Spermatozoal KO studies for the highly present paternal Potential maternal interactions Conclusions of the KO studies on the maternal gene transcripts detected by GeneMANIA candidates for interaction with the paternal Fbxo2 Selective cochlear degeneration in mice lacking Cul1, Fbxl2, Fbxl3, Fbxl5, Cul-1 KO causes early embryonic lethality at E6.5 before Fbxo2 Fbxo34, Fbxo5, Itgb1, Rbx1, the onset of gastrulation http://www.jneurosci.org/content/27/19/5163.full Skp1a http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3641602/. Loss of Cul1 results in early embryonic lethality and Another KO study showed the following: Loss of dysregulation of cyclin E F-box only 2 (Fbxo2) disrupts levels and http://www.ncbi.nlm.nih.gov/pubmed/10508527?dopt=Abs localization of select NMDA receptor subunits, tract. and promotes aberrant synaptic connectivity http://www.ncbi.nlm.nih.gov/pubmed/25878288. Fbxo5 (or Emi1): Regulates early mitosis. KO studies shown lethal defects in preimplantation embryo A third KO study showed that Fbxo2 regulates development http://mcb.asm.org/content/26/14/5373.full. amyloid precursor protein levels and processing http://www.jbc.org/content/289/10/7038.long#fn- Rbx1/Roc1: Rbx1 disruption results in early embryonic 1. lethality due to proliferation failure http://www.pnas.org/content/106/15/6203.full.pdf & Fbxo2 has been found to be involved in http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2732615/. neurons, but it has not been tested for potential decreased fertilization or pregnancy rates. It Skp1a: In vivo interference with Skp1 function leads to shows high expression levels in testis, indicating genetic instability and neoplastic transformation potential other not investigated functions http://www.ncbi.nlm.nih.gov/pubmed/12417738?dopt=Abs http://biogps.org/#goto=genereport&id=230904 tract. & http://www.informatics.jax.org/image/MGI:26820 38.

Map1lc3a Atg10, Atg3, Gabarapl2, Kif15, Atg3: Atg3-deficient mice were born and died 1 day after Map1b, Map1lc3b, Pias4, birth http://www.molbiolcell.org/content/19/11/4762.full.

Tubb2a, Wdfy3 Map1b is involved in neural-diseases. Perinatal lethality of -associated protein 1B-deficient mice expressing alternative isoforms of the protein at low levels. Homozygous mice died on the first day after birth, probably due to a severe abnormal development of the nervous system. They present alterations in the structure of several brain regions http://www.ncbi.nlm.nih.gov/pubmed/11085878?dopt=Abs tract. Pcbp4 Mice deficient in poly(C)-binding protein 4 are Pcbp1, Pcbp2, Qk, Hnrnpk Qk (Quaking): Mature mice may have seizures in which a

susceptible to spontaneous tumors through the motionless posture is maintained for many seconds. increased expression of ZFP871 that targets Females are viable and fertile and males are sterile due to for degradation defective spermatic differentiation. The entire CNS of http://genesdev.cshlp.org/content/early/2016/02/ quaking mutant mice is severely deficient in myelin and 24/gad.271890.115. there is a less severe myelin deficiency in the PNS. Homozygous quaking alleles show increased embryo lethality http://www.ncbi.nlm.nih.gov/pubmed/21189687.

Hnrnpk: Coordinates Transcriptional Silencing by SETDB1 in Embryonic Stem Cells. Zfp821 No KO studies found, but Zfp821 is expressed in Aggf1, Chchd3, Fam13c, Fchsd2, Rimklb: Females homozygous for the mutation appear to reproductive system Mob1a, Rimklb , Tspyl4, Zfc3h1 be infertile https://www.jax.org/strain/022346. http://www.informatics.jax.org/marker/MGI:1923 121.

Hdac11 HDAC11 KO studies showed immunological Aamp, Bub1b, Cdc20, Hdac2, Bub1b: Homozygous embryos do not survive beyond aberrations. No disease or developmental Hdac8, Mettl18, Nelfcd early gestation (E8.5) due to impaired blastocyst aberrations were reported, however, the effect of proliferation and extensive . Mouse embryonic KO males needs to be further investigated fibroblasts from heterozygous mice exhibit defective http://www.ncbi.nlm.nih.gov/pmc/articles/PMC42 spindle checkpoint activation, an important mechanism for 52813/. genomic stability. In addition, mice are susceptible to development of lung and colon adenocarcinomas No study investigated the homozygous C57/BL6 following challenge with carcinogen. Mice homozygous for KO Hdac11 regarding the fertilization and the mutation are embryonic lethal. This mutant mouse pregnancy rates. strain may be useful in studies of early embryonic development, hematopoiesis, megakaryopoiesis tumorigenesis and genomic instability https://www.jax.org/strain/010495.

Cdc20: Cdc20 is critical for meiosis I and fertility of female mice http://www.ncbi.nlm.nih.gov/pubmed/20941357?dopt=Abs tract.

Loss of Cdc20 causes a securin-dependent metaphase arrest in two-cell mouse embryos. http://www.ncbi.nlm.nih.gov/pubmed/17325031?dopt=Abs tract No homozygous mice were detected from heterozygous intercrosses at wean (n>28 pups) https://www.jax.org/strain/018590.

Hdac2: Homozygous KO mice are born at a 2-fold lower frequency than predicted by Mendelian ratios and showed compromised fertility. Both male and female homozygous knockout mice are approximately 25% smaller than wildtype and heterozygous littermates https://www.jax.org/strain/022625.

Supplementary Table 2