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Co-occurring hydrocephalus in autism spectrum disorder: a Danish population-based cohort study
Tina Nørgaard Munch
Copenhagen University Hospital
Corresponding Author
ORCiD: https://orcid.org/0000-0001-5938-000X
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Background The association between autism spectrum disorder and hydrocephalus is not well understood, despite demonstrated links between autism spectrum disorder and cerebrospinal fluid abnormalities. Based on the hypothesis that autism spectrum disorder and hydrocephalus may, at least in some cases, be two manifestations of a shared congenital brain pathology, we investigated the potential association between autism spectrum disorder and hydrocephalus in a large Danish population-based cohort.
Methods Patients and controls were obtained from the Lundbeck Foundation Initiative for Integrative Psychiatric Research iPSYCH2012 case-cohort, which includes all patients with selected psychiatric disorders born in Denmark 1981-2005 along with randomly selected population controls (end of follow-up, December 31, 2016). The associations between individual psychiatric disorders and hydrocephalus were estimated using binary logistic regression with adjustment for age and sex.
Results The cohort consisted of 86,571 individuals, of which 14,654 were diagnosed with autism spectrum disorder, 28,606 were population controls, and the remaining were diagnosed with other psychiatric disorders. We identified 201 hydrocephalus cases; 68 among autism spectrum disorder patients and 40 among controls (OR 3.77, 95% CI 2.48-5.78), which corresponds to an absolute risk of 0,46 %. The autism spectrum disorder-hydrocephalus association was significant over the entire subgroup spectrum of autism spectrum disorder.
Conclusions Given the considerable risk of hydrocephalus among patients with autism spectrum disorder, we suggest that patients with autism spectrum disorder should be evaluated for co-occurring hydrocephalus on a routine basis as timely neurosurgical intervention is important. Likewise, attention must be paid to traits of autism spectrum disorder in children with hydrocephalus. The results of this study call for future investigations on a potential shared aetiology between hydrocephalus and autism spectrum disorder, including the role abnormal CSF dynamics in the pathogenesis of autism spectrum disorder.
Keywords
Autism Spectrum Disorder, Hydrocephalus, Epidemiology, Cohort, Congenital
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Editorial decision: Major revision
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Received 19 Oct, 2020
Reviewer #2 agreed
On 08 Oct, 2020
Review #1 received
Received 29 Sep, 2020
Reviewer #1 agreed
On 08 Sep, 2020
Reviewers invited
Invitations sent on 02 Sep, 2020
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Subject Areas
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This preprint is under consideration at Journal of Neurodevelopmental Disorders. A preprint is a preliminary version of a manuscript that has not completed peer review at a journal. Research Square does not conduct peer review prior to posting preprints. The posting of a preprint on this server should not be interpreted as an endorsement of its validity or suitability for dissemination as established information or for guiding clinical practice.
Learn more about In Review
Research
Co-occurring hydrocephalus in autism spectrum disorder: a Danish population-based cohort study
Tina Nørgaard Munch
Copenhagen University Hospital
Corresponding Author
ORCiD: https://orcid.org/0000-0001-5938-000X
Badges
Prescreen
This manuscript passed our Prescreen assessment
Complete author information
Appropriate declaration statements
Potential risks to human health
Prescreen
Peer Review Timeline
CURRENT STATUS: Under Review
Version 2
Posted 20 Jan, 2021
No community comments so far
Review #1 received
Received 26 Feb, 2021
Reviewer #1 agreed
On 15 Jan, 2021
Reviewers invited
Invitations sent on 13 Jan, 2021
Editor assigned
On 12 Jan, 2021
Submission checks complete
On 12 Jan, 2021
Editor invited
On 12 Jan, 2021
No comments provided
Editorial decision: Major revision
On 10 Dec, 2020
Review #2 received
Received 19 Oct, 2020
Reviewer #2 agreed
On 08 Oct, 2020
Review #1 received
Received 29 Sep, 2020
Reviewer #1 agreed
On 08 Sep, 2020
Reviewers invited
Invitations sent on 02 Sep, 2020
Editor assigned
On 15 Jul, 2020
First submitted
On 14 Jul, 2020
Submission checks complete
On 14 Jul, 2020
Editor invited
On 14 Jul, 2020
Metrics
Comments: 0
PDF Downloads: ...
HTML Views: ...
Subject Areas
Neurology
License:
This work is licensed under a CC BY 4.0 License. Read Full License
Background The association between autism spectrum disorder and hydrocephalus is not well understood, despite demonstrated links between autism spectrum disorder and cerebrospinal fluid abnormalities. Based on the hypothesis that autism spectrum disorder and hydrocephalus may, at least in some cases, be two manifestations of a shared congenital brain pathology, we investigated the potential association between autism spectrum disorder and hydrocephalus in a large Danish population-based cohort.
Methods Patients and controls were obtained from the Lundbeck Foundation Initiative for Integrative Psychiatric Research iPSYCH2012 case-cohort, which includes all patients with selected psychiatric disorders born in Denmark 1981-2005 along with randomly selected population controls (end of follow-up, December 31, 2016). The associations between individual psychiatric disorders and hydrocephalus were estimated using binary logistic regression with adjustment for age and sex.
Results The cohort consisted of 86,571 individuals, of which 14,654 were diagnosed with autism spectrum disorder, 28,606 were population controls, and the remaining were diagnosed with other psychiatric disorders. We identified 201 hydrocephalus cases; 68 among autism spectrum disorder patients and 40 among controls (OR 3.77, 95% CI 2.48-5.78), which corresponds to an absolute risk of 0,46 %. The autism spectrum disorder-hydrocephalus association was significant over the entire subgroup spectrum of autism spectrum disorder.
Conclusions Given the considerable risk of hydrocephalus among patients with autism spectrum disorder, we suggest that patients with autism spectrum disorder should be evaluated for co-occurring hydrocephalus on a routine basis as timely neurosurgical intervention is important. Likewise, attention must be paid to traits of autism spectrum disorder in children with hydrocephalus. The results of this study call for future investigations on a potential shared aetiology between hydrocephalus and autism spectrum disorder, including the role abnormal CSF dynamics in the pathogenesis of autism spectrum disorder.
Figure 1