Recurrence risk for offspring of twins discordant for oral cleft: a population-based cohort study of the Danish 1936-2004 cleft twin cohort.
Recurrence risk for offspring of twins discordant for oral cleft: a population-based cohort study of the Danish 1936-2004 cleft twin cohort.
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DOI：
10.1002/ajmg.a.33608
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发表时间：
2010-10
影响因子：
2
通讯作者：
Christensen, Kaare
中科院分区：
文献类型：
--
作者：
Grosen, Dorthe;Bille, Camilla;Pedersen, Jacob Krabbe;Skytthe, Axel;Murray, Jeffrey C.;Christensen, Kaare
在这项以丹麦人群为基础的队列研究中，我们的目标是评估单纯性口腔裂(OC)不和谐双胞胎双胞胎未受影响的后代的复发风险，并将该风险与受影响的同卵双胞胎后代的复发风险以及背景人群的风险进行比较。在1936-2004年间，有207对双胞胎被确定，其中至少有一对双胞胎有OC。调查对象为有子女(N=117)及其子女(N=239)的双胞胎。参与者是通过丹麦面部裂隙数据库、丹麦双胞胎登记处和丹麦民事登记系统之间的联系确定的。在这项研究中，对患病和未患病双胞胎的后代的OC复发风险和相对风险与背景患病率进行了比较。我们发现，在54对受OC影响的双胞胎中的110名儿童中，有两名(1.8%)儿童患有OC，与背景人群中的频率相比，其相对风险显著增加(RR=10；95%可信区间1.2至35)。在63对双胞胎中的129名儿童中，有3名儿童(2.3%)受到影响，与背景患病率相比，相对风险显著增加(RR=13；95%可信区间2.6~36)。我们的结论是，在OC不协调的双胞胎中，在OC受影响的双胞胎和未受OC影响的双胞胎的后代中发现类似的复发风险增加。这为裂隙病因学中的遗传成分提供了进一步的证据，并为裂隙不协调的双胞胎的遗传咨询提供了有用的信息。
Our objective in this Danish population-based cohort study was to estimate the recurrence risk of isolated oral cleft (OC) for offspring of the unaffected co-twins of OC discordant twin pairs and to compare this risk to the recurrence risk in the offspring of the affected co-twin as well as to the risk in the background population. During 1936–2004, 207 twin pairs were ascertained, among whom at least one twin had an OC. The index persons were twins discordant for OC who had children (N=117), and their offspring (N=239). The participants were ascertained by linkage between The Danish Facial Cleft Database, The Danish Twin Registry and The Danish Civil Registration System. In the study OC recurrence risk for offspring of the affected and unaffected twin and relative risk were compared to the background prevalence. We found that among 110 children of the 54 OC affected twins, two (1.8%) children had OC corresponding to a significantly increased relative risk (RR = 10; 95% CI 1.2 to 35) when compared to the frequency in the background population. Among the 129 children of the 63 unaffected twins, three (2.3%) children were affected, corresponding to a significantly increased relative risk (RR = 13; 95% CI 2.6 to 36) when compared the background prevalence. We concluded that in OC discordant twin pairs similar increased recurrence risks were found among offspring of both OC affected and OC unaffected twins. This provides further evidence for a genetic component in cleft etiology and is useful information for genetic counseling of twin pairs discordant for clefting.
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DOI：
10.1002/ajmg.1320470620
发表时间：
1993-11-01
期刊：
AMERICAN JOURNAL OF MEDICAL GENETICS
影响因子：
--
作者：
CHRISTENSEN, K;FOGHANDERSEN, P
通讯作者：
FOGHANDERSEN, P
影响因子：
30.8
作者：
Sakuntabhai, A;Ruiz-Perez, V;Hovnanian, A
通讯作者：
Hovnanian, A
影响因子：
0.9
作者：
Kimani, Jane W.;Yoshiura, Koh-ichiro;Murray, Jeffrey C.
通讯作者：
Murray, Jeffrey C.
影响因子：
2
作者：
Neiswanger, Katherine;Weinberg, Seth M.;Marazita, Mary L.
通讯作者：
Marazita, Mary L.
影响因子：
1.9
作者：
DAVIE, AM
通讯作者：
DAVIE, AM