American Journal of Medical Genetics 4:21 5-21 8 (1979)
The Questionnaire to Parents of Children With the Down Syndrome: How to Inform the Parents and Psychological Responses to Counseling Hiroshi Shiono and Jun-lchi Kadowaki Department of Legal Medicine, Sapporo Medical College, Chuo-ku, Sapporo (H.S.I , Department of Pediatrics, National Nishi-Sapporo Hospital, Sapporo (J.-1. K.)
This study determined the experience of 137 sets of parents when they were informed that their child had Down syndrome and how they would have preferred this matter to have been handled. The survey revealed that the majority of parents would have preferred being told as soon as possible, with both of them present, and that they had suspected something wrong at the birth of the child. This information prompted us to analyze critically the parental experiences and to formulate a positive approach, with sensitive, supportive, and progressive counseling. Key words: questionnaire, counseling, parents, Down syndrome, Japan
INTRODUCTION The task of informing parents that their child has the Down syndrome (DS) is an exceedingly difficult and trying experience. A large number of parents of DS children have communicated t o us their experiences with professionals after the birth of the affected infant. Some reported that the physicians were understanding, empathic, informative, and truly helpful. Others described the physicians as kind but unable t o offer positive assistance because of sadness, insecurity, and limited knowledge of the syndrome. The vast majority of the parents related disappointing encounters with physicians who invoked a “subhuman” nature of the child and suggested institutionalization as the only appropriate course. We report the results of the questionnaire t o parents of DS children in the Hokkaido District of Japan.
Received for publication December 15, 1978; revision accepted February 5 , 1979. Address reprint requests to Dr. H . Shiono, Department of Legal Medicine, Sapporo Medical College, S-1 W-17, Chuo-ku, Sapporo 060, Japan.
0148-7299/79/0403-0215$01.10 0 1979 Alan R. Liss, Inc.
216
Shiono and Kadowaki
MATERIALS A N D METHODS
Over 300 DS patients are being followed as genetic outpatients at Sapporo Medical College. A questionnaire was sent during eight months to 220 sets of parents of DS children who were located in the Hokkaido District through the cooperation of “Kobato-Kai” community agencies, which are the parents’ associations for DS in Japan. The sample consisted of 137 fathers ranging in age from 23 t o 53 years, and 137 mothers who were between 19 and 45 years old. The DS children ranged in age from one month to 15 years. One hundred and thirty-seven (62.8%) sets of parents responded to the questionnaire.
RESULTS AND COMMENTS When Were the Parents Told?
Thirty-eight (27.7%) sets of parents were told of the diagnosis within the first week after the birth of an affected child, whereas 121 (88.5%) were told sometime after the first week and before the end of the first year. It has been discussed when the parents of DS children should be told the diagnosis [l-61. We think that the parents should be notified as soon as a definite diagnosis has been made. Delay in informing the parents may well lead to misunderstanding and may undermine their confidence in the physicians. Drotar et a1 [7] described parental psychologic adaptation as progressing in five stages (shock, denial, sadness and anger, adaptation, and reorganization) in dealing with a congenitally malformed child during the course of its development and care. Observations of these patients suggest that early crisis counseling in the first months of the child’s life may be particularly crucial in parental attachment and adjustment. As Drillien and Wilkinson [3] noted, a full explanation, given early, with support thereafter, appears to facilitate the mother’s acceptance of the handicap. Are the Parents Satisfied With the Time of Telling?
Eighty-one (59%) sets of parents were satisfied with the time of telling; 35 (26%) sets of parents were dissatisfied; they were those who were told after the end of the first year. The time of telling for 2 1 sets of parents (15%) is unknown. This result supports informing the parents as soon as possible. Together or Alone?
Only 37 (27%) sets of parents were told together; 73 (53%) mothers, and 19 (41%) fathers were told alone. The situation in the case of eight couples (6%) is unknown. The survey revealed that the majority of parents would have preferred being told as soon as possible with both of them present. The largest single group is made up of mothers. This contrasts with the result of Jacobs [ l ] . When the diagnosis of the DS has been confirmed, it seems most appropriate for both parents to be present, as they can offer support to each other, The meeting with both parents should be arranged to allow privacy and adequate time for discussion. But we have also encountered a case when lactation ceased when the mother was told the diagnosis. In some cases, therefore, we prefer to tell the father initially, and only later the mother. Preferably we would avoid placing the entire burden of stress on one parent, or making him or her responsible for informing the spouse.
Down Syndrome Questionnaire
217
Who Does the Telling?
One hundred five (77%) sets of parents were told by their pediatrician, 16 (12%) by their obstetrician, six (4%) by their family doctor, one (0.7%)by the midwife, and nine (5%) by others; 9 3 (68%) parents indicated that they were satisfied with the way in which they were informed, and 40 (29%) parents were dissatisfied with the counseling they were given. Reasons for dissatisfaction included 1) too pessimistic - eg, “Your child may die prematurely,” 2 ) the physician does not know the pattern of physical and mental development in DS, and 3) a few physicians described the child as “subhuman” and suggested institutionalization as the only appropriate course. The initial counseling might well be brief, without going into details; ordinarily the troubled parents can assimilate only a few of the implications presented to them. For most parents, initial shock, disbelief, and a period of intense emotional upset were followed by a period of gradual adaptation, so the physician’s remarks should be timed to coincide with increasing parental adaptive capacity [ 5 , 8 ] . Was I t a Shock When Told?
In 80% of cases the news, or the discovery, that her child had the DS came as a shock to the mother, even though she already noticed that there was something wrong. Only one mother said that she was not shocked, because she had a foreboding when she became pregnant. Some 19% of the parents did not comprehend the meaning of the term DS, because the physicians’ information was insufficient. Characteristics of the DS and the mental and physical retardation need to be explained; the relative frequency of the condition should be pointed out, as well as the implications of major congenital anomalies. Mention should be made of the hypotonia and decreased motor activity often present in these children. The mother was asked if she had suspected something wrong when she gave birth to the child; 62 (45%) mothers reported being suspicious. The mothers thought the baby might have some illness, baby was blue, too floppy, or too sleepy, or fed poorly. Some mothers sensed something from the attendants’ attitudes. Did You Become Pregnant Again After the Birth of the DS Child?
The high frequency of induced abortion (28,20%) is striking in subsequent pregnancies. One child was stillborn, nine were aborted spontaneously, 28 were born normally, and one abnormal child was born subsequently. These results might be caused by inaccurate counseling. After chromosome analysis has been performed, the result should be explained accurately and in detail, and information should be provided about the risk involved in future pregnancies and the availability of amniocentesis. Was Amniocentesis Performed in Pregnancy After the Birth of the DS Child?
Only 12 (8.8%)mothers had amniocentesis. Recently, parents have begun t o ask if they can have additional pregnancies with monitoring of the fetus by prenatal cytogenetic study. CONCLUSIONS
Counseling cannot end with informing the parents of the diagnosis of the Down syndrome. Here physicians play a significant role in their continuing care of the child, focusing on developmental aspects as well as the realistic adjustment of the family.
218
Shiono and Kadowaki
Some physicians still recommend institutionalization of DS children without being informed of the availability, quality, and cost of facilities and care, not to mention the detrimental effects; this occurs less often today than it did in the past. Parents wellinstructed in training techniques and recognition of developmental readiness are able to provide appropriate stimulation and can engage constructively in teaching their DS child the basic skills of daily living [9, 101. REFERENCES 1. Jacobs J: Mentally handicapped children. Lancet 2:805, 1962. 2. Carr J: Mongolism: Telling the parents. Dev Med Child Neurol 12:213-221, 1970. 3. Drillien CM, Wilkinson EM: Mongolism: “When should parents b e told.” Br Med J 2: 1306-1307, 1964. 4. Gayton WFL, Walker MSW: Down’s syndrome: Informing the parents. Am J Dis Child 127:510512, 1974. 5. Pueschel S, Murphy A: Counseling parents with Down’s syndrome. Postgrad Med 58:90-95, 1975. 6 . Gilmore DW, Oates RK: Counseling about Down’s syndrome: The parents’ viewpoint. Med J Aust 21600-603, 1977. 7. Drotar D, Basklewica A, Irvin BA, Kenne N , Klaus M: The adaptation of parents t o the birth of an infant with a congenital malformation. Pediatrics 56:710-7 17, 1975. 8. Antley RM, Hartlage LC: Psychological responses t o genetic counseling for Down’s syndrome. Clin Genet 9:257-265, 1976. 10. Golden WAB, Pashayan M : The effect of parental education on the eventual with Down’s syndrome. J Pediatr 89:603-605, 1976. 11. Shipe D, Shotwell A: A continuing study. Am J Ment Defic 69:649, 1965.
Edited by John M. Opitz
The Questionnaire to Parents of Children With the Down Syndrome: How to Inform the Parents and Psychological Responses to Counseling Hiroshi Shiono and Jun-lchi Kadowaki Department of Legal Medicine, Sapporo Medical College, Chuo-ku, Sapporo (H.S.I , Department of Pediatrics, National Nishi-Sapporo Hospital, Sapporo (J.-1. K.)
This study determined the experience of 137 sets of parents when they were informed that their child had Down syndrome and how they would have preferred this matter to have been handled. The survey revealed that the majority of parents would have preferred being told as soon as possible, with both of them present, and that they had suspected something wrong at the birth of the child. This information prompted us to analyze critically the parental experiences and to formulate a positive approach, with sensitive, supportive, and progressive counseling. Key words: questionnaire, counseling, parents, Down syndrome, Japan
INTRODUCTION The task of informing parents that their child has the Down syndrome (DS) is an exceedingly difficult and trying experience. A large number of parents of DS children have communicated t o us their experiences with professionals after the birth of the affected infant. Some reported that the physicians were understanding, empathic, informative, and truly helpful. Others described the physicians as kind but unable t o offer positive assistance because of sadness, insecurity, and limited knowledge of the syndrome. The vast majority of the parents related disappointing encounters with physicians who invoked a “subhuman” nature of the child and suggested institutionalization as the only appropriate course. We report the results of the questionnaire t o parents of DS children in the Hokkaido District of Japan.
Received for publication December 15, 1978; revision accepted February 5 , 1979. Address reprint requests to Dr. H . Shiono, Department of Legal Medicine, Sapporo Medical College, S-1 W-17, Chuo-ku, Sapporo 060, Japan.
0148-7299/79/0403-0215$01.10 0 1979 Alan R. Liss, Inc.
216
Shiono and Kadowaki
MATERIALS A N D METHODS
Over 300 DS patients are being followed as genetic outpatients at Sapporo Medical College. A questionnaire was sent during eight months to 220 sets of parents of DS children who were located in the Hokkaido District through the cooperation of “Kobato-Kai” community agencies, which are the parents’ associations for DS in Japan. The sample consisted of 137 fathers ranging in age from 23 t o 53 years, and 137 mothers who were between 19 and 45 years old. The DS children ranged in age from one month to 15 years. One hundred and thirty-seven (62.8%) sets of parents responded to the questionnaire.
RESULTS AND COMMENTS When Were the Parents Told?
Thirty-eight (27.7%) sets of parents were told of the diagnosis within the first week after the birth of an affected child, whereas 121 (88.5%) were told sometime after the first week and before the end of the first year. It has been discussed when the parents of DS children should be told the diagnosis [l-61. We think that the parents should be notified as soon as a definite diagnosis has been made. Delay in informing the parents may well lead to misunderstanding and may undermine their confidence in the physicians. Drotar et a1 [7] described parental psychologic adaptation as progressing in five stages (shock, denial, sadness and anger, adaptation, and reorganization) in dealing with a congenitally malformed child during the course of its development and care. Observations of these patients suggest that early crisis counseling in the first months of the child’s life may be particularly crucial in parental attachment and adjustment. As Drillien and Wilkinson [3] noted, a full explanation, given early, with support thereafter, appears to facilitate the mother’s acceptance of the handicap. Are the Parents Satisfied With the Time of Telling?
Eighty-one (59%) sets of parents were satisfied with the time of telling; 35 (26%) sets of parents were dissatisfied; they were those who were told after the end of the first year. The time of telling for 2 1 sets of parents (15%) is unknown. This result supports informing the parents as soon as possible. Together or Alone?
Only 37 (27%) sets of parents were told together; 73 (53%) mothers, and 19 (41%) fathers were told alone. The situation in the case of eight couples (6%) is unknown. The survey revealed that the majority of parents would have preferred being told as soon as possible with both of them present. The largest single group is made up of mothers. This contrasts with the result of Jacobs [ l ] . When the diagnosis of the DS has been confirmed, it seems most appropriate for both parents to be present, as they can offer support to each other, The meeting with both parents should be arranged to allow privacy and adequate time for discussion. But we have also encountered a case when lactation ceased when the mother was told the diagnosis. In some cases, therefore, we prefer to tell the father initially, and only later the mother. Preferably we would avoid placing the entire burden of stress on one parent, or making him or her responsible for informing the spouse.
Down Syndrome Questionnaire
217
Who Does the Telling?
One hundred five (77%) sets of parents were told by their pediatrician, 16 (12%) by their obstetrician, six (4%) by their family doctor, one (0.7%)by the midwife, and nine (5%) by others; 9 3 (68%) parents indicated that they were satisfied with the way in which they were informed, and 40 (29%) parents were dissatisfied with the counseling they were given. Reasons for dissatisfaction included 1) too pessimistic - eg, “Your child may die prematurely,” 2 ) the physician does not know the pattern of physical and mental development in DS, and 3) a few physicians described the child as “subhuman” and suggested institutionalization as the only appropriate course. The initial counseling might well be brief, without going into details; ordinarily the troubled parents can assimilate only a few of the implications presented to them. For most parents, initial shock, disbelief, and a period of intense emotional upset were followed by a period of gradual adaptation, so the physician’s remarks should be timed to coincide with increasing parental adaptive capacity [ 5 , 8 ] . Was I t a Shock When Told?
In 80% of cases the news, or the discovery, that her child had the DS came as a shock to the mother, even though she already noticed that there was something wrong. Only one mother said that she was not shocked, because she had a foreboding when she became pregnant. Some 19% of the parents did not comprehend the meaning of the term DS, because the physicians’ information was insufficient. Characteristics of the DS and the mental and physical retardation need to be explained; the relative frequency of the condition should be pointed out, as well as the implications of major congenital anomalies. Mention should be made of the hypotonia and decreased motor activity often present in these children. The mother was asked if she had suspected something wrong when she gave birth to the child; 62 (45%) mothers reported being suspicious. The mothers thought the baby might have some illness, baby was blue, too floppy, or too sleepy, or fed poorly. Some mothers sensed something from the attendants’ attitudes. Did You Become Pregnant Again After the Birth of the DS Child?
The high frequency of induced abortion (28,20%) is striking in subsequent pregnancies. One child was stillborn, nine were aborted spontaneously, 28 were born normally, and one abnormal child was born subsequently. These results might be caused by inaccurate counseling. After chromosome analysis has been performed, the result should be explained accurately and in detail, and information should be provided about the risk involved in future pregnancies and the availability of amniocentesis. Was Amniocentesis Performed in Pregnancy After the Birth of the DS Child?
Only 12 (8.8%)mothers had amniocentesis. Recently, parents have begun t o ask if they can have additional pregnancies with monitoring of the fetus by prenatal cytogenetic study. CONCLUSIONS
Counseling cannot end with informing the parents of the diagnosis of the Down syndrome. Here physicians play a significant role in their continuing care of the child, focusing on developmental aspects as well as the realistic adjustment of the family.
218
Shiono and Kadowaki
Some physicians still recommend institutionalization of DS children without being informed of the availability, quality, and cost of facilities and care, not to mention the detrimental effects; this occurs less often today than it did in the past. Parents wellinstructed in training techniques and recognition of developmental readiness are able to provide appropriate stimulation and can engage constructively in teaching their DS child the basic skills of daily living [9, 101. REFERENCES 1. Jacobs J: Mentally handicapped children. Lancet 2:805, 1962. 2. Carr J: Mongolism: Telling the parents. Dev Med Child Neurol 12:213-221, 1970. 3. Drillien CM, Wilkinson EM: Mongolism: “When should parents b e told.” Br Med J 2: 1306-1307, 1964. 4. Gayton WFL, Walker MSW: Down’s syndrome: Informing the parents. Am J Dis Child 127:510512, 1974. 5. Pueschel S, Murphy A: Counseling parents with Down’s syndrome. Postgrad Med 58:90-95, 1975. 6 . Gilmore DW, Oates RK: Counseling about Down’s syndrome: The parents’ viewpoint. Med J Aust 21600-603, 1977. 7. Drotar D, Basklewica A, Irvin BA, Kenne N , Klaus M: The adaptation of parents t o the birth of an infant with a congenital malformation. Pediatrics 56:710-7 17, 1975. 8. Antley RM, Hartlage LC: Psychological responses t o genetic counseling for Down’s syndrome. Clin Genet 9:257-265, 1976. 10. Golden WAB, Pashayan M : The effect of parental education on the eventual with Down’s syndrome. J Pediatr 89:603-605, 1976. 11. Shipe D, Shotwell A: A continuing study. Am J Ment Defic 69:649, 1965.
Edited by John M. Opitz
