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Parent Experiences and Preferences When Dysmelia Is Identified During the Prenatal and Perinatal Periods: A Qualitative Study Into Family Nursing Care for Rare Diseases

January 2018
Journal of Family Nursing 24(2)

Authors:

University of Leeds

Several rare diseases are regularly identified during the prenatal and perinatal periods, including dysmelia. How these are communicated to parents has a marked emotional impact, but minimal research has investigated this. The purpose of this study was to explore parent experiences and preferences when their baby was diagnosed with dysmelia.Twenty mothers and fathers were interviewed. Data was analyzed using thematic analysis. The overriding emotion parents experienced was shock, but the extent of this was influenced by several factors including their previous experience of disability. Four key needs of parents were identified, including the need for signposting to peer support organizations, for information, for sensitive communication, and for a plan regarding their child’s care. Parents wanted immediate information provision and signposting to peer support, and for discussions regarding possible causes of the dysmelia or termination (in the case of prenatal identification) to be delayed until they had processed the news.

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PUBLISHED IN JOURNAL OF FAMILY NURSING

http://journals.sagepub.com/doi/full/10.1177/1074840718772808

Parent Experiences and Preferences when Dysemlia is Identified During the Prenatal

and Perinatal Periods: A Qualitative Study into Family Nursing Care for Rare Diseases

Judith Johnson, PhD, ClinPsyD

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Olivia Johnson, BSc(Hons)

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

Jane Heyhoe, PhD

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Charlotte Fielder

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Alice Dunning, BSc(Hons)

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

Abstract



1"

&12"3

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""1$",12

3"&3"&

3&1!

",

41

"

&+

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Funding Acknowledgements/2"&( % 

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Declaration of Conflicting Interests/2

1

Introduction

"79666+#

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$

1$&

&"&&

&1

$"&

"1$

?0<76666&+@A9677B'@A

9667-&860;6C+0

#1966:BD1967?-1'"

&B&"&

&+'@19667-1

&&B&

+@19677-1 "&E

3

,E1

+.A'9666-1&



&+11

2-+ 966?-1

3"

&"

3&+%F A966<B

!.A)967=-1

&&+%'AG

9679B$A966=-B&"&

&+%,1966:B%19679BA@966<-B

3&"&

+%,1966:-1"

E3+%F1966<B

'966:B)HA966?-B+%19679B

*@A"&967;-B+!1

967=B'966:-B+'.A2,967?-1

2I3

&"

31

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"&&&

"""0

&+%") A%966J-123

+A9667-+%"1

966J-101

E30

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+@19677B0#1966:B

'@19667-1E

"&"1

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"&1

*"&"

&

,K4+D1967?B.A

'9666-1"04

3"

"

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""&B

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3&"&&

+)0'A967;-1



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&"

+D1967?-12""

+ ",A

966;-"

"12

&000"3

3"&&""&

1""&&4&

&1

Methods

2E33"&&

""&&

"&1

Participants and Recruitment

""";

12"&."

1""&

,""+11-

+2"!&-1"1

Research Design

%0""0

E"&E12"/

43&&&B3

"&&"&B

"&&

10""

""1

""&%$*DDD"

"""D !1 !3

3""012

""1

Data Collection

""&"(&967?'967=1

2""&"1%

""&&E

&"&1!

"E

&00"&

+A966:-1"9;=;

"0&1

Data Analysis

2",123&"

3

+A 966?-12"&

431

*"3& +966?-1!"

,""&"0"

"1"B"

",

12"&DD"&&*D

%$76C&D1$

"012"

1!"&"

+DD%*%$D-12"

31!"

+&9667-3"

3

+A 966?-1%"

"&",

312"3

31

Ethics

2"&# 

.+/7?069=<B/9;0760967?-1"

"

1@"

3

1(

&"

,1

Results

Participants

%;798

"1*""&"""&

;96"1

"3&

""&&1(

"77""8;18;+L:19=-1%

1".

##1("0

"B1(

"&"7;=+L9;166L7<1;7-"

&"1'&+L7=B<;C-"

"+L9B76C-+L7B;C-&1 

%+L8B7;C-

&+L8B7;C-+L9B76C-2%&+

L7B;C-12+L77B;;C-1#

+:6C-""79+?6C-

&1

Thematic Analysis

2+2&7-1

2&7



2 $ & #3E





234

"



31

2



+1

&-







 +7B'B

-

2



3



!"!#

$!!#

 +:B'B

-

#3



&

""

%&'($"

)!

$

*!

$ +'B78B

&-

2







""

""

 +'7:B&-

(











1

&)%%+

!%%

 +'B7B

-

(



'"



&

&&41

!,"

""%

 +'B:B-

(





&





E1

"!""

&)-%

"%

"-

!%"""+

!$

"!$%

" +'B78B&-

( 3



41

!#$#

""

"

#%""#%"

"#

$"

 +'B77B

&-

Influencing factors. '4&

"1"3

31

Timing of detection. 2"

B"&&

"&"&1"&&4&

"&"

"&"&

,1



./0112

"&&4&"&&

&&3&1!

&"

"&

&&12"&"

/

!"""!"

$!#%""3!#%"!$

""!"

./04151!2

1

"!#$

%"./06151!2

The influence of previous experiences on reactions. &"

3"1#3"

"&"&"3B

3&"&

1

!3"""

!"""

./7112



!"!#$!

!#./811'2



&"&

&"&/

""$"9

%%"#%"#%./0:1

5!2

"!"""!"

""!"%./811'

2

Expectations of the scan or birth. &"

&"3&

3&1"3

"/

""%&'($"

)!$

*!$.1/0;1!

2

The influence of other complications on reactions. &

&&4&"3

&""&&1

"3"

&&4&"/

""""

./081!2

Need for signposting to peer support. ("4&

"12&

"&4&&"

&1&

&&K4

/

%$<#.5/0:1

!2

"+!+"

"%"+

".1/01'2

1'"

,&

1&"&

"&4/

&)%%+!%%

.1/01'2

%&")

#%

.51/0=1!2

"&)*"

#$5%#$$

.51

/41'2

&&&"

,&

&"&,&K&41

'1"

&"",1

"""%&

*)!"%!#%""1

%

.51/0>1!2

',"1"

""&

&/

!!#$%"&*)?&)!#$

3!"$

.1/:1!2

Need for information. *"4&

"&1'

&

&/

!,"""%

.1/81'2

!""%&#)"

.1/001!2

""&

""/

!""&#)"

"%"#%

"./001!2

3

1&"E

"1

/

"#%"!

!#"#""

,.51/41'2

""+%

,%"

.1/0@1!2

3"

"E

1

,"&I/

$"

%.51/7@1!2

3&&4

&1"&

"/

%%""$%

$"$".1/71

'2

Need for clear and sensitive communication. "

""

31"3

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31""

&,/

<!%+

#<%"+$#$$

!"&)"#

""%.51/>1

'2

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4&/

"#""+%""

.1/0;1!2

'"&"""

"12

""

E&4&1

$#"#

%"<%

$#.1/=1'

2

Need for a plan. '&3

12

&&B"

B&1%&

,4""

&K4&41

!$#$"#$

"!#&)"!#

$%"!%"&*).1/=1

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&"+ -B3

 "&&&4&"" 

1" 

"&

&1

""&)"<"#B#

D!"<""###"<

!#!#$$$#!"%#%

%""B.1/=1

'2

3I0&"1

%""

0001%

&""& 

"E&41""

313

12""""

&""&&1"

""""3"

"&31

!#$#"

""#%""#

%""#$"

.1/001!2

3"

"1&

&K&4"B"

"&4

3312""

/

%!

"!%""!#$

".51/>1

'2

Discussion

'""&



3+D1967?-1

2E33"&&"

"&&

"&1

"+'.1967?-

3""

&&4&"1"3&

3"&

3&"""&"

&&1!

&

,

41

2E33

""

&3

3"&+D1

967?-1"&

B",

"1"

&"""3&1

!&"

&

1"&

B&&

E

&1

2"""&&

3"

1%&

"&"&

&1%"&

3"1'&

,&&",

"E&&1"

&&

"""1

&

3B"

"""

""E12"

"&1+966<-""

"K4K&41%&

&"

&"K4&

"K&4"1"

&&&""

&1

&

""+!1967=B1

966=-123&""

"""

1!"&",

K"4+19666-

"""

12"3&

&&

3K41"

"K"43&&

&1"

+ 1966;B!1967=-

E&E

E&&

12

"&

1"&

"

1

"1!""

""

"&E1

!,

"+D1

967?B.A'9666-3&

1!,"

&""

1

B"

"3"&12

"&""

"&41

"

"&&,1

Implications for Family Nursing Practice and Future Research

2"

12

3"&

3&

1"

1

3&&&"

&,

&"123""

&""&"

K"4123""",

K"4"+19666-

""3

1!""&

E&

+-

1



""&

"3""

4"&12"



,1

!&0

"+A9678-"

&

,31'.+967?-3

7;0'!"+A9678-0

1&

!(%+!(%-

+9678-"+967;-12

"

"1

!3"

1

%0

"+DA967<-&

3"

1&

1

Limitations

2"

"3+"-1

"""&0"&

,1!

1

Conclusions

'3"&&

"12

3"&&"

",&&

1 "&&

"1"3&

3"&3&

""1"

"&"&123

3"&/



&&41"

&

&&+

-&"

1'&



1

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%,!12'1@&D1@1$1DD1M

1+966:-1""/N&

"&1'EF

,7;+7-;?0?91/7617669O1J6?

%"#1#1D11) 1A%D11+966J-1#3

"&"/%

E1G/=8+9-97=1/761768=O667;?;7

%F1 1A@1+966<-1!&/N

3"3"1D$H

IAEJHDHH6+?-8:<08;:1

/76176J=O671%( 166668:9=?<1J:=8:198

%(11'1AG1@1+9679-1"43

&&

1FG$K;+9-=80=<1/

761767?OI1196771791669

1!11,1@&@1#1%1A.%11+9666-1

.#P30&"/%"

1E=+:-86908771/7617?8:O1;0:0869

&11+9667-1 E/%

"QLJ;77+=9J:-777;1

D1'1+967?-12/ 

&QR#S1J5H77+:-:8J0::J1/

76177==O76=:<:6=7??<6<8=

)1A )1+966?-11M$G

/;+9-==07671/76177J7O7:=<6<<=6?E6?8

D1",1A1+966;-1#3

&""1

J,I08+9-7;707?91/761766=O76<J=066;06:<<0J

1+966?-1$+&$O-D,

I+18790899-12"(D/1

#1+967:-1Q

/OO"""11OO"00

!1..1A)'1+967=-1"4

"1J5H7;+:-

;7?0;881/76177==O76=:<:6=7==8;:JJ

@(11'1(1A11+9677-1%

&1DJ,/D0==+?-

799;0798;1/7617669OI1188JJJ

0#'1$12$1*1)1!$1M

'01+966:-10&/%

76=1DJ,/D078+:-:7=0:991/

7617669OI11968;J

!(%+!(%-1+967;-1T!5HD/-

,I5H/T

T/OO1O"O"0OO967;O6=O

@ 0 0$!0000"00#01

!(%+!(%-1+967=-1T!5HD/-

D$/-I5HT

T/OO1O967=O6;O7JO00

O

DD1%0@1%21I$1D1A21+967?-1

0/

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9?:<1966<16:==<13

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;8?>1966?1667:<13

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8:J1/76177<?O79<<:067;06=J703

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&""1/

/AG/48+9-9;;09=91/

76178:<O666=779677?6<?=

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Parents’ Psychological Adaptation after Receiving a Fetal Diagnosis: A Systematic Review

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Catherine Rogers Gaspar

A fetal diagnosis places an unexpected psychological burden on parents and triggers a complex pregnancy experience. Parents who choose to continue the pregnancy have unique perspectives as they prepare for birth. It is crucial to understand these families’ experiences to inform their care and support. This qualitative systematic review explored the psychological adjustment of parents who continued gestation after they received a fetal diagnosis. A systematic database search was conducted with subsequent thematic analysis of fourteen included studies. Parents experienced intense initial emotional reactions to the diagnosis including shock and grief, followed by a complex processing period influenced by personal and social factors. The findings demonstrate a need for improved multidisciplinary parental support for families who receive a fetal diagnosis and add rationale for the addition of psychological services to the care teams of prospective parents. Keywords: prenatal, fetal diagnosis, pregnancy, parents, emotions, process

Swedish parents' experiences of their role in treatment for children with congenital limb reduction deficiency: Decision-making and treatment support

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Full-text available

Aug 2020

Background Parents of children with congenital limb reduction deficiency have an essential role in making treatment decisions during their child's first years of life. Treatment options usually concern surgical and/or prosthetic treatment. To tailor treatment options to fit different family values and priorities, the family‐centered approach indicates the importance of understanding the parental role in partnership with health care professionals. The aim of this study was to describe parents’ experiences of their role in decision‐making and treatment for children with congenital limb reduction deficiency. Methods A descriptive design with a qualitative approach was used. Semi‐structured interviews were conducted with 17 parents (12 mothers and 5 fathers) of children with upper and/or lower limb deficiency (mean age 5.9 years). The interview data were analyzed using qualitative content analysis with an inductive approach. Results Two major themes emerged from the data. The first theme, being a decision‐maker for someone else, was described as an ambivalent parental role, including collaboration within the family and with health care professionals. The second theme, becoming and being a treatment supporter in the child's everyday life, was made up of four categories: being a supporter of the child in everyday activities, mentoring the child to handle encounters with others, becoming a coordinator of information, and, being an “extended arm” of the health care provision for the child. Conclusions This study enhances our understanding of the parental role in decision‐making and treatment for children with congenital limb reduction deficiency. The results may contribute to the continued development of the family‐centered service approach by providing guidelines for treatment programs, with the goal of improving decision support and broadening the support for parents during treatment for these children.

UK consensus guidelines for the delivery of unexpected news in obstetric ultrasound: The ASCKS framework

Article

Full-text available

Aug 2020

Background Studies indicate there is a need to improve the delivery of unexpected news via obstetric ultrasound, but there have been few advances in this area. One factor preventing improvement has been a lack of consensus regarding the appropriate phrases and behaviours which sonographers and ultrasound practitioners should use in these situations. Aims To develop consensus guidelines for unexpected news delivery in Early Pregnancy Unit and Fetal Anomaly Screening Programme NHS settings. Methods A workshop was conducted to identify priorities and reach consensus on areas of contention. Contributors included interdisciplinary healthcare professionals, policy experts, representatives from third-sector organisations, lay experts and academic researchers ( n = 28). Written and verbal feedback was used to draft initial guidance which was then circulated amongst the wider writing group ( n = 39). Revisions were undertaken until consensus was reached. Results Consensus guidelines were developed outlining the behaviours and phrases which should be used during scans where unexpected findings are identified. Specific recommendations included that: honest and clear communication should be prioritised, even with uncertain findings; technical terms should be used, but these should be written down together with their lay interpretations; unless expectant parents use other terminology (e.g. ‘foetus’), the term ‘baby’ should be used as a default, even in early pregnancy; at the initial news disclosure, communication should focus on information provision. Expectant parents should not be asked to make decisions during the scan. Conclusions These recommendations can be used to develop and improve news delivery interventions in obstetric ultrasound settings. The full guidelines can be accessed online as supplemental material and at https://doi.org/10.5518/100/24 .

UK consensus guidelines for the delivery of unexpected news in obstetric ultrasound: the ASCKS framework

Article

Full-text available

May 2020

Background: studies indicatethere is a need to improve the delivery of unexpected news via obstetric ultrasound, but there have been few advances in this area. One factor preventing improvement has been a lack of consensus regarding the appropriate phrases and behaviours which sonographers and ultrasound practitioners should use in these situations. Aims: to develop consensus guidelines forunexpected news delivery inEarly Pregnancy Unit (EPU) and Fetal Anomaly Screening Programme (FASP) NHS settings. Methods: a workshop was conducted to identify priorities and reach consensus on areas of contention.Contributors included interdisciplinary healthcare professionals, policy experts, representatives from third-sector organisations, lay experts and academic researchers (n = 28). Written and verbal feedback was used to draft initial guidance which wasthen circulated amongstthe wider writing group (n = 39). Revisionswereundertaken untilconsensus was reached. Results: consensus guidelines were developed outliningthe behaviours and phrases which should be used during scans where unexpected findings areidentified. Specific recommendations included that: honest andclear communication should be prioritised, evenwith uncertain findings; technical terms should be used, but these should be written down together with their lay interpretations; unless expectant parents use other terminology (e.g., ‘fetus’), the term ‘baby’ should be used as a default, even in early pregnancy; atthe initial news disclosure, communication should focus oninformationprovision. Expectant parents should not be asked to make decisions during the scan. Conclusions: these recommendations can be used to develop and improve news delivery interventions inobstetric ultrasound settings.

Breaking bad and difficult news in obstetric ultrasound and sonographer burnout: Is training helpful?

Article

Full-text available

Dec 2018

Background: Sonographers report high levels of burnout. For those working in obstetric ultrasound, one frequently cited stressor is the delivery of bad or difficult news. Training in news delivery may reduce sonographer stress levels, but no studies have investigated sonographer experiences of this training. Aims: To investigate sonographer experiences of difficult news delivery training and preferences for training techniques, and to assess whether news delivery training is associated with lower burnout and higher wellbeing. Methods: A cross-sectional survey measured occupational characteristics, news delivery training experiences and preferences, burnout (on two dimensions of exhaustion and disengagement) and general mental wellbeing. Results: Ninety sonographers (85 female; mean age = 47) responded. The majority of participants thought training in difficult news delivery had improved their practice. Preferred training techniques were observation of clinical practice and receiving service-user input. Eighty per cent of participants were experiencing exhaustion, 43.3% were experiencing disengagement and 88.9% could be classed as having a minor psychiatric disorder. Having received difficult news delivery training was associated with lower levels of disengagement, even when other variables were controlled for. Discussion: News delivery training is perceived to be effective by sonographers and may help to reduce sonographer burnout levels.

R E V I E W Living through liminality? Situating the transitional experience of parents of children with mucopolysaccharidoses | Aoife Brinkley DClinPsych, MSc, BA, Senior Clinical Psychologist 2 | Professor of Clinical Nursing [Palliative Care] 1

Preprint

Full-text available

Aug 2021

Background: Mucopolysaccharidoses (MPS) are rare inherited metabolic disorders

Living through liminality? Situating the transitional experience of parents of children with mucopolysaccharidoses

Article

Full-text available

Aug 2021

Background Mucopolysaccharidoses (MPS) are rare inherited metabolic disorders that come under category three of life-limiting conditions. Children born with this condition show no symptoms at birth, but its effects show as a progressive disease in subsequent years. The severity of the condition varies according to the specific type, ranging from very mild symptoms to, in most cases, complex healthcare needs, including mental and physical disabilities. Aims This study aimed to elucidate the meanings of the transition experience of being a parent of a child with MPS. Van Gennep’s three-stage rite of passage theory and Turner’s theory of liminality were utilised to understand these families’ transition experiences are learning to live with their child’s ongoing progressive illness trajectory. Methods A qualitative design utilising hermeneutic phenomenology was used. Longitudinal qualitative in-depth interviews were carried out with eight parents at a three-time point over 17-months period. Results This study provided an interpretation of the lived experience of parents of children and young adults with MPS. As such, it embraces a liminal experience of living with a rare life-limiting illness and the unique passage to becoming a parent of a child with MPS. Parents reported their experience of transition from being the parent of a normal healthy child to be the parent of a child with MPS. They described their transition experience as multi-faceted and complex, neither linear nor time-bound, but rather cyclical. Conclusion The rites of passage conceptual framework helped to identify specific and significant unmet supportive and social care needs of these families and their children. This information will enhance the development of a substantial support system to meet the family’s emotional, psychological and social needs during the illness transitions from diagnosis and throughout their illness journey.

Managing the unmanageable: A qualitative study exploring sonographer experiences of and training in unexpected and difficult news delivery

Article

Full-text available

Oct 2020
Radiography

Introduction Ultrasound is used to diagnose pregnancy complications such as miscarriage and fetal health conditions. Within the UK, findings identified during ultrasound examination are delivered by sonographers as standard. However, little is known about the experiences of sonographers when delivering unexpected news (DUN), the impact this has on them, or their preferences for training on news delivery. Methods Qualitative interviews were completed with fourteen sonographers and were analysed using an inductive thematic approach. Key themes were identified. Results Participants said that obstetric ultrasound often involves ‘managing’ the patient encounter, including: navigating (unrealistic) patient expectations; handling their own responses to unexpected findings; and managing interaction by moderating emotional expression and communication practices to deliver patient-centred and empathic care. Persistent uncertainty of outcomes, prognosis and patient reactions, alongside high workloads, and frequent siloed working, makes DUN challenging for sonographers. DUN was experienced as emotionally burdensome, and sonographers employed personal coping strategies to reduce stress/burnout. However, the greatest mitigation for stress/burnout was support from peers, though accessing this was challenging. Peers were also described as key sources of learning, especially through observation. Conclusion Challenges associated with DUN are an enduring experience for sonographers. Facilitating regular ongoing support and training would enable sonographers to cope with negative aspect of the role, including the emotional burden of DUN. Implications for practice Long patient lists are prioritised to deal with high demand for services. However, sonographer wellbeing needs to be a key priority to avoid stress and burnout. This means facilitating protected time to access support from colleagues, multidisciplinary working where possible, and regular access to training to support DUN. Training focusing on communication practices, alongside dealing with emotional burdens of the role would be beneficial.

Waiting for a diagnosis in Rubinstein-Taybi: The journey from "ignorance is bliss" to the value of "a label"

Article

Jan 2021

The journey to receiving a diagnosis for rare genetic disease can be long and emotionally impactful. This study describes parental experiences of receiving their child's diagnosis of Rubinstein-Taybi syndrome (RTS), a rare genetic condition characterized by growth and developmental delay together with dysmorphic features. Parents from the RTS Australia support group participated in qualitative, semi-structured phone interviews, which were transcribed verbatim and thematically analyzed. Questions focused on psychosocial challenges and benefits pre and post-diagnosis. Ten mothers and three fathers participated, with the mean age of diagnosis being 8 months. Parents reported positive psychological effects from a slight delay in diagnosis, and negative effects from an extended diagnostic delay, suggesting the ideal time for a parent to receive a diagnosis lies in the post attachment stage, prior to the development of significant parental concerns. This stage would vary depending on condition severity. Parents desired a diagnosis to reduce uncertainty; however, uncertainty remained post diagnosis, and shifted its focus from broadly encompassing etiology and prognosis, to specifically focusing on concerns regarding severity within the spectrum. Perceived benefits of a diagnosis mainly centered on the provision of a label. Parents articulated that a label increased social acceptance, enhanced coping, promoted communication, and improved access to medical, financial, and support services. This study provides insights into the experience of families prior to and following receipt of a diagnosis. It also highlights the possibility of an optimal time window to receive a diagnosis; in which bonding is maximized and parental distress is minimized.

Learning how to deliver bad and challenging news: Exploring the experience of trainee sonographers – A qualitative study

Article

Full-text available

Sep 2019

Background Previous studies suggest there is a need to improve the delivery of bad and challenging news in obstetric ultrasound settings. However, no research has explored the experiences of trainee sonographers when learning how to deliver challenging news. Understanding this could identify gaps in current provision and inform future training interventions. Aims To explore the experiences of trainee sonographers when learning how to deliver challenging news. Methods Semi-structured interviews were conducted with trainee sonographers ( n = 7) from four training centres to explore their experiences and preferences for news delivery training. Results Learning how to deliver difficult news was a journey where trainees developed their confidence over time. Most learning occurred in clinical settings, but classroom teaching complemented this. Trainees appreciated the opportunity to observe clinical practice and to hear from patient representatives. However, quality of teaching varied between centres and trainees reported uncertainty regarding the specific language and behaviours they should use. They described building their own personal protocol for news delivery through the course of their training. Discussion An ultrasound-specific news delivery protocol which details the words and behaviours sonographers can employ could help reduce uncertainty in trainees. Trainees may also benefit from receiving structured feedback on their news delivery performance.

Interventions to Improve the Breaking of Bad or Difficult News by Physicians, Medical Students, and Interns/Residents: A Systematic Review and Meta-Analysis

Article

Full-text available

Jun 2018
ACAD MED

Purpose: To assess the effectiveness of news delivery interventions to improve observer-rated skills, physician confidence, and patient-reported depression/anxiety. Method: MEDLINE, EMBASE, CINAHL, PsycINFO, and Cochrane Register of Controlled Trials databases were searched from inception to September 5, 2016 (updated February 2017). Eligible studies included randomized controlled trials (RCTs), non-RCTs, and controlled before-after studies of interventions to improve the communication of bad or difficult news by physicians, medical students, and residents/interns. The EPOC risk of bias tool was used to conduct a risk of bias assessment. Main and secondary meta-analyses examined the effectiveness of the identified interventions for improving observer-rated news delivery skills and for improving physician confidence in delivering news and patient-reported depression/anxiety, respectively. Results: Seventeen studies were included in the systematic review and meta-analysis, including 19 independent comparisons on 1,322 participants and 9 independent comparisons on 985 participants for the main and secondary (physician confidence) analyses (mean [SD] age = 35 [7] years; 46% male), respectively. Interventions were associated with large, significant improvements in observer-rated news delivery skills (19 comparisons: standardized mean difference [SMD] = 0.74, 95% CI = 0.47-1.01) and moderate, significant improvements in physician confidence (9 comparisons: SMD = 0.60, 95% CI = 0.26-0.95). One study reported intervention effects on patient-reported depression/anxiety. The risk of bias findings did not influence the significance of the results. Conclusions: Interventions are effective for improving news delivery and physician confidence. Further research is needed to test the impact of interventions on patient outcomes and determine optimal components and length.

How the Child’s Gender Matters for Families Having a Child With Congenital Adrenal Hyperplasia

Article

Full-text available

Oct 2017
J FAM NURS

Children with congenital adrenal hyperplasia (CAH) are exposed to high levels of testosterone in utero often resulting in nontypical genitalia at birth for girls. The purpose of this analysis, which draws on data from a larger study, was to examine, based on the gender of the child, the family experiences of having a child with CAH. Sixteen parents were interviewed, and comparisons were made across all categories coded in the parents’ interviews to examine similarities and differences in the experiences of families based on the child’s gender. Families having a daughter with CAH experienced additional challenges when compared with families having a son. These include fear of stigmatization, challenging surgical decisions, and concerns regarding disclosure of the condition. Acknowledging this and creating support programs tailored for girls that address surgical procedures and complications resulting from being born with elevated testosterone are critical to promoting a healthy family and child response.

Providing Family-centred Care for Rare Diseases in Maternity Services: Parent Satisfaction and Preferences when Dysmelia is Identified

Article

Full-text available

May 2016

Problem and background: Dysmelia is usually detected prenatally or postnatally in maternity services. The provision of family-centred care for parents at the time of initial diagnosis is crucial to facilitate decision making, access to appropriate services, and the provision of parental care-giving, but no research has investigated parent experiences or preferences in this population. Aims: The current research aimed to address this by investigating satisfaction with service, occurrence of signposting and preferences in this group. Methods: Two online surveys were conducted. In the first survey (n=417), parents reported whether they were offered signposting information and their level of satisfaction with the service they received when initially diagnosed. In the second survey (n=130), a subgroup of participants who completed the first survey reported their preferences for signposting and health service access after diagnosis. Findings: On average, participants were less than satisfied with the service they received and only 27% were offered signposting information. Satisfaction was higher amongst parents who had been offered signposting information. 91% of parents said they would have wanted signposting information and 67% would have wanted access to a support group. Conclusions: There is a need to improve the family-centeredness of care when dysmelia is identified. Offering signposting information to relevant third-sector organisations may increase parent satisfaction and address parent preferences. These findings could have implications for parents of children with other rare diseases identified in maternity services.

Walking the "Emotional Tightrope" From Pregnancy to Parenthood: Understanding Parental Motivation to Manage Health Care and Distress After a Fetal Diagnosis of Complex Congenital Heart Disease

Article

Full-text available

Dec 2015
J FAM NURS

Advances in medical technology account for increasingly more couples receiving fetal diagnoses of complex congenital heart disease. Theory on internal working models of caregiving during parenting transitions informed this prospective, exploratory study. Data included conjoint interviews and measures of anxiety, trauma, and depression collected from six couples after diagnosis and after birth. Severity of illness was described using infant health records. Directed content analysis furthered understanding of the caregiving motivation to manage health care that included three categories of parental efforts: (a) to determine expectations of health care providers, (b) to reconcile illness- and non-illness-related care, and (c) to express agency as a parent. Synthesis of qualitative findings transformed into categorical ratings with parents' levels of distress resulted in two profiles characterizing types of internal working models. Findings extend theory on internal working models of caregiving and offer direction for future research regarding parental management of health care for their chronically ill offspring. Implications for practice with families are offered.

When fetal hydronephrosis is suspected antenatally—a qualitative study

Article

Full-text available

Dec 2015

Abstract Background: The information about fetal malformation findings during the ultrasound examination often comes unexpectedly, and the women and their partners may not necessarily receive any conclusive statement on the prognosis. A finding such as fetal hydronephrosis range from being a soft markers or mild anomaly, to a serious condition associated with neonatal morbidity and mortality. The aim of this study was to explore women’s reactions to the discovery of fetal hydronephrosis in the context of uncertainty regarding the prognosis. Methods: Ten women were interviewed and the interviews were conducted six to twelve months after the women gave birth. They had experience of suspected fetal hydronephrosis in gestational week 18–20. The interviews were recorded, transcribed verbatim and analysed using constant comparative analysis. Results: The core category, ‘Going through crisis by knowing that you are doing the right thing’ illustrates the meaning of women’s reactions and feelings. It illuminates the four categories: ‘When the unexpected happens’– on the one hand, women had positive views that the suspicious malformation could be discovered; however, on the other hand, women questioned the screening. ‘To live in suspense during pregnancy’ – the suspicious malformation caused anxiety and was a stressful situation. ‘Difficulties in understanding information’ – the women thought they had limited knowledge and had difficulties in understanding the information. ‘Suppress feelings and hope for the best’ – the women tried to postpone the problem and thought they should deal with it after delivery. Conclusions: Women are worried irrespective of suspicious or severe malformations, and in need of information and counselling tailored to their individual needs. Other sources of support could be: written information, links to reliable sources on the Internet and possibilities for ongoing follow-ups.

Using thematic analysis in psychology

Article

Full-text available

Jan 2006
Qual Res Psychol

Thematic analysis is a poorly demarcated, rarely acknowledged, yet widely used qualitative analytic method within psychology. In this paper, we argue that it offers an accessible and theoretically flexible approach to analysing qualitative data. We outline what thematic analysis is, locating it in relation to other qualitative analytic methods that search for themes or patterns, and in relation to different epistemological and ontological positions. We then provide clear guidelines to those wanting to start thematic analysis, or conduct it in a more deliberate and rigorous way, and consider potential pitfalls in conducting thematic analysis. Finally, we outline the disadvantages and advantages of thematic analysis. We conclude by advocating thematic analysis as a useful and flexible method for qualitative research in and beyond psychology.

The Central Importance of Therapeutic Conversations in Family Nursing: Can Talking Be Healing?

Article

Nov 2016
J FAM NURS

Janice M. Bell

Atlas of Genetic Diagnosis and Counseling

Article

Jan 2006

Harold Chen

Many birth defects, although rare individually, are encountered in clinical practice and have now become treatable if properly diagnosed. In the Atlas of Genetic Diagnosis and Counseling, Harold Chen, MD, shares his almost 40 years of clinical genetics practice in a comprehensive pictorial atlas of 203 genetic disorders, malformations, and malformation syndromes. The author provides a detailed outline for each disorder, describing its genetics, basic defects, clinical features, diagnostic tests, and counseling issues, including recurrence risk, prenatal diagnosis, and management. Numerous color photographs of prenatal ultrasounds, imagings, cytogenetics, and postmortem findings illustrate the clinical features of patients at different ages, patients with varying degrees of severity, and the optimal diagnostic strategies. The disorders cited are supplemented by case histories and diagnostic confirmation by cytogenetics, biochemical, and molecular techniques, when available. Also available in a CD-ROM edition (ISBN: 1-58829-974-5). Authoritative and up-to-date, the Atlas of Genetic Diagnosis and Counseling will help all physicians to understand and recognize genetic diseases and malformation syndromes, and consequently better evaluate, counsel, and manage affected patients.

SPIKES--A Six-Step Protocol for Delivering Bad News: Application to the Patient with Cancer

Article

Aug 2000

Walter Baile

We describe a protocol for disclosing unfavorable information—“breaking bad news”—to cancer patients about their illness. Straightforward and practical, the protocol meets the requirements defined by published research on this topic. The protocol (SPIKES) consists of six steps. The goal is to enable the clinician to fulfill the four most important objectives of the interview disclosing bad news: gathering information from the patient, transmitting the medical information, providing support to the patient, and eliciting the patient’s collaboration in developing a strategy or treatment plan for the future. Oncologists, oncology trainees, and medical students who have been taught the protocol have reported increased confidence in their ability to disclose unfavorable medical information to patients. Directions for continuing assessment of the protocol are suggested. The Oncologist 2000;5:302-311

Comparing Telephone and Face-to-Face Qualitative Interviewing: A Research Note

Article

Apr 2004

This research note reports the results of a comparison of face-to-face interviewing with telephone interviewing in a qualitative study. The study was designed to learn visitors’ and correctional officers’ perceptions of visiting county jail inmates. The original study design called for all face-to-face interviews, but the contingencies of fieldwork required an adaptation and half of the interviews were conducted by phone. Prior literature suggested that the interview modes might yield different results. However, comparison of the interview transcripts revealed no significant differences in the interviews. With some qualifications, we conclude that telephone interviews can be used productively in qualitative research.

Parent Experiences and Preferences When Dysmelia Is Identified During the Prenatal and Perinatal Periods: A Qualitative Study Into Family Nursing Care for Rare Diseases

Abstract

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