What are some psychosocial implications of congenital craniofacial anomalies?

8/9/2019 What are some psychosocial implications of congenital craniofacial anomalies?

1/69

What are some psychosocial impl i ca t ions of

congeni ta l

cran io fac ia l

anomalies?

What fac to rs con t r ibu te to

the

cur ren t

s i tua t ion? b r i e f

h i s t o r i c a l

compara t ive survey

of

per t inen t sc ience and noted

t h inkers .

I

j u s t wanna be normal Doctor, give me t ha t

old

fashioned

normali ty

- -Ac t re s s

ma

Thurman

as

Sissy Hankshaw pleading

to

Dr.

Dreyfus in the f i lm Even Cowgirls ge t t he

Blues


2/69

Dedicat ion

To Dr. Robert

W Blakeley

Professor of

Speech

Pathology

Director Craniofacial Disorders Program

Oregon Health Sciences

University

Child

Development

Rehabi l i t a t ion enter

Thank you

Your e f fo r t

and

t reatment on

y

behal f have

given me the voice to

m ke

this

oral

presentat ion


3/69

Tab le o f Con ten t s

Page In t roduc t i on

Page 2

i r t h

Page

Freud

Page

7

Hi s t o r y

Page 9 Teas ing

Page 2 Speech

Page 5 Bi

b l i ography

Fina l

Page Reasearch Out l ine


4/69

In t roduc t ion

y

i n t e re s t in the sub jec t of the psychosocia l

impl ica t ions o f congen i ta l c ran io fac ia l anomalies i s

persona l .

I am a f f l i c t e d

with

a un i l a t e r a l c l e f t

l i p

and

pa la te .

I welcome the oppor tuni ty

to wr i t e and

speak

on

the

subjec t to

promote fu r the r

publ ic unders tanding.

The

process a l so provides me with g re a t e r

se l f -awareness .

This repor t wil l focus

on

persons with

congeni ta l

anomalies

as opposed to

acquired disf igurement . Acquired

cran io fac ia l

disf igurement r e s u l t s

from

acc iden t s f i r e s and

i l l ness .

The psychosocia l

impl ica t ions for

each group vary

and

I

wil l

note them

throughout

the

r epor t .

y presen ta t ion wi l l include photographs

of

persons

wi th

var ious examples of c ran io fac ia l

d iso rder s .

Unlike

other d i s a b i l i t i e s f a c i a l disf igurement i s to

a

la rge

extent

v i sua l

and

a

p ic tu re

r e a l l y

i s

worth

a

thousand

words.

I

wil l begin

t h i s

paper

with the

s to ry

of a

young

mother and

the b i r t h of

her son.

I ll a l so discuss

Freudian

impl ica t ions

h i s to r i c a l background the subject of t eas ing

and

speech

concerns .

In

America about

4

mil l ion bab ies are born each

year.

Between

the

7th

and

8th

week of ges ta t ion the nasomedial

process completes the fus ing o f t he phi l t rum of the l i p .

This

f a i l s

to

occur

in

about

of

every

700 l i ve b i r t h s

r e s u l t i n g

in about 5 700

new

c l e f t

cases

per year .

Page- l


5/69

Bir th ,

the

beginning

of mother -ch i ld re l a t ionsh ip

I would

l i ke

to begin t h i s pro jec t by

cons ider ing the

thoughts of a young mother ,

Rita Brzozowski,

and

her

reac t ion to the

b i r t h

of her

son,

Adam.

Ri t a ' s

s to ry

appeared in the May/June 1992 i s sue of AboutFace, a

cran io fac ia l support

group

news le t t e r .

Rita begins ,

For most people,

having t h e i r f i r s t ch i ld

i s

an ex c i t i n g

event .

There are the

usual

concerns

about

what

could

go

wrong, but a

normal, hea l thy baby i s expected. When our

f i r s t

ch i ld ,

Adam was born t h i s was

not

the

case . I t s t a r t e d with a d i sce rn ib le hush in

the

de l ive ry

room. Jus t

a

hare

l i p ,

r ep l ied my

obs t e t r i c i a n . Not able

to

see the

baby 's face, I t r i ed to r e c a l l anything I

knew

about

t h i s

condi t ion

When I

saw

him,

my hear t

sank.

This

was

not

the per fec t baby

I had envi s ioned -

the

one with

the

rosy

checks,

de l ica te l i p s

and

upturned nose.

This baby ' s face was

d is f igured

As I held

my

newborn

son,

a l l I could see was t h i s

de fec t I f e l t I was in a dream and held

someone e l s e ' s ch i ld .

Later

Rita

would say, My ego had su f fe red a major

blow. I did

not

fee l pr ide in showing my baby to o ther s .

Even a

t r i p

to

the

p e d i a t r i c i a n was a cha l l enge to my pr ide

Page-2


6/69

and

vani ty . In the

wai t ing

room

I

would keep Adam s

face

hidden over

my shoulder .

My in format ion

i n d i c a te s t h a t

R i t a ' s

exper ience i s

not

unusual .

Brant ley

and

C l i f for d (1979b)

found

t h a t

mothers

o f ch i l d ren with

c l e f t l i p and/or p a la t e

reported

s i g n i f i c a n t l y g rea t er

n e gat ive r e c o l l e c t i o n s o f the

postnata l

per iod than mothers o f normal c h i ld r e n . Other

rea c t i o n s have

been

more n e gat ive , inc lud ing abandonment

o f

the

ch i ld . This

i s

par t i cu l a r l y t r ue

in Russia Blakeley) , ~

~

t

·

{:r>

Korea and to a

l e s se r extent , China Li ) .

ll

of t h i s

informat ion po in t s to

an

important fac t : The in te rac t ion

between

mother

and c h i l d i s

a

c r i t i c a l f ac t or in determin ing d \

j » »

Nv

,LA

the psycho log ica l adjus tment o f t h e s e c h i ld r e n . ~ \ · ~

recent

s tudy

e n t i t l e d The Role o f

Maternal

Factors in

the Adaptat ion

o f

Children with Cra n i o f a c i a l

Dis f igurement

was completed a t Harvard

Medical

School and Chi ldren ' s

Hosp i ta l ,

Boston,

M

(Campis,

DeMaso,

Twente, 1993)

and

reported

in

The

C l e f t

P ala t e - Cr an io f ac ia l

Journal

(January

1995) .

This s tudy hypothes ized t h a t maternal

adjustment ,

percept ions ,

and s o c i a l

support

would b e t t e r p r e d ic t c h i l d

adapta t ion

to

c r an i o fac i a l dis f igurement than

medical

se ve r i ty .

Of the 77 ch i l d ren (ages 6-12) in the s tudy, 33

had c l e f t l i p

and/or p a l a te , the o ther 44 had a

more

s ev ere

deformity . The

s tudy found

tha t maternal adjus tment

and

maternal

percept ions

o f

the

mother -ch i ld r e l a t i on sh i p were

more potent pred ic tors of c h i l d r e n ' s emot ional adjus tment

than e i t h e r

medical s e v e r i t y

or maternal s o c i a l support .

This

s tudy a l s o

reported

t h a t the degree o f f a c i a l

Page-3


7/69

disf igurement

had

no

re l a t ionsh ip to ch i ld

o r

maternal

percept ions ,

but

t ha t

having a

comorbid

severe

medical

cond i t ion

was re l a t ed

to grea te r behavior problems in

chi ldren . This s tudy c i t e d nine pr io r

s tud ies

which

indica ted

t h a t

ch i ld ren with c ran io fac ia l dis f igurement

have

d i f f i c u l t i e s in

psychological

adjustment and

two

s tud ies to

the cont ra ry . The s tudy a l so c i t e d o ther c o n f l i c t i n g

information in almost every

ca tegory .

The major l i m i t a t i o n

of

t h i s

study was t ha t

the

eva lua t ion of ch i ld adjus tment

relied." on, p ~ ~ l e ~ t r ~ ~ ~ E t t _ Also, g e d o . n n ~ ~ ~ .upper r S ~ )i th

a ~

r - f ~ Vf J

yv

f 1 N ~ 1(1 01\ L VI

d


8/69

infant i n t e rac t ion ; (3)

mothers

of CFA ch i ld ren with v i s i b l e

de fec t s

repor ted

l e s s favorab le soc ia l suppor t than mothers

of CFA ch i ld ren

without

v i s i b l e defec t s .

One

i n t e r e s t i ng

aspec t of t h i s survey was t h a t

the

po ten t ia l p red ic to r va r iab le s

used

were based on

observa t iona l

measures of

mother- infant

play and t each ing

i n t e rac t ions . These

measures

are in f requen t ly used in CFA

psychological

research

desp i te t h e i r widespread app l ica t ion

in o ther a reas

of

developmental psycholog y. Also, mothers

of CFA ch i ld ren

repor ted

higher l eve l s

of

emotional d i s t r e s s

and

grea te r mari ta l c onf l i c t than

con t ro l s , as

repor ted

on

s tandard ized ques t ionnaires . In

the

Child

Behavior

Checkl is t

CBCL)

por t ion of t h i s s tudy, g i r l s with CFA had

mean scores above the 85th p e rc e n t i l e fo r t h e i r normative

group, but

boys

with CFA had

mean

scores

very

near t h e i r

normative average . In

add i t ion

18 o f the CFA ch i ld ren had

CBCL

scores

above

the

95th

p e rc e n t i l e ,

ind ica t ing the

need

for c l i n i c - r e f e r r a l fo r psych ia t r i c

problems (compare with

non-CFA ch i ld ren

a t

cons iderab ly l e s s

than 5 ). The

researchers

suggested t ha t

the mo ther s

ch i ld -d i rec ted

or i en ta t ion dur ing play with he r

in fan t

o r todd le r may

pred ic t

maternal

behavior problem

r epor t s

up to 4 years

l a t e r ; higher

l eve l s

o f

ch i ld -d i rec ted

play s k i l l

were

assoc ia ted with lower

subsequent

CBCL

score s .

As we

have seen

from

the

foregoing informat ion, the re

are

ea r ly psychologica l

impl i ca t ions

fo r persons with

congeni ta l

c ran io fac ia l anomal ies . Juxtaposed with

acquired

disf igurement

in

l a t e r

l i f e , t he se impl i ca t ions a re

unique.

Page-5


9/69

Freudian

Cons idera t ions

From

a Freudian

psychosexual developmental perspect ive ,

c l e f t l i p and

pa la te of f e r s

some

i n t e r e s t i ng

cons idera t ions .

These cons ide ra t ions

involve

the f i r s t psychosexual

s tage ,

the Oral Stage.

According

to

the theory , from b i r t h

to

age one, the

mouth, tongue,

and gums

are the focus of plea surab le

sensa t ions in the

baby ' s body, and

feeding

i s

the

most

s t imula t ing ac t i v i t y .

(Berger , 1994)

Feeding an in fan t with

c l e f t

l i p

and

pa la te presen t s a

cha l l eng ing s e t of physical c i rcumstances . Children with a

c le f t

p a l a t e

cannot c rea te s u f f i c i e n t

nega t ive

pre ssure to

suck

milk, which

i s

expressed from the n ipple

between

t he

upper

and

lower gum pads, because

of

the absence

of

a

pa la ta l

sea l (Berkowitz, 1994).

One

mother

descr ibed each

feeding of her c l e f t pa la te in fan t as a

nightmare

(AboutFace

January /February 1993).

In add i t ion to feeding

problems,

surgery o f

the

l i p ,

pa la te and gums

of an i n fan t presen t s an

oppor tuni ty

for

pain and

t rauma. Presurgica l or thoped ic al ignment

procedures as wel l as pos t su rg ica l concerns are another

source of poss ib le

in t e r f e r en ce

with

ora l

s t age development.

In add i t ion

pos t su rg ica l feedings

are

sometimes espec ia l ly

d i f f i c u l t .

fee l t ha t the

above i s sues

place an a f f l i c t e d

in fan t

a t r i sk for poss ib le

o ra l

s t age f ixa t ion . This i s

not

a

concern with

c ran io fac ia l disf igurement acquired

l a t e r

in

l i f e .

Page-6


10/69

His to r i ca l Perspec t ive

Dr. Benjamin M

Spock,

in h i s

l a t e s t

book

B et t e r

World

For Our

Children, provides

a poignant

example

o f

the

negat ive a t t i t u d e s

confront ing

persons wi th

congeni ta l

de fo rmi t i e s .

On page

21 of

h i s book, Dr.

Spock

r e l a t e s a

s to ry t o ld by h i s mother and the impact the remarks made on

him.

She taught us tha t

s i n fu l

thought s were

as

harmful as

deeds,

and

to touch ourse lves down

the re"

was

not

j u s t

s in fu l

but might

cause b i r t h

defec t s

in

our ch i ld ren .

After

four

years o f medical school

and

four r es idenc ies , I thought

I had long outgrown such

t eachings ,

but I

reca l l

when our

f i r s t

ch i ld

was born I re turned from the h o s p i t a l ' s nursery

to my w i fe ' s room

to

exclaim happ i ly , Mike

has t en

f inger s

and t en toes "

Attorney

Allen

Fagin

spoke

a t the 1992 ~ o n f r n

on fac ia l disf igurement

and noted

t ha t un t i l r ecen t ly a

number

of

major

American

c i t i e s had

ugly

laws

tha t

imposed

f ines on "unsight ly" people who were seen i n pub l i c p l ace s .

An

example was t he Chicago municipal code

which, un t i l

1974,

f ined

persons

who appeared i n pub l i c who were "d i seased ,

maimed, muti la ted

or in any

way

deformed,

so

as

to be an

uns igh t ly o r d isgus t ing

ob jec t" .

In addi t ion , I found

examples

da t ing from Medieval

England.

One example da t ing from the l a t e

e leventh or

twe l f th cen tu ry

i s

the f i r s t

ev idence

for

both c l e f t l i p and

p a l a t e

in Br i t i s h

archaeology.

Desp i te

the i n a b i l i t y to

b reas t - f eed , and

the

poss ib le

soc ia l

s t igma,

the indiv idua l

had survived

in to

adul thood (CPJ) .

Page-7


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12/69

Teasing

I ' d

l i ke

to begin t h i s sec t ion with a br i e f quote from

the book

Beauty i s

the Beast , Appearance-Impared Chi ldren

in

America ,

by Ann

Hil l

Beuf.

On Monday,

March

I , 1988,

an American

s ix th-grade s tudent

walked

in to

h i s

elementary

school c lassroom

and

shot himse l f .

He did

t h i s because h i s

classmates had

t eased him

about

being overweight .

Clear ly

the

t ime has

come

to address the problem of

t eas ing .

The

f i r s t

s tep in

confront ing

the i s sue of

t eas ing

i s

to give

t h i s ac t i v i t y

a

more appropr i a t e name: Verbal

Assaul t , based on d i s a b i l i t y

or

appearance.

In

a

sense, t

i s easy to unders tand

why

ch i ld ren

verba l ly assau l t those who look d i f f e r e n t . As

Dr.

McCurdy

s t a t e s in

h is

book The Complete

Guide to

Cosmetic

Surgery ,

Young ch i ld ren

are

extremely pe rcep t ive

of

d i f fe rences

in

appearance ,

and,

as they a re r e l a t i ve l y uninh ib i t ed in t h e i r

soc ia l i n t e rac t ions ,

such

d i f fe rences a re f ree ly poin ted

out .

Soc io log i s t ~ r e g o r notes t ha t der i s ive

l aughte r

i s

a l so a poten t

and

des t ruc t ive force . She wri tes These

reac t ions

to der i s ive

l augh te r

appear to be un iver sa l .

The

Hopi Indians , well

aware

of i t s

e f fec t ,

could and did

de l ibe ra te ly dr ive

an

offender in the community

to

i n san i ty

by

t he

s imp le

punishm nt

o f

l augh i ng

a t

h im .

;vi

~

Two othe r groups

o f

un l ike ly

offenders

are hea l th

care

prov iders

and

teachers .

When a ch i ld

i s

born

with

impaired

appearance,

many hosp i ta l s t a f f

members

employ

the term

F.L.K. ( funny

looking kid ) to desc r ibe

the

in fan t to one

another

(Beuf) .

Use of the F.L.K. term

c o n s t i t u t e s an

Page-9


13/69

act

o f

o b j e c t i f i c a t i o n . So does

any

use o f language that

r e fe r s to

the

c h i l d by h i s or her

d i s o rd er such

as the

c l e f t p a la t e in room 320

(Beuf ) .

That

i s , by

focus ing

on

the master- s tatus

o f

person-with- impaired-appearance and

thus

ignor ing

the t r a i t s p o s s e s s ed

by

the

c h i l d

as an

ind iv idual ,

the

s t i gm at i z e r manages to reduce

the

v ic t im to

the p os i t i on o f a th ing ra ther

than

a person

(Beuf ) .

T r i v i a l i z a t i o n was used

by

some doctors to

remind

the

ch i ldren of people who were worse o f f than they were, with

comments

such as ,

You're lucky you don' t have cancer .

Sure ly doctors do

not

s e t

out

to wound

the f e e l i n g s of t h e i r

young p at i e n t s , but they have been

taught

in

medical

schoo l

to judge

the

s e r iou s n e s s

o f

a medical problem in terms

of

i t s l i f e - t h r e a t e n i n g nature .

In a New York

Times s t or y

on phys ica l dis f igurement ,

author J i l l

Krementz

sa id that

whi le many o f the d i s f i gu r e d

ch i ldren

she

in terv iewed rece i v ed

support

from

t h e i r

p eers ,

a few were t ea s ed m e r c i l e s s l y or even

at tacked

by

schoolmates because o f how

they look.

The only

c h i ld r e n

who had r e a l l y pa in fu l t ime ~ m t h e i r peers were the ones

who

had

fac ia l

disf igurements

;

rementz

sa id

adding

t h a t ch i ldren

who

are miss ing l imbs

or

have other

d i s a b i l i t i e s

are more l i k e l y

to r e c e i ve comments

on

how wel l

they are

doing.

Krementz's o b s erv a t i o n s have been

noted by Macgregor

who s t a t es ;

there are two other handicapping aspect s

as s oc ia t e d

with d e n to - fac i a l

deformity .

In

the

Page-IO


14/69

f i r s t place , the a rea in

and

around the

mouth i s

both emot ional ly charged and s t rong ly connected

with

one ' s

se l f - image .

As an ins t rument

of speech

and

ea t ing , as wel l as

a mirror of emotions, t

a l so has unique soc ia l and psychological

impl ica t ions

and

symbolic meaning. Any

abnormali ty in t h i s area , t he re fo re ,

i s not on ly

high ly v i s ib l e and obtrus ive but - as

research

has

shown -

t ends to

evoke a type

of

avers ion

which

i s

both

e s t h e t i c

and sexua l . ~

Teachers

a re another

source of problems

fo r

a f f l i c t e d

chi ldren (The Providence Sunday Journa l ) . Examples

c i t e d

by

Beuf include a

t eacher

r i d i c u l i n g a

s tudent

who l o s t a

contac t lense

and another who's home-room t eacher

d i dn ' t

see

the harm

of

a

s tudent

being ca l l ed Dumbo ears because

of

pro t rud ing

ea rs .

Legis la t ion

such

as

t he

American 's with

Di sab i l i t i e s

Act,

the

Rehab i l i t a t ion Act of 1973, and

the

r ecen t ly

passed

Washington Sta te b i l l

# 5474

on

D is a b i l i t y

Discr imina t ion

should be u t i l i z e d to prevent t he abuses c i t e d in t h i s

sect ion . Paren t s must be wi l l ing to f igh t fo r t he i r chi ld ,

Beuf

emphasized, and

why

they don ' t i s sometimes a

mystery .

Page- I I


15/69

Speech

Speech i s

a

mirror o f

the soul ;

as

a

man speaks , so i s he.

Publ i l iu s Syrus , c i r c a

42 B.C.

Speech

i s a concern tha t

separa tes c l e f t

pa la te

from

some othe r c ran io fac ia l d iso rder s . The psychological

impl ica t ions

of speech present

cha l lenges

to

the

a f f l i c t ed

indiv idua l

from infancy

through

adulthood.

Clef t

pa la te

ch i ld ren

are a t

r i sk

for

language

development

problems.

screen ing

device to address t h i s

i ssue was the

subject

of a recent

s tudy.

The Parent

Ques t ionna i re

for

Screening

Ear ly

Language Development in

Chi ld ren wi th Clef t

Pala te

i s

the

titl of

t he paper .

Thi r ty sub jec t s , 16 to

30

months

of age rece ived the

MacArthur Communicative Development

Inventory:

Toddler

(CDI:Toddler) . cont ro l group was

a l so

t e s ted . Both

groups

received a speech language sc reening.

Resu l t s

indica ted t ha t the

CDI:Toddler was

a

va l id

sc reener

of

language development.

The

c l e f t

group demonstrated evidence

of

delays in

express ive

language

development . The

c l e f t

group had

a

mean

vocabulary of 177 words, compared with

288

words

for the

cont ro l group. The

c l e f t group

used

shor t e r ,

l e s s

complex

sen tences . I n t e l l i g i b i l i t y was poorer in

the

c l e f t

group.

Within

the

c l e f t

group,

hypernasa l i ty ra t ings of moderate

and severe were assoc ia ted with

express ive language de lays .

Page-12


16/69

Adult

c l e f t pa la te

popula t ions

are a l so a t

r i sk

for

impaired speech

concerns , when r e l a t e d to employment, as

demonst ra ted by two s tud ies . From a psychological

s t andpo in t Neiman

and

Duncan emphasized the importance o f

speech. This s tudy revealed t ha t speech

was

t he

s in g l e

fac to r

tha t

adverse ly a f f ec t ed t he se lec t ion

of

pres t ig ious

jobs even in

the

presence

of

a f a c i a l dis f igurement . I t

would

appear

t ha t speech

should be given

top pr i o r i t y .

(Lehman,

J r . , MD 1993)

I spoke with Dr.

Lehman

about t h i s s tudy, where photos

of persons

wi th

both un i l a t e r a l

and

b i l a t e r a l c l e f t l i p

and

pa la te were

shown to a

personnel manager.

Also presen ted

were t ape recorded

voices ,

both

normal

and hypernasal

speech.

The hypernasal

speech

was

much

l e s s

l i ke ly

to be

viewed as

having the

communication s k i l l s

needed

in

the

marketplace.

Another

s tudy

was

conducted

by

Dr.

Jane

Scheuer le

a t

the Tampa Bay Craniofac ia l Center . In t h i s t e s t adu l t

c l e f t

pa la te

sub jec t s voices were t ape

recorded, both

preopera t ive

and

pos tope ra t ive . The

recorded

voices

were presen ted to

a

panel

of th ree bus iness persons

for

eva lua t ion as

employees.

The

r e s u l t s favored

the

pos tope ra t ive voices unanimously.

Adult

speech i s sues were a l so

noted

a t the F i r s t

In te rna t iona l

Symposium

fo r Long Term

Treatment

in C le f t Lip

and

Pa la t e

a t the Univers i ty

of

Bern,

Swi tzer land . When an

adu l t

does

not

speak

cor rec t ly , those around

him

no t ice

t

immediately,

and

specu la t e whether or

not

the

af fec ted

person

i s

of

normal i n t e l l i g e n c e .

For t h i s

reason,

we

fee l

Page-13


17/69

t h a t cor rec t

speech has

many important

consequences . (J .

Weissen, 1979) From

the

beginning our team

considered

speech eva lua t ion

and

speech therapy as most important ,

because recep t ive speech, i . e . t ha t which one hears , i s

dependent on the

en t i r e

environment ( i . e . 36 degrees)

as

opposed to

the

ope ra t ive cosmetic r e s u l t which i s only

v isua l ,

i . e . maximal f i e l d

of

180 degrees .

(Weissen

M Bet tex ,

1979)

Goffman's view of c ran io fac ia l handicapping condi t ions

expressed

the

subject e sp e c i a l l y wel l . The c lose r the

de fec t

i s to

the communication

equipment

upon

which the

l i s t ene r

must

focus

h i s

a t t e n t ion , the smal le r the

defec t

needs

to

be to throw the l i s t e n e r off balance.

These

de fec t s

tend to

shut o f f the a f f l i c t e d indiv idua l from the

st ream of da i ly con tac t s , t rans fo rming

him

i n to a fau l ty

in te rac tan t , e i t h e r

in

h i s eyes o r

in

the eyes

of

o ther s .

Sigmund Freud was

an

i nd iv idua l who

su f fe red

acquired

speech impairment. In Apri l , 1923, Freud underwent surgery

for pa la ta l cancer . More ope ra t ions fol lowed in the

f a l l

and Freud

was compelled

to

wear a pros thes i s . He

had

Page-14


18/69

Bib l iography

1. Robert

W Blakeley ,

Ph.D. ,

Pro fes s o r

o f

Speech

Pathology,

Direc to r , Cran io fac i a l Disorders

Program,

Oregon Heal th Sciences

Univers i ty ,

CDRC Por t l and , OR

Personal communication

and

photographs .

2. The

Clef t Pa la t e -Cran io fac i a l

Journa l

(CPJ) ,

January

1995, Volume 32,

number

I , American Clef t

Pa la t e -Cran io fac i a l

Associa t ion (ACPA). "The Role

of

Maternal Facto rs in

the

Adapta t ion o f C hi ld ren wi th

Craniofac ia l

Disfigurement" by Les l i e Campis, Ph.D. ,

David Ray DeMaso,

M.D.,

and

Al l i son

White Twente,

Ph.D.

3.

CPJ, January

1995,

Volume

32,

number I ,

ACPA

Paren t

Ques t ionnai re

for Screening Ear ly

Language Development

in

Chi ld ren wi th C le f t Pala te 1993, Nancy Scherer , Ph.D

and Linda L.

D'Antonio,

Ph.D.

4. CPJ, November 1994, Volume

31, Number 6, ACPA

"Medieval

Example o f Cle f t Lip

and

Pa la t e from St . Gregory ' s

Pr io ry , Canterbury" , by Trevor Anderson, M.A.

5.

CPJ, September 1993, Volume 30, Number 5, ACPA

"Psychological

Funct ioning

of

Chi ld ren wi th

Craniofac ia l

Anomalies and Thei r

Mothers:

Follow-Up

from Late Infancy to

School

Entry"

by

Matthew

L.

Spel tz , Ph.D. ,

Kathi Morton,

Ph.D. ,

El izabe th

W Goodel l , Ph.D. , S t e r l i n g

K.

Clar ren , M.D.

Page-IS


19/69

6. Dr. Benjam in M. Spock,

A

Bet te r World For Our Children"

Nat ional

Book

Network,

1994

7. National

Foundation For Fac ia l

Reconst ruc t ion

(NFFR),

Conference

Proceedings of 11/18/92, "Specia l Faces:

Understanding Fac ia l

Disf igurement . "

8. AboutFace news le t t e r , May/June 1992, Vol. 6, No.3 ,

Rita Brzozowski,

A

Mother ' s F i r s t

Lesson",

cover s to ry .

9.

Weissen, J . ,

Speech

Therap i s t , Department of

Ped ia t r i c

Surgery,

Inse l sp i t a l ,

CH-3010

Berne.

Proceedings of

the

Fi r s t In ternat ional Symposium, Long Term Treatment

in

Cle f t

Lip and

Pala te , August,

1979, Univers i ty

of

Bern,

Switzer land.

10

Jim

Lehman,

J r . ,

MD, AboutFace

Newsle t t e r ,

March/Apri l ,

1993,

"Ask a Profess iona l

column

and persona l

communication.

11

Ningyi Li, MD,

DDS, Professor

and Chairman,

Department

of

Stomatology,

Qingdao

Univers i ty Medical

College and

Hospi ta l ,

Qingdao,

Shandong,

The Peoples Republ ic of

China. Personal communication.

12 The Complete

Guide to

Cosmetic Fac ia l

Surgery ,

John A.

McCurdy,

J r . ,

M

FACS, 1981, Freder ick

Fel l

Publ i shers ,

Inc .

Page-16


20/69

13. Ann Hil l Beuf, "Beauty i s the Beast ;

Appearance

Impaired

Children in America", 1990,

Univers i ty of

Pennsylvania

Press .

14.

Kathleen

Stassen

Berger, "The Developing

Person

Through

the Li fe Span", t h i rd

ed i t ion 1994, Worth

Publ i shers

15.

Samual

Berkowitz, DDS,

MS

FICD,

"The Clef t

Pala te

Story" , 1994, Quintessence

Publ i sh ing

Co.,

Inc.

16. The New

York

Times, Thursday, October 1, 1992, "Parent

& Child" by

Lawrence

Kutner.

17. Frances

Cooke Macgregor,

M.A.,

Social

and Psychological

Impl ica t ions of

Dento-Facia l Disf igurement ,

1969

18. The Providence Sunday Journa l October 7, 1990,

by

Rosemary Jones

of

the

Allentown

Morning Cal l .

19. Thurston County Works in Progess , November 1994,

Legis la t ive

Report

Card,

D is a b i l i t y b i l l

20.

Clef t Pa la te

and Clef t Lip: A Team

Approach to

Clin ica l

Management

and Rehab i l i t a t ion of the

Pat i en t .

1979,

W.B. Saunders

Company

21. Sigmund Freud, In t roduc tory Lectures

on Psycho-Analys is

Page-17


21/69

1966, W.W. Norton Company,

I n c .

Pe t e r Gay, i n t ro .

22. Dr. Jane Scheuer le Co-Direc to r Tampa Bay

Craniofac ia l

Center , Tampa,

Flor ida

personal communicat ion 1993.

23.

Goffman, E. Al iena t ion from i n t e r a c t i o n ~

Rela t ions 1957.

Page-18


22/69

LIFESPAN HUMAN DEVELOPMENT

J e r ry Shulenbarger , Facu l ty

Winter Q u ar t e r , 1995

Research

paper

t op ic :

What a re

some

psychosocia l i mp l i ca t i o n s

o f

congen i ta l

c ra n i o f a c i a l anomalies? What f ac to r s c o n t r i b u t e t o the

cu r ren t s i t u a t i o n ?

br i e f

h i s t o r i c a l ,

compara t ive

survey

o f

per t inen t

sc ience

and noted t h in k e r s .

At t h i s poin t I am submi t t ing my t o p i c

as

out l ined

above,

as opposed to a diagram, because I m n o t exac t ly sure

where the research wi l l t ake me. I have ordered

some

exce l l en t

books

on

the

sub jec t

o f c r an io f ac i a l anomalies and

need to read

them.

I

w i l l

a l so r e f e ren ce s ev e ra l cur ren t

mainst ream

publ i ca t ions

as

p a r t o f my

p r o j ec t .

My research

wi l l

a l so

inc lude inpu t from h ea l t h ca re p ro fe s s i o n a l s i n

the Pie ld and

t h e i r

r e s ea rch .

Submit ted

by:

Neil

J . G i l l e s p i e

866-7400


23/69


24/69


25/69


26/69


27/69

.

r ~ r f P4//lte.. C; /I#iOMU/1 /

)'dvfULv9/

.rl1

l.lr J 7 /916

Vel

~ I

The Role of Maternal Factors

n

the Adaptation of Children

with Craniofacial Disfigurement

LESLIE BIRON CAMPIS,

PH.D.

DAVID RAY

DEMASO, A.D.

ALLISON WHITE TWENTE,

PH.D.

This study hypothesized that maternal adjustment, perceptions, and social

.

support would better predict child adaptation to craniofacial disfigurement than

_

medical severity. Mothers of 77 children (ages 6-12 completed the Child Behav

ior Checklist, Beck Depression Inventory, Spielberger Trait Anxiety Scale, Social

Support Questionnaire Revised, and Parenting Stress Index. Medical severity

was assessed by the number of operations (craniofacial and other), comorbid

medical conditions, and the Hay Attractiveness Scale. The children and moth

ers in our sample resembled a normal population n terms of their psychologi

cal functioning and quality of the mother-child relationships. Maternal adjustment

and maternal perceptions of the mother-child relationship were more Dotent

predictors of children s emotional adjustment than either medical severity or mater

(

nal social support.

KEY

WORDS:

adaptation child adjustment craniofacial disfigurement

maternal adjustment medical severity mother-child

relationship social support

Craniofacial reconstructive surgery has progressed dra-

The variability in children's adjustment as

described

in

matically in the last several decades. New surgical tech-

these studies may in part be accoun ed for by the hetero:

nology

has

given children

with

craniofacial anomalies

geneity in diagnosis, age, methodology, and outcome mea-

significantly improved appearances and social acceptance

However, children with craniofacial conditions do

(Murray et aI., 1979). Along with these advances, inter-

appear to have

the

potential for healthy

adjustment.

est

in

examining

the

emotional adjustment of children

Researchers have identified protective factors in other

with

craniofacial abnormalities

has

generated increased

vulnerable populations which help children cope with

research. To date, the findings

of

these studies have

been

challenging life events

(Garmezy,

1981, 1985; Beardslee,

highly variable.

1989; DeMaso et aI., 1991). The protective factors which

A

number

of

studies have reported

that

children with may ameliorate the negative aspects

of

living

with

facial

craniofacial disfigurement have difficulties in psycho disfigurement have

not been well

defined

(Bennett

logical

adjustment, which include a diminished self-con

Stanton, 1993).

cept (Arndt et aI., 1986; Kapp-Simon, 1986: Pillemer and

Maternal emotional adjustment may be an influential

f

Cook, 1989), lowered school achievement (Brantley and

determinant

of

the emotional adaptation of children with

Clifford, 1979a; Kommers and Sullivan, 1979), marked

craniofacial

abnormalities. The substantial emotional

,social

inhibition (Kapp-Simon, 1986; Pertschuk

and

impact on parents when they learn of their child's mal-

Whitaker, 1988; Pillemer and Cook, 1989), and greater

formation has been documented in the literature. Brant-

dependence on adults (Pillemer and Cook, 1989).. ley and Clifford (1979b) found that mothers of children

trasL other researchers found these children to

have

psy

_with cleft lip

and/or

palate reported significantly greater

chosocial adjustment comparable to that of nonnal children_

negative recollections of the postnatal period than moth-

(Richman, 1983; Arndt et aI., 1987). Postoperative mea

,

ers of normal children. Similarly, Speltz

et al. (1990)

sures revealed improvement in

children's

self-concept,

reported that mothers of children with facial abnormali

adaptive

behavior,

and school

functioning. ties

experienced significantly greater

stress, felt less com-

petent, and experienced more marital conflict than a control

group. In contrast, Palkes et ai. (1986) found that children

Dr. Campis is Instructor in Psychology,

Dr

DeMaso is Assistant Profes-

with craniofacial disfigurement did not have a negative

sor of Psychiatry and Dr. Twente is Instructor in Psychology, Department

of Psychiatry at Harvard Medical School and Children's Hospital, Boston,

impact on parent's adjustment or a family's structure.

Massachusetts.

The interaction between mother and child is another criti-

This study was funded in part by a research grant from the Foundation for

cal factor in detennining the psychological adjustment

of

Faces of Children.

children (Rutter, 1986; Speltz et aI., 1993). Mothers' per-

Submitted October 1993; Accepted August 1994.

ceptions regarding their parenting skills and interactions with

Reprint requests: Dr. Leslie Biron Campis, Department of Psychiatry,

Children's Hospital, 300 Longwood Avenue, Boston, MA 02115.

their children may facilitate

or

hinder a child's adjustment

55


28/69

56 Cleft Palate-Craniofacial Journal, January 1995,

Vol

32 No. 1

(Holroyd and Guthie, 1986; OeMaso et aI., 1991). There has

been some examination of mothers' feelings and perceptions

regarding their interactions with their children with cranio

facial conditions (Field and Vaga-Lahr, 1984; Palkes et aI.,

1986; Barden et aI., 1989; Speltz et aI., 1990). Field and

Vega-Lahr (1984) found that these mothers were less active

in their interactions and their infants made less eye contact

·than nonnal children. Consequently, their interactions were

getennined to be significantly different from those of nonnal

infants and mothers.

~ e n

et at (1989) found that the moth

ers

of

children with craniofacial malformations reported more

personal and general life satisfaction than mothers

of

a healthy

control group. However, behavioral obselVations

of

the mother

· infant interactions demonstrated less nurturant relationships.

In contrast, Speltz et aI (1990) found no qualitative differ

ences in the interactions of children with craniofacial condi

tions (ages 1-3 years) and their mothers when compared to

a normal sample. The two groups of mothers were observed

to be equally responsive, critical, and instructive to their chil

dren. Their children were also equally responsive and active.

Similarly, Palkes et

aI

(1986) described parents as no less

accepting of their facially disfigured child or their parenting

responsibilities.

The extent and quality of a mother s social support net

· work

has been

identified as a m e i t i n ~

variable that

buffers the stress of having a child with a physical hand

icap (Barakat and Linney, 1992). Researchers have found

greater

social

support

to correlate with both

better parent

(Jessop

et

aI., 1988; Kronenberger and Thompson, 1992)

and child adjustment (Barakat and Linney, 1992). This vari

able is particularly

important

to study in children with

craniofacial

anomalies

as this parent

group

appears to

be

at greater risk for limited social support. Benson et aI.

(1991) and Speltz

et

aI. (1993) found that parents of chil

dren with visible facial deformities reported less social sup

12 2IL

Parents in the Benson et al. study were also less

satisfied with the

quality

of their social support. In con

,trast, Krueckeberg and Kapp-Simon (1993) found no dif

ferences in the

social

support

networks

of

parents

of

children with craniofacial conditions and a control group.

These studies assessed social support of parents whose chil

dren ranged in age from preschool to 7 years. Speltz et

ai. (1993) hypothes ized that social support decreases

over

time because of the negative effect of greater social expo

sure. It is important to study this variable in a school age

population.

It has been assumed that children with severe facial dis

figurement are at

greater

risk for disabling psychopath

ology. However, Macgregor (1979) disputes the validity

of

this assumption.

The

severity of medical impairment

has not proven to be a significant contributor to the

emotional adjustment

of

children with other physical ill

nesses (Stein and

Jessop,

1984; Mullins et al., 1991).

Researchers who have investigated medical severity or

its occurrence have found maternal adjustment (Walker et

aI., 1989), social support (Barakat and Linney, 1992), and

maternal perceptions (OeMaso et aI., 1991) to. be more

strongly associated with better child adjustment. Kruecke

berg and Kapp-Simon (1993) noted the need for more

studies to clarify whether severity

of

a craniofacial prob

lem or the mere presence of a

problem

is a crucial factor

in these families.

Maternal

emotional adjustment, relationship with their

children, and social support along with medical severity

of

a craniofacial condition have been identified as potential

influential factors in the adaptation of children with cran

iofacial conditions. This study uniquely examines the rela

tive contributions of all these variables to child adjustment

in a school age sample

of

these children. The maternal vari

ables were hypothesized to be significantly more predictive

of child adjustment than measures of medical severity.

METHODS

Subjects

The study was conducted in three outpatient settings

at Children s Hospital: (1)

Craniofacial Centre

Clinic,

(2) Cleft Lip and Palate Clinic, and (3) Outpatient Plas

tic Surgery Clinic. The subjects were 77 mothers whose

children

(ages from 6 through 12 years) were diagnosed

with an observable craniofacial anomaly. The children

(44 boys and 33 girls) had a mean age

of

8.1 years (SO

=

2.0). Table 1 lists

additional

sample

characteristics.

Mothers of children in a self-contained special

needs

classroom were excluded from the study. This study sample

was

part

of a larger sample of 100

mothers

and

children

between

the ages

of

6 and

17

years. In this

larger

study,

TABLE Sample Characteristics 0=77)

haracteristic

n

ojSample

Ethnic Group

White

67 87

Hispanic 4

5

Black

3

4

Other

3

4

Marital Status

Married

56

73

Divorced

15 19

Separated

3 4

Single 3 4

SES*

Major Business/Professional 21

27

Medium Businessrrechnical

23 30

Skilled Craft/Clerical 18 23

Semi-skilled

6 8

Unskilled

5

7

Craniofacial Diagnosis

Cleft lip and/or palate

33 43

Facial microsomia

8 23

Unilat. coronal synostosis

11

5

Vascular ano mal ies

6

8

Syndromic

craniosynostosis

4

5

Frontonasal dysplasia

4 5

Acquired facial deformity 1 1

*Dala

nor availahle on four subjecrs,


29/69

1 5

mothers were approached consecutively with five declin

ing to participate in the study.

Prior

to their clinic appointment, potential

participants

were mailed a letter informing them

of

the study's purpose.

The

research assistant invited mothers to participate in the

study while they waited

for their child's appointment.

Informed consent was obtained from all mothers, who then

completed

the

questionnaires

and

received

five

dollars

for

~ h e i r

participation.

Dependent Variable Measure

Child djustment

Measure

The Child Behavior Checklist (CBCL)

was

completed

by each mother (Achenbach

and

Edelbrock,

1983).

The

behavior problems measure of

the

CBCL

was used in this

analysis.

This measure

consists of 118 items which assess

internalizing

and

externalizing behavior

problems; an

overall

T

score

of

psychological adjustment

is

calculated

from these

subscales. Achenbach

and

Edelbrock (1983)

reported high test-retest

reliability

and good discriminant

validity.

Predictor Variable Measures

Demographic Variables

The

following

demographic

information was collected:

child's age, sex, and race; parent's occupation and level

of

education (Hollingshead, 1975); and marital status.

Maternal djustment

The Beck Depression Inventory (BDI) is a self-report mea

of depression (Beck Steer, 1987). The BDI has

21

syn1ptom-attitude categories which represent characteris

tic manifestations

of

depression. It has clinically validated

severity cutoff scores and extensive data regarding its accept

able psychometric properties (Derogatis, 1982).

. The Spielberger Trait Anxiety Scale (STAS) is a self-report

symptom mood inventory (Spielberger et aI., 1983). The

STAS, which contains 20 statements concerning how an indi

vidual feels, is designed to provide a measure

of

an endur

ing

personality

characteristic,

as

opposed

to a

transient

emotional experience. This scale has good internal stability

and construct validity (Derogatis, 1982).

Maternal Perceptions

The Parenting Stress Index (PSI) identifies parent-child sys

tems under excessive stress and at risk for the development

of dysfunctional parenting behaviors

or

child behavior prob

lems (Abidin, 1986). This 126-iten1 measure is divided into

Child and Parent domain subscales. The Child domain sub-

Campis et al.. MATERNAL

FACTORS IN

DISFIGUREMENT

57

scale assesses child temperament and the extent to which

these child characteristics are stressful to the parent. High

scores are associated with child qualities that interfere with

parenting.

The

Parent domain subscale evaluates parents'

personal characteristics and social support system as they

correspond to the demands

of

parenting. High scores indicate

that the parent characteristics are a potential source

of

dys

function in the mother-child relationship.

Although each mother comple ted the entire PSI, four

of

the

six Child domain subscales (Adaptability, Demandingness,

Mood, and Distractibility) were deleted because

of

their sub

stantial overlap with CBCL items. The remaining subscales

of

Child Acceptability and Child Reinforces Mother were

used as a more accurate measure

of

mother-child interaction

(DeMaso et aI., 1991). The Parent domain subscale was left

unchanged. The PSI has a reliability coefficient of .95 for the

total stress score.

Maternal Social Support

The Social Support Questionnaire Revised (SSQ) is a mea

sure

of

an adult's perceived level

of

social support available

from family and friends (Sarason et aI., 1987). The SSQ mea

sures the extent of the social network as well as the individ

ual's

degree

of satisfaction with their social support. The

questionnaire items posit different situations and the respon

dent lists supportive persons (up to nine people), character

izes their relationship with supportive persons (whether a

family

member or

friend), and rates their degree of satisfac

tion with the support provided. Two scale scores are derived

from the average number of persons listed (the number score)

and the average satisfaction rating (the satisfaction score).

Acceptable reliability coefficients for the total scale have

been reported at

or

above .75.

Medical Severity Measures

Medical data were collected

on

all children from medical

chart review.

The

craniofacial diagnosis was also recorded.

Information was obtained regarding: (1) number of cranio

facial operations, (2) number of noncraniofacial operations,

and (3) comorbid medical conditions. Comorbid medical con

ditions were defined as other problems requiring ongoing

monitoring or medication.

They

were

coded

none, minor

(e.g., otitis media),

or

major (e.g., seizures).

The

Hay Attractiveness Scale (HAY) was used to provide

an objective rating of physical appearance (Hay and Heather,

1973). The scale has nine points ranging from perfect features

(1) to very marked imperfection (9).

Each

child was assigned

an appearance rating by the research assistant at their hospi

tal visit. Interrater consistency was also assessed. Another

researcher familiar with the craniofacial population provided

appearance ratings for 25 randomly selected p r t i c i p ~ n t s

Comparison of the ratings revealed an 85% agreement WIthIn

one point on the scale.


30/69

7

58 Cleft Palate-Craniofacial Journal, January 1995,

Vol.

32 No. 1

TABLE 2

Means

and Standard Deviations·

Measure

Mean

SD

Maternal Adjustment

BDI

5.04

5.00

STAS

36.45 7.88

Maternal Perceptions

PSI Child Domin

24.30

6.40

PSI Parent Domain

151.56 39.82


SSQ Family Support

t

2.59

1.53

SSQ Friend Support

t

1.18

1.00

SSQ Satisfaction

5.21

1.09

Medical Severity

Total Operations

3.45

2.77

Craniofacial Operations

2.60

2.37

Comorbid Medical Problem

1.71

0.69

HAY

3.75

1.82

Child Adjustment

CBCL

55.84 11.00

*BDI = Beck Depression Inventory: STAS = Spielberger Trait Anxiety Scale; PSI = Parenting

Stress Index: SSQ = Social Suppon Questionnaire - Revised; HAY = Hay Attractiveness Scale:

CBCL =Child Behavior Checklist.

t

p

< .01.

RESULTS

Descriptive Data

Nine measures were used in these analyses: BDI, STAS, three

subscales of the SSQ, child and parent domains of the PSI,

HAY,

and the behavior problems measure of the CBCL. Oper

ations and comorbid medical problems were additional mea

sures used to assess medical severity. Means and standard

deviations are reported in Table 2.

The sample mean scores on the BDI, STAS, PSI, and CBCL

were all within the normal range. When the mean scores of

the SSQ were compared with the nonnative SSQ data, this

sample reported significantly more Family support and less

Friend support (p < .01). This sample's overall Satisfaction

rating was comparable to that of the normative group.

Intercorrelations between Maternal Factors

Intercorrelations were calculated to measure associations

between maternal adjustment and maternal perceptions. Given

the large number of correlations in this study, the signifi

cance level was set at the more stringent

.01

level for this and

subsequent calculations. The maternal adjustment and per

ception variables were only minimally correlated (Table 3).

Both Parent and Child domains

of

the PSI were significantly

linked to maternal reports of anxiety and depression, such that

mothers who reported more stress also reported increased

levels of anxiety and depression. The social support vari

ables were not related to maternal adjustment or maternal

perception. Global satisfaction with social support was only

significantly related to social support provided by family.

Influences on Maternal

Factors

Demographic Variables. Pearson correlation coefficients

were calculated to measure associations between demographic

TABLE

3

Intercorrelations among Matern al Adjustment

and

Maternal Perception

Variables·

Maternal Perception

Adjustment

Variables

1

2

3

4

5 6

PSI-C

PS -P SSQ- SSQ- SSQ- ST S BD

F FR

S

1. PSI-C

2. PSI-P

.58'

3.

SSQ-FA -.27 -.05

4. SSQ-FR -.11 -.19 -.05

5. SSQ-SA -.23 -.15 .34

t

.26

6. STAS .312

t

.30

t

-.01 -.14 -.10

7. BDI

.44

t

-.28 -.14 -.10

*PSI = Parenting Stress Index. Child Domain (PSI-C) and Parent Domain (PSI-P):

SSQ

=

Social

Suppon

Questionnaire - Revised Family (SSQ-FA) Friend (SSQ-FR).

and Global Satisfaction (SSQ·SA); STAS =Spielberger Trait Anxiety Scale;

BOI = Beck Depression Inventory.

p

< .01.

.p< 001

variables and the following measures: BDI, STAS, SSQ, and

PSI. There was a significant negative correlation p

<

.00 1

between SES and amount of social support from friends.

Mothers in lower SES families reported less social support

from friends. No other correlations were significant.

Medical Severity Measures. Since the medical severity

measures (operations, comorbid medical conditions, and

HAY) were skewed by some extreme values, Speannan rank

correlation coefficients were calculated to measure associa

tions between medical severity variables and the measures of

maternal adjustment and perception. No statistically signifi

cant correlations were found.

Predictors of Child Adjustment

Demographic Variables. Pearson correlation coefficients

were computed to detennine the relationship between the

demographic variables and child a d j u s t m e n t : l ~ ~ o n e of the

demographic variables were significantly correlated with

CBCL scores. Analysis of variance models using categorized

demographic variables to predict CBCL scores were con

structed and showed no significant effects for these variables.

Maternal Adjustment and Perceptions. Correlations between

the maternal adjustment, maternal perception, and child adjust

ment variables were also completed. Several significant pos

itive correlations were found (Table 4). The STAS, the BDI,

and the PSI (both Child and Parent domains), were all highly

correlated with the CBCL. Mothers who reported symptoms

of anxiety and depression described their children as more

TABLE 4 Pearson Correlation Coefficients for Child Adjustment

and

Maternal Adjustment,

Perception,

and

Social

Support Variables·

Child STAS BDI SSQ- SSQ-

SSQ- PS -C PSI-P

djuSTment F FR S

CBCL

-.17 -.14 -.26

*STAS =Spielberger Trait Anxiet)' Scale: BDI =Beck Depression Inventory: SSQ =Social Sup

pon

QUt'stionnaire - Revised Family (SSQ-FA). Fncnd(SSQ-F R). and Global Satisfaction (SSQ

SA): PSI

=

Parenting Suess Index. Child Domain (PSI-C) and Parent Domain (PSI-P): CBCL

=

Child Behavior Checklts .

p

< 001


31/69

maladjusted.

In

addition, the relationship between the PSI and

the CBCL indicates that stress within the mother-child rela

tionship was associated with child maladjustment.

Medical Severity Measures.

Speannan rank correlation coef

ficients showed no significant correlations between the med

ical severity measures and child adjustment. Additionally,

analysis

of

variance models using categorized medical sever

ity variables were tested. A significant effect was found for one

medical severity variable, comorbid medical conditions F=4.39,

p

<

.016).Aseries of planned comparisons revealed that

the

group

. with the most severe comorbid medical conditions had the

est CBCL scores. This group had more adjustment problems

than did children with mild or moderate comorbid medical con

ditions. The groups with mild and moderate comorbid medical

problems did not differ statistically.

Differential Contributors to

Child

Adjustment

The relative importance of specific measures in predicting

child adjustment was detennined next. First, the large set

of

predictor variables was reduced by creating a single sum

mary score (accomplished by summing standardized scores

of the individual variables) to represent each of the four vari

able domains: (a) maternal adjustment, STAS and BDI; (b)

maternal perception, PSI Child and Parent domains; (c) mater

nal social support,

SSQ

family, friend, and satisfaction; (d)

medical severity, operations, comorbid medical conditions,

HAY appearance rating.

A series

of

iterative multiple-regression analyses (Cohen and

Cohen, 1983) were then conducted using the set of four key

variables. Iterative analyses were completed so that each of

the four variables were entered last into the regression equa

tion. This approach provided an examination

of

the unique

contribution of each specific variable above and beyond the

variance explained by the other summary variables. The strat

egy also allowed for a test

of

the central hypothesis that

maternal adjustment and maternal perceptions would be more

predictive of child adjustment than medical severity.

The total variance in child adjustment explained by the set

of four summary variables was 38% (Table 5). As hypothe

sized, both maternal adjustment and maternal perceptions

made unique contributions to the explanation

of

child adjust

ment. Neither medical severity nor maternal social support

TABLE 5 Summary of Multiple Regression Analyses Predic ting

Child Adjustment·

Predictor Correlation Beta Uniqul

Variables with DV

Contribution

Maternal Perception .53' .35

.087§

Maternal Adjustment .47' .34 .086'

Medical Severity .13 .18

.030


-.27 -.06

.003

*TotaJ variance accounted for

by

model. R2 =

.38;

tCalculated as incremental change in multiple

R;

'P<

.001;

'Significant incremental change at p

< 05

level.

Campis et al.. MATERNAL FACTORS

IN

DISFIGUREMENT 59

contributed significantly above and beyond all other vari

ables to the total model.

DISCUSSION

The findings

of

this study support the psychological resiliency

and adaptive potential

of

children with craniofacial conditions

and their mothers. Mothers' emotional adjustment and per

ceptions of their relationship with their children were more

potent predictors of children's emotional adjustment than the

severity of the craniofacial anomaly. This finding corresponds

to other studies in which illness severity was less important

to a child's adjus tment than maternal functioning (Spaulding

and Morgan, 1986; Walker et aI., 1989). Furthermore, the

children and mothers in our sample resembled a normal pop

ulation in tenns of their psychological functioning and qual

ity

of

the mother-child relationships.

While there were no significant differences in maternal

adjustment between these mothers and a normal population,

this finding must be interpreted cautiously. This sample had

,a slightly higher means SES than the normative samples of

_the predictor variable measures. Mothers who did report clin

ically significant levels of depression and anxiety also rated

their children as having more behavior problems. This find

ing is consistent with that of Barakat and Linney (1992) who

found that maternal psychological functioning was related to

child adjustment. An impaired mother-child relationship,

including under involvement or overprotection, has been con

ceptualized as an important etiologic factor in childhood

depression (Weller and Weller, 1991). The presence

of

mater

nal maladjustment in a family with a chronic medical illness

may place a child at greater risk for the development of emo

tional difficulties.

The Parenting Stress Index (PSI) was found to have a strong

relationship to children's emotional adjustment. Within the

Child domain on the PSI, mothers reported their interactions

with their children to e reinforcing and felt acceptance of their

child. As measured by the Parent domain, the mothers of the

well-adjusted children described greater emotional closeness

and empathy in their relationships, despite the degree of med

ical severity. Speltz et a1 (1993) hypothesized that child adjust

ment in this population can be predicted from observations

of

parent-child interactions. These researchers discovered that

mother's child-directed orientation in a play task was predic

tive

of

lower

CBCL

scores at 4 -year follow-up.

Contrary to expectations, maternal social support was not

highly associated with child adjustment. Other researchers

(Speechley and Noh, 1992) have found the extent of social

support to be inversely related to psychological distress for

mothers. The population in this study appears to be different

in this regard. The mothers had small social networks which

were primarily comprised of family members. Benson et a1

(1991) and Speltz et a1 (1993) also found that parents of

children with craniofacial conditions reported decreased social

support systems. Despite this, mothers appeared to be con

tent with the extent and composition of their social network.


32/69

60 CleftPalate-CraniofacialJournal,January1995,Vol. 32No. 1

Suchinsularsocialnetworksmay be relatedtotheunique

socialstresseswithwhichthispopulationmustcontend.Par

ents

of

childrenwithcraniofacialdisfigurement!paybeless

emotionallyrelianton socialsupportbecausesoci,alrejection

of theirchildisgreaterintheelementaryschoolageyears.

Thismayalsoaccountformothers'tendencyto relyon rel

ativesforsocialsupport,instead of friends. Limitingsocial

contactoutsidethefamilymayserveaprotectivefunctionin

reducingthechildandparent'ssenseof stigmatization.How

ever,thedependenceon familymembersmay alsobestress

ful, in thattheserelationshipsarepotentially more conflict

riddenandlessamenableto change (Coyneand Delongis,

1986;Sarasonetal.,1990).Beforeconclusionscan bedrawn

aboutthequalitativedifferencesinthis

group's

socialsup

port system replicationof this finding with a similar age

groupisnecessary.

The

of

facial disfj2urement

had

0 0 relationship

.tochild

or

maternalpsychologicalfunctioning or tomater

nalperceptions.However,

having

a

comorbid severe med

.ical

condition

was

related to

greater behavior problems

in

_children.The impact of additional

illnesses may

multiply

thestressto children

and

their family

systems.

The rna'or limitation of thiss tud is that

the

evaluation

of child adjustment relied

on

parent report.There were

no

other

sources

of information

of child

behavior

to

corrob

orate the mother's view. In

addition,

emotional malad

justment

inmothersis related tonegative

maternal

reports

of

children's

behavior

(Brody

and

Forehand,

1986;Fried

lander etal.,1986).Despite thisfinding,these

researchers

found thatdepressed mothers werestillable to differenti

ate children with and without

emotional problems,

sup

portingthecriterionvalidityof theCBCL. However,causal

relationsbetween maternal variablesand child

adjustment

can notbederivedfromthepresentstudy

given

itsmethod

ological limitations.

Future

studies

should

use

multiple

informants ina longitudinal assessment of the children

withcraniofacial conditions.

Despitethepresenceof acraniofacialmalformation, chil

dren have thecapacity for healthy psychological adjust

ment in

the school age years.

This study supports the

. important association

between

the

mother-child

relation

ship and children's psychological

adaptation.

Given the

small nUITlber of minority families

and

the

predominance

of upper

SESfamiliesinthisstudy 2eneraJjzatioD

Of these

findingstoamore

heterogeneous population

must

be

done

with caution.

The

findingsare

strengthened

bythe

sample

sizeand theinclusionof alimitedagerange to control for

theeffectsof developmentaldifferences.Itisimportantthat

future studies assess whether the mother-child relation

shiphas adirectand moderating effect

on

the

psycholog

icalandsocialstress of facial disfigurement.

Acknowledgment

TheauthorswouldliketothankJohn

B.

Mulliken.

M.D.andthestaff

of

theCraniofacialCentre,Children'sHospitalfortheir

support

of

this project.Thecontributions

of

SarahBynumand Melissa

Brodieare appreciated.

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I

/793

l/oL 30 - S-

Psychological Functioning of h i l ~ r n with Craniofacial Anomalies and

Their Mothers: Follow-Up from Late Infancy to School Entry

MATTHEW

L

SPELTZ,

PH.D.

KATHI MORTON, PH.D.

ELIZABETH

W

GOODELL,

PH.D.

STERLING K. CLARREN,

M.D.

Twenty-three mothers and their 5- to 7-year-old children with craniofacial

anomalies

(CFA)

who were assessed during the child s infancy were followed.

Three types

of

CFA were Included:

cleft

lip and palate (CLP), isolated cleft

palate (CP). and sagittal synostosis. Measures

of

child status focused on

behaylor-problem frequency and self-concept. Moth9rs completed self-re

port

measures of emotional well-being, marital satisfaction, and social sup

port. Results Indicated that (1) a sizable

minority

(18%) of the children with

CFA had clin icall y significant behavior-problem scores shown in concordant

repgrts by parent and teacher of behavior problems; (2) individual differences

in child func'lioning within the

CFA

group were predicted by observational

measures of earlier mother-infant interaction during play and teaching situ

,ations;

(3)

mothers of children with

CLP

reported less fayorable social

support than mothers of children wltb Cp

gf

sagittal synostosis

KEY WORDS:

craniofacial anomalies, infancy and early childhood,

psychological functioning

In a previous study (Speltz et aI., 1990) the maternal predictor variables examined in our study were based on

interactions and parenting characteristics affecting infants observational measur s

of

moth r-inf nt

and toddlers with craniofacial anomalies (CFA) were in- ing interactions. Such measures have been infreQuently

vestigated. The present paper will describe a follow-up

of

used in previous CFA

p s y h o l o ~ i l

research. despite their

the same children at ages 5 to 7 years. The objectives

of

widespread application in other areas

of

pediatric and de-

the follow-up were to determine the psychological status y"e]opmental psycholoiY (e.g., Barnard et aI., 1989)..

IhiI:d.

of

the children and their mothers at a later age and to the time period encompassed by the follow-up (infancy to

identify measures from infancy associated with later vari- the

child's

entO' into irade schoo]) is one jn whjch re]a

ation among the children. tively little is known about the development

of

children

Although small sample size and

a

high rate

of

control- with CFA, despi te the intensity

of

the medical care and

. group attrition were factors limiting the methods

of

this family emotional factors operative

durini

this period

follow-up, the data were collected for several reasons. (e.g., parental adjustment to the anomaly, surgeries, feed

First ,prev ious longitudinal research on the psychological ing and speech difficulties, early peer encounters).

development

of

CFA children and adolescents is almost The original sample studied by Speltz et

a1

(1990) com

nonexistent. There has been only one previous study

of

a prised infants and toddlers (ages 12 to 36 months), with

longitudinal nature to our knowledge (Allen et aI., 1990)

three

types

of repaired

anomalies

including clef

Documents

What are some psychosocial implications of congenital craniofacial anomalies?