ANNOTATIONS

7. McClure. H. M., Belden. K . H., Pieper. W. A. (1969) ‘Autosomal trisomy in a chimpanzee: resemblance to Down’s syndrome.’ Science. 165, 1010-1012.

8. Benirschke, K., Bogart, M. H.. McClure, H . M., Nelson-Rees, W. A. (1974) ‘Fluorescence of the trisomic chimpazee chromosomes.’ Journal of Medical Primatology, 3, 31 1-314.

9. Andryle, M., Fiedler, W., Rett, A., Ambros, P., Schweitzer, D. (1979) ‘A case of trisomy 22 in fongo pygmaeus. ’ C.vtogenics and Cell Genetics. 24, 1-6.

10. Fredga, K. (1968) ‘Idiogram and trisomy of the water vole (Arvicola terrestris L . ) , a favourable animal for cytogenetic research.’ Chromosoma. 25, 75-89.

I I . Gropp, A,, Putz, B.. Zimmerman, U. (1976) ‘Autosomal monosomy and trisomy causing develop- mental failure.’ Currenf Topics in Pathology. 62 , 177-192.

12. White, B. J . , Tjio, J.-H.. Van d e Water, L. C . , Crandall, C . (1974) ‘Trisomy 19 in the laboratory mouse. I . Frequency in different crosses at specific developmental stages and relationship of trisomy to cleft palate.’ Cytogenetics and Cell Genetics. 13, 217-231.

13. White, B. J., Tjio. J.-H., Van d e W z w , L. C., Crandal!, C. (1974) ‘Trisomy 19 in the laboratory mouse. 11. Intrauterine growth and ~iistological studies qf trisomics and their normal littermates.’ Cy togenetics and Cell Genetics, 13, 232-245.

14. Miyapara, F., Gropp. A. (1979) Personal communication. 15. Donald, L. J., Hamerton, J . L. (1978) ‘Asummaryof the humangenemap, 1973-1977.’1n Bergsma, D.,

Hamerton, J. L., Klinger, H. P., McKusick, V. A,, Evans, J . (Eds.) Human Gene Mapping 4. Birth Defects: Original Article Series. Vol. XIV. pp. 5-1 I . New York: Alan R. Liss.

16. Davisson, M. T., Roderick, T. H. (July 1979) ‘Linkage map of the mouse.’ Mouse News Letter. 61, 19. 17. Francke. U.. Taggart, R. T. (1979) ‘Assignment of the gene for cytoplasmic superoxide dismutase

(Sod-1) to a region of chromosome 16 and of Hprt to a region of the X chromosome in the mouse.’ Procecdings of the National Academy of Science. USA, 76, 5230-5233.

18. Epstein, C. J . , Epstein, L. B., Cox, D., Weil, J . (1979) ‘Functional implications of gene dosage effects in trisomy 21 .’ Paper presented at the International Symposium on Trisomy 21, Rapallo. 8-10 November 1979.

19. Sidman, R. L., Green, M. C . , Appel, S. H. (1965) Catalogue ofNeurologica1 Mutants ofrhe Mouse. Cambridge, Massachusetts: Harvard University Press.

20. Sotelo, C . , Changeux, J . P. (1974) ‘Bergmann fibers and granule cell migration in the cerebellum of homozygous weaver mutant mice.’ Brain Research. 77, 484-491.

21. Bignami, A,, Dahl, D. (1974) ‘The development of Bergmann glia in mutant mice with cerebellar malformations: reeler, staggerer and weaver. Immunofluorescence study with antibodies t o the glial fibrillary acidic protein.’ Journal of Comparative Neurology. 155. 219-229.

22. Mallet. J . , Hutchet, M.. Shelanski, M., Changeux. J. P. (1974) ‘Protein differences associated with the absence of granular cells in the cerebellum of mutant weaver mouse a n d X-irradiated rat.’ FEBS Letters. 76, 243-247.

23. Longstreth, J . D., Morse, H. C. I11 (1980) ‘Expression of murine leukemia viruses in inbred strains of‘ mice.’ In Berry, R. J . (Ed.) The Biology of the House Mouse. London: Academic Press (in press).

24. Krivit, W., Good, R . A. (1956) ‘The simultaneous occurrence of leukemia a n d mongolism: report of 4 cases.’ American Journal of Diseases of Children. 91, 218-222.

25. Todaro, G . J., Martin, G. M. (1967) ‘Increased susceptibility of Down’s syndrome fibroblasts t o transformation by SV,,,.’ Proceedings of the Society of Experimental Biology. 124, 1232-1236.

26. Polani, P. E. (1974) ‘Chromosomal and other genetic influences o n birth weight variation.’ In Elliott, K . , Knight, J . (Eds.) Size at Birrh. Ciba Foundation Symposium 27 (New Series). Amsterdam: Elsevier/Excerpta Medica/North Holland. pp. 127-164.

TOILET TRAINING THE MENTALLY HANDICAPPED CHILD

IT can be argued that toilet training is the most important skill for the mentally handicapped child to master. New learning opportunities may be open to him when he spends less of his day being toiletted, and there may be increased self-confidence. He is less likely to develop skin infections and more likely to be socially acceptable. He is also less likely to be admitted to long-term care’**. Furthermore, toiletting skills, together with other self-help skills, are relatively easy ones to teach by means of behaviour modification methods and are frequently rewarding for the person teaching them, as well as for the child.

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Before embarking on a toilet training programme it is necessary to determine (a) whether the child is incontinent of urine or faeces or both and (6) whether this occurs only during the day, only during the night or at all times. As well as the basic skills of bladder and bowel control and elimination in the appropriate place, full toilet training includes the child’s being able to indicate the need to use the toilet or to go to the toilet alone, and adjusting clothing, bottom-wiping, flushing the lavatory and hand-washing. These extra skills may be taught concurrently with the basic skills or may need to be introduced a t a later stage.

Although as a general rule it is best to be guided by the developmental level of the mentally handicapped child, this is not always possible with toilet training. Most normal children are dry by day at two years of age, but some severely retarded people never reach a mental age of two years. Nevertheless, i t is possible to toilet train them3, but they may need a longer period of training4. I t is also worth observing and recording for a week or two just what the handicapped child does and does not do. For example, does he always urinate or defacate into his pants or does he sometimes use the toilet? Does he open his bowels at the same time each day? Is he constipated? As well as observing carefully, the toilet trainer must ensure that suitable conditions for learning are provided. The child should be comfortable on the potty or lavatory: his feet should reach the floor or rest on a platform. He may need something to hold onto. It may be necessary to toilet at certain times of the day when ‘accidents’ are most likely to occur, and this is where the record sheets may prove useful. If the child goes for long periods without urinating it may be helpful to provide extra fluids shortly before toiletting him. Whenever any appropriate toiletting behaviour occurs, such as approaching the toilet, pulling down pants or eliminating, the child should be rewarded. Praise and smiles may be sufficient, but if he is indifferent to these something else should be sought, perhaps a sweet, a finger game or letting him flush the toilet. Musical potties are often powerful reinforcers for children, and also inform the teacher immediately urination has occurred.

There are various procedures for teaching toilet training and which one is chosen will depend on the problems of the particular child involved, the teacher’s preferences and the time and resources available. For the child who is wet during the day, the simplest strategy-and probably the most commonly used-is the habit-training procedure. This involves toiletting the child at regular and frequent intervals (every half-hour or so, depending on how often he empties his bladder), sitting him on the potty or lavatory and leaving him for a few minutes: if he urinates he is rewarded. This simple strategy is often all that is required, but the biggest problem appears to be to persuade the person caring for the child to make the intervals short enough at the beginning of training. Ideally, the child should be taken just before the next ‘accident’ is due. The observation period may indicate quite reliably how frequently the child urinates and the trips to the toilet can be adjusted appropriately. Once the child is used to eliminating in a certain place at a certain time the intervals can be extended very gradually. For the child who reliably urinates in the right place each time he is taken but who is also frequently wet in between, the intervals need to be shortened. If the child never uses the potty when taken, and so never gets rewarded for using it, it is worth giving extra fluids a few minutes beforehand to see if this will help matters. If the child refuses to sit on the potty or lavatory it will be necessary to shape this behaviour first before attempting toilet training proper. This can be done by rewarding him for sitting for a brief period, perhaps 20 or 30 seconds, then increasing this very slowly until he is sitting for five minutes or so.

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ANNOTATIONS

If the habit training method proves unsuccessful, an intensive toilet training procedure, such as that described by AZRIN and Foxx3 may be called for. Adaptations of their approach have also been used, with various S U C C ~ S S ~ ’ ~ . The essential features of the AZRIN and FOXX intensive toilet training programme are increased opportunities for correct toiletting, immediate and consistent reinforcement, reprimands and withdrawal of rewards following incorrect toiletting, and a post-training procedure to reduce likelihood of relapse. In practice, the trainee spends a large part of his waking life (for a short period) on a potty or lavatory. Many people find the idea of this upsetting, but in my experience many mentally handicapped children positively enjoy the undivided one-to-one attention they receive. However, the procedure needs to be carefully planned beforehand and would not be practicable in a home setting since a considerable number of helpers are needed to ensure success. There is no doubt, however, that rapid and dramatic success can be achieved with even severely mentally handicapped people.

Nocturnal enuresis is another widely encountered problem among the retarded popula- tion, and there appear to be three main methods for dealing with this: (a) bell and pad; (6) retention control; and (c) dry-bed training. MEADOW’ has clearly described the principle behind the bell-and-pad method, and some of the problems frequently en- countered in using it. Probably the major problem for many mentally retarded children is that they interfere with the equipment, so it may be worthwhile to attempt to teach the child to accept the device, since the method can be effective*. Other problems include misunderstanding by parents about the equipment and the way it works, reluctance to wake the child during the night, and fear of disturbing other siblings. The misunderstanding can be overcome by careful explanation, and disturbance to others in the bedroom may be avoided by adapting the equipment so that the buzzer sounds in the parents’ room rather than tht child’s. Alternatively, it is possible to substitute a device which vibrates rather than buzzes. However, it is not possible to use the bell-and-pad technique without some co-operation from the child.

Retention control is a method which attempts to increase the amount of urine a child will retain before urinating. Once the child indicates a need to use the toilet he is rewarded for delaying elimination for a short interval, then the interval is gradually increased. However, there is only limited evidence for the effectiveness of this method. A similar and under-investigated procedure, but perhaps worth considering, is the progressive retention technique. The child is lifted each night before he becomes wet, but it may be necessary to spend a few nights finding a time when he is reliably dry. The next step is to delay lifting him by a few minutes every two or three nights, the idea being to accustom him to an increasingly full bladder but toiletting him before he eliminates.

The final main method for treating nocturnal enuresis-dry-bed training-is the night-time equivalent of the intensive toilet training procedure described above. Again, it is a very demanding procedure (for a week or so) for both trainers and trainees, but the results reported by AZRIN et aL9 are remarkable. A group of 12 profoundly retarded adults showed an 85 per cent reduction in incontinence during the first week and no relapses occurred during a three-month follow-up period.

Sometimes teaching bladder control will lead indirectly to bowel controllo, but obviously this does not always occur. As with enuresis, a simple habit-training procedure is worth trying first for bowel training, but as bowel movements are less frequent, feweropportunities for correct toiletting occur. However, it is possible (under a doctor’s supervision) to provide some extra opportunities by using suppositories; several studies have reported success

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with a combination of medical and behavioural treatments to establish bowel control” Is.

The strategies include positive reinforcement used alone, positive reinforcement used with a laxative or suppository, and positive reinforcement used with punishment for inappropriate toiletting. Punishment typically involves the child washing soiled under- clothes or being placed alone in his room for a short period. An adaptation of AZRIN and Foxx’s intensive toilet training procedure was used successfully for an eight-year-old retarded, encopretic boy treated by DOLEYS and A R N O L D ~ ~ . The boy’s pants were checked every quarter of an hour, and he was awarded for clean pants and for any attempts to defaecate into the toilet, to which he was taken every hour, Star charts were also used as reinforcers, and a mild laxative was used to make defaecation easier. When he did soil his pants the boy had to wash them for 15 minutes, clean himself and face his parents’ displeasure.

Of course there are other toiletting problems encountered with some handicapped children, e.g. smearing faeces, deliberate urination or defaecation in inappropriate places, or even using the toilet too frequently in the absence of any physical cause for doing so. It is not possible to discuss these problems here, but the interested reader is referred to WILSON~ for more information.

The purpose of this annotation has been to convince everyone caring for the mentally handicapped that toilet training should be taught, or at least attempted, with all retarded people and to outline some of the methods for doing so. No single method can be expected to succeed with everyone, and the approach chosen must be considered in the light of the handicapped child’s particular difficulties, as well as the preferences, time and resources of those caring for him.

Rivermead Rehabilitation Centre, Abingdon Road, Oxford OX 1 4XD.

BARBARA WILSON

REFERENCES 1. McCoull, G. (1969-197 1) Report on the Newcastle-upon-Tyne Regional Aetiological Survey (Mental

RetardationJPrudhoe Hospital, Northumberland. 2. Wing, L. (1971) ‘Severely retarded children in the London area: prevalence and provision of services.’

Psychological Medicine, 1, 405-41 5. 3. Azrin, N. H., Foxx, R. M. (1971) ‘A rapid method of toilet training the institutionalized retarded.’

Journal of Applied Behaviour Analysis, 4, 89-99. 4. Smith, P. S., Smith, L. J. (1977) ‘Chronological age and social age as factors in intensive daytime

toilet training of institutionalized mentally retarded individuals.’ Journal of Behavior Therapy and Experimental Psychiatry, 8, 269-273.

5. Smith, P. S., Britton, P. G., Johnson, M., Thomas, D. A. (1975) ‘Problems involved in toileting training profoundly mentally handicapped adults.’ Behaviour Research and Therapy, 15, 301-307.

6. Wilson, B. A. (1980) ‘Toilet training.’ I n Yule, W., Carr, J. (Eds.) Behaviour Modification for the Severely Retarded. London: Croom Helm.

7. Meadow, R. (1977) ‘How to use buzzer alarms to cure bedwetting.’ British Medical Journal, 2 ,

8. Sloop, E. W., Kennedy, W. A. (1973) ‘Institutionalized retarded enuretics treated by a conditioning

9. Azrin, N. H., Sneed, T. J., Foxx, R. M. (1973) ‘Dry-bed: a rapid method of eliminating bedwetting

10. Epstein, L. H., McCoy, J. F. (1977) ‘Bladder and bowel control in Hirschsprung’s disease.’ Journal

11. Neale, D. H.( 1963) ‘Behaviour therapy and encopresis in children.’ Behaviour Research and Therapy.

12. Tomlinson, J. R. (1970) ‘The treatment of bowel retention by operant procedures: a case study.’

13. Ashkenazi, Z. (1975) ‘The treatment of encopresis using a discriminitive stimulus and positive

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1073-1075.

technique.’ American Journal of Mental Deficiency. 77, 717-721.

(enuresis) of the retarded.’ Behaviour Research and Therapy. 11. 427-434.

of Behaviour Therapy and Experimental Psychiatry, 8, 97-99.

1, 139-149.

Journal of Behaviour Therapy and Experimental Psychiatry. 1, 83-88.

reintorcement.’ Journal of Behaviour Therapy and Experimental Psychiatry. 6 , 155- 157.

ANNOTATIONS

14. Crowley, C. P., Armstrong, P. M. (1977) ‘Positive practice, over-correction and behavior rehearsal in the treatment of three cases of encopresis.’ Journal of Behavior Therapy and Experimental Psychiatry. 8, 41 1-416.

15. Wright, D. F.. Bunch, G. (1977) ‘Parental intervention in the treatment of chronic constipation.’ Journal of Behavior Therapy and Experimental Psychiatry, 8, 93-95.

16. Doleys, D. M., Arnold, S. (1975) ‘Treatment of childhood encopresis: full cleanliness training.’ Mental Retardation. 13, (6). 14- 16.

FETAL BREATHING

NO subsequent event in the life of man is more dramatic or more essential to survival than the prompt initiation at birth of co-ordinated respiratory movements leading to adequate gas exchange. For many years interest was focused on this ‘first breath’, but it is now generally accepted that breathing movements normally occur in utero for a considerable period before birth. Although these movements contribute nothing towards gas exchange, they are important for normal pulmonary development’ and there is evidence that their presence is an indication of fetal well-being.

Detailed accounts of early studies of fetal breathing have been given by W I L D S ~ and by DUENHOLTER and PRITCHARD3. As long ago as 1781, WINSLOW observed respiratory movements in animal fetuses through the intact uterine wall. In 1888, AHLFELD4 described periodic rhythmic fetal movements which were visible in the periumbilical area of some pregnant women. With kymograph tracings, he showed that they were irregular, occurring at rates of 36 to 78/min and he concluded that they were due to fetal respiratory activity. REIFFERSCHEID~, in 191 1, confirmed that these movements were not synchronous with maternal pulse or respiration.

In the 1930s several groups investigated fetal breathing in acute experiments using different animal species, experimental techniques and anaesthetic agents. They showed that fetuses could be observed to make breathing movements in certain circumstances, but not that they normally did so. For example BARCROFT and B A R R O N ~ recorded spontaneous breathing movements in exteriorized fetal lambs only up to the fiftieth day of gestation (term in the lamb is 145 days), though gasping could be provoked by hypoxia after this time. Those authors concluded that after a certain gestational age the respiratory centre was actively inhibited. However, SNYDER and ROSENFELD’ observed respiratory movements in term rabbits in an intact uterus bathed in warm saline if spinal section was used for maternal analgesia, but noted that barbiturate anaesthesia abolished them completely.

In 1970, DAWES and coworkerss and MERLET and coworkers9 directly demonstrated episodic breathing activity by recording tracheal flow and pressure in lambs with chronically implanted catheters in utero. The breaths were noted to be shallow and irregular, producing insufficient alteration in lung volume to clear the tracheal dead space, and to be unaffected by spontaneous variations in fetal carotid blood-gas values or by section of the cervical vagi. They also explained the conflicting results of earlier workers by showing that breathing was readily depressed by surgery, temperature changes, tactile stimuli and maternal sedation or anaesthesia.

Animal studies Since that time, studies of chronically implanted fetal lambs have shown a changing

pattern of breathing as gestation advances. Early in gestation (<lo0 days), before 229