The Use of Fluoxetine in an Adolescent with Prader-Willi Syndrome

Dech, B.; Budow, Lauren

doi:10.1097/00004583-199103000-00020

Cited by 63 publications

(27 citation statements)

References 21 publications

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“…The 5-HT-induced restoration of a stable RRG activity in Ndn-KO suggests that pharmacological treatments targeting the 5-HT system might soften their breathing deficits. In PWS, treatments with the selective 5-HT uptake inhibitor fluoxetine alleviates some specific symptoms (Dech and Budow, 1991;Warnock and Kestenbaum, 1992;Benjamin and Buot-Smith, 1993;Hellings and Warnock, 1994;Goldstone, 2004), but nothing has been reported on breathing symptoms. However, chronic treatments with fluoxetine increase the respiratory response to hypercapnia in rats (Taylor et al, 2004), and short-term fluoxetine treatment improves respiration of obese non-PWS patients suffering from apnea/hypopnea periods during sleep (Kopelman et al, 1992).…”

Section: Central Respiratory Deficits and 5-ht Alterationsmentioning

confidence: 99%

Necdin Plays a Role in the Serotonergic Modulation of the Mouse Respiratory Network: Implication for Prader-Willi Syndrome

Zanella¹,

Watrin²,

Mébarek³

et al. 2008

J. Neurosci.

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Prader-Willi syndrome is a neurogenetic disease resulting from the absence of paternal expression of several imprinted genes, including NECDIN. Prader-Willi children and adults have severe breathing defects with irregular rhythm, frequent sleep apneas, and blunted respiratory regulations. For the first time, we show that Prader-Willi infants have sleep apneas already present at birth. In parallel, in wild-type and Necdin-deficient mice, we studied the respiratory system with in vivo plethysmography, in vitro electrophysiology, and pharmacology. Because serotonin is known to contribute to CNS development and to affect maturation and function of the brainstem respiratory network, we also investigated the serotonergic system with HPLC, immunohistochemistry, Rabies virus tracing approaches, and primary culture experiments. We report first that Necdin-deficiency in mice induces central respiratory deficits reminiscent of Prader-Willi syndrome (irregular rhythm, frequent apneas, and blunted respiratory regulations), second that Necdin is expressed by medullary serotonergic neurons, and third that Necdin deficiency alters the serotonergic metabolism, the morphology of serotonin vesicles in medullary serotonergic neurons but not the number of these cells. We also show that Necdin deficiency in neonatal mice alters the serotonergic modulation of the respiratory rhythm generator. Thus, we propose that the lack of Necdin expression induces perinatal serotonergic alterations that affect the maturation and function of the respiratory network, inducing breathing deficits in mice and probably in Prader-Willi patients.

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Section: Central Respiratory Deficits and 5-ht Alterationsmentioning

confidence: 99%

Necdin Plays a Role in the Serotonergic Modulation of the Mouse Respiratory Network: Implication for Prader-Willi Syndrome

Zanella¹,

Watrin²,

Mébarek³

et al. 2008

J. Neurosci.

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“…Dech & Budow descreveram caso de uma portadora de SPW que apresentou grande melhora no controle do peso e moderada melhora na tricotilomania e tricofagia após introdução de fluoxetina (43). Outros relatos de caso mostraram que a fluoxetina, além de auxiliar no controle do peso, reduziu o comportamento de automutilação (44)(45)(46).…”

Section: Drogas Antiobesidadeunclassified

Abordagem terapêutica da obesidade na Síndrome de Prader-Willi

Carvalho

Cercato

Almeida

et al. 2007

Arq Bras Endocrinol Metab

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RESUMOA Síndrome de Prader-Willi (SPW) é uma doença complexa, multissistêmica, caracterizada por hipotonia, retardo mental, características dismórficas, hiperfagia e compulsão alimentar devido à disfunção hipotalâ-mica. SPW ocorre pela perda de função de genes localizados no cromossomo 15q11-13, região que sofre imprinting genômico. Obesidade é a principal causa de morbidade e mortalidade entre pacientes com SPW. O objetivo desta revisão é analisar as opções terapêuticas disponíveis para o tratamento da obesidade na SPW, incluindo a terapia farmacológica e o tratamento cirúrgico.

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“…17 Pharmacological management of behavior and psychiatric problems are often used, particularly in depression and obsessive compulsion symptoms. Selective serotonin reuptake inhibitors are particularly effective, 52 fluoxetine is currently popularly used, 53,54 risperidone is effective in treating psychosis. 55 Most dental problems with PWS are decreased salivary flow and mouth breathing, these promote to dental decay, periodontal disease, crowded arches, severe wear of tooth.…”

Section: 51mentioning

confidence: 99%

Oral and General Findings: Management of Prader-Willi Syndrome

Umapathy¹,

K²,

Kathariya³

et al. 2013

JIAOMR

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It is a rare neurogenic illeness; because of its clinical characteristics it is important for dentists. PWS is first know human genomic imprinting disorder, microdeletion syndrome. It is caused by the absence of certain paternally inherited genes on long arm of chromosome 15. The clinical picture of Prader-Willi syndrome is complex and alters with age. It is mostly due to impaired hypothalamus function. The characteristic features include short stature; muscular hypotonia; hypopigmentation of the skin and hair; a triangular facies, frequently with a small mouth with a thin upper lip and down-turned corners; and diminished secretion of thick and sticky saliva. The article describes the phenotypic features of Prader-Willi syndrome (PWS) and their impact on the oral health status Primary care by pediatric dentist's play an important role in children's with PWS.

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The Use of Fluoxetine in an Adolescent with Prader-Willi Syndrome

Cited by 63 publications

References 21 publications

Necdin Plays a Role in the Serotonergic Modulation of the Mouse Respiratory Network: Implication for Prader-Willi Syndrome

Necdin Plays a Role in the Serotonergic Modulation of the Mouse Respiratory Network: Implication for Prader-Willi Syndrome

Abordagem terapêutica da obesidade na Síndrome de Prader-Willi

Oral and General Findings: Management of Prader-Willi Syndrome

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