Literature DB >> 22715259

Rapid-onset obesity with hypothalamic dysfunction, hypoventilation and autonomic dysregulation (ROHHAD): a case with additional features and review of the literature.

H B Chew1, L H Ngu, W T Keng.   

Abstract

A rare syndrome of rapid-onset obesity with hypothalamic dysfunction, hypoventilation and autonomic dysregulation (ROHHAD) has been recently described. We report the first patient with this syndrome in Southeast Asia and review reported cases to date. Our patient was good health with normal development until the age of 2. He then developed hyperphagic obesity, hypersomnolence, seizures, alveolar hypoventilation, central hypothyroidism, sodium and water dysregulation, gastrointestinal dysmotility, strabismus, disordered temperature and irregular heart rate, altered sweating, delayed puberty, mental retardation and recurrent respiratory tract infections. The cardiomyopathy with heart failure and abnormal cerebral spinal fluid (CSF) neurotransmitter analysis present in our patient have not been reported previously. Tumours of the sympathetic nervous system are known to be associated with this syndrome but had not been found in our patient at the time of reporting. We highlight the difficulty of achieving the diagnosis of ROHHAD syndrome and its overlap with other well-established disease entities. The mortality and morbidity resulting from the high incidence of cardiorespiratory arrest may be prevented by early ventilatory support.

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Mesh:

Year:  2011        PMID: 22715259      PMCID: PMC3062047          DOI: 10.1136/bcr.02.2010.2706

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  21 in total

Review 1.  Idiopathic hypothalamic dysfunction: a paraneoplastic syndrome?

Authors:  K Nunn; R Ouvrier; T Sprague; S Arbuckle; M Docker
Journal:  J Child Neurol       Date:  1997-06       Impact factor: 1.987

2.  Late onset hypoventilation syndrome: is there a spectrum of idiopathic hypoventilation syndromes?

Authors:  D Gothi; J M Joshi
Journal:  Indian J Chest Dis Allied Sci       Date:  2005 Oct-Dec

3.  The French Congenital Central Hypoventilation Syndrome Registry: general data, phenotype, and genotype.

Authors:  Ha Trang; Michel Dehan; François Beaufils; Isabelle Zaccaria; Jeanne Amiel; Claude Gaultier
Journal:  Chest       Date:  2005-01       Impact factor: 9.410

Review 4.  Hypothalamic dysfunction in a child: a distinct syndrome? Report of a case and review of the literature.

Authors:  F Proulx; M L Weber; R Collu; M Lelièvre; A Larbrisseau; M Delisle
Journal:  Eur J Pediatr       Date:  1993-06       Impact factor: 3.183

5.  Delineation of late onset hypoventilation associated with hypothalamic dysfunction syndrome.

Authors:  Loic De Pontual; Delphine Trochet; Sophie Caillat-Zucman; Othman A Abou Shenab; Pierre Bougneres; Yanick Crow; Steve Cunningham; Blandine Esteva; Lada Cindro Heberle; Juliane Leger; Graziella Pinto; Michel Polak; Magdy Helmy Shafik; Christian Straus; Ha Trang; Arnold Munnich; Stanislas Lyonnet; Isabelle Desguerre; Jeanne Amiel
Journal:  Pediatr Res       Date:  2008-12       Impact factor: 3.756

6.  Endocrine manifestations of the rapid-onset obesity with hypoventilation, hypothalamic, autonomic dysregulation, and neural tumor syndrome in childhood.

Authors:  Pierre Bougnères; Letitia Pantalone; Agnès Linglart; Anya Rothenbühler; Catherine Le Stunff
Journal:  J Clin Endocrinol Metab       Date:  2008-07-15       Impact factor: 5.958

7.  PHOX2B germline and somatic mutations in late-onset central hypoventilation syndrome.

Authors:  Delphine Trochet; Loïc de Pontual; Christian Straus; David Gozal; Ha Trang; Pierre Landrieu; Arnold Munnich; Stanislas Lyonnet; Claude Gaultier; Jeanne Amiel
Journal:  Am J Respir Crit Care Med       Date:  2007-12-13       Impact factor: 21.405

8.  Idiopathic hypothalamic dysfunction with dilated unresponsive pupils: report of two cases.

Authors:  K N North; R A Ouvrier; C A McLean; I J Hopkins
Journal:  J Child Neurol       Date:  1994-07       Impact factor: 1.987

9.  Rapid-onset obesity with hypothalamic dysfunction, hypoventilation, and autonomic dysregulation presenting in childhood.

Authors:  Diego Ize-Ludlow; Juliette A Gray; Mark A Sperling; Elizabeth M Berry-Kravis; Jeff M Milunsky; I Sadaf Farooqi; Casey M Rand; Debra E Weese-Mayer
Journal:  Pediatrics       Date:  2007-07       Impact factor: 7.124

10.  PRIMARY ALVEOLAR HYPOVENTILATION SYNDROME (ONDINE'S CURSE).

Authors:  L S FISHMAN; J H SAMSON; D R SPERLING
Journal:  Am J Dis Child       Date:  1965-08
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  17 in total

Review 1.  Genetic and Syndromic Causes of Obesity and its Management.

Authors:  Ildiko H Koves; Christian Roth
Journal:  Indian J Pediatr       Date:  2017-11-27       Impact factor: 1.967

2.  Images: Sleep-disordered breathing and hypoventilation in a child with obesity and hypothalamic dysfunction.

Authors:  Radhika Ghosh; Manisha Malik; Tanicia C Daley; Ajay S Kasi
Journal:  J Clin Sleep Med       Date:  2022-01-01       Impact factor: 4.062

3.  Improved Behavior and Neuropsychological Function in Children With ROHHAD After High-Dose Cyclophosphamide.

Authors:  Lisa A Jacobson; Shruti Rane; Lisa J McReynolds; Diana A Steppan; Allen R Chen; Ido Paz-Priel
Journal:  Pediatrics       Date:  2016-06-16       Impact factor: 7.124

4.  Nocturnal Anxiety in a Youth with Rapid-onset Obesity, Hypothalamic Dysfunction, Hypoventilation, and Autonomic Dysregulation (ROHHAD).

Authors:  Eugene Grudnikoff; Carmel Foley; Claudette Poole; Eva Theodosiadis
Journal:  J Can Acad Child Adolesc Psychiatry       Date:  2013-08

5.  ROHHAD Syndrome: Reasons for Diagnostic Difficulties in Obesity.

Authors:  Pınar Kocaay; Zeynep Şıklar; Emine Çamtosun; Tanıl Kendirli; Merih Berberoğlu
Journal:  J Clin Res Pediatr Endocrinol       Date:  2014-12

6.  ROHHAD syndrome and evolution of sleep disordered breathing.

Authors:  Diana Reppucci; Jill Hamilton; E Ann Yeh; Sherri Katz; Suhail Al-Saleh; Indra Narang
Journal:  Orphanet J Rare Dis       Date:  2016-07-30       Impact factor: 4.123

7.  Rapid-onset obesity, hypoventilation, hypothalamic dysfunction, autonomic dysregulation and neuroendocrine tumor syndrome with a homogenous enlargement of the pituitary gland: a case report.

Authors:  Lama Aljabban; Lina Kassab; Nour Alhuda Bakoura; Mohammad Fayez Alsalka; Ismaeil Maksoud
Journal:  J Med Case Rep       Date:  2016-11-22

8.  Demographic, Clinical, and Immunologic Features of 389 Children with Opsoclonus-Myoclonus Syndrome: A Cross-sectional Study.

Authors:  Michael R Pranzatelli; Elizabeth D Tate; Nathan R McGee
Journal:  Front Neurol       Date:  2017-09-11       Impact factor: 4.003

9.  Rapid-onset obesity, hypoventilation, hypothalamic dysfunction, autonomic dysregulation syndrome.

Authors:  Ismaeil Maksoud; Lina Kassab
Journal:  Avicenna J Med       Date:  2015 Jul-Sep

Review 10.  Less known aspects of central hypothyroidism: Part 2 - Congenital etiologies.

Authors:  Salvatore Benvenga; Marianne Klose; Roberto Vita; Ulla Feldt-Rasmussen
Journal:  J Clin Transl Endocrinol       Date:  2018-09-27
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