Pathophysiology – EXAM IV

An exam IV survival guide, hope this helps. (*) indicates it will be covered in Monday’s lecture, so the material I used came from the notes as is.

Rock on,

-Pat Clements

Codominance?

Achondroplasia

- autosomal dominant trait

- most adults are heterozygotes (homozygotes don’t survive)

- gain of function mutation in fibroblast growth factor receptor 3 (FGFR3)

à arginine substitution (i.e. for glycine)

à abnormal endochondral ossification

- dwarfism with disproportionate arms and legs

- abnormal pelvis, neuro and cardiopulmonary problems, hearing loss, spine deform

- somatotrophin (synthetic human growth hormone) can treat, in early years

Albinism

- lack of pigmentation

- defect is autosomal recessive

- defect in tyrosinase gene (catalyzes the reactions necessary to make dark pigment)

- heterozygous has normal phenotype, must be homozygous aa to have condition.


Angleman’s syndrome (aka Happy Puppet Syndrome)

- most patients have deletion in the AS region of maternal chromosome 15

- some have paternal uniparental disomy for chromosome 15

- few (3%) have defect in maternal imprinting

- candidate gene: UBE3A - transfers ubiquitin to protein

- targets protein for breakdown by proteasome

- characterized by mental retardation, speech impairment, excitability, inapprop. laughter

- unsteady gait and unsteady limbs

Anhidrotic extodermal dysplasia

- mosaicism, resulting from incomplete X-inactivation in females

- mutant X chromosome gene à lack sweat glands

- use electrochemical resistance test to find areas of skin that are mutant

Aniridia

- caused by a PAX6 mutation

- absence of the iris

Ataxia Telangiectasia

- a disease of DNA repair (nothing else mentioned in lecture)

Becker Muscular Dystrophy (see DMD)

Bloom’s syndrome

- defect in BLM helicase (but other helicases not affected)

à helicase works to unwind tetraplex DNA structures

- leads to chromosomal and genetic instability

Causes: Growth retardation Sunlight sensitivity

Decreased fertility Immunodeficiency

Cancer (both solid tumors and leukemias)

Bruton’s agammaglobulinemia

- defective tyrosine kinase à defective maturation of B-cells

à no Ig production à bacterial infection susceptibility at 4-8 months

(initially ok, b/c antibodies from mother’s breastmilk)

Campomelic dysplasia

- mutant SOX9 gene (homologies to SRY, sex determining region of Y chromosome)

- underdeveloped thorax, bent limbs

- sex reversal (i.e. female with XY karyotype)

Chronic atrophic gastritis – see pernicious anemia

Complete Androgen Insensitivity Syndrome (Testicular Feminization)

- XY female

- has testicular tissue (secretes androgens

- but defective androgen receptor à androgen insensitive

- phenotypically normal female, with some abnormalities

Congenital Adrenal Hyperplasia

- defect. adrenal cortex à overproduction of androgens à virilization of female infants

- often caused by deficiency 21 hydroxylase

Craniosynostosis

- caused by Fibroblast growth factor response genes (cell division/migrat/diff.)

- mutant FGFR 2

- irregular shape of skull

- i.e. Crouzon syndrome and Apert syndrome

Cri du Chat syndrome

- caused by deletion of the p region of chromosome 5

- classified as a contiguous gene syndrome (segmental aneusomy syndrome)

- characteristic “cat cry” in newborns, caused by a pharyngeal defect

- the following webpage has a link to hear what the cry sounds like, and other info

http://www.criduchat.asn.au/criduchat/video.htm

* Cystic Fibrosis

- autosomal recessive disorder (“horizontal” inheritance, heterozygotes not affected)

- single gene mutation, involving a 3 BP deletion (therefore in frame), Δ508 common

- mutant cystic fibrosis transmembrane regulator gene (CFTR)

- abnormal regulation of chloride ion passage through endothelial cells

- In skin, CFTR normally keeps Cl- ions out of sweat ducts

- mutant increased NaCl in sweat à “salty sweat” diagnosis

- In endoth. cells lining airways, Cl- not transported out of cells

à Na rushes in, followed by water à highly dehydrated mucus

- causes lower airway inflammation and infection, chronic breathing difficulties

- GI reabsorption problems, diabetes mellitus, defective pancreatic secretion, cirrhosis

- azoospermia, lack of vas deferens in men

- also decreased fertility in women

- average survival age of 33 yrs.

- more common in Caucasians

- no good molecular test for CF

DiGeorge anomaly of brachial arch defects (related to the other DiGeorge stuff below???)

- defect in thymus development (site of T-cell differentiation and selection)

à leads to T-cell deficiency

à susceptible to yeast and viral infections

DiGeorge Velocardiofacial syndrome

- actually a combo of 3 sydromes: DiGeorge Syndrome (DGS)

Conotruncal anomalies face (CTAF)

Velocardiofacial syndrome (VCFS)

- deletion in DiGeorge Syndrome critical region (DSCR) – [chromosome 22]

- Cardiac problems are the most life threatening

- also cognitive impairments and neonatal hypocalcemia

Down syndrome – see trisomy 21

* Duchene muscular dystrophy (DMD)

- x-linked recessive trait

- apparent non-carrier mother might produce offspring with germline mosaics

- increased risk of child passing on to their progeny

- carrier females are normal

- mutation in the dystrophin gene at Xp21

- the largest human gene

- most common mutation is deletion, though duplication or bad splicing also poss.

- frameshift mutation à absent protein

- dystrophin mainly found in skeletal, heart, smooth muscle tissue

- interfaces between cytoskeletal proteins and group transmembrane proteins

- protein helps maintain muscle calcium permeability

- also acts as shock absorber (prevents damage when fibers contract)

- characterized by progressive myopathy (muscle wasting), starting at age 3-5

- death at age 20-25 from respiratory failure

* Becker muscular dystrophy (BMD)

- less severe progressive myopathy than DMD, less common than DMD

- truncating mutations at Xp21

- in-frame mutation à partially functioning dystrophin protein

- typically later onset than DMD

- Molecular Testing:

- Immunofluorescence staining for dystrophin protein in muscle sample

- PCR amplification of gene

Edward’s syndrome – see trisomy 18

Emphysema

- environmental factors can influence genes

- α1-anti-trypsin, coded by the Serpina genes (multiple alleles)

-blocks degradation of proteins (inhibits trypsin and elastase)

- smoking introduces superoxide anion à inhibits this enzyme à can’t protect

à breakdown of structural proteins à emphysema

Faconi Anemia

- bone marrow failure, growth retardation

- skin, G.I., and kidney abnormalities

- increased cancer occurrences, particularly myolegnous leukemia

- increased chromosome damage (more susceptible)

- defect in a Faconi complementation group

à possibly knocks out important signal or repair pathway

* Factor V Leiden Trhombophilia

- single point mutation in factor V gene at nucleotide 1691

- Gà A base substitution leads to Arginine à to histidine

- occurs at an APC cleavage site (therefore Activated Protein C resistance)

- causes slower inactivation of Factor V by APC

à increases thrombin production

- increased risk of clotting (thrombosis)

- heterozygotes have slightly elevated risk

- detect mutation by MnI 1 enzyme digestion

(mutants have one less cleavage site, so produces 2 DNA fragments instead of 3)

Fetal Alcohol syndrome

- teratogen = ethanol

- causes low birthweight, growth delay, heart defects

Fetal Dilantin syndrome

- teratogen – dilantin (anti-epileptic drug)

- causes hypertelorism, short nose and low nasal bridge

Favism

- example of ecogenetics

- decreased activity in glucose-6-phosphate dehydrogenase

à less reduced glutathione à oxid. damage to RBC’s

- oxidant drugs can accelerate loss of glutathione

- or ingestion of fava beans

- So is it just me, or does half of what we learned in block 1 relate to Silence of the Lambs?

Folate deficiency - see homocysteinemia, neural tube defects, megaloblastic anemia


Fragile X syndrome

- classified as a DNA repeat expansion disorder, loss of function

- most common inherited form of mental retardation (remember, trisomy 21 is common, yet it is not caused by the inheritance of a specific gene, but rather by having an extra whole chromosome)

- some have mild facial dysmorphic features, while others have no symptoms at all

- self-injurious, hyperactive, and impulsive behaviors most common

- fragile site contains a CGG sequence, repeated in the 5’ region of FMR1 gene

- repeat occurs upstream of the start codon

- FMR1 encodes an RNA binding protein that interacts with polyribosomes

- with disease, characteristic low expression in brain and testes

- more repeats à greater inactivation (b/c highly unstable during mitosis)

(6-50 repeats is normal, 50-200 is permutation, 200+ is full mutation)

- repeats due to uneven crossing over or amplification b/c of defect replication

- repeats accumulate over generations

- to test for fragile X, run PCR of FMR gene, then separate using electrophoresis

- look for large DNA sequence (won’t move far in the gel)

Galactosemia

- autosomal recessive, equal gender and race ratios

- can’t metabolize galactose (serum levels higher than 3-4 mg/dl)

- defective galactokinase or uridine diphosphate hexose-4-epimerase

- Most common – defect galactose-1-phosphate uridyltransferase

- can’t convert to UDP-galactose and glucose-1-P

- severity depends on type of variant

- buildup of galactisol (toxic)

- causes cataracts, jaundice, liver enlargement

- TREAT: - restrict galactose in diet (i.e. milk)

Gout

- defective purine metabolism à excess uric acid

- Sodium urate crystal deposits in joints à painful attacks, often in toes

- multiple possible causes:

- defective PRPP synthase à can’t be regulated à oversynth of purines à

therefore many broken down)

- defective glucose-6-phosphatase à increased G6P à inc Ribose 5-P via PPP

à therefore oversynthesis of purines

- partial HGPRTase deficiency à can’t salvage purines properly à inc uric acid

(Gout is not actually caused by the evil gout demon with the stingray barb tail)

- Follow this link for an 18th century newspaper editorial regarding the treatment of gout

http://www.infopt.demon.co.uk/grub/gout.htm

Hand-foot-genital syndrome

- caused by HOX-A13 mutation

- just like it sounds, defects in the hands, feet, and genitals

* Hereditary Hemochromatosis

- iron overload disorder (transferrin saturation, increased serum ferritin)

- autosomal recessive trait, but phenotype is sex-influenced

- less common in females

(possibly b/c of less dietary iron uptake and losses via menstruation)

- molecular testing on HFE gene (common: C282Y and H63D mutations)

- HFE facilitates transferrin-bound iron uptake from gut

- if mutant, cell fails to sense circulating T.F.-bound iron

à increased absorption of dietary iron

à high expression of iron transporters, but low ferritin expression

- heterozygotes have elevated serum iron, but no major iron overload complications

- takes long time to develop pathology

- especially toxic to liver and kidneys


Hemophilia A

- X-linked recessive

- higher incidence in males (since hemizygous)

- deficient in factor VIII (clotting factor)

- traced in royal families of Europe

Hereditary Orotic Anemia

- caused by two deficiencies: à can’t make pyrimidines

- orotidine-5-P phosphorylase

- orotidine-5-P decarboxylase

- leads to decreased UTP (inhibitor of the pathway)

à high flux through à increased orotic acid

-Treatment: uridine

(+ kinase) à UMP à UDP à UTP synthesis à can downregulate pathway

Hermaphroditism (2 classifications)

PSUEDO: - XX male w/ gonadal dysgenesis (can’t undergo puberty)

- SRY factor was transferred to X chromosome

TRUE: - 46, XX

- has both testicular and ovarian tissue, both sex organs, or ambiguous

Heterogeneous Nonpolyposis Colon Cancer

- a disease of DNA repair (nothing else mentioned in lecture)

HIV (Human Immunodeficiency Virus)

- great variety among viruses b/c lacks reverse transcriptase 3’-5’ exonuclease activity

- polycistronic nuclear material, RNA spliced after transcription

à target for protease inhibitors!

Homocystinuria

- autosomal recessive

- homocysteine is produced from methionine.

- Classical Homocystinuria - defect serine cystathione synthase (chromosome 21)

- can’t covert homocysteine à cystathione

- also disease with B12 deficiency

- can’t re-methylate homocyteine to form methionine

- Methionine synthase deficiency

- converta homocysteine to methionine (requires methyl-THF cofactor)

- blocks the reconversion of methyl-THF à THF

- Characterized by developmental delay, childhood osteoporosis, myopia & ectopia lentis

- increased CHD (increased thrombus risk)

- TREAT with folic acid, pyroxidine

- restrict methionine in diet, and supplement cysteine.

Huntington’s Disease

- single gene mutation

- classified as a DNA repeat expansion disorder

- autosomal dominant (“vertical” inheritance, heterozygotes are affected)

- expansion of CAG repeat in the coding region for the protein Huntingtin

à causes a polyglutaminyl chain to be added (gain of function)

- extra glutamines causes Huntingtin aggregation in brain cell nuclei

- binds to Huntingtin associated protein (HAP) in brain

- later onset, usually well into reproductive age (30-50 years old)

- all heterozygotes will eventually express phenotype

- 50% express it by age 50

- chorea (abnormal voluntary movement)

- dementia (basal ganglia atrophy and dilation of lateral ventricle)

(degredation in cerebral cortex and basal ganglia)


Hyperammonemia

- autosomal recessive, equal gender ratios

- disorder of urea cycle, many enzyme deficiencies possible

- most common – deficient ornithine transcarbamylase

- can’t convert carbamoyl phosphate à citrilline, for shuttle out of mitochondria

- anorexia, hypothermia, edema, respiratory problems, tremors, poor coordination

- TREAT: - stop protein uptake

- supplement non-protein calories

- dialysis

Hyperphenylalaninemia

- autosomal recessive (equal gender ratios, but racial differences exist)

- generally classified with serum phenylalanine higher than 2mg/dl

- Classical Phenylketonuria Hyperphenylalaninemia (most common)

- caused by lack of phenyalanine-4-hydroxylase [PAH] (can’t catabolize)

- allelic heterogeneity allows for different levels of enzyme (different severities)

- Non-classical PKU (BH4 deficiency hyperphenylalaninemia)

- deficiency in tetrahydrobiopterin (BH4)

- this is a necessary cofactor for PAH

Hyperphenylalaninemia characterized by:

- increased serum phenylalanine

- decreased tyrosine, melanin, fumarate, and catecholamines

- irritability, eczema-like rash, musty urine odor

- microcephaly, mental retardation

- TREAT: - supplement BH4 and tyrosine

- avoid high protein foods

- high carbs and fats instead for energy