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LA COMUNICAZIONE DELLA DIAGNOSI

Dott.ssa Anna Zambon Hobart
Psicoterapeuta

Roma, 9 Marzo 2004

Sono una psicologa che appartiene alla Società Italiana di Psicoterapia Psicoanalitica e che dal ’79 al ‘97 ha fatto parte del Servizio di Informazione e Consulenza presso l'Associazione Italiana Persone Down di Roma.

Il mio lavoro in AIPD riguardava prevalentemente il supporto ai genitori alla nascita del neonato con SD sia con colloqui individuali che con la formazione di piccoli gruppi di genitori (non più di quattro o cinque coppie) che incontravo sistematicamente per un paio di anni.

I miei colloqui iniziavano sempre con la domanda "Come avete ricevuto la notizia della sindrome?" e questo non solo per aver dei dati che ho raccolto dall'86 all' ottobre 89 su 130 coppie di genitori di neonati, ma per rompere il ghiaccio e venire subito al centro dell'argomento figlio/a e SD.

Sono certa che attualmente le informazioni che hanno i medici permettano delle modalità più adeguate di quando nell'86 su 58 famiglie la notizia veniva data al padre da solo in 41 casi e il tempo in cui il padre riusciva a darla alla moglie variava da un'ora a 18 mesi. Poveri padri che si assumeva essere i più forti. Curiosamente in UK e in USA la notizia da sempre ed ancora oggi viene data prevalentemente alla madre quando in casi, fortunatamente sempre più rari, viene data solo ad un genitore a cui si delega la difficile incombenza di darla all'altro. Nell'89 su 80 famiglie la notizia viene data solamente in due casi in modo ottimale e dunque ai due genitori insieme. Un medico ricordato con gratitudine è quello che concede ai genitori   tempo e  spazio privato, uno spazio fisico e mentale che permetta di esprimere emozioni e fare domande. Le informazioni più apprezzate sono quelle legate ad un’immagine più simile che diversa tra un bambino\persona con SD da un bambino\persona senza SD. Naturalmente sempre nell'ambito di dati rigorosamente corretti e non di menzogne pietose.

Mi rendo conto perfettamente che per un medico il compito sia prevalentemente quello di avere informazione mediche.

Ma il medico e soprattutto il pediatra assai frequentemente riceve dai genitori, con e senza SD in famiglia, un ruolo che va ben oltre alle diagnosi mediche.

Per i genitori a parte l'impatto traumatico con la notizia vera e propria, le  parole del medico, le informazioni che darà, ma soprattutto il suo atteggiamento verso quel bambino o bambina, rimarranno scolpiti per sempre nella mente dei genitori. J. Carr, una studiosa inglese, ha intervistato un vasto gruppo di genitori di neonati alla nascita e dopo 20 anni, sul momento della diagnosi. Gli intervistati hanno descritto le parole del medico e le proprie emozioni usando le stesse parole usate 20 anni prima.

I genitori continueranno in qualche modo per tutta la vita a dare la diagnosi di SD e più che altro a descrivere agli altri il loro figlio o figlia. Ai fratelli prima di tutto, ai parenti, agli amici, a scuola e nella vita adulta..

Un medico che riesce a dare la notizia in modo propositivo (e ci riuscirà soprattutto se ne è convinto) e parlerà del problema Down come un problema affrontabile per quanto difficile e drammatico inizialmente, aiuterà i genitori ad affrontare  assai meglio una nascita così diversa da quella attesa. Potrà suggerire con le sue parole un modo adeguato di informare gli altri, soprattutto nei primi momenti che sono quelli più difficili.

Nei primi momenti  i genitori possono non riuscire a parlare tra di loro e questo avviene assai più spesso di quanto non si possa immaginare.

I pediatri o i genetisti che abbandonano la famiglia con una diagnosi lapidaria o quelli che sgridano le mamme per aver voluto un figlio in età avanzata e così via, sono forse quelli che più di altri hanno un immagine devastante di quello che può significare la presenza di una persona con SD in famiglia.

Io non sto affatto dicendo che un pediatra o un genetista debbano diventare psicologi o psicoanalisti. Assolutamente no.

Spero di essere in qualche modo utile a chi affronta il problema della comunicazione della diagnosi, sintetizzando al massimo i risultati delle ricerche longitudinali su famiglie di persone con SD dalle quali risulta che la presenza di una persona con SD è un problema affrontabile e non è dramma senza speranza tale da creare fughe o deleghe o sgridate da parte di alcuni medici particolarmente pessimisti.

Le ricerche longitudinali sulla famiglia dagli anni ‘70 ad oggi possono dividersi in due gruppi.

Un primo gruppo sempre meno numeroso ma ancora presente riguarda una raccolta di dati sulla salute fisica, mentale e sociale delle famiglie, incredibilmente senza gruppo di controllo. Come se una famiglia con una persona con SD non possa essere paragonabile per sofferenza a nessun'altra famiglia, presumendo dunque che una famiglia senza SD sia comunque più funzionale.

Sui fratelli delle persone con SD sono state fatte ricerche con questo metodo anche in Italia, attribuendo quindi qualsiasi problema sia a livello individuale che della famiglia in genere, alla presenza di una persona con SD.

Numerosi studiosi che hanno iniziato la ricerca senza gruppi di controllo, da A. Gath agli studi più recenti, hanno eseguito successivamente nuove ricerche con gruppi di controllo formati da famiglie in cui era nato un bambino/a senza SD ma analoghi per vari parametri. La ricerca attuale si basa ancora sulle ricerche storiche di Gath, Carr, Cunningham.

I risultati nelle ricerche più attuali sulla famiglia confermano ancora oggi quelli precedenti e cioè che i due gruppi con SD e senza SD per pari condizioni sociali, culturali (salute fisica e psichica dei genitori e dei fratelli, rapporti di coppia, dunque separazioni o meno, rapporti sociali fuori del gruppo familiare e così via) sono sovrapponibili.

Se ci sono alcune differenze sono spesso in positivo. Nelle famiglie in cui è presente una persona con SD, può esserci una migliore consapevolezza sociale e psicologica ad esempio. Ci sono meno separazioni contro uno dei tanti stereotipi che le persone si divorziano quando nasce un bambino con SD. Un evento traumatico viene affrontato dalla famiglia in base alla qualità del rapporto preesistente. Dunque un bambino con SD può contribuire a saldare un rapporto buono o venire strumentalizzato dai genitori per giustificare una rottura già in atto. Maggiore capacità riflessiva dunque, maggiore sensibilità, più frequente impegno sociale.

Nei miei piccoli gruppi di genitori assai frequentemente i genitori dicevano "Se avessi saputo questo o quello anche per gli altri figli!" Si tratta di dati assolutamente ovvi per chiunque abbia una lunga esperienza clinica sia medica che psicologica con le famiglie, assai meno ovvi per chi si avvicina inizialmente o comunque raramente al problema Down.

Per questo  penso che possa essere  utile per qualsiasi operatore in campo medico o psicologico o sociale avere delle informazioni sulla capacità che ha la famiglia di affrontare una esperienza inizialmente molto difficile, che in seguito può rivelarsi affrontabile al punto da rappresentare a qualche livello un arricchimento per la famiglia.

Spero che con informazioni realistiche sul destino della famiglia con una persona con SD sia possibile comunicare più facilmente con i genitori grazie a un’immagine  assai meno drammatica di quella che sarà la loro vita.

Può essere utile rivolgersi per riferimenti bibliografici alla NDSS (National Down Syndrome Society) che contribuisce più di altri gruppi alla ricerca in campo medico e psicologico.
Allego un buon articolo che riassume dati e ricerche e un’esauriente bibliografia sulla famiglia.

Anna Zambon Hobart

Questo lavoro è stato inviato come contributo alla preparazione delle
"Linee Guida Multidisciplinari per l'Assistenza Integrata
alle Persone con Sindrome Down e alle Loro Famiglie" (LGSD)

in collaborazione con L'Istituto Superiore di Sanità

Bibliografia di riferimento:

 

http://www.denison.edu/collaborations/dsq/vanriper.html

[Reprinted from Down Syndrome Quarterly, Volume 4, Number 1, March, 1999]

Living with Down Syndrome: The Family Experience

Marcia Van Riper1

Department of Community, Parent-Child, and Psychiatric Nursing

The Ohio State University

Our current understanding of well-being in families of children with Down syndrome is rather limited. What we do know is that these family experience increased stress, increased time demands, and changes in roles. It remains unclear why some families recover or adapt in the face of stressful circumstances, while others remain vulnerable, and some deteriorate. Future research needs to shift the focus from assessing stress and distress, to assessing resilience and adaptation. We have growing evidence that many families of children with Down syndrome are doing very well. What we need now is an understanding of the factors that contribute to resilience and successful adaptation.


It is estimated that over 350,000 families in the United States are affected by Down syndrome (National Down Syndrome Society, 1999). There is no "typical" family of a child with Down syndrome. Families of children with Down syndrome are from all races, religions, ethnic backgrounds and socioeconomic status.

So, what do we know about these families? How are they doing in terms of individual, dyadic, and family functioning? Are they suffering from psychological impairment as was predicted by most researchers and health care providers prior to the late 1970's? Or, are they responding to the challenges associated with raising a child with Down syndrome with resilience and adaptive functioning?

The purpose of this paper is twofold: (a) to present the key findings from this investigator's program of research concerning well-being in families that include a child with Down syndrome and (b) to compare these findings with the published findings from other investigators. It is hoped that this overview of research findings concerning well-being in families of Down syndrome will be help to decrease some of uncertainty experienced by families and professionals following awareness of an infant's diagnosis of Down syndrome. In addition, it is hoped that an awareness of what we know about well-being in families of children with Down syndrome, as well as an awareness of the gaps that exist in our current understanding, will stimulate further research in this area.

To date, this investigator has conducted six studies concerning the experience of living in a family that includes a child with Down syndrome. The direction and breadth of this research has been significantly influenced by interactions with, and feedback from, individuals with Down syndrome and their families. Key findings are presented below.

Study 1

The first study was a qualitative study concerning parental responses to the birth of a child with Down syndrome (Van Riper, 1987, Van Riper & Selder, 1989). The purpose of this study was to describe the responses of parents following the birth of an infant with Down syndrome. Sixteen parents of young children with Down syndrome were interviewed in order to explore and describe the experience of parenting a child with Down syndrome from their perspective. Uncertainty was found to be a major characteristic of the life transition experienced by parents following the birth of a child with Down syndrome. Initial sources of uncertainty included (a) the unexpected diagnosis of Down syndrome, (b) parental concern about the future, (c) apprehensions regarding parenting abilities, and (d) the initial responses of professionals. Ongoing sources of uncertainty included (a) the child's involvement in an educational program, (b) parental concern about the future, and (c) the child's health status. All parents reported that the experience of raising a child with Down syndrome had a profound impact on their life. More importantly, they reported that the positive consequences associated with this experience far outweighed the negative ones. Positive consequences included: bringing the family closer together, learning the true meaning of unconditional love, putting things in proper perspective, and appreciating diversity. According to one parent,

It is really not quite the tragedy...At the time you feel that this is the biggest tragedy that ever happened. If we could have known what it would be like to have M., we wouldn't have been nearly so sad. No one really mentioned the positive side. (Van Riper & Selder, 1989, p.66).

Study 2

The purpose of this study was to examine the effects of a child with Down syndrome on the individual functioning of both parents, marital functioning, and family functioning (Van Riper, Ryff, & Pridham, 1992). Thirty-four families of children with Down syndrome between three to ten years of age were compared to 41 families with nondisabled children between three to ten years of age. Mothers and fathers in both groups completed a series of valid and reliable measures designed to assess parental depression, six dimensions of parental psychological well-being, dyadic functioning, and family functioning. Based on prior research, it was predicted that families of children with Down syndrome would differ from families of nondisabled children on specific indices of individual, dyadic, and family functioning. The findings do not support these predictions, but rather indicate that families of children with Down syndrome are more comparable to than different from families with nondisabled children. There were no significant differences between the two groups of families on any of the measures of individual, marital, or family functioning. The results of this study provide support for a competence model in which families may respond to the challenges associated with raising a child with Down syndrome with resilience and adaptive functioning. Given the extensive prior literature on the negative effects suffered by families of children with disabilities (e.g., Birnbaum, 1970; Bristol, Gallagher, & Schopler, 1988; Childs, 1985; Cummings, 1976; Cummings, Bayley, & Rie, 1966; Damrosch & Perry, 1989; Friedrich & Friedrich, 1981; Friedrich, Wilturner, & Cohen, 1985; Gath & Gumley, 1984; Goldberg, Marcovitch, MacGregor, & Lojkasek, 1986; McAndrews, 1976; Olshansky, 1962; Wilker, Wasow, & Hatfield, 1981) such findings are significant.

Study 3

This study was a descriptive study (Van Riper, Pridham, & Ryff, 1992) that combined qualitative and quantitative methods to examine the impact of others on well-being in 90 families that include a child with Down syndrome. The guiding framework for this study was symbolic interactionism. Findings from this study provide support for prior research indicating that interactions with health care providers can have a critical impact on how families respond following the birth of a child with Down syndrome (e.g., Cunningham, Morgan, & McGucken, 1984; Murphy & Pueschel, 1975; Pueschel, 1985). One mother noted that the physician helped to set the tone for how she felt about her daughter and the experience of parenting a child with Down syndrome.

The pediatrician entered the room and sat down with us, held our hands, and explained the best that he could what having such a child meant. He reassured us that this was a time for celebration, that she would make us happier than we could ever imagine and that the only thing that made her different was that one (lousy) chromosome. She would be able to do anything she wants (read, write, talk, walk, etc), it might just take her a little longer than most. But give her time, you'll see... After his talk with us, I didn't cry anymore. I no longer felt sorry for myself. (Van Riper, Pridham, & Ryff, 1992, p. 29).

In contrast, another mother recalled that the initial responses of health care providers "made time stand still." According to this mother,

They generally ignored me and avoided talking about my daughter. They didn't realize what I wanted and needed was someone to say, "Now she's here, it's time to go on." Instead, they made time stand still. I couldn't move ahead. (Van Riper, Pridham, & Ryff, 1992, p. 35).

Despite notable changes in the care of children with Down syndrome, many parents in this study indicated that they were dissatisfied with how they were informed of their child's diagnosis. Less than one-half of the parents reported that the initial information they received was accurate and encouraging. One parent noted,

The only choices the doctor gave me were (1) I would take him home and love him. (2) Put him in an institution. (3) Order them to cut off his food supply... What no one told me and I desperately needed to hear, was that these children generate more love than is imaginable. I was scared to death of my baby... For the first few months, I felt suicidal. (Van Riper, Pridham, & Ryff, 1992, p. 30).

A mother who underwent an amniocentesis reported,

The doctor who told us, tried to convince us to get an abortion. He said that there was no guarantee that the baby would not be a "vegetable" and that having a handicapped child was going to be very hard on us and our family. (Van Riper, Pridham, & Ryff, 1992, p. 31).

While many parents initially viewed the birth of an infant with Down syndrome as tragedy, their opinions shifted dramatically over time. According to one parent,

All of us have learned to look beyond face value. Before our son was born, there were no disabilities in our very large family. I think it at first shook our perfect world, but now in three years our world has broadened. We have all watched something wonderful grow out of what was initially felt as a tragedy (Van Riper, Pridham, & Ryff, 1992, p. 34).

Study 4

The purpose of this study was twofold: (a) to describe parental perceptions of family-provider relationships, and (b) to explore linkages between parental perceptions of family-provider relationships and well-being in families with children who have Down syndrome (Van Riper, 1995). Mailed questionnaires were used to collect data from 127 parents (91 mothers and 36 fathers).

The findings indicate that when parents of children with Down syndrome believe that their family's relationship with health care providers is positive and family-centered, they feel more satisfied with the care that their child is receiving and they are more likely to seek-help from health care providers. In addition, the findings indicate that when a discrepancy exists between what parents want the family-provider relationship to be like and what they believe the relationship is like, parents feel less satisfied with the care that their child is receiving. Finally, the findings indicate that parents who want, and believe that they have, positive family-centered relationships with providers and who feel satisfied with care that their child is receiving report higher levels of psychological well-being and family functioning

Overall, scores for the well-being variables reflected positive parental and family functioning. The mean depression score for this sample ( M 9.82, SD = 9.59) fell well below 16, the score indicative of clinical depression (Radloff, 1977). Also the mean level of family functioning (M 48.01, SD = 6.96) was lower, suggesting better functioning, than the mean level (M 50.00) for the normative sample described by Skinner, Steinhaurer, and Santa-Barbara (1983).

Study 5

The purpose of this descriptive study was to describe the process by which family members became aware of a child's diagnosis of Down syndrome. In-depth interviews were used to collect data from 20 families of young children with Down syndrome. Findings revealed that most parents became aware of their child's diagnosis of Down syndrome prenatally, in contrast to soon after the child's birth, which was the case for most parents in prior research by this investigator. In general, parents did not feel that they were well informed about the purpose, limitations, and/or accuracy of the prenatal screening for Down syndrome. Parents described feeling shocked by the ramifications of a positive test result. Some of the ethical issues that parents were confronted with were concerns about disclosure of information, discrimination, social stigmatization, autonomous decision making, and insurability. One mother reported that her insurance company implied that they would not cover the infant if a decision were made to continue an affected pregnancy. Another mother described how she became severely depressed after the birth of her child with Down syndrome. According to this mother, she had been congratulated by her physician, because according to her physician, the results of her triple marker screening indicated that her risk of having a child with Down syndrome were less than that of most women her age. She was extremely shocked and unprepared when she ended up giving birth to a child with Down syndrome. The majority of parents in this study indicated that when their child with Down syndrome was born, they were able to celebrate the child's birth because they were over the initial shock of Down syndrome and they had already had a chance to grieve the loss of the "expected child".

Study 6

The purpose of this study was to explore how children respond to the experience of living in a family that includes a child with Down syndrome. The guiding framework for this research was the Resiliency Model of Family Stress, Adjustment, and Adaptation (McCubbin & McCubbin, 1993). Mailed questionnaires were used to collect data from 76 families of children with Down syndrome. The results indicate that for many siblings, the experience of living in a family that includes a child with Down syndrome may be a positive, growth producing experience. As a group, siblings in this study had above average self-concepts. In addition, maternal reports typically indicated that these siblings were socially competent, with a low incidence of behavior problems. Four family variables (i.e., family vulnerability, family appraisal, family resources, and family problem-solving communication) were significantly associated with sibling well-being.

When parents were asked to describe how well their family was doing, most reported that their family was doing well to very well. Many noted that they were doing much better than they had originally predicted. A recurrent theme was the need to constantly juggle multiple demands. According to one parent,

We as a family, I feel, are doing very well. We all try to be individuals and yet keep a close family relationship. We are all interested in each others' goals, talents, and challenges. We try to take an active part in these interests when we can. This means lots of juggling but we make it work. The key is connecting as a family and communicating. Health wise we are all very healthy and grateful.

Another parent wrote,

Overall, our family is doing okay. I say "okay" because there are days when I almost forget that M. has a disability and there are other days when I feel our life is the way it is because she has a disability. For instance, there are a number of hours I spend on the phone each month talking to doctors, teachers, therapists, advocacy groups, support groups etc - time that I would be spending otherwise if M. didn't have DS. Having a family is a balancing act, but when one member's needs are seemingly greater than the others, the juggling is a little more challenging and a little more demanding.

While most parents reported that their family was doing well to very well, a few parents indicated that one or more family members were having a difficult time. For example, one mother wrote,

Our family has been hanging from a string since N's birth. We have financially struggled - mentally struggled. Trying to keep the fact that we could lose our boy. We (my husband and I ) don't talk much. Our daughter had to grow up a little faster than others. She doesn't see me much lately. Trying to get enough money to pay a couple months worth of bills. My husband is the one that continues to worry about money no matter how much or how little we have. We've stayed married so far and kept our house - this is a major accomplishment.

Most siblings indicated that their life had changed or was somewhat different because their brother or sister had Down syndrome. According to one sibling,

Although J. being in my life has not always been easy, I know she has changed my life for the better.

Summary of Findings

Findings, to date, from this investigator's program of research indicate that while the birth of a child with Down syndrome involves a "change of plans" for families, it does not have to be a negative experience. In fact, for many families, it is a positive, growth producing experience. One parent noted,

Our entire family and marriage is stronger. It has changed our view of the world, our view of ourselves, and others. It has made use more giving and less selfish. It has drawn us closer to God. It has caused us to be more concerned about others who are different. It has shown us what we value in life --- relationships ---- not power and wealth. It has made us more content to just be!

Another parent wrote,

I think dealing with the fact of having a child with Down syndrome has deepened our ability to communicate, to talk, to trust each other, and to work together.

Siblings, both young and old, indicated that their brother or sister with Down syndrome had helped or taught them many important things. For example, one sibling wrote,

My brother has taught me more than he will ever know. He has taught me patience and how to be more accepting of others.

According to another sibling,

He has been a blessing to our family. We were getting way too comfortable and set in our ways. As soon as he came into our family we had to stretch ourselves. He has taught us the meaning of love and how the product of love and grow and become such a beautiful person.

Comparison Between These Findings And Findings By Other Investigators

There have been two large programs of research concerning families of children with Down syndrome, one led by Ann Gath and one led by Cliff Cunningham. Both of these were started in the early 1970's and they were conducted in England. A comparison between this investigator's findings and the findings from these two programs of research, especially the early findings, may seem somewhat inappropriate considering all the changes that have occurred in the philosophy of care for children with Down syndrome since the 1970's. On the other hand, findings from these two programs of research have permeated the literature for years and they have had a significant impact on our current understanding of families that include a child with Down syndrome. In addition, both of these investigators have updated their findings over the years.

Gath's program of research

Gath's program of research began in the early 1970's. Originally, Gath focused on behavior problems in siblings of children with Down syndrome (Gath, 1972, 1973, 1974, 1990; Gath & Gumley, 1987). Then, she conducted a prospective study of 30 families that include a child with Down syndrome and 30 families with nondisabled children (Gath, 1977, 1985; Gath & Gumley, 1984). In 1983, Gath reported findings from interviews with 169 substitute families of children with Down syndrome. Gath's 1986 study was a comparison study between 192 families of children with Down syndrome and a group of families with children who had a similar degree of retardation. In her most recent articles (Gath, 1990, 1993), Gath has summarized the findings from her program of research.

Findings from Gath's early work on siblings (Gath, 1973, 1974) indicated that siblings of children with Down syndrome, particularly older sisters, may be at increased risk for behavioral problems. In her later work Gath (1985) suggested that improvements in the provision of services may have lessened the "burden of care" for siblings. She also noted that her findings did not provide support for the earlier predictions by researchers and health care providers that siblings of children with Down syndrome would suffer from lack of parental care and attention.

Findings from Gath's prospective study (Gath, 1977, Gath & Gumley, 1984) revealed few differences between the mental and physical health of parents of children with Down syndrome and parents of nondisabled children. At time one (up to 2 years after the child's birth), similar numbers of marriages in both groups were characterized as warm and harmonious. The two groups of families did differ on the number of families experiencing marital breakdown or severe marital disharmony. According to Gath, three of the 30 families of children with Down syndrome had already broken down irretrievably and six others were very unhappy, with open discord and hostility. At time two, which was eight to nine years later, there had been no further marital breakdowns in the group that included families of children with Down syndrome, but one family in the control group had been divorced and the mother had died in another one of the families.

Gath (1985) noted that when things go wrong for a family, the very existence of the child with Down syndrome brings into sharp focus the vulnerable areas within the family, such as personality defects, immaturity, and precarious defects and precarious relationships. She went on to say that it is unjust to the child with Down syndrome to regard these problems as inevitable results of the increased stress associated with raising a child with Down syndrome. She concluded by saying that many families of children with Down syndrome are able to function in a healthy way and a significant number of families have found their lives enhanced by what they originally thought was going to be an intolerable burden.

In general, findings from Gath's program of research are very similar to findings from this investigator's work. Findings from this investigator's work did not provide support for Gath's early finding that siblings of children with Down syndrome, especially older sisters, may be at increased risk for behavioral problems. But, as noted previously, Gath suggested that changes in the provision of services for individuals with Down syndrome may have decreased the "burden of care" for siblings, thus decreasing the risk of increased behavioral problems.

Cunningham's Program of Research

Cunningham and colleagues started their longitudinal study of the Manchester Down Syndrome Cohort in 1973 (Cunningham, 1996). Initially, the study sample included 183 families of children with Down syndrome. Currently, over 100 families remain in the study. This is one of the largest and most detailed multifactorial programs of research concerning families of children with Down syndrome.

According to Cunningham (1998), there were five major studies in their program of research concerning the Manchester Down Syndrome Cohort. The first was an early development and intervention study (Cunningham, Aumonier, & Sloper, 1982; Sloper, Cunningham, & Arnljotsdottir, 1983). The second study was an exploration of maternal perceptions concerning family adaptation and functioning, using interviews and self-report instruments (Bryne & Cunningham, 1985). In the third study, families in the cohort were compared with families of non-handicapped children (Bryne Cunningham, & Sloper, 1988; Cunningham, 1987). The fourth and fifth study was conducted in 1986-1987 and 1991, respectively. These were comprehensive investigations of families of children with Down syndrome. In addition to being a follow-up study for the fourth study, the fifth study also included the collection of sibling data ( Sloper, Knussen, Turner, & Cunningham, 1991; Turner, Sloper, Knussen, & Cunningham, 1991)

In 1996, Cunningham published an overview of their findings in Down Syndrome Research and Practice. This paper was reprinted in 1998 and can be found at the following internet address: http://www.downsnet.org/library/dsrp/families/default.asp. According to Cunningham,

The overriding impression of the families and their child with Down syndrome is one of normality. The factors that influence the well-being of all members are largely the same as those influencing any child or family. The majority of families did not exhibit pathology as a consequence of having a child with Down syndrome. Indeed the evidence points to positive effects for many families when one member has Down syndrome.

The majority of the families (65%-70%) in the Manchester Down Syndrome Cohort had levels of functioning similar to those of families with nondisabled children. Their levels of psychological and physiological distress were lower than levels reported by families of children with other disabilities (Sloper & Turner, 1993). The divorce rate for the Manchester Down Syndrome Cohort was lower than the national average and only 14% of the parents indicated that having a child with Down syndrome had a detrimental effect on their marriage. The majority of parents indicated that their lives had changed for the better following the birth of their child with Down syndrome.

Once the child with Down syndrome reached adolescence, mothers in the Manchester Down Syndrome Cohort did report a decrease in perceived satisfaction with life (Cunningham, 1998). This decrease in perceived satisfaction with life was associated with a decrease in actual and perceived satisfaction with social support. There was also a trend for more mothers to perceive that the adolescent with Down syndrome had a negative impact on the family. According to Cunningham, this may be due to the fact that adolescents with Down syndrome may place greater restrictions on family life than do nondisabled adolescence.

The main finding from the sibling data collected by Cunningham and colleagues was positive adaptation (Cunningham, 1998). Approximately 80% of the siblings indicated that they had a positive relationship with their parents and their brother or sister with Down syndrome. They also had positive or neutral feelings about the effect of having a brother or sister with Down syndrome. In addition, siblings had positive perceptions of their own self-worth. Ninety-five percent of the siblings were actively involved in helping with the child with Down syndrome and this seemed to have a positive impact on the sibling relationship. Sixty percent of the siblings indicated that they did not take on more household responsibility than did their peers. The majority of mothers in the cohort felt that the experience of having a brother or sister with Down syndrome had a positive effect on other children in the family.

Cunningham (1998) was careful to point out that while most of the families in Manchester Down Syndrome Cohort were doing well, some families were vulnerable and at risk. Factors that seemed to have the greatest impact on well-being in these families were characteristics of the child with Down syndrome (e.g., mental ability, behavior problems) and family resources, especially utilitarian resources (e.g., finances, housing, employment).

As was true with the findings from Gath's work, findings from this investigator's work are very similar to the findings reported by Cunningham and colleagues. The work by Cunningham and colleagues is longitudinal and includes data from many more families that include adolescents with Down syndrome than does this investigator's work. Therefore, it is difficult to compare their findings concerning changes in satisfaction with life once the child with Down syndrome reaches adolescence with findings from this investigator's work.

Recent Findings From Other Investigators

Using a variety of research databases, 18 additional research studies concerning families with Down syndrome (published during the past five years) were identified. Of these, thirteen were conducted in countries other than the United States (e.g., Canada, China, England, Greece, Hong Kong, Japan, and a group of Nordic countries). The majority ( n = 11) focused on how parents respond to the birth and rearing of a child with Down syndrome. Concepts that were assessed in the 18 studies include: parental stress (Atkinson et al., 1994; Cahill & Glidden, 1996; Chen & Tang, 1997; Padeliadu, 1998; Sander & Morgan, 1997; Stores, Stores, Fellow, & Buckley, 1998), parental adjustment/adaptation (Cheng & Tang, 1995; Chiang & Yuh, 1997; Herbert & Carpenter, 1994; Hornby, 1995; Huang, 1996; Sander & Morgan, 1997), psychological distress/depression (Atkinson, Scott, Chishom, & Blackwell, 1994), Fisman et al., 1996; Scott, Atkinson, Minton, & Bowman, 1997), behavior problems (Stores, Stores, Fellow, & Buckley, 1998; Wolf, Fisman, Ellison, & Freeman, 1998), time demands (Barnett & Boyce, 1995; Padeliadu, 1998), parental blame (Hall, Bobrow, & Marteaun, 1997), marital adjustment (Fisman et al., 1996), coping style (Atkinson, Scott, Chishom, & Blackwell, 1994; Cheng & Tang, 1995), social support (Chen & Tang, 1997; Wolf, Fisman, Ellison, & Freeman, 1998), role perceptions (Crowe, Van Leit, Berghmans, & Mann, 1997), family stress (Duis, Summers, & Summers, 1997),and family strengths (Cahill & Glidden, 1996). Minimal attention was paid to the assessment of well-being in families of children with Down syndrome. In addition, mothers were the primary respondents. Few of the studies included data from fathers or siblings.

Findings from these studies indicate that parents of children with Down syndrome experience increased stress, increased time demands, and changes in roles. These findings are consistent with findings from this investigator's work. In addition, parental responses and/or stages of adaptation described in these studies were similar to those found in this investigator's work. The finding by Fisman et al (1996) that marital satisfaction, lack of parental depression, a cohesive family, and a warm, nonconflictual sibling relationship were protective for children with Down syndrome agrees with this investigator's findings that family variables such as family vulnerability, family appraisal, family resources, and family problem-solving communication are significantly associated with sibling well-being.

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1This research was supported by a National Research Service Award No. NR06338, a Helen Denne Schulte Research Award from the University of Wisconsin- Madison, a Seed Grant from The Ohio State University College of Nursing, and a grant from Sigma Theta Tau - Epsilon Chapter. Thanks are extended to the families who participated in this program of research. Correspondence concerning this article should be addressed to Marcia Van Riper, OSU College of Nursing, 1585 Neil Avenue, Columbus, OH 43210 email: [email protected]