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BMC Psychiatry
Published in: BMC Psychiatry 1/2016

Open Access 01-12-2016 | Research article

Nationwide population-based cohort study of psychiatric disorders in individuals with Ehlers–Danlos syndrome or hypermobility syndrome and their siblings

Authors: Martin Cederlöf, Henrik Larsson, Paul Lichtenstein, Catarina Almqvist, Eva Serlachius, Jonas F. Ludvigsson

Published in: BMC Psychiatry | Issue 1/2016

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Abstract

Background

To assess the risk of psychiatric disorders in Ehlers-Danlos syndrome (EDS) and hypermobility syndrome.

Methods

Nationwide population-based matched cohort study. EDS, hypermobility syndrome and psychiatric disorders were identified through Swedish national registries. Individuals with EDS (n = 1,771) were matched with comparison individuals (n = 17,710). Further, siblings to individuals with EDS who did not have an EDS diagnosis themselves were compared with matched comparison siblings. Using conditional logistic regression, risk of autism spectrum disorder (ASD), bipolar disorder, attention deficit hyperactivity disorder (ADHD), depression, attempted suicide, suicide and schizophrenia were estimated. The same analyses were conducted in individuals with hypermobility syndrome (n = 10,019) and their siblings.

Results

EDS was associated with ASD: risk ratio (RR) 7.4, 95 % confidence interval (95 % CI) 5.2–10.7; bipolar disorder: RR 2.7, CI 1.5–4.7; ADHD: RR 5.6, CI 4.2–7.4; depression: RR 3.4, 95 % CI 2.9–4.1; and attempted suicide: RR 2.1, 95 % CI 1.7–2.7, but not with suicide or schizophrenia. EDS siblings were at increased risk of ADHD: RR 2.1, 95 % CI 1.4–3.3; depression: RR 1.5, 95 % CI 1.1–1.8; and suicide attempt: RR 1.8, 95 % CI 1.4–2.3. Similar results were observed for individuals with hypermobility syndrome and their siblings.

Conclusions

Individuals with EDS and hypermobility syndrome are at increased risks of being diagnosed with psychiatric disorders. These risk increases may have a genetic and/or early environmental background as suggested by evidence showing that siblings to patients have elevated risks of certain psychiatric disorders.
Appendix
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Literature
1.
Sobey G. Ehlers-Danlos syndrome: how to diagnose and when to perform genetic tests. Arch Dis Child. 2015;100(1):57–61.CrossRefPubMed
2.
Beighton P, De Paepe A, Steinmann B, Tsipouras P, Wenstrup RJ. Ehlers-Danlos syndromes: revised nosology, Villefranche, 1997. Ehlers-Danlos National Foundation (USA) and Ehlers-Danlos Support Group (UK). Am J Med Genet. 1998;77(1):31–7.CrossRefPubMed
3.
Palmer S, Bailey S, Barker L, Barney L, Elliott A. The effectiveness of therapeutic exercise for joint hypermobility syndrome: a systematic review. Physiotherapy. 2014;100(3):220–7.CrossRefPubMed
4.
Smith TO, Easton V, Bacon H, Jerman E, Armon K, Poland F, Macgregor AJ. The relationship between benign joint hypermobility syndrome and psychological distress: a systematic review and meta-analysis. Rheumatology (Oxford). 2014;53(1):114–22.CrossRef
5.
Lumley MA, Jordan M, Rubenstein R, Tsipouras P, Evans MI. Psychosocial functioning in the Ehlers-Danlos syndrome. Am J Med Genet. 1994;53(2):149–52.CrossRefPubMed
6.
Bulbena A, Duro JC, Porta M, Martin-Santos R, Mateo A, Molina L, Vallescar R, Vallejo J. Anxiety disorders in the joint hypermobility syndrome. Psychiatry Res. 1993;46(1):59–68.CrossRefPubMed
7.
Gurer G, Sendur F, Bulent K, Mehmet E. The anxiety between individuals with and without joint hypermobility. Eur J Psychiat. 2010;24(4):205–9.CrossRef
8.
Martin-Santos R, Bulbena A, Porta M, Gago J, Molina L, Duro JC. Association between joint hypermobility syndrome and panic disorder. Am J Psychiatry. 1998;155(11):1578–83.CrossRefPubMed
9.
Baeza-Velasco C, Pailhez G, Bulbena A, Baghdadli A. Joint hypermobility and the heritable disorders of connective tissue: clinical and empirical evidence of links with psychiatry. Gen Hosp Psychiatry. 2015;37(1):24–30.CrossRefPubMed
10.
Bulbena A, Gago J, Pailhez G, Sperry L, Fullana MA, Vilarroya O. Joint hypermobility syndrome is a risk factor trait for anxiety disorders: a 15-year follow-up cohort study. Gen Hosp Psychiatry. 2011;33(4):363–70.CrossRefPubMed
11.
Murray B, Yashar BM, Uhlmann WR, Clauw DJ, Petty EM. Ehlers-Danlos syndrome, hypermobility type: A characterization of the patients’ lived experience. Am J Med Genet A. 2013;161A(12):2981–8.CrossRefPubMed
12.
Sirota P, Frydman M, Sirota L. Schizophrenia and Marfan syndrome. Br J Psychiatry. 1990;157:433–6.CrossRefPubMed
13.
Takei A, Mera K, Sato Y, Haraoka Y. High-functioning autistic disorder with Ehlers-Danlos syndrome. Psychiatry Clin Neurosci. 2011;65(6):605–6.CrossRefPubMed
14.
Ludvigsson JF, Andersson E, Ekbom A, Feychting M, Kim JL, Reuterwall C, Heurgren M, Otterblad Olausson P. External review and validation of the Swedish national inpatient register. BMC Public Health. 2011;11(1):450.CrossRefPubMedPubMedCentral
15.
Sellgren C, Landen M, Lichtenstein P, Hultman CM, Langstrom N. Validity of bipolar disorder hospital discharge diagnoses: file review and multiple register linkage in Sweden. Acta Psychiatr Scand. 2011;124(6):447–53.CrossRefPubMed
16.
Lundh A, Forsman M, Serlachius E, Lichtenstein P, Landen M. Outcomes of child psychiatric treatment. Acta Psychiatr Scand. 2013;128(1):34–44.CrossRefPubMed
17.
Ekholm B, Ekholm A, Adolfsson R, Vares M, Osby U, Sedvall GC, Jonsson EG. Evaluation of diagnostic procedures in Swedish patients with schizophrenia and related psychoses. Nord J Psychiatry. 2005;59(6):457–64.CrossRefPubMed
18.
Dalman C, Broms J, Cullberg J, Allebeck P. Young cases of schizophrenia identified in a national inpatient register--are the diagnoses valid? Soc Psychiatry Psychiatr Epidemiol. 2002;37(11):527–31.CrossRefPubMed
19.
Lichtenstein P, Bjork C, Hultman CM, Scolnick E, Sklar P, Sullivan PF. Recurrence risks for schizophrenia in a Swedish national cohort. Psychol Med. 2006;36(10):1417–25.CrossRefPubMed
20.
Lauritsen MB, Jorgensen M, Madsen KM, Lemcke S, Toft S, Grove J, Schendel DE, Thorsen P. Validity of childhood autism in the Danish Psychiatric Central Register: findings from a cohort sample born 1990–1999. J Autism Dev Disord. 2010;40(2):139–48.CrossRefPubMed
21.
Ludvigsson JF, Otterblad-Olausson P, Pettersson BU, Ekbom A. The Swedish personal identity number: possibilities and pitfalls in healthcare and medical research. Eur J Epidemiol. 2009;24(11):659–67.CrossRefPubMedPubMedCentral
22.
Ludvigsson JF, Almqvist C, Bonamy AE, Ljung R, Michaelsson K, Neovius M, Stephansson O, Ye W. Registers of the Swedish total population and their use in medical research. Eur J Epidemiol. 2016.
23.
24.
Klein RG, Mannuzza S, Olazagasti MA, Roizen E, Hutchison JA, Lashua EC, Castellanos FX. Clinical and functional outcome of childhood attention-deficit/hyperactivity disorder 33 years later. Arch Gen Psychiatry. 2012;69(12):1295–303.CrossRefPubMedPubMedCentral
25.
Leibson CL, Katusic SK, Barbaresi WJ, Ransom J, O’Brien PC. Use and costs of medical care for children and adolescents with and without attention-deficit/hyperactivity disorder. JAMA. 2001;285(1):60–6.CrossRefPubMed
26.
Polanczyk G, de Lima MS, Horta BL, Biederman J, Rohde LA. The worldwide prevalence of ADHD: a systematic review and metaregression analysis. Am J Psychiatry. 2007;164(6):942–8.CrossRefPubMed
27.
Kessler RC, Adler L, Barkley R, Biederman J, Conners CK, Demler O, Faraone SV, Greenhill LL, Howes MJ, Secnik K, et al. The prevalence and correlates of adult ADHD in the United States: results from the National Comorbidity Survey Replication. Am J Psychiatry. 2006;163(4):716–23.CrossRefPubMedPubMedCentral
28.
Ludvigsson JF, Haberg SE, Knudsen GP, Lafolie P, Zoega H, Sarkkola C, von Kraemer S, Weiderpass E, Norgaard M. Ethical aspects of registry-based research in the Nordic countries. Clin Epidemiol. 2015;7:491–508.CrossRefPubMedPubMedCentral
Metadata
Title
Nationwide population-based cohort study of psychiatric disorders in individuals with Ehlers–Danlos syndrome or hypermobility syndrome and their siblings
Authors
Martin Cederlöf
Henrik Larsson
Paul Lichtenstein
Catarina Almqvist
Eva Serlachius
Jonas F. Ludvigsson
Publication date
01-12-2016
Publisher
BioMed Central
Published in
BMC Psychiatry / Issue 1/2016
Electronic ISSN: 1471-244X
DOI
https://doi.org/10.1186/s12888-016-0922-6

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