Retinoblastoma

 

-RB gene is an antioncogene

-oncogenesis occurs with mutational inactivation of both copies

-RB gene product functions to inhibit DNA synthesis and cell proliferation

-gene located on 13q14 region

-tumorogenesis requires homozygosity of mutant allele

 

-inheritance of predisposition is dominant

-tumor formation is a recessive trait

-inheritance of one allele imparts predisposition

 

-CELL OF ORIGIN OF Rb is likely immature neural epithelial cell (have potential to differentiate into photoreceptors or Muller cells)

-retina matures in a wave from posterior pole to periphery

-peripheral retina most likely to be affected by late-onset tumor

 

-DNA analysis is available for detecting mutations of RB1 gene

-some mutations have higher penetrance than others (overall about 80%)

 

-40% hereditary, 60% sporadic

-only 10 percent have familhy history

 

-incidence 1/18000 live births

-assoc with advanced paternal age

-recent evidence for inc incidence in developing countries

 

eval:

-correct diagnosis can be made in >99% of cases

-presenting signs/symptoms:

age <5

leukocoria, greater than 50 percent

abnl red reflex test by pediatrician

strabismus

red, painful eye

orbital cellulitis

 

DDX:

persistent fetal vasculature

coats' disease

ocular toxocariasis

congenital cataract

ROP

 

w/u:

to confirm the diagnosis

to stage the tumor for treatment planning

 

w/u:

physical exam by pediatrician

imaging study

staging EUA

 

CT vs. MRI

CT less expensive and faster

CT better at detecting calcified lesions

MRI more sensitive for optic nerve and pineal lesions

MRI has no radiation exposure

 

U/S

dependent on echographer

good sensitivity for calcification

allows for accurate measurement of size

 

EUA complete routine w/u:

slit lamp for rubeosis and ant vit seeds

fundus drawing

photography

 

Metastatic workup:

done only when there is evidence of extraocular disease

-bone marrow bx, LP, bone scan

 

other ancillary tests:

fine needle bx - WARNING: viable tumor cells demonstrated in needle tracks

aqueous neuron specific enolase and lactate dehydrogenase

 

atypical manifestations:

presentation at older age

hyphema

vit hemorrhage

glaucoma

diffuse infiltrating rb: hypopyon, granulomatous appearance, orbital cellulitis

 

RB in older children:

34 of 400 greater than 5 years old

active RB in 26 and retinoma in 8 (benign)

misdiagnosis common: 5 of 26 had undergone prev vitrectomy

 

Reese-Ellsworth

developed in 50's to predict survival of eye after external beam radiation

indirect ophthalmoscope was just being introduced

anterior lesions, even if small, classified as more advanced

any vitreous seeding places eye into worst category

 

Murphree:

based on likelihood of salvage based on todays treatment modalities (chemo, laser, cryo, EBR)

 

Endophytic grow in vs. exophytic grow out towards RPE:

no difference in optic nerve invasion, orbital recurrence, or survival

 

diffuse infiltrating:  no calcium, no mass, just thickened retina, almost always unilateral, may have pseudohypopyon

 

Retinoma: benign variant, no growth or mets

 

Treatments:

Focal treatments:

photocoagulation (if less than 1 DD)

hyperthermia (TTT, using diode laser for long time)

cryotherapy - for lesions up to 2.5mm in thickness (free-thaw 3x)

often require sequential treatments every 2-3 weeks

increase cellular uptake of carboplatin so used in conjunction with chemo

 

Brachytherapy

-I125 plaque

-dosimetry calculated for 40 Gy to apex if primary treatment (different from melanoma=100)

-use 20 Gy for eyes previously treated with chemo

 

Chemotherapy:

-sig advance in management of intraocular RB

-primary (neoadjuvant) chemotherapy: first therapeutic intervention, can be followed by focal therapy (chemoreduction)

-adjuvant chemotherapy: given after enuc to prevent mets, used when path specimen shows ON invasion beyond lamina or massive choroidal invasion

 

agents used (CEV protocol = carboplatin, etoposide, vincristine)

complications/side effects: bone marrow toxicity, anemia, neutropenia; renal, hepatic, ototoxicity; nausea, vomiting, alopecia

etoposide likely inc risk of future leukemia

 

periocular carboplatin combined with focal treatment:

higher vit levels and lower serumxxx

 

external beam radiotherapy:

most effective still

inc chances of second malignant neoplasms in field of treatment

mortality rate of SMNs at 40 years in bilateral RB: 6% without EBR, 35% with EBR (these are if given before age 1)

 

enucleation:

indications

-tumor filling >half to two-thirds of the eye

-no likelihood of salvageable vision

-if giving chemo anyway for the other eye, can wait and judge response before enucleation

technique

-leave low stumps of LR and MR for traction

-lateral canthotomy

-goal is >10mm of optic nerve

do not clamp nerve before dividing

medpor implant wrapped in sclera, muscles attached

 

Follow-up:

-appearance of treated tumors (regression patterns)

type I - calcified scar, looks like cottage cheese, II - semitransparent, fish flesh appearance, III - combination of I and II, IV- flat chorioretinal scar (best)

these patterns originally defined with use of EBR

type I and IV are the most likely to be stable long term

 

screening for trilateral RB (in both eyes and pineal gland)

-3% of patients

-question of benefit from screening on ultimate survival

-recs vary from no screening to MRI scanning every 3-6 months x 9 years

we currently scan everyone at diagnosis and bilateral cases yearly thereafter

 

genetic counseling:

important numbers

-offspring have 50% chance of inheriting RB1 gene

-penetrance is 80-90%, so 40-45% chance of having RB

-10% of heritable cases are unilateral

-risk for sibling of new bilateral patient is 2% if parents are clear

-risk for sibling of new unilateral patient is 1% if parents are clear

 

 

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