Literature DB >> 23292759

Intrathecal synthesis of oligoclonal bands in rapid-onset obesity with hypothalamic dysfunction, hypoventilation, and autonomic dysregulation syndrome: new evidence supporting immunological pathogenesis.

Stefano Sartori1, Elena Priante, Andrea Pettenazzo, Piero Marson, Agnese Suppiej, Franca Benini, Giorgio Perilongo, Irene Toldo.   

Abstract

Rapid-onset obesity with hypothalamic dysfunction, hypoventilation, and autonomic dysregulation syndrome (ROHHADS) is a rare, but potentially lethal, pediatric disorder. To date, nearly 80 patients have been reported in the literature; however, the etiopathogenesis is still unclear and debated. Both genetic and paraneoplastic or immune-mediated causes have been supposed to be involved in this syndrome. Nonetheless, at this time, a diagnostic biomarker is not available and diagnosis is based exclusively on clinical criteria. Aiming to establish the immune-mediated pathogenesis, we report 2 children with a clinical picture consistent with ROHHADS and whose cerebrospinal fluid analysis disclosed an intrathecal synthesis of oligoclonal bands. Even if many aspects remain to be explained, this finding suggests that ROHHADS could share similar pathogenetic mechanisms with other immune-mediated central nervous system disorders, and even more important, it might pave the way to a therapeutic chance for these patients by means of immunotherapy.

Entities:  

Keywords:  ROHHADS; central hypoventilation; hypothalamic dysfunction; oligoclonal bands

Mesh:

Substances:

Year:  2013        PMID: 23292759     DOI: 10.1177/0883073812469050

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  12 in total

1.  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

2.  Obesity Does Not Influence Outcomes in Hepatocellular Carcinoma Patients following Curative Hepatectomy.

Authors:  Zhe Guo; Jun Zhang; Jing-Hang Jiang; Le-Qun Li; Bang-De Xiang
Journal:  PLoS One       Date:  2015-05-12       Impact factor: 3.240

3.  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

4.  Rapid-Onset Obesity with Hypoventilation, Hypothalamic, Autonomic Dysregulation, and Neuroendocrine Tumors (ROHHADNET) Syndrome: A Systematic Review.

Authors:  Jiwon M Lee; Jaewon Shin; Sol Kim; Heon Yung Gee; Joon Suk Lee; Do Hyeon Cha; John Hoon Rim; Se-Jin Park; Ji Hong Kim; Ahmet Uçar; Andreas Kronbichler; Keum Hwa Lee; Jae Il Shin
Journal:  Biomed Res Int       Date:  2018-11-21       Impact factor: 3.411

5.  Natural history of ROHHAD syndrome: development of severe insulin resistance and fatty liver disease over time.

Authors:  Abdel Wahab Jalal Eldin; Dilara Tombayoglu; Laura Butz; Alison Affinati; Rasimcan Meral; Mehmet Selman Ontan; Kelly Walkovich; Maria Westerhoff; Jeffrey W Innis; Neehar D Parikh; Elif A Oral
Journal:  Clin Diabetes Endocrinol       Date:  2019-07-09

6.  Case Report: COVID-19-Associated ROHHAD-Like Syndrome.

Authors:  Irina N Artamonova; Natalia A Petrova; Natalia A Lyubimova; Natalia Yu Kolbina; Alexander V Bryzzhin; Alexander V Borodin; Tatyana A Levko; Ekaterina A Mamaeva; Tatiana M Pervunina; Elena S Vasichkina; Irina L Nikitina; Anna M Zlotina; Alexander Yu Efimtsev; Mikhail M Kostik
Journal:  Front Pediatr       Date:  2022-03-31       Impact factor: 3.418

7.  Rapid-Onset Obesity with Hypothalamic Dysfunction, Hypoventilation, and Autonomic Dysregulation (ROHHAD): exome sequencing of trios, monozygotic twins and tumours.

Authors:  Sarah F Barclay; Casey M Rand; Lauren A Borch; Lisa Nguyen; Paul A Gray; William T Gibson; Richard J A Wilson; Paul M K Gordon; Zaw Aung; Elizabeth M Berry-Kravis; Diego Ize-Ludlow; Debra E Weese-Mayer; N Torben Bech-Hansen
Journal:  Orphanet J Rare Dis       Date:  2015-08-25       Impact factor: 4.123

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

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

Review 9.  Genetic Predisposition to Neuroblastoma.

Authors:  Erin K Barr; Mark A Applebaum
Journal:  Children (Basel)       Date:  2018-08-31

10.  ROHHAD Syndrome, a Rare Cause of Hypothalamic Obesity: Report of Two Cases

Authors:  Ülkü Gül Şiraz; Deniz Ökdemir; Gül Direk; Leyla Akın; Nihal Hatipoğlu; Mustafa Kendirci; Selim Kurtoğlu
Journal:  J Clin Res Pediatr Endocrinol       Date:  2018-03-19
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