Familial aggregation of hypospadias: a cohort study

Tine H Schnack, Slobodan Zdravkovic, Charlotte Myrup, Tine Westergaard, Kaare Christensen, Jan Wohlfahrt, Mads Melbye

Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

Resumé

Udgivelsesdato: 2008-Feb-1
OriginalsprogEngelsk
TidsskriftAmerican Journal of Epidemiology
Vol/bind167
Udgave nummer3
Sider (fra-til)251-256
Antal sider5
ISSN0002-9262
DOI
StatusUdgivet - 1. feb. 2008

Fingeraftryk

Hypospadias
Cohort Studies
Odds Ratio
Confidence Intervals
Mothers
Nuclear Family
Siblings
Linear Models

Citer dette

Schnack, T. H., Zdravkovic, S., Myrup, C., Westergaard, T., Christensen, K., Wohlfahrt, J., & Melbye, M. (2008). Familial aggregation of hypospadias: a cohort study. American Journal of Epidemiology, 167(3), 251-256. https://doi.org/10.1093/aje/kwm317
Schnack, Tine H ; Zdravkovic, Slobodan ; Myrup, Charlotte ; Westergaard, Tine ; Christensen, Kaare ; Wohlfahrt, Jan ; Melbye, Mads. / Familial aggregation of hypospadias: a cohort study. I: American Journal of Epidemiology. 2008 ; Bind 167, Nr. 3. s. 251-256.
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title = "Familial aggregation of hypospadias: a cohort study",
abstract = "Hypospadias is one of the most common birth defects. However, its etiology remains largely unknown. The authors investigated the contribution of genetic and environmental factors to familial aggregation of hypospadias. Using Danish health registers, they identified 5,380 boys diagnosed with hypospadias in a cohort of 1,201,790 boys born in 1973-2005. Using binomial log-linear regression, they estimated recurrence risk ratios of hypospadias for male twin pairs and first-, second-, and third-degree relatives of a hypospadias case, which were 50.8 (95{\%} confidence interval [CI]: 34.2, 75.5), 11.6 (95{\%} CI: 9.75, 13.7), 3.27 (95{\%} CI: 2.47, 4.34), and 1.33 (95{\%} CI: 0.94, 1.88), respectively. Recurrence risk ratios did not differ for family members of a hypospadias case related to the same degree. In addition, the authors found no difference in the recurrence risk ratio for maternal compared with paternal second- and third-degree relatives of a hypospadias case. In conclusion, hypospadias was found to have a strong familial component and also to aggregate within more-distant relatives. Importantly, hypospadias was equally transmitted through the paternal and maternal sides of a family, and recurrence risk ratios for brothers and sons of a hypospadias case were similar. These findings indicate that genetic rather than intrauterine environmental factors have a principal role in causing familial hypospadias.",
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Schnack, TH, Zdravkovic, S, Myrup, C, Westergaard, T, Christensen, K, Wohlfahrt, J & Melbye, M 2008, 'Familial aggregation of hypospadias: a cohort study', American Journal of Epidemiology, bind 167, nr. 3, s. 251-256. https://doi.org/10.1093/aje/kwm317

Familial aggregation of hypospadias: a cohort study. / Schnack, Tine H; Zdravkovic, Slobodan; Myrup, Charlotte; Westergaard, Tine; Christensen, Kaare; Wohlfahrt, Jan; Melbye, Mads.

I: American Journal of Epidemiology, Bind 167, Nr. 3, 01.02.2008, s. 251-256.

Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

TY - JOUR

T1 - Familial aggregation of hypospadias: a cohort study

AU - Schnack, Tine H

AU - Zdravkovic, Slobodan

AU - Myrup, Charlotte

AU - Westergaard, Tine

AU - Christensen, Kaare

AU - Wohlfahrt, Jan

AU - Melbye, Mads

PY - 2008/2/1

Y1 - 2008/2/1

N2 - Hypospadias is one of the most common birth defects. However, its etiology remains largely unknown. The authors investigated the contribution of genetic and environmental factors to familial aggregation of hypospadias. Using Danish health registers, they identified 5,380 boys diagnosed with hypospadias in a cohort of 1,201,790 boys born in 1973-2005. Using binomial log-linear regression, they estimated recurrence risk ratios of hypospadias for male twin pairs and first-, second-, and third-degree relatives of a hypospadias case, which were 50.8 (95% confidence interval [CI]: 34.2, 75.5), 11.6 (95% CI: 9.75, 13.7), 3.27 (95% CI: 2.47, 4.34), and 1.33 (95% CI: 0.94, 1.88), respectively. Recurrence risk ratios did not differ for family members of a hypospadias case related to the same degree. In addition, the authors found no difference in the recurrence risk ratio for maternal compared with paternal second- and third-degree relatives of a hypospadias case. In conclusion, hypospadias was found to have a strong familial component and also to aggregate within more-distant relatives. Importantly, hypospadias was equally transmitted through the paternal and maternal sides of a family, and recurrence risk ratios for brothers and sons of a hypospadias case were similar. These findings indicate that genetic rather than intrauterine environmental factors have a principal role in causing familial hypospadias.

AB - Hypospadias is one of the most common birth defects. However, its etiology remains largely unknown. The authors investigated the contribution of genetic and environmental factors to familial aggregation of hypospadias. Using Danish health registers, they identified 5,380 boys diagnosed with hypospadias in a cohort of 1,201,790 boys born in 1973-2005. Using binomial log-linear regression, they estimated recurrence risk ratios of hypospadias for male twin pairs and first-, second-, and third-degree relatives of a hypospadias case, which were 50.8 (95% confidence interval [CI]: 34.2, 75.5), 11.6 (95% CI: 9.75, 13.7), 3.27 (95% CI: 2.47, 4.34), and 1.33 (95% CI: 0.94, 1.88), respectively. Recurrence risk ratios did not differ for family members of a hypospadias case related to the same degree. In addition, the authors found no difference in the recurrence risk ratio for maternal compared with paternal second- and third-degree relatives of a hypospadias case. In conclusion, hypospadias was found to have a strong familial component and also to aggregate within more-distant relatives. Importantly, hypospadias was equally transmitted through the paternal and maternal sides of a family, and recurrence risk ratios for brothers and sons of a hypospadias case were similar. These findings indicate that genetic rather than intrauterine environmental factors have a principal role in causing familial hypospadias.

KW - Cohort Studies

KW - Family

KW - Humans

KW - Hypospadias

KW - Inheritance Patterns

KW - Male

KW - Odds Ratio

KW - Registries

KW - Twins

U2 - 10.1093/aje/kwm317

DO - 10.1093/aje/kwm317

M3 - Journal article

C2 - 18042671

VL - 167

SP - 251

EP - 256

JO - American Journal of Epidemiology

JF - American Journal of Epidemiology

SN - 0002-9262

IS - 3

ER -

Schnack TH, Zdravkovic S, Myrup C, Westergaard T, Christensen K, Wohlfahrt J et al. Familial aggregation of hypospadias: a cohort study. American Journal of Epidemiology. 2008 feb 1;167(3):251-256. https://doi.org/10.1093/aje/kwm317