Are Indian Parents of Children with Down Syndrome engaged in the blame game

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ORIGINAL ARTICLE
Are Indian Parents of Children with Down Syndrome
Engaged in the Blame Game?
Mamta N. Muranjan & Sweta R. Budyal & Henal R. Shah
Received: 3 April 2012 /Accepted: 11 September 2012 /Published online: 19 October 2012
# Dr. K C Chaudhuri Foundation 2012
Abstract
Objectives To study blame ascription among parents of chil-
dren with Down syndrome and to study its correlation with
sociodemographic factors, parental perception of dysmor-
phisms and parents’ knowledge about Down syndrome. This
is a prospective, observational, non-interventional case con-
trol study.
Methods Interview of biological parents of children with
Down syndrome less than 12 y of age was taken. Dysmor-
phism and parents’ feeling of blame was assessed and graded
by Likert’s scale. Controls were parents of age and gender
matched children with non-genetic chronic disorders.
Results During the study period, 50 mothers and 46 fathers of
cases and 50 control parents were interviewed. Parents in the
study group were older; the mothers were better educated and
had more frequent antenatal visits. There was no significant
difference in the proportion of parents counseled but genetic
counseling was associated with a significantly higher propor-
tion of parents having knowledge about Down syndrome. A
higher proportion of parents perceived their child with Down
syndrome being dysmorphic. Blame ascription was not sig-
nificantly different among the two groups and was seen only
in a small proportion of parents of cases. When it did occur, it
was directed at health professionals.
Conclusions Blame ascription is not frequent in a cohort of
Indian parents of children with Down syndrome even when
dysmorphism is perceived by parents. Genetic counseling
was associated with better knowledge about Down syn-
drome in the parents.
Keywords Antenatal screening . Down syndrome .
Dysmorphism . Genetic counseling . Trisomy 21
Introduction
Disclosure of diagnosis of a chronic medical condition in a
child is a crisis for the parents. They pass through phases of
mourning, grieving and adjustment. Mourning arises out of
“grieving the loss of the perfect child” [1]. Blame ascription
can occur acutely in response to grief or out of anger from the
loss or it can become chronic if the grieving process is
obstructed, dysfunctional or complicated by depression [2].
Blame ascription can be expected in families with genetic
disorders which are chronic and associated with experience of
chronic sorrow. [3]
Birth of a child with Down syndrome (DS) is akin to an
accidental event due to lack of antenatal screening. The diag-
nosis is often delayed and benefits of genetic counseling are not
available to themajority. Moreover, the facial appearance easily
recognizable by lay persons and the invariable association with
mental retardation may stigmatize the child and the parents.
These factors may promote blame ascription in DS. The present
study was undertaken to document blame ascription among
parents of children with DS and to study the correlation of
blaming with sociodemographic factors, parental perception of
dysmorphisms and parents’ knowledge about DS.
M. N. Muranjan : S. R. Budyal
The Genetic Clinic, Department of Pediatrics,
Seth GS Medical College & KEM Hospital,
Parel, Mumbai 400012, India
H. R. Shah
Department of Psychiatry,
Seth GS Medical College & KEM Hospital,
Parel, Mumbai 400012, India
M. N. Muranjan (*)
Flat No. 301, Suman Apartments, 16 – B, Naushir Bharucha Road,
Tardeo, Mumbai 400007, India
e-mail: muranjanmamta@gmail.com
Indian J Pediatr (July 2013) 80(7):560–564
DOI 10.1007/s12098-012-0888-4
Material and Methods
A prospective case control study was conducted in a Genetic
Clinic of a tertiary care public hospital after approval of the
protocol by the Institutional Ethics Committee. Consenting
biological parents of children with DS (confirmed by cytoge-
netic study) and less than 12 y of age were enrolled. Couples
with multiple gestations, previous offspring or another family
member with DS and death of a child with DS were excluded.
The parents were interviewed independently. After reading
out the semi-structured proforma, the parents’ responses were
recorded in writing and audio-taped. Recorded data included
demographic information, prenatal screening, prior genetic
counseling and parents’ knowledge about DS assessed by
asking them to name the condition and describe features.
Perception of dysmorphism was assessed by asking if the
parent felt their child was different from other children. If
the answer was ‘Yes’, they were asked to describe the way
they felt the child was different. The degree of dysmorphism
was graded as mild, moderate and severe using ‘Likert’s five
point subject centered scale’.
To assess parents’ feeling of blame, they were asked if they
blame any person or factor for having a child with Down
syndrome. The degree of blaming was graded using ‘Likert’s
five point subject centered scale’ on the basis of frequency
with which such thoughts were experienced (‘Not at all’,
‘Rarely’, ‘Occasionally’, ‘Most of the times and Always’).
After the interview, parents inappropriately informed
about Down syndrome were counseled and those found to
have underlying psychological issues were referred to the
Psychiatry service.
Parents of children with non-genetic chronic disorders
were selected as controls matched for age and gender. For
the control group, instead of perception of dysmorphism,
‘perception of disease’ was assessed. Control parents were
also counseled about their child’s disease.
The data was analyzed to determine the percentage of
parents with blame ascription. The association between
blaming by parents with respect to the demographic and
non-demographic variables were analyzed. The data was
analyzed using the Chi-square test, Unpaired t test and
Fisher’s exact test for univariate analysis. The analysis was
carried out at 5 % significance (p<0.05).
Results
During the study period of 10mo, 57 consecutive children were
screened. The final study population consisted of 50 cases (after
excluding seven cases: twomore than 12 y old, two had expired
and one child each had an affected family member, was born of
multiple gestations and the parents declined to participate) and
50 age and gender matched controls. Only 46 fathers of cases
could be interviewed as one had expired, one was non-
ambulatory due to a physical disability and in two cases, the
parents were separated. Controls had congenital heart disease,
rheumatic heart disease, aortoarteritis with hypertension, hy-
drocephalus, epilepsy, stroke, nephrotic syndrome, chronic kid-
ney disease, asthma, recurrent lower respiratory infections,
chronic diarrhea, chronic liver disease, extrahepatic portal vein
obstruction and lymphoma.
Mean age of cases and controls was 35.2±36.2mo (range 2
to 144mo). Therewere 28 boys and 22 girls (M:F ratio 1.27:1).
Mothers and fathers of cases were significantly older [mean
31.5±8y(range20–48y)andmean35.7±7.2y(range25–50y)
respectively] as compared to controls [mean 27.9±4.9 y (range
21–40 y) and mean 31.7±4.7 y (range 24–45 y) respectively],
p00.0039 and 0.0014 respectively.Mean duration ofmarriage
in cases [9.5±6.6 y (range 1 to 30 y)] was not significantly
different from controls [7.9±5.4 y, (range 1.5 to 28 y)], p0
0.205. The mothers of cases were better educated (p00.0005)
but there was no difference in the educational status of fathers
(p00.084). The cases and controlswerematched for frequency
of outpatient visits and prior hospitalizations, however antena-
tal visits were significantly higher among cases (94 %) than
controls (78%), p00.021. None of the mothers had undergone
antenatal screening for Down syndrome.
Parental knowledge of DS, perception of dysmorphisms
and frequency of blaming are presented in Table 1. Knowl-
edge about Downsyndrome [naming the disease by mothers
and fathers (p00.0005 in each) and enumerating features
(p00.0055, 0.00005 respectively)] was significantly associ-
ated with prior counseling whereas only enumerating fea-
tures by mothers of controls (p00.02) was seen to be
significantly associated with counseling. The parent charac-
teristics and excerpts from the interview of parents of cases
who blamed are described in Table 2. Among the controls,
one mother and father experienced blame directed at self
and the doctor respectively.
Discussion
Several studies have documented feelings of guilt and self-
blame among parents with children having or testing posi-
tive for a genetic disease [4]. To date blaming in DS has not
been extensively studied.
One study reports 16 % of mothers of children having DS
blaming others [5]. Another study documented 20 % of
parents not offered antenatal screening for DS and 50 % of
those with false negative results blaming health professio-
nals or the health care system. In those with false negative
results, blaming was due to failure of expectation of an
unaffected child or refusal of confirmatory tests [6]. In those
not offered screening, blaming resulted from the expectation
that sonography would detect an affected child or from
Indian J Pediatr (July 2013) 80(7):560–564 561
denial of serum screening to all irrespective of maternal age
[6]. Such unmet expectations from health professionals or
the health care system elicit anger leading to blaming. High
levels of anger in response to crisis correlate with poor
physical health, emotional distress and Post–Traumatic
Stress Disorder (PTSD) symptoms [2]. Blaming can disrupt
parenting by causing high levels of parenting stress and
negative attitudes towards the child. Depression and anxiety
Table 1 Parental knowledge of
disease, perception of dysmor-
phisms and blaming
aN046
Figures in parentheses represent
percentages
Parameter Mothers Fathers
Cases Controls p value Casesa Controls p value
Received genetic counseling 22 (44) 25 (50) 0.689 25 (54.3) 21 (42) 0.973
Named condition 27 (54) 8 (16) 0.002 24 (52.1) 6 (12) 0.0009
Enumerated features 30 (60) 26 (52) 0.546 28 (60.8) 17 (34) 0.775
Perception of dysmorphisms 20 (40) 9 (18) 0.015 23 (50) 8 (16) 0.0003
Degree of dysmorphism
Mild 10 (50) 9 (100) 13 (56.5) 8 (100)
Moderate 4 (20) 6 (26.1)
Severe 6 (30) 4 (17.4)
Blaming 5 (10) 1 (2) 0.204 3 (6.5) 1 (2) 0.347
Table 2 Profiles of parents of children with Down syndrome experiencing blame
Case no Age in
years
Education Named
disease
No. of
features
enumerated
Counseling Dysmorphism
perceived
Blame Interview excerpts
3 (Mother) 42 8th standard, housewife Yes 3 Yes Mild Yes Private practitioner consulted
with expectation of healthy child,
Condition not detected on antenatal
sonogram, Liked challenged children,
child god’s gift, Child having DS
attributed to god, mental stress,
excessive vomiting in 3rd trimester
Always
Doctor
5 (Mother) 38 7th standard, housewife Yes 2 Yes No Yes Failure of tubal ligation, Affected
pregnancy not detected early, hence
could not be terminated.
Always Child looked better than siblings, Regret
for inability to provide therapy and
special education due to financial
constraints
Doctor
33 (Mother) 35 Bachelor of Arts, housewife Yes 1 Yes Mild Yes Condition not being diagnosed in
pregnancy by doctor, Occurrence
of Down syndrome attributed
to chance
Always
Doctor
40 (Mother) 23.5 9th standard, housewife Yes Yes No No Yes Baby with DS attributed to some
defect in selfAlways
Self
42 (Mother) 42 5th standard, housewife Yes 2 Yes Mild Yes Condition attributed to god, fate and
extra chromosomeAlways
Herself and
spouse
26 (Father) 39 10th standard, business No 1 No No, despite doctors
informing that
child looked
different
Yes Not diagnosed on antenatal sonogram
Always
Doctor
31 (Father) 40 Graduate, service No 1 Yes No Yes Frustration, junior doctor giving
improper treatment to wife during
delivery
Most of the
time
Doctor
43 (Father) 35 10th standard, service Yes 2 No No Yes Condition not diagnosed antenatally
Always
Doctor
562 Indian J Pediatr (July 2013) 80(7):560–564
was also found to be more frequent among parents who
blamed [6]. There are reports of parents being angry at the
birth of a child with DS and health professionals or the health
care system were blamed for not preventing the births [6].
Ninety-four percent of mothers with DS in the present
study were visiting antenatal clinics and yet had not under-
gone antenatal screening for DS. This reflects the lack of a
national policy for antenatal screening for DS in India or an
outcome of late antenatal registration. Customarily, >80 %
of pregnant women in India register for the first antenatal
visit after the first trimester and beyond the eligible period
for screening [7]. Moreover, many practicing obstetricians
may not have knowledge about availability, utility and in-
terpretation of these tests [8]. Therefore unlike their counter-
parts in UK, France and USA, parents in India are denied
their right to ensure an unaffected baby by exercising a
choice for termination of a pregnancy affected by DS [9,
10]. This is highlighted by the present study with parents
who blamed reporting that they would have opted for ter-
mination of the affected pregnancy.
In the present study, blaming observed in a very small
proportion of parents did not differ significantly from parents
of children with chronic diseases. Additionally, despite 40 %
of mothers and 50 % of fathers of cases perceiving their child
as being dysmorphic, it did not result in blame ascription.
These reactions to the child’s appearance were not severe.
This contrasts with earlier observations where parents have
expressed concern about children with DS being recognized
by the facial appearance as having mental retardation or being
pointed out in society [11]. Thus, it was not the genetic nature
of Down syndrome or the physical attributes that contributed
to blaming. However, it is pertinent that in six out of eight
parents who did blame, the physician was blamed for not
having detected the condition antenatally. On the other hand,
it is possible that ignorance about DS being preventable by
antenatal screening or testing or about the implications of the
condition and the complexities of caring for these children
may be the reason for lack of blaming in the majority. In this
context, earlier studies from India have documented that
parents may be ignorant about presence of DS in their child
or are not well informed about DS, it’s preventive strategies
and options to deal with recurrence in future pregnancies [11].
Another aspect to the absence of blaming could be that parents
of childrenwith DS experience less stress andmore rewards as
compared to children with other disabilities [12]. Two parents
in the present study felt gifted by the birth of a child with DS
believing that their daughter had brought them good fortune
and five parents felt that their child with DS looked prettier
than others. Such positive perceptions about the child could be
healthy coping mechanisms encouraging success at parenting.
Blaming could also be gender specific. Self blame was
reported more frequently by mothers in a study by Dam-
rosch and Perry [13]. Mothers in India and Turkey being
solely blamed for a child having DS reveals bias against the
female gender. [14, 15] However, it was heartening that
none of the mothers in the present study were blamed by
their spouse and the father was blamed in just one case.
In the present study, the authors speculate that counseling
received by the parents in both the groups could be the reasons
for lack of blaming. However, this may not be the only factor
as ≤50 % of mothers and fathers in both groups hadbeen
counseled. Also, a predisposing personality of the perpetrator
or victim could be a contributory factor accompanied by
physical, demographic or sociocultural characteristics [2, 16,
17]. Other factors such as older and better educated parents of
cases or constant contact with the heath care system in both
groups could have contributed. Nevertheless, it underscores
the need for referral of families with DS to a genetic counsel-
ing service. Genetic counseling facilitates obtaining factual
information, understanding the condition and addressing the
psychosocial needs of affected families. It provides an oppor-
tunity for exploring parents’ reactions and blame ascriptions
[18]. It also seems prudent to educate the general public for
first trimester antenatal registration and realistic expectations
from antenatal screening for DS if nationwide antenatal
screening for DS were to be successful in India.
Acknowledgments The authors thank Dr. Sanjay Oak, Director (ME
and HE) for granting permission to publish the paper.
Contributing MM conceptualized the study, approved the protocol,
analyzed the data and drafted the manuscript, SB recorded and ana-
lyzed the data and performed literature search and HS gave intellectual
inputs to the study design and protocol.
Conflict of Interest None.
Role of Funding Source None.
References
1. Sheeran T, Marvin RS, Pianta RC. Mothers’ resolution of their
child’s diagnosis and self-reported measures of parenting stress,
marital relations, and social support. J Pediatr Psychol.
1997;22:197–212.
2. O’Connor N, Kotze B, Wright M. Blame and accountability1:
understanding blame and blame pathologies. Australas Psychiatry.
2011;19:113–8.
3. Bennett RL. Genetic counseling. In: Runge MS, Patterson C, eds.
Principles of molecular medicine. 2nd ed. New Jersey: Humana
Press; 2006. pp. 46–52.
4. Arribas-Ayllon M, Sarangi S, Clarke A. Managing self-
responsibility through other oriented blame: family accounts of
genetic testing. Soc Sci Med. 2008;66:1521–32.
5. Hall S, Marteau T. Causal attribution and blame: associations with
mothers’ adjustment to the birth of a child with Down syndrome.
Psychol Health Med. 2003;8:415–23.
Indian J Pediatr (July 2013) 80(7):560–564 563
6. Hall S, Bobrow M, Marteau TM. Psychological consequences
for parents of false negative results on prenatal screening for
Down syndrome: retrospective interview study. B Med J.
2000;320:407–12.
7. Colah RB, Gorakshakar AC, Nadkarni AH. Invasive and non-
invasive approaches for prenatal diagnosis of hemoglobinopathies:
experiences from India. Indian J Med Res. 2011;134:552–60.
8. Hall S, Chitty L, Dormandy E, et al. Undergoing prenatal
screening for Down’s syndrome: presentation of choice and
information in Europe and Asia. Eur J Hum Genet.
2007;15:563–9.
9. Bittles AH, Bower C, Hussain R, Glasson EJ. The four ages of
Down syndrome. Eur J Pub Health. 2007;17:221–5.
10. Julian-Reynier C, Aurran Y, Dumaret A, et al. Attitudes towards
Down’s syndrome: follow up of a cohort of 280 cases. J Med
Genet. 1995;32:597–9.
11. Girisha KM, Sharda SV, Phadke SR. Issues in counseling for
Down syndrome. Indian Pediatr. 2007;44:131–3.
12. Hodapp RM, Ly TM, Fidler DJ, Ricci LA. Less stress, more
rewarding: parenting children with Down syndrome. Parenting.
2001;1:317–37.
13. Damrosch SP, Perry LA. Self-reported adjustment, chronic sorrow,
and coping of parents of children with Down syndrome. Nurs Res.
1989;38:25–30.
14. Lakshminarayana P, Ibrahim S, Venkataraman P, Jagatheesan T,
Kamala KG. KAP study on mothers of children with Down syn-
drome. Indian Pediatr. 1991;28:997–1001.
15. Durmaz A, Cankaya T, Durmaz B, et al. Interview with parents of
children with Down syndrome: their perceptions and feelings.
Indian J Pediatr. 2011;78:698–702.
16. Alicke MA. Culpable control and the psychology of blame. Psy-
chol Bull. 2000;126:556–74.
17. Ly TM. Asian American parents’ attributions of children with
Down syndrome: connections with child characteristics and cul-
ture. Intellect Dev Disabil. 2008;46:129–40.
18. Phadke SR. Genetic counseling. Indian J Pediatr. 2004;71:151–6.
564 Indian J Pediatr (July 2013) 80(7):560–564
	Are Indian Parents of Children with Down Syndrome Engaged in the Blame Game?
	Abstract
	Abstract
	Abstract
	Abstract
	Abstract
	Introduction
	Material and Methods
	Results
	Discussion
	References