Ondine’s Curse with Accompanying Trigeminal and Glossopharyngeal Neuralgia Secondary to Medullary Telangiectasia

Kapnadak, Siddhartha G.; Mikolaenko, Ivan; Enfield, Kyle B.; Gress, Daryl R.; Nathan, Barnett R.

doi:10.1007/s12028-009-9321-x

Cited by 15 publications

(7 citation statements)

References 12 publications

(13 reference statements)

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“…Similar set of symptoms, also referred to as Ondine's curse, was described in an adult female patient admitted to the hospital for diagnostic of relapsing acute severe central hypoventilation, coexisting with trigeminal and glossopharyngeal neuralgia. Although the symptoms she presented with were almost identical to those present in the 2 years old girl case, the patient was diagnosed with medullar capillary telangiectasia and was successfully treated with diaphragmatic pacing system placement (Kapnadak et al 2010).…”

Section: Resultsmentioning

confidence: 65%

Ondine’s Curse – Genetic and Iatrogenic Central Hypoventilation as Diagnostic Options in Forensic Medicine

Susło

Trnka

Siewiera

et al. 2015

Advances in Experimental Medicine and Biology

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In the Nordic mythology a man lost his ability to breathe without remembering it after he was cursed by water nymph - referred to as 'Ondine's curse' - and then he died as soon as he fell asleep. Family medicine specialists are familiar with many sleeping disorders that their patients commonly call by the term Ondine's Curse. In medical sciences this term is historically related to the group of conditions that have as the common denominator seemingly spontaneous onset of life-threatening hypoventilation. The physiology and genetics specialists focus mainly on congenital central hypoventilation syndrome (CCHS), which was proven to be linked to several genetic mutations. Anesthesiologists tend to be more interested in similarly manifesting iatrogenic condition. Typically, patients that were previously subjected to general anesthesia, after temporarily waking up and regaining the spontaneous respiratory drive, later fall back into unconsciousness and develop hypoventilation. Anesthesiologists also call it Ondine's curse because of the sudden and unexpected sleep onset. The iatrogenic Ondine's curse is proven to be precipitated by delayed anesthetics release from patients' fat tissue - where it was deposited at the time general anesthesia was administered - back into bloodstream. Forensic medicine has to consider the latter form of Ondine's curse called scenario more often, as they investigate sudden deaths related to surgery and general anesthesia in the post-operational care period. These cases may also fall into the category of medical malpractice-related deaths.

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Section: Resultsmentioning

confidence: 65%

Ondine’s Curse – Genetic and Iatrogenic Central Hypoventilation as Diagnostic Options in Forensic Medicine

Susło

Trnka

Siewiera

et al. 2015

Advances in Experimental Medicine and Biology

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“…[13][14][15] Symptoms may be related to the size of the lesion. In a study by Sayama et al, 9 7/105 BCTs (6.7%) were Ͼ1 cm; 2 of these lesions (28.6%) were identified as likely causing symptoms.…”

Section: Discussionmentioning

confidence: 99%

Susceptibility-Weighted MR Imaging for Diagnosis of Capillary Telangiectasia of the Brain

El-Koussy¹,

Schroth²,

Gralla³

et al. 2011

AJNR Am J Neuroradiol

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BACKGROUND AND PURPOSE:BCT is a benign entity, whose appearance on conventional MR imaging makes its differentiation from neoplastic, inflammatory, or subacute ischemic disease challenging. SWI is sensitive to susceptibility effects from deoxyhemoglobin with excellent spatial resolution. Only scarce case reports have described the utility of SWI in cases of BCT. Our aim was to show the diagnostic value of SWI applied to a larger series of cases.

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“…Therapeutic symptomatic options range from pharmacologic approaches [5] and use of bilevel positive airway pressure to implantation of diaphragmatic pacemaker [8, 9]. As was seen in patients 2 and 3, spontaneous recovery can occur [10].…”

Section: Discussionmentioning

confidence: 99%

Central Neurogenic Respiratory Failure: A Challenging Diagnosis

et al. 2011

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Background: Central nervous system lesions are rare causes of respiratory failure. Simple observation of the breathing pattern can help localize the lesion, but the examiner needs to be aware of potential pitfalls such as metabolic or pulmonary alterations. Methods: We describe 3 cases in which central neurogenic respiratory failure occurred simultaneously with other alterations or in an unusual presentation. Results: All patients were diagnosed with central neurogenic respiratory failure and treated for it with good recovery. Conclusion: Central neurogenic respiratory failure is a challenging diagnosis and needs to be reminded in difficult-to-wean patients carrying inconclusive evidences of metabolic or pulmonary alterations.

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Ondine’s Curse with Accompanying Trigeminal and Glossopharyngeal Neuralgia Secondary to Medullary Telangiectasia

Abstract: Placement of a diaphragmatic pacing system was warranted and became lifesaving as the patient was deemed to be severely incapacitated by chronic ventilatory insufficiency.

Cited by 15 publications

References 12 publications

Ondine’s Curse – Genetic and Iatrogenic Central Hypoventilation as Diagnostic Options in Forensic Medicine

Ondine’s Curse – Genetic and Iatrogenic Central Hypoventilation as Diagnostic Options in Forensic Medicine

Susceptibility-Weighted MR Imaging for Diagnosis of Capillary Telangiectasia of the Brain

Central Neurogenic Respiratory Failure: A Challenging Diagnosis

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