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Survival outcome in neonatal intensive care
Volume 83 Number 5
Letters to the Editor
9. Summitt, R. L.: Turner syndrome and Noonan's syndrome, J. PEDIATR. 74: 155, 1969. 10. Levy, E. P.: Unpublished review of 49 persoi'lal cases. 11..Abdel-Salam, E., and Temtamy, S. A.: Familial Turner phenotype, J. PEDIATR. 74: 67, 1969. 12. Levy, E. P., Pashayan, It., Fraser, F. C., and 9 Pinsky, L.: X X and XY Turner phenotypes in a family, Am. J. Dis. Child. 120: 36, 1970.
Reply To the Editor: We were very interested in the suggestion by Pinsky and Levy that the four young boys who developed malignant hyperpyrexia and were described in our paper resemble the X X - X Y Turner phenotype (Noonan's syndrome), since this was one of the possible diagnoses which we had originally considered. However, although these patients do have some features which have been described in association with Noonan's syndrome, the latter may well be a heterogeneous collection of entities, and after some consideration it seemed to us that the four children did not fit completely into any previously recognized genetic syndrome. With this in mind we thought that we should record the clinical findings in these children in detail. It will be interesting to see what characteristics will be found in other young patients susceptible to malignant hyperpyrexia in the future.
M. A. Denborough, M.D., D.Phil., F.R.C.P., F.R.A.C.P. Department o[ Medicine Royal Melbourne Hospital Victoria 3050, Australia
Survival outcome in neonatal intensive care To the Editor: I should like to take exception to Schlesinger's 1 comment concerning outcome among survivors. He states, "Present evidence strongly suggests that the lowered neonatal mortality rate after intensive care is accompanied by a more favorable outcome even among infants of very low birth weight." To support this statement he quoted studies from University College Hospital in London by Rawlings and associates 2 and from the University of Washington in Seattle by Alden and associates, a However, on closer scrutiny of these articles Schlesinger would have discovered that although
89 7
the summaries give glowing results, the body of these articles are more sobering. T h e summary of Rawlings and associates 2 of surviving babies under 1,500 Gm. stated 87 per cent were normal, 7 per cent abnormal, and 6 per cent doubtful. However, closer scrutiny of this paper indicates the mean "conceptual age" at time of testing was only 2 years, 3 months, with a range from 9 months to 4 years, 3 mon}hs. There is some question about the validity of using a Stanford-Binet or Merrill-Palmer test at this age. Furthermore, in those children under 2 years of age, the 80 to 90 developmental quotient is listed in the "normal" rather than in the "doubtful" area, whereas in the over 3 years of age group the distribution in "average" range of 84 to 115 is never stated. In the summary of Alden and associates '~ of outcome of survivors of babies weighing less than 1,000 grains, they state, "otlly two infants of the twenty surviving had definitely abnormal developmental quotients." ttowever, they fail to specify the developmental tests used or the ages at which the last tests were administered, although the children would have to have been quite young. Furthermore, the body of the article reveals that only twelve of the infants had a normal I.Q., six had a "high-abnormal" to borderline I.Q., and two were retarded. There were other significant problems as well. These are only two of a number of instances that I mention in an article written for the Long Island Pediatrician, September, 1973, which demonstrate discrepancies between the summary and the body of the article. A plea was made then, and a plea is being made now, for more critical reporting and more critical review of various findings on the long-term status of survivors of prematurity.
Ralph Cobrinik, M.D. Medical Director Developmental Disabilities Center The Brookdale Hospital Medical Center Brooklyn, N. Y. 11212
REFERENCES 1. Schlesinger, Edward R.: Neonatal intensive care: Planning for services and outcomes following care, J. PEDIATm 82: 916, 1973. 2. Rawlings, G., Reynolds, E. O. R., Stewart, A., and Strang, L. B.: Changing prognosis for infants of very low birth weight, Lancet 1: 516, 1971. 3. Alden, E. R., Mandelkorn, T., Woodrum, D. E., Wennberg, R. P., Parks, C. R., and ttodson, W. A.: Morbidity and mortality of infants
8 98
Letters to the Editor
weighing less than 1000 grams in an intensive care nursery, Pediatrics 50: 40, 1972.
Reply To the Editor: I am afraid that Cobrinik has set up a straw man which he has proceeded to demolish effectively. My point was not that neonatal intensive care necessarilr produces glowing results but simply that there is highly suggestive evidence that intensive care is accompanied by more favorable outcomes than were previous methods of care. This means that the findings from the many excellent long-range follow-up studies of lowbirth-weight infants must now be considered essentially as baseline information against which the findings of future definitive studies after intensive care must be measured. Findings evaluating intensive care up to this time must necessarily relate only t o young children. The point of the study by Rawlings and associates 1 is that the proportion of severely handicapped children, particularly those with severe physical handicaps, was much lower after intensive care of very low-birth-weight infants than after previous methods of care. These handicaps would almost certainly be evident even at the early age at the time of follow-up examination. On the other hand, as Cobrinik points out, the results of developmental testing are more debatable at these early ages. However, among the 17 children with adequate psychometric assessments at 3 or 4. years of age in the series of
The Journal of Pediatrics November 1973
Rawlings and associates, a only one was found to have an intelligence quotient of 84 or less. Even if two or three children in the 84 to 115 I.Q. group fell into the upper range of the borderline level, this would still constitute considerably more favorable outcomes than in earlier studies. Similarly, the admittedly less desirable outcomes among infants with a birth weight of 1,000 Gm. or less, as reported by Alden and associates, 2 are still considerably better than expected for these highly vulnerable infants. Finally, Cobrinik fails to take notice of the low prevalence of severe physical handicaps among very low-birth-weight infants in the Toronto experience reported by Swyer and Linsao. a
Edward R. Schlesinger, M.D. Professor of Maternal and Child Health Department of Public Health Practice University of Pittsburgh Pittsburgh, Pa. 15213
REFERENCES
1. Rawlings, G., Reynolds, E. O. R., Stewart, A., and Strang, L. B.: Changing prognosis for infants of very low birth weight, Lancet I: 516, 1971. 2. Alden, E. R., MandeIkorn, T., Woodrum, D. E., Wennberg, R. P., Parks, C. R., and Itodson, W. A." Morbidity and mortality of infants weighing less than 1000 grains in an intensive care nursery, Pediatrics 50" 40, 1972. 3. Swyer, P. R., and Linsao, L. S.: Personal communication, 1972.
Letters to the Editor
9. Summitt, R. L.: Turner syndrome and Noonan's syndrome, J. PEDIATR. 74: 155, 1969. 10. Levy, E. P.: Unpublished review of 49 persoi'lal cases. 11..Abdel-Salam, E., and Temtamy, S. A.: Familial Turner phenotype, J. PEDIATR. 74: 67, 1969. 12. Levy, E. P., Pashayan, It., Fraser, F. C., and 9 Pinsky, L.: X X and XY Turner phenotypes in a family, Am. J. Dis. Child. 120: 36, 1970.
Reply To the Editor: We were very interested in the suggestion by Pinsky and Levy that the four young boys who developed malignant hyperpyrexia and were described in our paper resemble the X X - X Y Turner phenotype (Noonan's syndrome), since this was one of the possible diagnoses which we had originally considered. However, although these patients do have some features which have been described in association with Noonan's syndrome, the latter may well be a heterogeneous collection of entities, and after some consideration it seemed to us that the four children did not fit completely into any previously recognized genetic syndrome. With this in mind we thought that we should record the clinical findings in these children in detail. It will be interesting to see what characteristics will be found in other young patients susceptible to malignant hyperpyrexia in the future.
M. A. Denborough, M.D., D.Phil., F.R.C.P., F.R.A.C.P. Department o[ Medicine Royal Melbourne Hospital Victoria 3050, Australia
Survival outcome in neonatal intensive care To the Editor: I should like to take exception to Schlesinger's 1 comment concerning outcome among survivors. He states, "Present evidence strongly suggests that the lowered neonatal mortality rate after intensive care is accompanied by a more favorable outcome even among infants of very low birth weight." To support this statement he quoted studies from University College Hospital in London by Rawlings and associates 2 and from the University of Washington in Seattle by Alden and associates, a However, on closer scrutiny of these articles Schlesinger would have discovered that although
89 7
the summaries give glowing results, the body of these articles are more sobering. T h e summary of Rawlings and associates 2 of surviving babies under 1,500 Gm. stated 87 per cent were normal, 7 per cent abnormal, and 6 per cent doubtful. However, closer scrutiny of this paper indicates the mean "conceptual age" at time of testing was only 2 years, 3 months, with a range from 9 months to 4 years, 3 mon}hs. There is some question about the validity of using a Stanford-Binet or Merrill-Palmer test at this age. Furthermore, in those children under 2 years of age, the 80 to 90 developmental quotient is listed in the "normal" rather than in the "doubtful" area, whereas in the over 3 years of age group the distribution in "average" range of 84 to 115 is never stated. In the summary of Alden and associates '~ of outcome of survivors of babies weighing less than 1,000 grains, they state, "otlly two infants of the twenty surviving had definitely abnormal developmental quotients." ttowever, they fail to specify the developmental tests used or the ages at which the last tests were administered, although the children would have to have been quite young. Furthermore, the body of the article reveals that only twelve of the infants had a normal I.Q., six had a "high-abnormal" to borderline I.Q., and two were retarded. There were other significant problems as well. These are only two of a number of instances that I mention in an article written for the Long Island Pediatrician, September, 1973, which demonstrate discrepancies between the summary and the body of the article. A plea was made then, and a plea is being made now, for more critical reporting and more critical review of various findings on the long-term status of survivors of prematurity.
Ralph Cobrinik, M.D. Medical Director Developmental Disabilities Center The Brookdale Hospital Medical Center Brooklyn, N. Y. 11212
REFERENCES 1. Schlesinger, Edward R.: Neonatal intensive care: Planning for services and outcomes following care, J. PEDIATm 82: 916, 1973. 2. Rawlings, G., Reynolds, E. O. R., Stewart, A., and Strang, L. B.: Changing prognosis for infants of very low birth weight, Lancet 1: 516, 1971. 3. Alden, E. R., Mandelkorn, T., Woodrum, D. E., Wennberg, R. P., Parks, C. R., and ttodson, W. A.: Morbidity and mortality of infants
8 98
Letters to the Editor
weighing less than 1000 grams in an intensive care nursery, Pediatrics 50: 40, 1972.
Reply To the Editor: I am afraid that Cobrinik has set up a straw man which he has proceeded to demolish effectively. My point was not that neonatal intensive care necessarilr produces glowing results but simply that there is highly suggestive evidence that intensive care is accompanied by more favorable outcomes than were previous methods of care. This means that the findings from the many excellent long-range follow-up studies of lowbirth-weight infants must now be considered essentially as baseline information against which the findings of future definitive studies after intensive care must be measured. Findings evaluating intensive care up to this time must necessarily relate only t o young children. The point of the study by Rawlings and associates 1 is that the proportion of severely handicapped children, particularly those with severe physical handicaps, was much lower after intensive care of very low-birth-weight infants than after previous methods of care. These handicaps would almost certainly be evident even at the early age at the time of follow-up examination. On the other hand, as Cobrinik points out, the results of developmental testing are more debatable at these early ages. However, among the 17 children with adequate psychometric assessments at 3 or 4. years of age in the series of
The Journal of Pediatrics November 1973
Rawlings and associates, a only one was found to have an intelligence quotient of 84 or less. Even if two or three children in the 84 to 115 I.Q. group fell into the upper range of the borderline level, this would still constitute considerably more favorable outcomes than in earlier studies. Similarly, the admittedly less desirable outcomes among infants with a birth weight of 1,000 Gm. or less, as reported by Alden and associates, 2 are still considerably better than expected for these highly vulnerable infants. Finally, Cobrinik fails to take notice of the low prevalence of severe physical handicaps among very low-birth-weight infants in the Toronto experience reported by Swyer and Linsao. a
Edward R. Schlesinger, M.D. Professor of Maternal and Child Health Department of Public Health Practice University of Pittsburgh Pittsburgh, Pa. 15213
REFERENCES
1. Rawlings, G., Reynolds, E. O. R., Stewart, A., and Strang, L. B.: Changing prognosis for infants of very low birth weight, Lancet I: 516, 1971. 2. Alden, E. R., MandeIkorn, T., Woodrum, D. E., Wennberg, R. P., Parks, C. R., and Itodson, W. A." Morbidity and mortality of infants weighing less than 1000 grains in an intensive care nursery, Pediatrics 50" 40, 1972. 3. Swyer, P. R., and Linsao, L. S.: Personal communication, 1972.