI Brain commonly shows hyperintense signal involving the cerebral cortex, corpus striatum, caudate, and putamen. EEG may capture pattern of periodic bi-or triphasic period sharp wave complexes. CSF might detect 14-3-3 protein with elevation of tau protein but real-time quaking-induced conversion (RT-QuIC) has the highest specificity for diagnosis for CJD. Though brain biopsy is the sole method of definitive diagnosis, results of MRI, EEG, and CSF analysis along with presenting signs and symptoms are sufficient for clinical diagnosis of CJD. Our patient's dementia, myoclonus, ataxia, hypokinesia, bradykinesia, dystonia, and rigidity all progressing to akinetic mutism within three months are classic presentation of CJD. EEG was normal, but MRI with hyperintensity of basal ganglia and cerebral cortices and CSF analysis with positive 14-3-3 and elevated tau proteins are all lead to diagnosis of CJD. CONCLUSION: This case illustrates a classic case of a Creutzfeldt-Jakob Disease, a rare prion disease marked by rapidly progressive dementia with neuropsychiatric features.

RESTING TREMOR AFTER COVID-19 INFECTION

CASE: The patient is a 66 year-old woman with history of hypertension and recovered COVID-19 presenting to the outpatient clinic for eight months of persistent resting tremor of her left arm. The tremor started shortly after she developed headache, fatigue, and epistaxis found to have COVID-19. The tremor is mild, occurs multiple times throughout the day, and usually resolves spontaneously after several seconds. The patient denies any paresthesias, muscle weakness, motor slowing, or ataxia. She has no family history of Parkinson's disease or essential tremor. On physical exam, vital signs are normal. Motor strength is 5/5 and sensation is intact throughout. Brachioradialis deep tendon reflex is 1/4 bilaterally though slightly increased on the right side. Cranial nerves II through XII are intact. Gait is normal with no evidence of shuffling. No pronator drift is evident. No cogwheel rigidity is noted. Finger-to-nose motion is normal. Throughout the appointment, the patient is noted to have an intermittent mild resting tremor in her left arm that lasts several seconds and resolves spontaneously. Laboratory results including a basic metabolic panel and thyroid stimulating hormone level are normal. Incidentally, the patient underwent a recent brain MRI for chronic sensorineural hearing loss that showed normal appearance of the internal auditory canals/ middle ear structures and no evidence of intracranial pathology. The patient was subsequently started on daily propranolol. A subsequent telemedicine visit one month later revealed that her resting tremor had nearly resolved. IMPACT/DISCUSSION: The outpatient presentation of resting tremor warrants consideration of a broad differential that includes Parkinson's disease and other causes of parkinsonism, including neurodegenerative diseases and essential tremor, among others. Furthermore, previous studies have demonstrated new onset movement disorders associated with COVID-19 including myoclonus, ataxia, action/postural tremor, catatonia, dystonia, chorea, and functional movement disorders. The exact pathophysiology of COVID-19 related movement disorders is not well understood. Of note, these prior studies did not specifically address evaluation of COVID-19 related movement disorders in the outpatient setting. CONCLUSION: The patient described above likely developed new onset left arm tremor secondary to COVID-19. Her reassuring physical exam findings, laboratory results, and head MRI suggest against other etiologies. The patient was successfully treated with propranolol. This case demonstrates the importance of neurologic assessment in the outpatient setting, particular in patients with a history of COVID-19 diagnosis. Though limited data exists on outpatient evaluation and management of movement disorders secondary to COVID-19, it is important to recognize this phenomenon as a potential diagnosis.

Sleep disturbances in craniopharyngioma: a challenging diagnosis

Introduction: The hypothalamus plays a crucial role in regulating vital functions and circadian rhythms. Both the tumor involving the hypothalamic area and its treatment can lead to hypothalamic dysfunction, resulting in disturbances in sleep-wake patterns, sleep fragmentation, and increased daytime sleepiness. We describe two patients with craniopharyngioma who came to our attention due to the occurrence of episodes characterized by psychomotor slowing and afinalistic limb movements, temporal and spatial disorientation, psychomotor agitation, and oneiric stupor like episodes diagnosed as severe sleep disturbances. Case reports: Patient 1 is a 19-year-old male diagnosed with surgically treated craniopharyngioma. Subsequently, episodes of psychomotor slowing, afinalistic movements of the upper limbs diagnosed as seizures in another neurological center appeared;antiepileptic treatment was started without improvement. At the first examination in our center, excessive daytime sleepiness (EDS), fragmented nighttime sleep, episodes characterized by bimanual automatic gestures occurring during drowsy state, hypnagogic hallucinations, and sudden loss of muscle tone while awake were recognized. Actigraphy demonstrated irregular bedtimes, frequent nocturnal activity, and inappropriate daytime rest episodes. The Epworth Sleepiness Scale (ESS) showed subjective EDS (ESS=19). At PSG, hypersomnolence, severe sleep-related breathing disorder (SRBD), and no interictal and ictal seizure abnormalities were found. A BiPAP NIV was started, and antiepileptic therapy was discontinued. In the following months, PSG revealed marked improvement in SRBD and 1 SOREMP, and the MSLT a mean SOL of 6 min and 10 sec and 3 SOREMPs. These data allowed the diagnosis of secondary narcolepsy, and treatment with pitolisant was initiated with clinical improvement and reduced daytime sleepiness (ESS=9). Patient 2 is a 12-year-old male, surgically treated for craniopharyngioma at the age of 4 years, who developed episodes of myoclonic jerks, temporal and spatial disorientation, and psychomotor agitation during the lockdown period for COVID-19 emergency. Surmising paroxysmal epileptic episodes, the patient was hospitalized. The anamnestic data collection revealed a sleep-wake rhythm dysregulation, fragmented nighttime sleep, EDS, oneiric stupor-like episodes during which the patient performed simple automatic gestures mimicking daily-life activity, and severe impairment of alertness. The Long-term video-EEG, including polygraphic measurements, showed destruction of the wake-NREM sleep-REM sleep boundaries, episodes of undetermined state of vigilance, and concurrence of elements typical of different sleep stages. Moreover, a severe SRBD (AHI 19/h) has been observed. The MRI showed a volumetric increase in the post-surgical interpeduncular fossa and right paramedian cysts. Therefore, a multifactorial therapeutic plan including sleep hygiene and slow-release melatonin was started with improvement in nighttime sleep, but EDS persisted. Surgical treatment of cyst fenestration improved sleep-wake rhythm and behavior;BiPAP NIV was initiated with very poor adherence. Discussion: We aim to focus on sleep disorders as a possible complication of tumors involving the hypothalamic region. Our cases highlight that the clinical manifestation of these dysfunctions can be challenging to diagnose and can lead to misdiagnosis and inappropriate treatment that can harm patients' health and the quality of life of patients and their families. Conclusion: These findings support the need to incorporate comprehensive sleep assessment in survivors from childhood brain tumors involving the suprasellar/hypothalamic region.