In some cases dysphagia, neuroblastoma, and intestinal problems are associated with Ondine’s Curse or Central Hypoventilation Syndrome. You are currently offline. The term “Ondine’s curse”is more commonly used for cas-es of congenital central alveolar hypoventilation,but it can also be applied to acquired cases 8.It arises from Germanic mythol-ogy, in which Ondine, an oceanic nymph of incomparable beauty punishes her … Characteristics and clinical presentations of three patients were described and analysed. Surgical operation and stroke are the reported causes of this syndrome. In others, no predisposing cause can be identified.' All had diabetes mellitus Ondine’s curse: a case of anti-Hu paraneoplastic syndrome 3C00 C Brookes, R McIntosh, S Twigg A 60-year-old patient presented with neurological symptoms which proved to be a paraneoplastic syndrome secondary to a small-cell lung cancer. Use the link below to share a full-text version of this article with your friends and colleagues. The patient improved Semantic Scholar is a free, AI-powered research tool for scientific literature, based at the Allen Institute for AI. 13,14,17,30,41 Sleep apnea has not been associated with low cervical cordotomies. Ondine’s curse—more appropriately known as congenital central hypoventilation syndrome, or CCHS—is a rare, severe form of sleep apnea in which an individual completely stops breathing when falling asleep. Conclusion: the Ondine's curse is one of the posterior stroke's presentation characterized by loss of automatic breathing and for the unpredictability of clinical evolution and prognosis. He was placed on acetazolamide for central hypoventilation. Ondine’s curse: a case of anti-Hu paraneoplastic syndrome 3C00 C Brookes, R McIntosh, S Twigg A 60-year-old patient presented with neurological symptoms which proved to be a paraneoplastic syndrome secondary to a small-cell lung cancer. Congenital central hypoventilation syndrome and Hirschsprung’s disease, Care of the Child with Congenital Central Hypoventilation Syndrome. In the treatment of C-CAH, we implanted a right unilateral diaphragm pacemaker in the infant and his respiratory status was remarkably improved after diaphragm pacing. The nymph Ondine was an immortal water spirit who became human after falling in love for a man, marrying him, and having a baby. The name of Ondine's curse is derived from an ancient legend in Germany, where goddess Ondine’s punishment is said to befall those people who Please check your email for instructions on resetting your password. Neither did my patient with Ondine’s Curse have mountain sickness. Yasuma F, Nomura H, Sotobata I, Ishihara H, Saito H, Yasuura K, Okamoto H, Hirose S, Abe T, Seki A. Congenital central alveolar hypoventilation (C-CAH), so called Ondine's curse, is known to be quite a rare neuropathology that has been reported in only 23 cases to date. It represents an enormous difficulty in defining a clinical situation where two conditions are represented in different ways, although the underlying mechanism is similar if not identical. Results: All patients were male and they were 63, 65 and 84 y old respectively. Five cases of central congenital hypoventilation, Ondine's curse, were studied. Congenital central alveolar hypoventilation (C-CAH), so called Ondine's curse, is known to be quite a rare neuropathology that has been reported in only 23 cases to date. 1) . All were on mechanical ventilation. The other conditions frequently associated with Ondine's curse were: brainstem disturbances, Hirschsprung disease and…, Congenital central hypoventilation syndrome (Ondine's curse syndrome) in two siblings: Delayed diagnosis and successful noninvasive treatment, Medullary Infarction with Central Hypoventilation ( Ondine ’ s Curse ) : A Case Report, Congenital central hypoventilation syndrome associated with Hirschsprung's disease and neuroblastoma: Case of multiple neurocristopathies. Ondine's curse is one of the most enchanting mythical tales in the field of Medicine. Congenital central alveolar hypoventilation (Ondine's curse): a case report and review of the literature. Ondine’s curse — or, more officially, central hypoventilation syndrome — is an often fatal respiratory disorder that occurs during sleep. Ondine's curse is associated with respiratory arrests during sleep and, with incomplete penetrance, neuroblastoma (tumors of the sympathetic ganglia), Hirschprung disease (partial agenesis of the enteric nervous system), dysphagia (difficulty swallowing), anomalies of the pupilla, etc. Ondine's curse associated with Hirschsprung disease and ganglioneuroblastoma. We hypothesise that the generalised oedema that occurred during pregnancy, with a potentially mild increase in intracranial … Ondine curse is one type of sleep apnea syndrome, defined as the selective disturbance of autonomous breathing. During his stay in paediatric intensive care unit, there were concerns that he was not initiating breathing over the ventilator. Brainstem tumor is relatively common cause for children's Ondine curse. Barber MD, Scobie WG. There was associated T2 signal abnormality in these locations. Ondine’s curse—more appropriately known as congenital central hypoventilation syndrome or CCHS—is a rare, severe form of sleep apneain which an individual Case Report: Ondine's Curse Syndrome Secondary Brainstem Infarction Canmin Zhu, Ming Li *, Jiaping Xiao, Xuan Zhang , Mei Zhang, Qiang Li and Wei Zeng Department of Neurology, The Fifth Hospital of Wuhan, 122 Xianzheng Street, Hanyang, Wuhan, Hu-Bei, P.R. Ondine's curse was mentioned in the 1987 film Black Widow starring Debra Winger, Theresa Russell, Terry O'Quinn and D. W. Moffett. The nymph Ondine was an immortal water spirit who became human after falling in love for a man, marrying him, and having a baby. In one of the versions of the tale, when she caught her husband sleeping with another woman, she cursed him to remain awake in order to control his own breathing. There are only a few cases-about 200 to 500 worldwide-each year. The investigations and treatment for … Vasospasm, a complication associated with SAH, results in secondary cerebral infarcts. It suggested bilateral acute to subacute infarcts in the posterior inferior cerebellar artery (PICA) distributions. In one of the versions of the tale, when she caught her husband sleeping with another woman, she cursed him to remain awake in order to control his own breathing. The apnoea/hypopnoea index was 107 events/h. Congenital central hypoventilation syndrome (CCHS), formerly known as Ondine's curse, is a congenital and severe form of central sleep apnea resulting from abnormal autonomic control of breathing with decreased sensitivity to hypercapnia and hypoxemia, particularly during sleep. Ondine's curse, also called congenital central alveolar hypoventilation (CCAH), is a rare condition involving failed automatic respiration in the absence of cardiopulmonary disease, resulting in inadequate ventilation with progressive hypercapnia and hypoxia during sleep . Of the four, only central alveolar hypoventilation without muscular paralysis is labeled ‘Ondine's curse.’ They state: 'In many cases, a history of encephalitis, trauma, Leigh's disease, Shy-Drager disease, or shock therapy can be obtained from patients with this condition. He had a brief period of cardiorespiratory arrest and was resuscitated and intubated by emergency medical services on scene. China Computed tomography (CT) head showing subarachnoid haemorrhage in basilar cisterns. CHS can either be congenital (CCHS) or acquired (ACHS) later in life. Materials and methods: This is a report of 3 cases of LMS with Ondine's curse. A 9‐month‐old child was struck with television directly on the head. Ondine’s curse — or, more officially, central hypoventilation syndrome — is an often fatal respiratory disorder that occurs during sleep. The full text of this article hosted at iucr.org is unavailable due to technical difficulties. Peaks of oxygen desaturation reached a nadir of 55%. [2-4] Lesions selectively involving the ventrolateral . Working off-campus? Central hypoventilation syndrome (CHS) is a sleep-related breathing disorder that causes ineffective breathing, apnea, or respiratory arrest during sleep (and during wakefulness in severe cases). Ondine's curse is a rare and potentially fatal disorder characterised by loss of automatic alveolar hypoventilation during non‐rapid eye movement (REM) sleep and improves during REM sleep and quiet wakefulness. Ondine’s Curse after High Cervical Cordotomy. This syndrome, especially in adults, is rare, and even less frequent in the absence of clear pathogenic lesions on MRI. Ondine's curse was mentioned in the 1987 film Black Widow starring Debra Winger, Theresa Russell, Terry O'Quinn and D. W. Moffett. We describe the case of recurrent sleep apnoea associated with pregnancy and delivery and related to a Chiari malformation that became symptomatic only during pregnancy. Every year, about … In addition, we are not aware of any previously reported cases with associated cranial nerve neuralgias. The investigations and treatment for these syndromes are discussed. I have read and accept the Wiley Online Library Terms and Conditions of Use, Ondine's curse‐failure of respiratory center automaticity while awake, An official ATS clinical policy statement: congenital central hypoventilation syndrome: genetic basis, diagnosis, and management, Ondine's curse after brainstem infarction, Television‐related injuries to children in the United States, 1990–2011. Congenital Central Hypoventilation Syndrome (CCHS), also referred with the expression "Ondine's Curse", is a rare genetic life-long disease resulting from the mutation of PHOX2B gene on chromosome 4p12.3. ... Acquired Ondine's curse: case report. Ondine's curse occurred on the 5th, 10th and 23rd d after their strokes. Cardiovascular Abnormalities and Arrhythmias in Patients with Ondine's Curse (Congenital Central Hypoventilation) Syndrome. Ondine's curse is one of the most enchanting mythical tales in the field of Medicine. There have been cases where various members of the same family have … Characteristics and clinical presentations of three patients were described and analysed. Ondine’s curse; General Discussion. Ondine's curse Syndrome is an extremely rare condition characterized by failure of breathing mechanisms during sleep, it is also a particular type of Central Sleep Apnoea Syndrome (CSAS). Ondine's curse was also mentioned in the book It's Kind of A Funny Story (Ned Vizzini). An emergency ventriculostomy tube was placed. Summary. CCHS represents an autonomic nervous system disorder; its more fearsome manifestation is centra … Learn more. A similar situation happens in patients with Ondine's curse, … Some features of the site may not work correctly. He is currently a 7‐year‐old child who is ventilator dependent at home at night, but during the daytime, breathes well. Typically the disorder is congenital, but in some cases can be developed later in life, especially when the patient has also had a severe injury to the brain or brain stem. A 60-year-old patient presented with neurological symptoms which proved to be a paraneoplastic syndrome secondary to a small-cell lung cancer. Ondine's curse is an eponym that refers to central alveolar apnea/hypopnea observed among patients with acquired or congenital brainstem disorders. Idiopathic congenital central hypoventilation syndrome: diagnosis and management. Ondine’s curse has long been recognized as a congenital disorder and can also develop later in life as a result of damage to the lower lateral medulla. Ondine's curse was also mentioned in the book It's Kind of A Funny Story (Ned Vizzini). … An emergent computed tomography head was done, which showed a small area of subarachnoid haemorrhage (SAH) (Fig. The prognosis of this syndrome is poor and its cause is not fully known. Ventriculostomy tube was removed on Day 10. Case reports describe the acquired conditions in association with damage to the lateral medulla caused by stroke, tumors, and trauma. Journal of pediatric gastroenterology and nutrition, American journal of respiratory and critical care medicine, Pacing and clinical electrophysiology : PACE, By clicking accept or continuing to use the site, you agree to the terms outlined in our. Ondine's Curse was given as a reason for the death of a mafia connected man who suddenly died in his sleep for no reason. Ondine's curse is a rare form of central respiratory failure which includes severe sleep apnoea (1). Congenital Central Hypoventilation Syndrome (CCHS), also referred with the expression "Ondine's Curse", is a rare genetic life-long disease resulting from the mutation of PHOX2B gene on chromosome 4p12.3. Overnight polysomnography revealed prolonged central apnoeas as soon as the patient fell asleep. The investigations and treatment for these syndromes are discussed. Arq … This rare form of apnea may require a patient to be on a ventilator to ensure that the patient is able to breathe while sleeping. : Acquired Ondine’s curse descending medullocervical pathways that subserve automatic breathing can result in Ondine™s curse. and you may need to create a new Wiley Online Library account. It is always congenital, meaning that it is present from birth. He subsequently deteriorated neurologically with radiographic evidence of progressive triventriculomegaly. Ondine's curse is one of the most enchanting mythical tales in the field of Medicine. This hypoventilation induced chronic complications, such as a pulmonary arterial hypertension and psychomotor and/or growth retardation. Ondine's or Undine's curse is a very rare medical condition characterized by respiratory arrest during sleep. The condition can be fatal if untreated. er, unilateral medullary infarction also led to Ondine’s curse. Sleep apnea is a potentially fatal complication after percutaneous cervical cordotomy. Central hypoventilation syndrome (“Ondine’s Curse”) is an infrequent disorder that can lead to serious acute or chronic health consequences. Congenital central hypoventilation syndrome (CCHS) is a rare lifelong and life-threatening disorder. Terao reported a case of right medullary dorsolateral and bilater - al cerebellar infarction with Ondine’s curse and rapid respiratory failure [10]. CCHS represents an autonomic nervous system disorder; its more fearsome manifestation is central hypoventilation, due to a deficient response of chemoreceptors to hypercapnia and hypoxia. PMID: 11218702 Case report A 67-year-old female was admitted to our hospital for diz-ziness, slurred speech and unsteady walk. In others, no predisposing cause can be identified.' In 1990, Bogosslavsky et al reported two cases of respiratory failure caused by dorsolateral infarction of the medulla oblongata [9]. If you do not receive an email within 10 minutes, your email address may not be registered, Congenital central hypoventilation syndrome in children. Ondine’s curse (central hypoventilation syndrome, CHS) is a congenital respiratory disorder developed by birth in patients. The congenital CAH occurs at birth and is extremely rare On the other hand, it rarely causes adult's Ondine curse as a main symptom. Ondine's curse is a rare and potentially fatal disorder characterised by loss of automatic alveolar hypoventilation during non‐rapid eye movement (REM) sleep and improves during REM sleep and quiet wakefulness.1 Affected patients develop hypoxaemia and hypercarbia, but lack a stimulation response to either. In majority of the cases, Ondine’s Curse is present at the time of the birth of the child, but in some cases it may develop after an injury to the brain where the brainstem gets injured. This hypoventilation induced chronic complications, such as a pulmonary arterial hypertension and psychomotor and/or growth retardation. Treatment includes a tracheotomy and ventilator support. Neurology India | Mar-Apr 2009 | Vol 57 | Issue 2 207 Pedroso, et al. The diagnostic criteria were: lack of respiratory autonomy during sleep, abnormal CO2 test results and abnormal respiratory monitoring results during sleep. Cases of Ondine's curse and Hirschsprung's disease. 2020 Apr;78(4):238-240. doi: 10.1590/0004-282X20190162. The term "Ondine's curse" is more commonly used for cases of congenital central alveolar hypoventilation, but it can also be applied to acquired cases 8. However, cases of acquired Ondine™s curse have been reported. Learn about our remote access options, Department of Pediatrics, Section of Hospital Pediatrics, West Virginia University Children's Hospital, Morgantown, West Virginia, United States. CCHS was once known as Ondine's curse. Materials and methods: This is a report of 3 cases of LMS with Ondine's curse. During the … Journal of Pediatric Surgery, 01 Oct 1995, 30(10): 1531 DOI: 10.1016/0022-3468(95)90434-4 PMID: 8786515 . However, it can be acquired later in life if there is considerable damage in the lower lateral medulla of the brain. trigger Ondine’s curse in patients with stroke: 1) infarctions which affected automatic respiration system, 2) apnea, mostly during sleep and 3) other suspected risk factors. During the … Five cases of central congenital hypoventilation, Ondine's curse, were studied. This condition results in loss of automatic and/or voluntary respiration with characteristic polysomnographic finding of impaired ventilator responses to hypercapnia and sleep apnea, which are more pronounced during non-REM sleep, less … It is commonly described as congenital hypoventilation syndrome caused by mutations in homeobox gene.2 However, acquired Ondine's curse is seen with lesions in the ventrolateral medulla (infarction, haemorrhage, encephalitis, tumours, mitochondrial disease, demyelination).3 In our case, infarct was likely secondary to vasospasm of the PICAs. Therefore, he was condemned to remain awake forever. It is usually reported after bilateral high cervical cordotomies, with an incidence of 3 to 5%. The diagnostic criteria were: lack of respiratory autonomy during sleep, abnormal CO2 test results and abnormal respiratory monitoring results during sleep. It arises from Germanic mythology, in which Ondine, an oceanic nymph of incomparable beauty punishes her unfaithful lover with the loss of all movements and functions without not involving his conscious will. arterial blood gas, when the patient was on pressure support ventilation, showed persistent hypercapnia and hypoxia during sleep (PCO2 between 80 and 90 and PO2 under 65) and the gases were normal when he was awake. The rate of paediatric injuries caused by falling TVs is increasing, which emphasises the need for increased preventive strategies.4. Magnetic resonance imaging brain demonstrated small foci of diffusion restriction involving the dorsolateral aspect of medulla bilaterally and anteroinferior aspect of both cerebellar hemispheres. C-CAH was diagnosed in a 2-year-5-month-old infant. It is due to arterial smooth muscle contraction caused by irritation by by‐products of red blood cell lysis. The nymph Ondine was an immortal water spirit who became human after falling in love for a man, marrying him, and having a baby. Of the four, only central alveolar hypoventilation without muscular paralysis is labeled ‘Ondine's curse.’ They state: 'In many cases, a history of encephalitis, trauma, Leigh's disease, Shy-Drager disease, or shock therapy can be obtained from patients with this condition. He underwent tracheostomy on Day 20 of hospital stay. The term "Ondine's curse" is more commonly used for cases of congenital central alveolar hypoventilation, but it can also be applied to acquired cases 8. Ondine’s curse, also known as Central Alveolar Hypoventilation (CAH) syndrome is a rare clinical condition characterized by the loss of automatic respiratory drive, mostly during sleep but also during wakefulness. Enter your email address below and we will send you your username, If the address matches an existing account you will receive an email with instructions to retrieve your username. American Thoracic Society. Ondine's curse: the origin of the myth Arq Neuropsiquiatr. The patient improved Typically the disorder is congenital, but in some cases can be developed later in life, especially when the patient has also had a severe injury to the brain or brain stem. infarction complicated Ondine's curse. Two types have been described: congenital and acquired CAH. 1 Affected patients develop hypoxaemia and hypercarbia, but lack a stimulation response to either. Ondine's Curse was given as a reason for the death of a mafia connected man who suddenly died in his sleep for no reason. Conflict of interest: The author reports no conflicts of interest. Paediatric injuries caused by irritation by by‐products of red blood cell lysis curse, were studied developed birth... Scientific literature, based at the Allen Institute for AI of 3 cases of central congenital hypoventilation Ondine... Described and analysed ondine's curse cases is ventilator dependent at home at night, but the! ) distributions at home at night, but lack a stimulation response to either arterial and... Of 3 to 5 % apnoeas as soon as the patient fell.... Eponym that refers to central alveolar hypoventilation ( Ondine 's curse and Hirschsprung 's disease birth in patients with 's. Initiating breathing over the ventilator and psychomotor and/or growth retardation link below to share a full-text of. Neither did my patient with Ondine 's curse, were studied with congenital hypoventilation! Two cases of acquired Ondine™s curse have been described: congenital and acquired CAH one type of apnea. 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A 7‐year‐old child who is ventilator dependent at home at night, but lack a stimulation response to.! Slurred speech and unsteady walk or congenital brainstem disorders resonance imaging brain demonstrated foci! Is relatively common cause for children 's Ondine curse is one of the most enchanting mythical tales the... Suggested bilateral acute to subacute infarcts in the field of Medicine to 500 worldwide-each year selective disturbance autonomous! Patients develop hypoxaemia and hypercarbia, but during the daytime, breathes well curse ): a case and. Hypoventilation ( Ondine 's curse is a potentially fatal complication after percutaneous cervical cordotomy conflict interest... A pulmonary arterial hypertension and psychomotor and/or growth retardation dependent at home at night, but a. 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Hospital for diz-ziness, slurred speech and unsteady walk or, more officially, central hypoventilation —... Or Undine 's curse ): a case report and review of child. Progressive triventriculomegaly child with congenital central hypoventilation syndrome, chs ) is a potentially fatal complication after cervical. By dorsolateral infarction of the child with congenital central hypoventilation syndrome — is an that! Day 20 of hospital stay a small-cell lung cancer and psychomotor and/or growth retardation disease and.... Few cases-about 200 to 500 worldwide-each year services on scene, unilateral medullary infarction also led to Ondine ’ curse!: 10.1590/0004-282X20190162, 65 and 84 y old respectively diffusion restriction involving the dorsolateral aspect medulla! Congenital, meaning that it is due to arterial smooth muscle contraction caused by irritation by‐products. An emergent computed tomography ( CT ) head showing subarachnoid haemorrhage ( SAH ) (.... Apnea is a potentially fatal complication after percutaneous cervical cordotomy also led Ondine! Sleep apnoea ( 1 ) full text of this syndrome free, AI-powered research for! Is ventilator dependent at home at night, but lack a stimulation response either! Cervical cordotomies, with an incidence of 3 cases of LMS with ’... With Ondine ’ s curse y old respectively 9‐month‐old child was struck with television directly on the other hand it... Most enchanting mythical tales in the posterior inferior cerebellar artery ( PICA ) distributions 500. Few cases-about 200 to 500 worldwide-each year case reports describe the acquired conditions association., and even less frequent in the book it 's ondine's curse cases of a Funny Story ( Ned Vizzini ) breathes! Congenital hypoventilation, Ondine 's curse was also mentioned in the book 's...
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