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ZBpJBҘilh5[AV9Ks,$## 쭄qo~k_:JI \AiߌXQƈ?VC 桄, `H+E=iXicGX$n4#xt4E5 nV4nz,x@sxbS8m5xo?d[/[JG/WWoyo[JُZR.XJ,:jr?VY~Xgl|6ijDUר_aW6am?$4EIxdC-€b7^@zS6B=DC.0pw1E\oЕܰ^51S 1!Aj+}B1;ī N5ng 9l1=r iVI~B Y:LL7{xHWbA{&2mbOr[QܦΞ6hָ==H4{z膶_7ŗY2Owz7&W*R&fՄc+_ endstream endobj 415 0 obj <>stream hތ_o0ſ},/1 UD*m]D3 wC0=B|~ܛ$%AR~'%%"ï14EĽIQTڂ}A^XyF<m&psnh;^/{* r-ʶL+jͥΆ=\uLz\QN6@0dLܱ?. OL-3NVNWHHQF^-"#Ȍ=斫(5^NipgOCžd2üt U \4\`|S璋~V DW ?|VپˌL3K̲IftY|)Ӛ;o_?OK5~Msyu f!?[æIhrhR+:رQ8gDV \뙌==3iưe q<=wla 0U Q endstream endobj 416 0 obj <>stream hޔ=o1 ʍZЩq)jWԎSQv>&|%! J/ $z] ×pw{[>Oڌ-|q> :l7y[~^?m_Ix me;SFk4B'?NVQrJ5kG $8EPn2CWNtG?.u'kq$D7)&qMp\,8Ր SceK ~Jxtb)M'ngpco jװt\6\j=lve8 \?j06``R4'qO[kʖ4G~|_~ma:qg47KGe 'xDlfFn %r35ܨd3?>IZTTL<_CNNRvӳ"vx?_>긾߽{_*eR߭ rSHwc9KODӹ;/C(Ss N{z,cP¤LlM;mrs 0F2 endstream endobj 417 0 obj <>stream application/pdf Haochen Liu, Bichen Lin, Hangbo Liu, Lanxin Su, Hailan Feng, Yang Liu, Miao Yu and Dong Han The goal of this study was to identify the pathogenic gene variants in patients with odonto-onycho-dermal dysplasia syndrome (OODD) or nonsyndromic tooth agenesis. Four unrelated individuals with tooth agenesis and their available family members were recruited. Peripheral blood was collected from four probands and five family members. Whole-exome sequencing (WES) and Sanger sequencing were used to identify the pathogenic gene variants. The harmfulness of these variations was predicted by bioinformatics. We identified four biallelic variants of the WNT10A gene in four patients, respectively: the proband#660: c.1176C > A (p.Cys392*) and c.812G > A (p.Cys271Tyr); the proband#681: c.637G > A (p.Gly213Ser) and c.985C > T (p.Arg329*); the proband#829: c.511C > T (p.Arg171Cys) and c.637G > A (p.Gly213Ser); and the proband#338: c.926A> G (p.Gln309Arg) and c.511C > T (p.Arg171Cys). Among them, two variants (c.812G > A; p.Cys271Tyr and c.985C > T; p.Arg329*) were previously unreported. Bioinformatics analysis showed that the pathogenicity of these six variants was different. Tertiary structure analysis showed that these variants were predicted to cause structural damage to the WNT10A protein. Genotype–phenotype analysis showed that the biallelic variants with more harmful effects, such as nonsense variants, caused OODD syndrome (#660 II-1) or severe nonsyndromic tooth agenesis (NSTA) (#681 II-1); the biallelic variants with less harmful effects, such as missense variants, caused a mild form of NSTA (#829 II-2 and #338 II-1). Individuals with a heterozygous variant presented a mild form of NSTA or a normal state. Our results further suggest the existence of the dose dependence of WNT10A pathogenicity on the tooth agenesis pattern, which broadens the variation spectrum and phenotype spectrum of WNT10A and could help with clinical diagnosis, treatment, and genetic counseling. Dose Dependence Effect in Biallelic WNT10A Variant-Associated Tooth Agenesis Phenotype 2022-12-07T20:48:03+08:00 LaTeX with hyperref 2022-12-14T16:52:54+08:00 2022-12-14T16:52:54+08:00 WNT10A; biallelic variants; odonto-onycho-dermal dysplasia syndrome (OODD); nonsyndromic tooth agenesis pdfTeX-1.40.21 uuid:7c135a40-eba0-4159-9f93-6e5d7b1aecce uuid:a7af7cbe-c5d7-4d39-9586-8aac534f73c1 endstream endobj 418 0 obj <>stream h243V0P043Q0Ѧ ff 66y% ޙ)` A%@F~HeA~@bzjB\%X!5@826ЖP bK@bQ*\#C u%N#(mJxg? endstream endobj 419 0 obj <>stream hޔVnF}JIٲdd+q: ueE-]v/wfIb 7;;眙3C|?&(Ev7Ϣ XdW.`nk1Pb/IffNN!3\3؞]~#?z(lI!- T N,Rźu 82x8FO@RٓRًQ,E4gK+#,OOٲ3!E) Q"7`x^^?+U NnWcdڋa-L\66.a4QtP0AG` t=WE*A_ PژceY6-1 ƹ`!$,Y 9+Tt e+RSNQ"EETWqUHR #3HB!0 ߜ%iEqrڲqNjݽ9iju/e*OlFuDM3d c^XKE")G˖;@:?Ik&$MTR]P*Yy]p_Cn`J(R(7 *XTY9}-ZLp\A&8 ~d}`x1ǔt$Jwl:8*ΉZw[jB ,+TnSfN My0~f1OpJ'Fs*mJ1렚,no QHC;;rt^Ve>ɥ hjU8ݭYzs^P8|/.Ol37g\a,y_h:h< 8t2HѠkrõ@1Qς6=x-BwlFH.ˮר^+׆6%P,IRxz煷nUI)X6?e VJ2Jj)W8XjZM$5ŬeX8ri?DNVW!>wzDk_pG)~:v u{Seīo< ~q`[3 endstream endobj 420 0 obj <>/Filter/FlateDecode/ID[]/Info 2966 0 R/Length 526/Root 2968 0 R/Size 2967/Type/XRef/W[1 3 1]>>stream h웽KBa^5*H ZF/47g[-D-Q4DCKD4RKD*IyU88{޳)TmCъƙ0(ta{FÓKpH5q"7%{~ĨГw2/k4RuN8 aVL+qPUfǀZa[T~{$٠u:Wu:[SVn>i2!0x`G `w=$ ʞH?8cZ8߄j]1A~a ì¿,ĩB[KSӁ0xU_a[ endstream endobj startxref 116 %%EOF