Genetics – Abnormalities of Chromosome number 105-113

Polyploidy

Euploidy- a cell that has a multiple of 23 chromosomes (46, 69, 92)

-Humans can be polyploid, complete sets of extra chromosomes. Humans can be triploid (69) or tetraploid (92)

-Large surplus gene product causes multiple anomalies such as heart and CNS defects

-triploidy (69 XXX) is seen 1 in 10,000 live births

-triploidy is one of the most common causes of fetal loss at conception (spontaneous abortion)

-if they survive until birth, die shortly after.

-Triploidy has 3 main causes:

1.) Dispermy – fertilization of egg by 2 sperm

2.) fusion of an ovum and polar body + fertilization

3.) Meiotic failure – diploid sperm or egg can be produced

-Tetraploidy is very rare, only a few live births have been recorded, causes are

1.) mitotic failure of early embryo, all duplicated chromosomes migrate to 1 cell

2.) fusion of 2 diploid zygotes

Aneuploidy – cells that contain missing or additional individual chromosomes (not a multiple of 23)

-Usually only 1 chromosome is affected, mostly of autosomes

Monosomy – presence of only one copy of a chromosome in an otherwise diploid cell)

-almost always incompatible with life if in an autosome

Trisomy – three copies of one chromosome

-autosomal trisomies are seen quite a bit

Non-disjunction - Most common cause of aneuploidy, failure of chromosomes to disjoin normally during meiosis

-Can occur either during meiosis 1 or 2

-both chromosomes migrate to one cell instead of splitting

Trisomy 21 – seen in approximately 1 out of every 800 to 1000 live births, most common aneuploidy condition

-Produces Down Syndrome

-Features: low nasal root, upslanting palpebral fissures, small ears, flattened maxillary and malar region, round cheeks, neck is short, skin is redundant at the nape of the neck

-50% of patients

-3% of infants develop duodenal atresia

-Respiratory problems are higher

-Increased risk for leukemia

Most Significant Medical Problem: 40% of Down Syndrome patients present with structural heart defects

-Most common is atrioventricular (AV) canal defect (interatrial and interventricular septa fail to fuse normally during development.

-Results in blood flow from left to right side of the heart and then to pulmonary circulation, causing pulmonary hypertension

-Ventricular septal defects (VSD) are also common

-Can also have hearing loss, hypothyroidism, eye abnormalities

-Males are sterile, females an reproduce (high risk of producing Down’s offspring 50%)

-Mostly caused by nondisjunction, remainder by chromosome translocations

-Mother contributes the extra chromosome in 90% of the case

-Mosaicism- Patients with some normal somatic cells and other cells with trisomy 21.

-Nomenclature: 47, XY + 21[10]/46, XY[10] (number in brackets indicate cells counted with the Down’s karyotype

-Most common cause is trisomic conception followed by loss of the extra chromosome during mitosis in some embryonic cells

-results in milder clinical symptoms

Tissue-specific mosaicism – mosaicism confined only to certain tissues

-mosaicism in germline of parent causes recurrent risk for Down Syndrome in patients

-Candidate gene for mental retardation in Down’s Patients is DYRK1A, a kinase gene that causes learning and memory defects when overexpressed in mice

-Another candidate gene is APP (amyloid B precursor protein), a 3rd copy of APP in a trisomy 21 patient accounts for the Alzheimer’s symptoms seen in nearly all Down’s patients by age 40.

-Partial trisomies not affecting the APP region do not produce alzheimer’s symptoms.

Trisomy 18 – Also known as Edwards Syndrome, second most common trisomy (1:6000 live births)

-most common chromosomal abnormality among stillborns

-prenatal growth deficiency, characteristic facial features, and a distinctive hand abnormality (index finger on top of middle finger)

-Congenital heart defects (VSD) are most common in 90% of children

-Omphalocele – protrusion of bowel into umbilical cord

-Radial Aplasia – missing radius bone

-Diaphragmatic hernia, and spina bifida

-50% of infants with trisomy 18 die within the first several weeks of life

-aspiration pneumonia, predisposition to infections, heart defects account for mortality.

-developmental abnormalities – children not able to walk independently.

-very little mosaicism, mostly complete trisomy.

-Nondisjunction of maternal chromosome 18 in 90% of all cases

Trisomy 13 – also called Patau Syndrome, seen 1:10000 live births, 95% of infants die within first year of life

-oral/facial clefts, micropthalmia (small eyes), polydactyly

-CNS malformations

-Heart defects

-Renal anomalies

-Cutis aplasia (scalp defect on posterior occiput)

-Can progress in development to communicate with parents, unlike trisomy 18

-80% of Patau syndrome patients have complete trisomy, with the other 20% having a translocation of the long arm of chromosome 13

-95% of Patau patients spontaneously lost during pregnancy

Nondisjunction Risk and Maternal Age –

Less than 30 years – 1/1000

35 years - 1/400

40 years -1/100

45 years -1/25

-the older the woman, the older her ova are, since they are arrested in prophase until ovulation. Environmental factors (smoking, alcohol consumption, and radiation can play a role)

Turner Syndrome – phenotype associated with sex chromosome aneuploidy of a single X chromosome (45X)

-usually female with the following characteristics:

-short stature

-sexual infantilism, ovarian dysgenesis (streaks of connective tissue seen instead of ovaries)

-major and minor malformations

-triangle shaped face, webbed neck, chest is broad and shield-like

-lymphedema of hands and feet

-congenital heart defects – most commonly obstructive lesions on left side of heart (bicuspid aortic valve in 50% of patients)

-50% of patients have kidney defects

-50% of patients have 45,X karyotype, 30-40% of patients have 45,X/46,XX and less commonly 45,X/46,XY

-10% of patients have structural X chromosome abnormalities involving deletion of some or all of XP.

-60-80% of cases involve loss of paternal X chromosome, occurring either in mitosis of embryo or early meiosis of father

-1-2% of all conceptions are 45,X, but the low number of births with 45,X (1:1000) suggests majority are lost prenatally.

-many survivors are mosaics, or placental mosaics (of placenta alone)

-mutations in SHOX gene, which encodes transcription factor for embryonic limbs, produces short stature, thought to be associated with Turner’s Phenotype

-located on distal tip of X and Y short arms (escaping inactivation), thus 2 copies are normally transcribed, Turner’s patients would only have one copy transcribed, resulting in haploinsufficiency

-Pseudoautosomal Region: During normal meiosis, crossover occurs between tip of short arm of Y chromosome and tip of short arm of X chromosome. These regions have homology, and therefore behave similar to autosomes. Distal portion of Y chromosome is called Pseudoautosomal region

-SRY gene is just proximal to pseudoautosomal region (determines male phenotype)

-Acts antagonistically against DAX1, which normally represses differentiation of embryo into a male

-absence of SRY causes DAX1 to act unopposed, producing females

-males produced when SRY upregulates SOX9

Klinefelter Syndrome (47, XXY) – 1/500 to 1/1000 births

-primary cause of hypogonadism in males

-taller than average, sterile, testosterone levels are low

-Gynecomastia is common (breast development)

-reduction in verbal IQ

-Extra chromosome is mostly derived maternally, and mosaicism can be seen in 15% of patients

-Can also be XXXY and XXXXY – degree of mental and physical deficiency increases with more X’s

-Can be treated with testosterone therapy

Trisomy X (47, XXX) – 1/1000 females, benign consequences, sometimes suffering from sterility, menstrual irregularity, mild mental retardation, can be found with XXXX or XXXXX

47, XXY Syndrome – males with this karyotype tend to be taller, lower IQ, few physical irregularities, found with high prevalence in prisons, minor behavioral disorders, hyperactivity, ADD< and learning disabilities