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HANDICAP AND PREMATURITY
1087
SIR,-Dr Levin and colleagues suggested that the haemorrhagic shock and encephalopathy syndrome is a "fulminating and often fatal disease", and subsequent case reportst°2 seem to confirm this. A child admitted to this unit may have had the condition in a less severe form. The second of non-identical male twins, born at 35-36 weeks’ gestation, birthweight 2040 g, was admitted at the age of 5 months. He had been a little off colour for 2 days, was not feeding well, and had passed loose stools. On the morning of admission he was found convulsing and very pale and was rushed to hospital where he was noted to be shocked and limp with a temperature of 41 oc. A few minutes later he again began convulsing. Both tympanic membranes were reddened and there were coarse rhonchi in the left lung field. Heart rate was 140/min. Treatment was started with intravenous fluids, penicillin, and gentamicin and the seizures were controlled with diazepam and paraldehyde. 3 h after admission the aspirate from a nasogastric tube was noted to be bloodstained, and profuse bloody diarrhoea developed. A firm liver was now palpable 3 cm below the costal margin. On admission Hb was 12-55 g/dl, and this fell to 9-88 g/dl. White cell count was 31 - 6 x 109/1 (16% neutrophils, 78% lymphocytes). The platelet count fell from 621 to 55 x 109/1. Other most abnormal laboratory results were: prothrombin time 31s (control 14 s), partial
thromboplastin time 65 s (normal range 38-45 s). pH 7-03, PC02 3’6 kPa, standard bicarbonate 10 -6 mmol/l, base deficit 16 mmol/1. Plasma sodium 143, potassium 6 -6, and blood urea 9 -8mmol/l. Plasma alanine aminotransferase 474 UII and y-glutamlytransferase 173 VII. O’rantitrypsin 1-7g/l (normal 1-8-3-0). CSF analysis showed no abnormality. Routine bacteriological studies, including blood culture, revealed no pathogens. A stool specimen grew type 2 poliovirus, thought to be a vaccine strain. Serological studies for viruses were negative. After resuscitation the child improved but he remained pyrexial and unresponsive with a staring expression, and he exhibited intermittent jerking convulsive movements for the next 5 days. Thereafter he steadily improved and was discharged 20 days after admission. Developmental assessment 1 month later suggested mild developmental delay, and follow-up continues. Precise criteria for establishing the diagnosis of haemorrhagic shock and encephalopathy syndrome have not been agreed, but the previously reported cases may represent one end of a spectrum. Recognition of milder forms would then be of considerable importance in providing a complete epidemiological picture and would also permit further laboratory investigation of the syndrome. Paediatric Department, Royal Albert Edward Infirmary,
R. B. MCGUCKEN
Wigan WN1 2NN
HANDICAP AND PREMATURITY
SIR,-Dr Koppe concludes (July 2, p 49) that among very immature infants treated at the Wilhelmina Gasthuis Hospital over the period 1959-78 there "has been a steady reduction in perinatal mortality and long term morbidity". The data presented do not support the assertion about morbidity. The reduction in the prevalence of handicap in the 1975-78 birth cohort is only seen when births to non-resident mothers are excluded. Among the births to non-resident mothers, however, the prevalence of handicap rose from 0 to 20% in the same time period. Is it fair to attribute only the favourable trends in handicap to "improvements in care"? When all infants are considered, no clear downward trend in
handicap is seen: 1983 report
Period 1959-64 1965-69 1970-74 1975-78 1. Morris JA, Matthews TS.
Births 251 160 156 176
(births 25-31 wk) Handicap prevalence per 100 liue births ]].0 15-6 10-9 8-0
Haemorrhagic shock and encephalopathy: a new syndrome young children. Lancet 1983; ii: 278. 2. Schrager GP, Shah A. Haemorrhagic shock/encephalopathy syndrome in infancy. Lancet 1983; ii: 396. in
These handicap trends are very different from those in Koppe and colleagues’ earlier Lancet report (Sept 5, 1981, p 527). 1981 report
Penod 1959-64 1965-69 1970-74 1975-79
(births 501-1501 g) Handicap
rate
per
100 lIVe births 9-0 0 9-0 0 11-6 2-1
Births 245 188 138 243
The 1981 report revealed a reduction in handicap prevalence among the most recently born infants, but in the latest report the impression of a sharp decline in the handicap rate after 1975 is not sustained. The likely explanation for this change is that in the earlier report, which appeared less than two years after the birth of the youngest infants reported on, ascertainment of handicap was
incomplete.
Stanley
has warned against reporting on handicap rates in low birthweight infants too soon. She pointed out that the prevalence of ascertained cerebral palsy in a cohort of low birthweight infants tends to rise until the age of five. In the analysis of time trends, the most recent cohorts contain the youngest children, and a false impression of declining handicap prevalence may therefore be obtained if the children are examined at too young an age. Secular trends in childhood handicap rates, and particularly their relationship to a perinatal care, are subjects of considerable public health importance. Detailed reporting of methods should accompany the results of studies on this subject. The procedures used in ascertainment, the ages of children at examination, the characteristics of infants lost to follow-up, and evidence that diagnostic categories are stable over time are just some of the data needed properly to evaluate the evidence. Gertrude H. Sergievsky Center,
Faculty of Medicine, Columbia University,
NIGEL PANETH
New York, NY 10032, USA
**This letter has been shown follows.-ED L. SIR,-Our "resident"
to
Dr
Koppe,
whose
reply
group is the best area-based group under
primary prenatal care that we have. Mixing with non-residents means introducing a bias of selection. Since obstetric policies influence the number of intrauterine deaths and live births we thought it better to study all births, including intrauterine deaths, instead of live births alone in our 1983 report. In my July 2 letter the figure shows perinatal mortality in the resident group, including these intrauterine deaths (the legends should read "perinatal" not "neonatal"). When all births of the resident group are considered a clear downward trend in handicap prevalence is seen: Penod 1959-64 1965-60 1970-74 1975-78
Births 334 217 178
Handicap prevalence per 100 births
110
8-0 0 11.5 9-6 2-7
The explanation for the difference in handicaps between the 1981 and 1983 reports is not ascertainment of handicap but a change to a gestational age cut-off point of 25-31 weeks (instead of birth weight below 1500 g). In our total material one-third of handicapped children under 32 weeks have birthweights over 1500 g. Department of Neonatology, University of Amsterdam, Academisch Medisch Centrum, 1105AZAmsterdamZ0,Netherlands
J. G. KOPPE
F. Using cerebral palsy data in the evaluation of neonatal intensive care; a warning. Devel Med Child Neurol 1982; 24: 93-94. 1. Sinclair JH, Torrance GW, Boyle M, Herwood SP, Saigal S. Evaluating neonatal 1.
Stanley
intensive
care.
Lancer 1981; ii: 1052.
SIR,-Dr Levin and colleagues suggested that the haemorrhagic shock and encephalopathy syndrome is a "fulminating and often fatal disease", and subsequent case reportst°2 seem to confirm this. A child admitted to this unit may have had the condition in a less severe form. The second of non-identical male twins, born at 35-36 weeks’ gestation, birthweight 2040 g, was admitted at the age of 5 months. He had been a little off colour for 2 days, was not feeding well, and had passed loose stools. On the morning of admission he was found convulsing and very pale and was rushed to hospital where he was noted to be shocked and limp with a temperature of 41 oc. A few minutes later he again began convulsing. Both tympanic membranes were reddened and there were coarse rhonchi in the left lung field. Heart rate was 140/min. Treatment was started with intravenous fluids, penicillin, and gentamicin and the seizures were controlled with diazepam and paraldehyde. 3 h after admission the aspirate from a nasogastric tube was noted to be bloodstained, and profuse bloody diarrhoea developed. A firm liver was now palpable 3 cm below the costal margin. On admission Hb was 12-55 g/dl, and this fell to 9-88 g/dl. White cell count was 31 - 6 x 109/1 (16% neutrophils, 78% lymphocytes). The platelet count fell from 621 to 55 x 109/1. Other most abnormal laboratory results were: prothrombin time 31s (control 14 s), partial
thromboplastin time 65 s (normal range 38-45 s). pH 7-03, PC02 3’6 kPa, standard bicarbonate 10 -6 mmol/l, base deficit 16 mmol/1. Plasma sodium 143, potassium 6 -6, and blood urea 9 -8mmol/l. Plasma alanine aminotransferase 474 UII and y-glutamlytransferase 173 VII. O’rantitrypsin 1-7g/l (normal 1-8-3-0). CSF analysis showed no abnormality. Routine bacteriological studies, including blood culture, revealed no pathogens. A stool specimen grew type 2 poliovirus, thought to be a vaccine strain. Serological studies for viruses were negative. After resuscitation the child improved but he remained pyrexial and unresponsive with a staring expression, and he exhibited intermittent jerking convulsive movements for the next 5 days. Thereafter he steadily improved and was discharged 20 days after admission. Developmental assessment 1 month later suggested mild developmental delay, and follow-up continues. Precise criteria for establishing the diagnosis of haemorrhagic shock and encephalopathy syndrome have not been agreed, but the previously reported cases may represent one end of a spectrum. Recognition of milder forms would then be of considerable importance in providing a complete epidemiological picture and would also permit further laboratory investigation of the syndrome. Paediatric Department, Royal Albert Edward Infirmary,
R. B. MCGUCKEN
Wigan WN1 2NN
HANDICAP AND PREMATURITY
SIR,-Dr Koppe concludes (July 2, p 49) that among very immature infants treated at the Wilhelmina Gasthuis Hospital over the period 1959-78 there "has been a steady reduction in perinatal mortality and long term morbidity". The data presented do not support the assertion about morbidity. The reduction in the prevalence of handicap in the 1975-78 birth cohort is only seen when births to non-resident mothers are excluded. Among the births to non-resident mothers, however, the prevalence of handicap rose from 0 to 20% in the same time period. Is it fair to attribute only the favourable trends in handicap to "improvements in care"? When all infants are considered, no clear downward trend in
handicap is seen: 1983 report
Period 1959-64 1965-69 1970-74 1975-78 1. Morris JA, Matthews TS.
Births 251 160 156 176
(births 25-31 wk) Handicap prevalence per 100 liue births ]].0 15-6 10-9 8-0
Haemorrhagic shock and encephalopathy: a new syndrome young children. Lancet 1983; ii: 278. 2. Schrager GP, Shah A. Haemorrhagic shock/encephalopathy syndrome in infancy. Lancet 1983; ii: 396. in
These handicap trends are very different from those in Koppe and colleagues’ earlier Lancet report (Sept 5, 1981, p 527). 1981 report
Penod 1959-64 1965-69 1970-74 1975-79
(births 501-1501 g) Handicap
rate
per
100 lIVe births 9-0 0 9-0 0 11-6 2-1
Births 245 188 138 243
The 1981 report revealed a reduction in handicap prevalence among the most recently born infants, but in the latest report the impression of a sharp decline in the handicap rate after 1975 is not sustained. The likely explanation for this change is that in the earlier report, which appeared less than two years after the birth of the youngest infants reported on, ascertainment of handicap was
incomplete.
Stanley
has warned against reporting on handicap rates in low birthweight infants too soon. She pointed out that the prevalence of ascertained cerebral palsy in a cohort of low birthweight infants tends to rise until the age of five. In the analysis of time trends, the most recent cohorts contain the youngest children, and a false impression of declining handicap prevalence may therefore be obtained if the children are examined at too young an age. Secular trends in childhood handicap rates, and particularly their relationship to a perinatal care, are subjects of considerable public health importance. Detailed reporting of methods should accompany the results of studies on this subject. The procedures used in ascertainment, the ages of children at examination, the characteristics of infants lost to follow-up, and evidence that diagnostic categories are stable over time are just some of the data needed properly to evaluate the evidence. Gertrude H. Sergievsky Center,
Faculty of Medicine, Columbia University,
NIGEL PANETH
New York, NY 10032, USA
**This letter has been shown follows.-ED L. SIR,-Our "resident"
to
Dr
Koppe,
whose
reply
group is the best area-based group under
primary prenatal care that we have. Mixing with non-residents means introducing a bias of selection. Since obstetric policies influence the number of intrauterine deaths and live births we thought it better to study all births, including intrauterine deaths, instead of live births alone in our 1983 report. In my July 2 letter the figure shows perinatal mortality in the resident group, including these intrauterine deaths (the legends should read "perinatal" not "neonatal"). When all births of the resident group are considered a clear downward trend in handicap prevalence is seen: Penod 1959-64 1965-60 1970-74 1975-78
Births 334 217 178
Handicap prevalence per 100 births
110
8-0 0 11.5 9-6 2-7
The explanation for the difference in handicaps between the 1981 and 1983 reports is not ascertainment of handicap but a change to a gestational age cut-off point of 25-31 weeks (instead of birth weight below 1500 g). In our total material one-third of handicapped children under 32 weeks have birthweights over 1500 g. Department of Neonatology, University of Amsterdam, Academisch Medisch Centrum, 1105AZAmsterdamZ0,Netherlands
J. G. KOPPE
F. Using cerebral palsy data in the evaluation of neonatal intensive care; a warning. Devel Med Child Neurol 1982; 24: 93-94. 1. Sinclair JH, Torrance GW, Boyle M, Herwood SP, Saigal S. Evaluating neonatal 1.
Stanley
intensive
care.
Lancer 1981; ii: 1052.