ArticleAnnals of neurology2022
ZSCAN1 Autoantibodies Are Associated with Pediatric Paraneoplastic ROHHAD.
Article in Annals of neurology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 37 papers.
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Who cites it
37 citing papers in PubMed, 48 citations in OpenAlex.
- Use of Fluid Biomarkers in NMOSD and MOGAD: Clinical and Research Applications.Neurology(R) neuroimmunology & neuroinflammation · 2026Review
- A 2.5-year-old obese girl with persistent hypoventilation post-pneumonia: when is it not just the lungs?Breathe (Sheffield, England) · 2026Article
- Chromatin regulator expression patterns predict prognosis and guide neoadjuvant therapy in esophageal squamous cell carcinoma.Fundamental research · 2026Article
- AEGIS reveals epitope- and clone-resolved convergence of CNS B and T cell autoreactivity in ROHHAD.bioRxiv : the preprint server for biology · 2026Article
- Gain-of-function mutation inbioRxiv : the preprint server for biology · 2026Article
- Immune profiling links autoimmune hepatitis to human herpesvirus 6 and relaxin receptor antigens.The Journal of experimental medicine · 2026Article
- ROHHAD syndrome: an interdisciplinary perspective.Frontiers in pediatrics · 2026Article
- Case Report: First description of an intracranial "NET" in ROHHAD syndrome; time to rename to the ROHHAD-NeCT syndrome?Frontiers in endocrinology · 2026Article
- PhIP-Seq: unveiling the complexity of antibody repertoires in health and disease.Frontiers in immunology · 2026Review
- ROHHAD NET in Retrospect: Key Lessons Learned from Five Cases.Indian journal of pediatrics · 2025Article
- Rapid-onset obesity, hypothalamic dysfunction, hypoventilation, and autonomic dysregulation as a paraneoplastic syndrome in neuroblastoma: case report.Translational pediatrics · 2025Article
- Genetic evaluation of five patients with ROHHAD-NET using whole genome sequencing and optical genome mapping.Orphanet journal of rare diseases · 2025Article
- ROHHAD syndrome in an adult: presence of ZSCAN1 antibodies.ERJ open research · 2025Article
- Permissive central tolerance plus defective peripheral checkpoints license pathogenic memory B cells in CASPR2-antibody encephalitis.Science advances · 2025Article
- Novel Meningoencephalomyelitis Associated With Vimentin IgG Autoantibodies.JAMA neurology · 2025Article
- Management of obesity in an individual with ROHHAD syndrome with semaglutide 2.4 mg/week: a case report.Clinical autonomic research : official journal of the Clinical Autonomic Research Society · 2025Article
- Ten Years ofNeurology(R) neuroimmunology & neuroinflammation · 2025Article
- Ocular Mucous Membrane Pemphigoid Demonstrates a Distinct Autoantibody Profile from Those of Other Autoimmune Blistering Diseases: A Preliminary Study.Antibodies (Basel, Switzerland) · 2024Article
- Autoimmunity related to anti-NaEndocrine journal · 2024Review
- Novel autoantibody targets identified in patients with autoimmune hepatitis (AIH) by PhIP-Seq reveals pathogenic insights.medRxiv : the preprint server for health sciences · 2024Article
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Authors and funding
24 authors at 7 institutions in 2 countries.
Funding
Abstract
objectiveRapid-onset Obesity with Hypothalamic Dysfunction, Hypoventilation and Autonomic Dysregulation (ROHHAD), is a severe pediatric disorder of uncertain etiology resulting in hypothalamic dysfunction and frequent sudden death. Frequent co-occurrence of neuroblastic tumors have fueled suspicion of an autoimmune paraneoplastic neurological syndrome (PNS); however, specific anti-neural autoantibodies, a hallmark of PNS, have not been identified. Our objective is to determine if an autoimmune paraneoplastic etiology underlies ROHHAD.
methodsImmunoglobulin G (IgG) from pediatric ROHHAD patients (n = 9), non-inflammatory individuals (n = 100) and relevant pediatric controls (n = 25) was screened using a programmable phage display of the human peptidome (PhIP-Seq). Putative ROHHAD-specific autoantibodies were orthogonally validated using radioactive ligand binding and cell-based assays. Expression of autoantibody targets in ROHHAD tumor and healthy brain tissue was assessed with immunohistochemistry and mass spectrometry, respectively.
resultsAutoantibodies to ZSCAN1 were detected in ROHHAD patients by PhIP-Seq and orthogonally validated in 7/9 ROHHAD patients and 0/125 controls using radioactive ligand binding and cell-based assays. Expression of ZSCAN1 in ROHHAD tumor and healthy human brain tissue was confirmed.
interpretationOur results support the notion that tumor-associated ROHHAD syndrome is a pediatric PNS, potentially initiated by an immune response to peripheral neuroblastic tumor. ZSCAN1 autoantibodies may aid in earlier, accurate diagnosis of ROHHAD syndrome, thus providing a means toward early detection and treatment. This work warrants follow-up studies to test sensitivity and specificity of a novel diagnostic test. Last, given the absence of the ZSCAN1 gene in rodents, our study highlights the value of human-based approaches for detecting novel PNS subtypes. ANN NEUROL 2022;92:279-291.
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