Infantile Hemangiomas in Twins: A Prospective Cohort Study

M. Fernanda Greco, Ilona J. Frieden, Beth A. Drolet, María C. Garzon, Anthony J. Mancini, Sarah L. Chamlin, Denise Metry, Denise Adams, Anne Lucky, Mary Sue Wentzel, Kimberly A. Horii, Eulalia Baselga, Catherine C. Mccuaig, Julie Powell, Anita Haggstrom, Dawn Siegel, Kimberly D. Morel, M. Rosa Cordisco, Amy J. Nopper, Alfons Krol

Research output: Contribution to journalArticle

5 Citations (Scopus)

Abstract

Background: Twins have a higher-than-expected risk of infantile hemangiomas (IHs), but the exact reasons for this association are not clear. Comparing concordant and discordant twin pairs might help elucidate these factors and yield more information about IH risk factors. Methods: A prospective cohort study of twin pairs from 12 pediatric dermatology centers in the United States, Canada, Argentina, and Spain was conducted. Information regarding maternal pregnancy history, family history of vascular birthmarks, zygosity (if known), and pregnancy-related information was collected. Information regarding twins (N = 202 sets) included birthweight, gestational age (GA), presence or absence of IHs, numbers and subtypes of IHs, presence of other birthmarks, and other medical morbidities. Results: Two hundred two sets of twins were enrolled. Concordance for IH was present in 37% of twin pairs. Concordance for IH was inversely related to gestational age (GA), present in 42% of GA of 32 weeks or less, 36% of GA of 33 to 36 weeks, and 32% of GA of 37 weeks or more. Twins of GA of 34 weeks or less were more than two and a half times as likely to be concordant as those of GA of 35 weeks or more (odds ratio (OR) = 2.66, 95% confidence interval (CI) = 1.42-4.99; p <0.01). In discordant twins, lower birthweight conferred a high risk of IH; of the 64 sets of twins with 10% or greater difference in weight, the smaller twin had IH in 62.5% (n = 40) of cases, versus 37.5% (n = 24) of cases in which the higher-birthweight twin was affected. Zygosity was reported in 188 twin sets (93%). Of these, 78% were dizygotic and 22% monozygotic. There was no statistically significant difference in rates of concordance between monozygotic twins (43%, 18/42) and dizygotic twins (36%, 52/146) (p = 0.50). In multivariate analysis comparing monozygotic and dizygotic twins, adjusting for effects of birthweight and sex, the likelihood of concordance for monozygotic was not appreciably higher than that for dizygotic twins (OR = 1.14, 95% CI = 0.52-2.49). Female sex also influenced concordance, confirming the effects of female sex on IH risk. The female-to-male ratio was 1.7:1 in the entire cohort and 1.9:1 in those with IH. Of the 61 concordant twin sets with known sex of both twins, 41% were female/female, 43% were female/male, and 16% were male/male. Conclusions: These findings suggest that the origin of IHs is multifactorial and that predisposing factors such as birthweight, sex, and GA may interact with one another such that a threshold is reached for clinical expression.

Original languageEnglish (US)
JournalPediatric Dermatology
DOIs
StateAccepted/In press - 2016

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Hemangioma
Cohort Studies
Prospective Studies
Gestational Age
Dizygotic Twins
Monozygotic Twins
Odds Ratio
Confidence Intervals
Reproductive History
Argentina
Dermatology
Causality
Spain
Canada
Blood Vessels
Multivariate Analysis

ASJC Scopus subject areas

  • Dermatology
  • Pediatrics, Perinatology, and Child Health

Cite this

Greco, M. F., Frieden, I. J., Drolet, B. A., Garzon, M. C., Mancini, A. J., Chamlin, S. L., ... Krol, A. (Accepted/In press). Infantile Hemangiomas in Twins: A Prospective Cohort Study. Pediatric Dermatology. https://doi.org/10.1111/pde.12781

Infantile Hemangiomas in Twins : A Prospective Cohort Study. / Greco, M. Fernanda; Frieden, Ilona J.; Drolet, Beth A.; Garzon, María C.; Mancini, Anthony J.; Chamlin, Sarah L.; Metry, Denise; Adams, Denise; Lucky, Anne; Wentzel, Mary Sue; Horii, Kimberly A.; Baselga, Eulalia; Mccuaig, Catherine C.; Powell, Julie; Haggstrom, Anita; Siegel, Dawn; Morel, Kimberly D.; Cordisco, M. Rosa; Nopper, Amy J.; Krol, Alfons.

In: Pediatric Dermatology, 2016.

Research output: Contribution to journalArticle

Greco, MF, Frieden, IJ, Drolet, BA, Garzon, MC, Mancini, AJ, Chamlin, SL, Metry, D, Adams, D, Lucky, A, Wentzel, MS, Horii, KA, Baselga, E, Mccuaig, CC, Powell, J, Haggstrom, A, Siegel, D, Morel, KD, Cordisco, MR, Nopper, AJ & Krol, A 2016, 'Infantile Hemangiomas in Twins: A Prospective Cohort Study', Pediatric Dermatology. https://doi.org/10.1111/pde.12781
Greco MF, Frieden IJ, Drolet BA, Garzon MC, Mancini AJ, Chamlin SL et al. Infantile Hemangiomas in Twins: A Prospective Cohort Study. Pediatric Dermatology. 2016. https://doi.org/10.1111/pde.12781
Greco, M. Fernanda ; Frieden, Ilona J. ; Drolet, Beth A. ; Garzon, María C. ; Mancini, Anthony J. ; Chamlin, Sarah L. ; Metry, Denise ; Adams, Denise ; Lucky, Anne ; Wentzel, Mary Sue ; Horii, Kimberly A. ; Baselga, Eulalia ; Mccuaig, Catherine C. ; Powell, Julie ; Haggstrom, Anita ; Siegel, Dawn ; Morel, Kimberly D. ; Cordisco, M. Rosa ; Nopper, Amy J. ; Krol, Alfons. / Infantile Hemangiomas in Twins : A Prospective Cohort Study. In: Pediatric Dermatology. 2016.
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title = "Infantile Hemangiomas in Twins: A Prospective Cohort Study",
abstract = "Background: Twins have a higher-than-expected risk of infantile hemangiomas (IHs), but the exact reasons for this association are not clear. Comparing concordant and discordant twin pairs might help elucidate these factors and yield more information about IH risk factors. Methods: A prospective cohort study of twin pairs from 12 pediatric dermatology centers in the United States, Canada, Argentina, and Spain was conducted. Information regarding maternal pregnancy history, family history of vascular birthmarks, zygosity (if known), and pregnancy-related information was collected. Information regarding twins (N = 202 sets) included birthweight, gestational age (GA), presence or absence of IHs, numbers and subtypes of IHs, presence of other birthmarks, and other medical morbidities. Results: Two hundred two sets of twins were enrolled. Concordance for IH was present in 37{\%} of twin pairs. Concordance for IH was inversely related to gestational age (GA), present in 42{\%} of GA of 32 weeks or less, 36{\%} of GA of 33 to 36 weeks, and 32{\%} of GA of 37 weeks or more. Twins of GA of 34 weeks or less were more than two and a half times as likely to be concordant as those of GA of 35 weeks or more (odds ratio (OR) = 2.66, 95{\%} confidence interval (CI) = 1.42-4.99; p <0.01). In discordant twins, lower birthweight conferred a high risk of IH; of the 64 sets of twins with 10{\%} or greater difference in weight, the smaller twin had IH in 62.5{\%} (n = 40) of cases, versus 37.5{\%} (n = 24) of cases in which the higher-birthweight twin was affected. Zygosity was reported in 188 twin sets (93{\%}). Of these, 78{\%} were dizygotic and 22{\%} monozygotic. There was no statistically significant difference in rates of concordance between monozygotic twins (43{\%}, 18/42) and dizygotic twins (36{\%}, 52/146) (p = 0.50). In multivariate analysis comparing monozygotic and dizygotic twins, adjusting for effects of birthweight and sex, the likelihood of concordance for monozygotic was not appreciably higher than that for dizygotic twins (OR = 1.14, 95{\%} CI = 0.52-2.49). Female sex also influenced concordance, confirming the effects of female sex on IH risk. The female-to-male ratio was 1.7:1 in the entire cohort and 1.9:1 in those with IH. Of the 61 concordant twin sets with known sex of both twins, 41{\%} were female/female, 43{\%} were female/male, and 16{\%} were male/male. Conclusions: These findings suggest that the origin of IHs is multifactorial and that predisposing factors such as birthweight, sex, and GA may interact with one another such that a threshold is reached for clinical expression.",
author = "Greco, {M. Fernanda} and Frieden, {Ilona J.} and Drolet, {Beth A.} and Garzon, {Mar{\'i}a C.} and Mancini, {Anthony J.} and Chamlin, {Sarah L.} and Denise Metry and Denise Adams and Anne Lucky and Wentzel, {Mary Sue} and Horii, {Kimberly A.} and Eulalia Baselga and Mccuaig, {Catherine C.} and Julie Powell and Anita Haggstrom and Dawn Siegel and Morel, {Kimberly D.} and Cordisco, {M. Rosa} and Nopper, {Amy J.} and Alfons Krol",
year = "2016",
doi = "10.1111/pde.12781",
language = "English (US)",
journal = "Pediatric Dermatology",
issn = "0736-8046",
publisher = "Wiley-Blackwell",

}

TY - JOUR

T1 - Infantile Hemangiomas in Twins

T2 - A Prospective Cohort Study

AU - Greco, M. Fernanda

AU - Frieden, Ilona J.

AU - Drolet, Beth A.

AU - Garzon, María C.

AU - Mancini, Anthony J.

AU - Chamlin, Sarah L.

AU - Metry, Denise

AU - Adams, Denise

AU - Lucky, Anne

AU - Wentzel, Mary Sue

AU - Horii, Kimberly A.

AU - Baselga, Eulalia

AU - Mccuaig, Catherine C.

AU - Powell, Julie

AU - Haggstrom, Anita

AU - Siegel, Dawn

AU - Morel, Kimberly D.

AU - Cordisco, M. Rosa

AU - Nopper, Amy J.

AU - Krol, Alfons

PY - 2016

Y1 - 2016

N2 - Background: Twins have a higher-than-expected risk of infantile hemangiomas (IHs), but the exact reasons for this association are not clear. Comparing concordant and discordant twin pairs might help elucidate these factors and yield more information about IH risk factors. Methods: A prospective cohort study of twin pairs from 12 pediatric dermatology centers in the United States, Canada, Argentina, and Spain was conducted. Information regarding maternal pregnancy history, family history of vascular birthmarks, zygosity (if known), and pregnancy-related information was collected. Information regarding twins (N = 202 sets) included birthweight, gestational age (GA), presence or absence of IHs, numbers and subtypes of IHs, presence of other birthmarks, and other medical morbidities. Results: Two hundred two sets of twins were enrolled. Concordance for IH was present in 37% of twin pairs. Concordance for IH was inversely related to gestational age (GA), present in 42% of GA of 32 weeks or less, 36% of GA of 33 to 36 weeks, and 32% of GA of 37 weeks or more. Twins of GA of 34 weeks or less were more than two and a half times as likely to be concordant as those of GA of 35 weeks or more (odds ratio (OR) = 2.66, 95% confidence interval (CI) = 1.42-4.99; p <0.01). In discordant twins, lower birthweight conferred a high risk of IH; of the 64 sets of twins with 10% or greater difference in weight, the smaller twin had IH in 62.5% (n = 40) of cases, versus 37.5% (n = 24) of cases in which the higher-birthweight twin was affected. Zygosity was reported in 188 twin sets (93%). Of these, 78% were dizygotic and 22% monozygotic. There was no statistically significant difference in rates of concordance between monozygotic twins (43%, 18/42) and dizygotic twins (36%, 52/146) (p = 0.50). In multivariate analysis comparing monozygotic and dizygotic twins, adjusting for effects of birthweight and sex, the likelihood of concordance for monozygotic was not appreciably higher than that for dizygotic twins (OR = 1.14, 95% CI = 0.52-2.49). Female sex also influenced concordance, confirming the effects of female sex on IH risk. The female-to-male ratio was 1.7:1 in the entire cohort and 1.9:1 in those with IH. Of the 61 concordant twin sets with known sex of both twins, 41% were female/female, 43% were female/male, and 16% were male/male. Conclusions: These findings suggest that the origin of IHs is multifactorial and that predisposing factors such as birthweight, sex, and GA may interact with one another such that a threshold is reached for clinical expression.

AB - Background: Twins have a higher-than-expected risk of infantile hemangiomas (IHs), but the exact reasons for this association are not clear. Comparing concordant and discordant twin pairs might help elucidate these factors and yield more information about IH risk factors. Methods: A prospective cohort study of twin pairs from 12 pediatric dermatology centers in the United States, Canada, Argentina, and Spain was conducted. Information regarding maternal pregnancy history, family history of vascular birthmarks, zygosity (if known), and pregnancy-related information was collected. Information regarding twins (N = 202 sets) included birthweight, gestational age (GA), presence or absence of IHs, numbers and subtypes of IHs, presence of other birthmarks, and other medical morbidities. Results: Two hundred two sets of twins were enrolled. Concordance for IH was present in 37% of twin pairs. Concordance for IH was inversely related to gestational age (GA), present in 42% of GA of 32 weeks or less, 36% of GA of 33 to 36 weeks, and 32% of GA of 37 weeks or more. Twins of GA of 34 weeks or less were more than two and a half times as likely to be concordant as those of GA of 35 weeks or more (odds ratio (OR) = 2.66, 95% confidence interval (CI) = 1.42-4.99; p <0.01). In discordant twins, lower birthweight conferred a high risk of IH; of the 64 sets of twins with 10% or greater difference in weight, the smaller twin had IH in 62.5% (n = 40) of cases, versus 37.5% (n = 24) of cases in which the higher-birthweight twin was affected. Zygosity was reported in 188 twin sets (93%). Of these, 78% were dizygotic and 22% monozygotic. There was no statistically significant difference in rates of concordance between monozygotic twins (43%, 18/42) and dizygotic twins (36%, 52/146) (p = 0.50). In multivariate analysis comparing monozygotic and dizygotic twins, adjusting for effects of birthweight and sex, the likelihood of concordance for monozygotic was not appreciably higher than that for dizygotic twins (OR = 1.14, 95% CI = 0.52-2.49). Female sex also influenced concordance, confirming the effects of female sex on IH risk. The female-to-male ratio was 1.7:1 in the entire cohort and 1.9:1 in those with IH. Of the 61 concordant twin sets with known sex of both twins, 41% were female/female, 43% were female/male, and 16% were male/male. Conclusions: These findings suggest that the origin of IHs is multifactorial and that predisposing factors such as birthweight, sex, and GA may interact with one another such that a threshold is reached for clinical expression.

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