PERSPECTIVES 1211 Arch Dis Child: first published as 10.1136/adc.2005.077057 on 21 November 2005. Downloaded from Congenital anomalies The movements of the tongue during ...... infant feeding have been studied by cine-radiography and more recently by ultrasound.12 13 Ultrasound reveals some Tongue tie similarities between the movements made by the baby when either breast D M B Hall, M J Renfrew or bottle feeding,14 but also some impor- tant differences.15 The tongue is pro- ...... jected further forward in breast 16 Common problem or old wives’ tale? feeding and the human nipple elon- gates during each suck in a way that an artificial teat cannot do.14 During feed- he resurgence of interest in breast tongue tie is an important issue—she ing, the artificial teat, or the nipple feeding has been accompanied by a experienced pain for many weeks while together with some breast tissue, is held Tlively debate about the significance breast feeding her first child, who fully in the mouth with the tongue of ‘‘tongue tie’’ or ankyloglossia. exhibited features said to be typical of covering the lower gum ridge. The Symptoms attributed to tongue tie tongue tie, and has since discussed this nipple is protected from damage and include nipple pain and trauma, diffi- issue widely with lactation specialists pain at the back of the baby’s mouth.16 culty in the baby attaching to the breast, and women having similar problems. The baby’s lower jaw is then elevated, frequent feeding, and uncoordinated The other (DH) accepted that ankylo- compressing the artificial teat, or the sucking. These problems may result in glossia occurs in dysmorphic infants10 breast immediately behind the nipple, the mother deciding to terminate breast and occasionally in otherwise normal while the front of the tongue moves up 11 feeding prematurely, slow weight gain babies, but was sceptical about the to aid the expression of milk. In breast for the baby, and even hypernatraemic high prevalence of the condition now feeding, this is by compression of the dehydration. Speech defects have also being described by several authors. milk ducts under the areola. A wave of been attributed to tongue tie. Strong upward movement of the medial part of views have been expressed by many ANATOMY AND PHYSIOLOGY the tongue progresses backwards, and eminent authors on the subject (box 1). The tongue is a highly mobile organ the expression of the milk is further This paper reviews what is known made up of longitudinal, horizontal, facilitated by negative pressure gener- about tongue movements and the sig- vertical, and transverse intrinsic muscle ated by downward movement of the nificance and treatment of tongue tie. It bundles. The extrinsic muscles are the back of the tongue and the lower jaw is based on two literature reviews, one fan-like genioglossus which is inserted and, in breast feeding, by the active conducted on behalf of NICE12by one of into the medial part of the tongue and expulsion of milk once the let down us (MR) and updated by further the styloglossus and hyoglossus into the occurs. searches of published and grey literature lateral portions. The sub-lingual frenu- In coordinated feeding, the sucking, and conference abstracts. The publica- lum is a fold of mucosa connecting the swallowing and breathing movements tions reviewed for this paper are sum- midline of the inferior surface of the follow in a 1:1:1 sequence. This can marised in table 1. tongue to the floor of the mouth. take several days to become established As our review found little high quality Tongue tie is the name given to the in healthy full term infants. In pre- objective evidence, we begin by making condition arising when the frenulum is term infants and in some term infants http://adc.bmj.com/ explicit the personal experience and bias unusually thick, tight, or short. There a variety of poorly coordinated feed- with which we commenced the review. are many variations and differing ing movement patterns are observed One of the authors (MR) felt that degrees of severity (fig 1). and sometimes persist.17 Antenatal

Box 1: Quotes from the past on September 24, 2021 by guest. Protected copyright. ‘‘In observing a very large series of newborn babies, we have never seen a tongue that had to be clipped’’ (McEnery and Gaines, Chicago,1940) ‘‘While tongue tie is not nearly as common as members of the public believe, nevertheless a genuine case is occasionally seen and the condition is not entirely mythical although surrounded by an aura of superstition and old wives’ tales’’ (Cullum, UK, 1959) ‘‘Tongue tie…has been described as a myth of hoary antiquity…but it is probably wrong to suggest that it never causes symptoms. A case is reported in which a tight fraenum ruptured spontaneously during feeding…this baby remained a slow feeder and…(had not been) disabled by his tongue tie’’ (Smithells, London, 1959) ‘‘Tongue tie is a rare but definite congenital deformity’’ (Browne, London,1959) ‘‘Tongue tie is a rare cause of dysarthria, though it is often blamed for slow speech development…most patients who have real limitation of movement as a result of tongue tie have a history of difficult milk feeding’’ (Ingram, Edinburgh, 1968) ‘‘I have never seen feeding difficulties in the first year resulting from tongue tie and I doubt whether it is ever necessary to carry out an operation on it till the age of two or three…There are still doctors who cut the frenulum in the newborn period. This is always wrong’’ (Illingworth, Sheffield, 1982) ‘‘Tongue tie where the tongue is forked can, very rarely, add to the baby’s difficulties in taking the breast with poor protractility’’ (Gunther, UK, 1970) ‘‘To some extent tongue tie is normal in every newborn baby and it should rarely interfere with either sucking or later speech development’’ (Davies et al, UK, 1972) ‘‘True tongue tie is a very rare condition. This condition has been over-diagnosed in the past because of the failure to recognise that the frenum passing from the tongue to the floor of the mouth is normally short in the newborn…Only in infants with severe limitation of the tongue movement and inability to suck is division of the frenum indicated’’ (Turner, Douglas, and Cockburn, UK, 1988)

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Table 1 Tongue tie; review of literature

How cases were Type of study and Author(s) Number and age group studied identified intervention Results

Messner et al3 Examined 1041 newborns. Identified 50 Screened by one Observational follow up study 30/36 TT cases and 33/36 controls TT cases (4.8%). M: F ratio 2.6:1 doctor, confirmed by but no intervention breast fed to 2 months (p = 0.29). 36 cases of TT enrolled and 36 controls one colleague 9 cases and 1 control experienced without TT breast feeding difficulties

Hogan et al4 Examined 1866 babies. Identified 201 TT Photos to assist staff in Randomised to immediate TT cases treated by frenulotomy; 27/28 cases (10.7% ). M:F ratio 1.6:1. 44% TT postnatal checks frenulotomy or support by marked improvement. Counselled cases cases had problems feeding. 57 TT babies lactation counsellor, at mean managed conservatively; 1/29 entered study (40 breast fed and 17 bottle age 20 days (3–70), median improved fed) age 14 days

Ballard et al5 Examined 2763 breast fed in-patient One observer examining 123 cases underwent Latch improved in all, pain scores fell babies and 273 attenders at lactation all babies. ATLFF used frenulotomy at age 1–2 days significantly clinic. M:F ratio 1.5:1. Identified TT in 3.2% in-patients and 12.8% clinic attenders

Ricke et al6 Examined 3490 babies, identified 148 TT Nurses assisted by Observational study, no Mothers of TT babies three times more cases (4.24%). M:F ratio 2.3:1. photos, ATLFF by team intervention likely to give up breast feeding by one Enrolled 49 TT babies for study with 2 week; however, 80% TT breast feeding matched non-TT breast fed babies as well at one week. TT and non-TT breast controls feeding in equal numbers at 1 month. Mothers with TT babies reporting more pain at one month but not statistically significant. Small numbers so type II errors possible

Ramsay7 Case series Referrals to paediatric Measured nipple tip to hard Distance changed from 7.99 mm to surgeon soft palate junction by 6.49 mm. Milk transfer increased from ultrasound, pre- and post- 3.3 to 7.2 ml/min. At least 7 day frenulotomy interval between frenulotomy and 2nd measurement

Messner and Case series of speech problems: 30 Measured tongue Frenulotomy Speech improved. 25 mothers had tried Lalakea8 children age 1–12 protrusion and inter- to breast feed; 21 said no problems incisal distance

Fernando9 Case series, n >200 Various; majority Frenulotomy Improved to varying degree. 20% had presenting with speech history of BF problems: 80% did not disorders

TT, tongue tie; ATLFF, Assessment Tool for Lingual Frenulum Function. http://adc.bmj.com/ ultrasound studies show that mouth N Division of the frenulum (frenulot- selection of photos (Hogan and collea- and tongue movements are already omy) is a low risk effective treatment gues,4 Ricke and colleagues6). We have well developed in association with N The condition is genetic not found any formal data on observer intra-uterine yawning and crying.18 19 agreement or variation in this process. The method of selection in the study by CASE DEFINITION Masaiti and Kaempf20 is unclear but cases Can tongue tie be defined—and to what were probably selected on the basis of on September 24, 2021 by guest. Protected copyright. HYPOTHESES extent do individual observers agree on breast feeding problems. Two authors Review of the literature and expert the diagnosis? The length of attachment (Messner,3 Ricke6)aimedtoreducebias opinion gave rise to the following of the frenulum varies widely. In some by trying to avoid specific mention of hypotheses: babies it extends to the tip of the tongue tie to mothers but acknowledge tongue. There may be an indentation that this was difficult. N Tongue tie is a definable condition of the anterior edge, referred to as a All authors agreed that function is Tongue tie affects 3–4% of infants heart shaped tongue. The appearance of N more important than appearance and the tongue is not sufficient on its own to N The tight frenulum prevents the Hazelbaker designed an Assessment Tool make a diagnosis, as the thickness and infant from getting the tongue over for Lingual Frenulum Function elasticity of the frenulum also vary the lower lip and gum ridge and (ATLFF).21 Ballard used this tool but did widely and affect the extent to which therefore can cause feeding pro- not examine inter-rater reliability. Ricke et normal tongue movements are inhibited blems, particularly affecting breast al found that the inter-rater agreement (see fig 1). feeding, leading to pain for the using the ATLFF was only moderate and In four published studies of tongue tie mother and poor infant weight gain; that many infants did not fit in any of the in babies, the initial selection of possible it can also affect bottle feeding categories defined by Hazelbaker. cases from the whole newborn population N The impact of a tight frenulum varies was based only on appearance and was between mother–baby dyads done by one individual (Ballard and BIRTH PREVALENCE OF TONGUE N A tight frenulum can also cause colleagues5), or by one individual with TIE problems in older children and adults, confirmation of positive diagnoses by one There is agreement among authors that involving speech, dental hygiene, lick- other observer (Messner and colleagues3), tongue tie is found in around 3–4% of ing ice cream, and French kissing or accomplished by providing staff with a babies, with the exception of Hogan et al

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Figure 1 Six examples of babies diagnosed as having tongue tie, showing the variation in the thickness and insertion of the frenulum (reproduced with kind permission from Carolyn Westcott, Princess Anne Hospital, Southampton).

www.archdischild.com 1214 PERSPECTIVES Arch Dis Child: first published as 10.1136/adc.2005.077057 on 21 November 2005. Downloaded from who report 10.7%. All report a male INTERVENTION AN OVERVIEW preponderance with ratios varying from All authors agree that frenulotomy in Tongue tie is at first glance a minor 1.5:1 to 2.6:1. the newborn is a low risk minor issue, but from the results of the only procedure, performed without anaes- randomised controlled trial yet con- THE IMPACT OF TONGUE TIE thetic. The presence of the deep lingual ducted, Hogan and colleagues4 suggest Maternal pain during feeding, some- vein just lateral to the midline means that at least 3% of newborns (57/1866) times accompanied by trauma, and that significant venous bleeding could would benefit from frenulotomy and difficulty in the baby taking the breast, occur if technique is not meticulous but that this would increase the rate of are the main breast feeding problems we found no reports of serious adverse continuing breast feeding. Most of the 3–6 attributed to tongue tie. Attributing events. In older children the procedure literature on tongue tie has been in pain during breast feeding to tongue tie needs an anaesthetic and sometimes a connection with breast feeding; how- is not straightforward, however, since frenuloplasty, which carries some risk of ever, Hogan et al report that of the 57 pain is a common problem that can scarring. babies in their study who benefited result from several other causes, includ- Ballard and colleagues5 reported a from frenulotomy, 40 were breast fed ing attachment problems unconnected marked fall in maternal pain scores but 17 were artificially fed. If they are with tongue tie, and infection.16 22–24 after the procedure. Hogan and collea- correct, this is a very common congeni- Ricke and colleagues6reported that more gues4 randomised their cases to immedi- tal anomaly that affects both breast fed tongue tied infants than controls were ate or delayed intervention and found and bottle fed babies, and up to 18 000 bottle fed at one week but there was no that frenulotomy was much more effec- such procedures should be performed significant difference at one month, tive than advice from a lactation coun- each year in the UK. It is therefore though attrition meant that numbers sellor. They reported dramatic and important to ask whether the evidence were small. Messner et al3 found no rapid, often immediate, improvement supports that rate of intervention and to difference in the rate of breast feeding after the procedure in most of their scrutinise the evidence with a particu- between tongue tied infants and con- larly critical eye. trols at 2 months but a significant cases; improvement was noted in 95% of babies. The measurement of outcomes There were a number of methodolo- difference in the numbers of mothers gical problems with most of the studies was not blinded. The precise criteria reporting problems with breast feeding. we reviewed. These included: Ramsay7 measured the distance from for improvement were not specified. nipple tip to the junction of the hard Improvement was not always imm- N Inadequate assessment of inter- and soft palate by sub-mental ultra- ediate, but this could be due to the observer reliability of the initial diag- sound. The distance decreased from need for sore nipples to heal or for the nosis, the dynamic assessment of 7.99 mm (¡2.80) to 6.49 mm (¡1.87) baby to re-learn optimal patterns of feeding and the maternal symptoms seven days after frenulotomy. This suckling. N Ethical and practical difficulties in change, though statistically significant, In case series of older children and concealing the suspected diagnosis is small and its practical significance is adults, some striking improvements from the mother, thus potentially unknown. The tongue movements were were noted after surgery, but many of introducing a bias by raising the said to become ‘‘more normal’’. the children showed only gradual or expectation of breast feeding pro- modest improvement, particularly blems and of improvement from VARIATION BETWEEN DYADS where the articulation of speech was intervention—this is, however, a 4 Hogan and colleagues found that more concerned; these children often needed common limitation in most studies than half of babies with tongue tie had continuing speech therapy and this was of breast feeding problems http://adc.bmj.com/ no problems breast feeding but could attributed to the need to un-learn N Poorly defined outcome measures; it not show any correlation between sever- 9 established patterns of articulation. is particularly difficult to establish an ity of tongue tie and feeding difficulty. The absence of any comparison or objective assessment of improve- This is perhaps surprising, but it may be control cases makes these reports ment, when the primary outcome that only a small shift in positioning on impossible to evaluate and we found measure is reduction in maternal the breast is sufficient to eliminate pain no comparative studies or randomised pain during breast feeding and improve feeding. Ricke and collea- trials addressing the role of tongue tie or on September 24, 2021 by guest. Protected copyright. 6 N The dilemma of when to assess and gues reported that 80% of tongue tied frenulotomy in older children. infants were breast feeding successfully intervene for tongue tie; if done very at one week. It is of interest that in two early, before breast feeding is estab- case series of older children presenting IS ANKYLOGLOSSIA INHERITED? lished, as in the Ballard et al study, with speech difficulties and other pro- To define the inheritance of a condition, improvements may be wrongly blems attributed by the authors to a robust case definition is needed, but attributed to the procedure (because tongue tie, 21/25 mothers (Messner tongue tie varies markedly in severity suckling efficiency improves over the 13 and Lalakea8) and 80% of an unspecified and is not an all-or-none condition. first few days and weeks ), but if number (Fernando9) who were asked When an anomaly is identified in a done later (as in Hogan et al), many about breast feeding reported no sig- newborn infant, the family searches its mothers may already have sore nip- nificant difficulties. collective memory for other similar ples or have given up breast feeding. cases but, in the case of tongue tie, it OTHER PROBLEMS would be impossible to assess the CONCLUSIONS Several case series report a range of validity of that diagnosis in retrospect. We began this review by stating our other problems in older children asso- None of the studies we reviewed con- personal bias. While DH confesses to ciated with ankyloglossia—speech sidered these issues and none had still being somewhat more sceptical defects, difficulty in licking the lips or gathered systematic family data across than MR, we are in complete agreement in kissing, dribbling, etc. These are a number of families with and without on the following conclusions: difficult to evaluate as the authors do the condition. Notwithstanding the not give details of the catchment popu- comments made in several papers, no N Individual case histories suggest that lation, referral patterns, or detailed conclusions can currently be drawn some babies do have a tight frenulum criteria for inclusion in the series. about family history. (tongue tie) which can inhibit breast

www.archdischild.com PERSPECTIVES 1215 Arch Dis Child: first published as 10.1136/adc.2005.077057 on 21 November 2005. Downloaded from feeding and, in some cases, bottle Arch Dis Child 2005;90:1211–1215. 9 Fernando C. Tongue tie—from confusion to doi: 10.1136/adc.2005.077065 clarity. Sydney: Tandem, 1998. feeding as well 10 Emmanouil-Nikoloussi E, Kerameos-Foroglou C. N Although frenulotomy is a simple low ...... Congenital syndromes connected with tongue risk procedure, it should be carried malformations. Bull Assoc Anat (Nancy) Authors’ affiliations 1992;76:67–72. out only by those who have been D M B Hall, Institute of General Practice and 11 Fitz-Dersorgher R. All tied up. Practising Midwife 25 trained in the procedure Primary Care, ScHARR, University of Sheffield, 2003;6:20–3. 12 Bosma JF, Hepburn LG, Josell SD, et al. N It can be justified only if it is likely to UK Ultrasound demonstration of tongue motions lead to significant improvement in M J Renfrew, Mother and Infant Research during suckle feeding. Dev Med Child Neurol the comfort and the continuation of Unit, Department of Health Sciences, 1990;32:223–9. University of York, UK 13 Weber F, Woolridge MW, Baum JD. An breast feeding, or of other longer ultrasonographic study of the organisation of term problems for the child sucking and swallowing by newborn infants. Dev Correspondence to: Prof. D M B Hall, Storrs Med Child Neurol 1986;28:19–24. N We do not know the true prevalence House Farm, Storrs Lane, Sheffield S6 6GY, 14 Nowak AJ, Smith WL, Erenberg A. of significant tongue tie UK; [email protected] Imaging evaluation of breast-feeding and bottle-feeding systems. J Pediatr N There is no evidence one way or the Competing interests: none declared 1995;126:S130–4. other about inheritance Parental consent was obtained for publication 15 Woolridge MW. The ‘anatomy’ of infant sucking. N On current evidence, there is no Midwifery 1986;2(4):164–71. of the babies in figure 1 16 Woolridge MW. Aetiology of sore nipples. justification for actively searching Midwifery 1986;2(4):172–6. for tongue tie during routine exam- REFERENCES 17 Bu’Lock F, Woolridge MW, Baum JD. ination, but when mothers are hav- Development of co-ordination of sucking, 1 Renfrew MJ, Dyson L, Wallace LW, et al. The swallowing and breathing: ultrasound study of ing difficulty in breast feeding this effectiveness of public health interventions to term and preterm infants. Dev Med Child Neurol should be considered as one of promote the duration of breastfeeding: a 1990;32:669–78. several possible causes systematic review. London: National Institute for 18 Gingras JL, Mitchell EA, Grattan KE. Fetal Health and Clinical Excellence, 2005. homologue of infant crying. Arch Dis Child Fetal N The diagnosis should rest primarily 2 Renfrew MJ, Woolridge MW, Ross McGill H. Neonatal Ed 2005;90:F415–18. on observation and analysis of feed- Enabling women to breastfeed. London: The 19 Kurjak A, Stanojevic M, Azumendi G, et al. The ing difficulties rather than the static Stationary Office, 2000. potential of four-dimensional (4D) 3 Messner AH, Lalakea ML, Aby J, et al. ultrasonography in the assessment of fetal appearance of the tongue Ankyloglossia: incidence and associated feeding awareness. J Perinat Med 2005;33:46–53. N It may be wise to be particularly difficulties. Arch Otolaryngol Head Neck Surg 20 Masaitis NS, Kaempf JW. Developing a 2000;126:36–9. frenotomy policy at one medical center: cautious in making this diagnosis in 4 Hogan M, Westcott C, Griffiths M. Randomized, a case study approach. J Hum Lact the first two or three days before controlled trial of division of tongue tie in infants 1996;12:229–32. lactation is established with feeding problems. J Paediatr Child Health 21 Hazelbaker AK. The assessment tool for lingual 2005;41:246–50. frenulum function (ATLFF): use in a lactation N The problem is of sufficient interest 5 Ballard JL, Auer CE, Khoury JC. Ankyloglossia: consultant private practice [thesis]. Pasadena, and importance to merit further assessment, incidence, and effect of frenuloplasty CA: Pacific Oaks College, 1993. on the breastfeeding dyad. Pediatrics 22 Gunther M. Sore nipples: causes and prevention. studies of both breast and bottle fed 2002;110:e63. Lancet 1945;ii:590–3. babies, in which more precise case 6 Ricke LA, Baker NJ, Madlon-Kay DJ, et al. 23 Amir LH, Pakula S. Nipple pain, mastalgia and definition, measures of inter-observer Newborn tongue tie: prevalence and effect on candidiasis in the lactating breast. Aust N Z J Obstet reliability of pre- and post-interven- breast-feeding. J Am Board Fam Pract Gynaecol 1991;31:378–80. 2005;18:1–7. 24 Hamlyn B, Brooker S, Olienkova K, et al. Infant tion assessment, and ultrasound ima- 7 Ramsay DT. Ultrasound imaging of the effect of Feeding Survey 2000. London: The Stationery ging are likely to play a key role frenulotomy on breast feeding infants with Office.

ankyloglossia [abstract]. Paper presented at 12th 25 Ballard J, Chantry C, Howard CR. Guidelines for http://adc.bmj.com/ N Given the evidence that breast feed- international conference of the International the evaluation and management of neonatal ing has many advantages for both Society for Research in Human Milk and ankyloglossia and its complications in the mother and baby, funding should be Lactation, Cambridge, UK, 2004. breastfeeding dyad. Academy of Breastfeeding 8 Messner AH, Lalakea ML. The effect of Medicine, 2004. http://www.bfmed.org/ sought for carefully planned defini- ankyloglossia on speech in children. Otolaryngol protocol/ankyloglossia.pdf. Accessed 21 July tive studies on the issue. Head Neck Surg 2002;127:539–45. 2005. on September 24, 2021 by guest. Protected copyright. Endocrinology cardiovascular health, adiposity in over- ...... weight youth, and blood pressure in mildly hypertensive adolescents. Physical activity also has a beneficial effect on Children with diabetes benefit from anxiety, depression, and self-concept. The 60 minutes or more of physical activity exercise can be achieved in a cumulative manner in school during physical education, J I Wolfsdorf recess, intramural sports, and before and after school programmes...... Exercise requires considerable altera- tions in fuel metabolism and presents Commentary on the paper by Massin et al (see page 1223) unique challenges for the person with type 1 diabetes mellitus (T1D).2 During ow much physical activity do school age youth should participate daily the first 5–10 minutes of moderate children require to obtain benefi- in at least 60 minutes of moderate to intensity exercise, skeletal muscle glyco- Hcial health and behavioural vigorous physical activity that is devel- gen is the major fuel for working effects? The recent report concerning opmentally appropriate, enjoyable, and muscle. With increasing duration of the effects of regular physical activity on involves a variety of activities.1 There is exercise, plasma glucose and non-ester- health and behavioural outcomes in strong evidence for beneficial effects of ified fatty acids (NEFA) predominate, 6–18 year old youth recommends that physical activity on: musculoskeletal and and to meet the increased demand for

www.archdischild.com 1216 PERSPECTIVES Arch Dis Child: first published as 10.1136/adc.2005.077057 on 21 November 2005. Downloaded from fuel, a complex hormonal and auto- is often difficult to implement consis- Despite its limitations, the study nomic response increases hepatic glu- tently. Extra snacks (for example, suggests that light and moderate physi- cose production and mobilisation of 10–15 g carbohydrate per 30 minutes cal activity may be associated with NEFA from adipose tissue. Plasma of vigorous physical activity depending better glycaemic control in school age insulin concentration decreases and on the child’s age) before and, if the children, but not in teenagers. What levels of the counter-regulatory hor- exercise is prolonged, during the activity might explain the difference between mones (adrenaline, noradrenaline, glu- are used to compensate for unplanned children and teenagers? Diabetes man- cagon, cortisol, and growth hormone) bursts of increased energy expenditure. agement is even more challenging dur- increase, resulting in enhanced hepatic Exercise may be more predictable in ing puberty, and glycaemia is typically production of new glucose from gluco- older children and adolescents, and less well controlled than before puberty neogenic substrates such as lactate and hypoglycaemia can usually be prevented and in adulthood. This is attributable to glycerol. Large quantities of the glucose by a combination of anticipatory reduc- a combination of the endocrinological transporter protein GLUT4 are recruited tion in the pre-exercise insulin dose or a changes characteristic of puberty9 and to the membrane of contracting muscle, temporary interruption of basal insulin less meticulous adherence to diet and independently of insulin, increasing infusion in patients who use continuous insulin administration. glucose transport into muscle.3 These subcutaneous insulin infusion (CSII) Although physical exercise is compli- changes result in the increased fuel together with supplemental carbohy- cated for the child with T1D by the need supply required to match glucose utili- drate before, during, and after physical to prevent hypoglycaemia, with proper sation by exercising muscle and prevent activity. The optimal strategy in the guidance and preparation, participation hypoglycaemia. After prolonged exer- individual child depends on the inten- in exercise can and should be a safe and cise, liver and muscle glycogen stores sity and duration of the physical activity enjoyable experience. Despite the lack of are low and hepatic glucose production and its timing relative to the child’s compelling evidence that physical train- is accelerated. Resynthesis of muscle usual dietary and insulin regimen. After ing and exercise per se improve glycae- glycogen is, initially, largely a result of prolonged or strenuous exercise in the mic control in children and adolescents increased GLUT4 transporter activity afternoon or evening, the pre-supper or with T1D,10–16 exercise clearly offers and insulin sensitivity. bedtime dose of intermediate acting many health and psychological benefits Glucose homoeostasis, which depends insulin should be reduced by 10–30% for people with and without diabetes. At on the balance between tissue glucose (or an equivalent temporary reduction least 60 minutes of moderate to vigorous uptake and hepatic glucose release, is in overnight basal insulin delivery in physical exercise daily should be a influenced by the plasma levels of patients using CSII). To further reduce component of a comprehensive pro- insulin and counter-regulatory hor- the risk of nocturnal or early morning gramme of diabetes management in mones. The normal regulation of insulin hypoglycaemia caused by the lag effect children. With the increased prevalence 67 secretion is lost in T1D, and current of exercise, the bedtime snack should of overweight and obesity in the popu- methods of replacing insulin do not be larger than usual and contain carbo- lation, children and adolescents with permit patients to mimic precisely the hydrate, protein, and fat. Frequent over- T1D may also be overweight or obese.17 exquisite complexity of the normal night blood glucose monitoring is For these children, exercise is a critical physiological adaptations to exercise. essential until sufficient experience has component of a weight management Consequently, the child with T1D fre- been obtained to appropriately modify strategy. Exercise ameliorates risk fac- quently experiences periods of either the evening dose of insulin after exer- tors (obesity, hypertension, and hyperli- 18 excessive or insufficient insulinaemia cise. pidaemia) for cardiovascular disease, http://adc.bmj.com/ during exercise. When plasma insulin In this issue, Massin et al report the but equally important, children with levels are relatively high, exercise causes resultsofanobservationalstudyofthe diabetes are likely to benefit from the blood glucose to decrease, whereas amount and intensity of physical exercise, enjoyment and enhanced feeling of self- when insulin levels are low, and espe- measured by 24 hour monitoring of heart worth derived from participation in cially if diabetes is poorly controlled, rate, in preschool, school age children, physical activity with their peers. vigorous exercise can aggravate hyper- and adolescents with T1D.8 The structured glycaemia and stimulate ketoacid pro- diabetes education programme at the Arch Dis Child 2005;90:1215–1217. duction.4 The child whose diabetes is out authors’ centre includes the recommen- doi: 10.1136/adc.2005.082446 on September 24, 2021 by guest. Protected copyright. of control (marked hyperglycaemia with dation to obtain regular physical activity. Correspondence to: Dr J I Wolfsdorf, Division of ketonuria) should not exercise until The message is reinforced by encouraging Endocrinology, Children’s Hospital Boston, 300 satisfactory glycaemic control has been regular physical exercise at clinic visits Longwood Avenue, Boston, MA 02115, USA; restored. and by attendance at diabetes camp [email protected] Exercise acutely lowers the blood where children learn about the effects of Competing interests: none declared glucose concentration to an extent that different types of physical activity on depends on its intensity and duration glycaemia. The study involved a ‘‘snap- and the concurrent level of insulinae- shot’’ of the lives of these children on a REFERENCES mia.2 In part, this results from acceler- single weekday, and it is possible that 1 Strong WB, Malina RM, Blimkie CJ, et al. ated insulin absorption from the subjects knowing that their physical Evidence based physical activity for school-age injection site owing to increased regio- activity was being measured affected the youth. J Pediatr 2005;146:732–7. nal blood flow and the massaging effect results. The majority of children with 2 Wasserman DH, Zinman B. Exercise in 5 individuals with IDDM. Diabetes Care of contracting limb musculature. If diabetes receiving care at this centre met 1994;17:924–37. exercise is planned, the preceding insu- the paediatric recommendations for phy- 3 Goodyear LJ, Kahn BB. Exercise, glucose lin dose should be reduced by 10–20% sical activity and compared favourably to transport, and insulin sensitivity. Annu Rev Med and the injection given in a site least their non-diabetic peers. The authors also 1998;49:235–61. 4 Berger M, Berchtold P, Cuppers HJ, et al. likely to be affected by exercise; for observed a significant inverse association Metabolic and hormonal effects of muscular example, the anterior abdominal wall in between mean annual glycated haemo- exercise in juvenile type diabetics. Diabetologia the morning preceding a sports event. globin and the amount of time spent in 1977;13:355–65. 5 Koivisto VA, Felig P. Effects of leg exercise on Because young children’s physical activ- light and moderate physical activity in insulin absorption in diabetic patients. ities tend to be spontaneous, this advice school age children. N Engl J Med 1978;298:79–83.

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6 MacDonald MJ. Postexercise late-onset children with insulin-dependent diabetes mellitus. 15 Huttunen NP, Lankela SL, Knip M, et al. hypoglycemia in insulin-dependent Diabetes Care 1984;7:57–62. Effect of once-a-week training program on diabetic patients. Diabetes Care 1987;10: 11 Arslanian S, Nixon PA, Becker D, et al. Impact of physical fitness and metabolic control in 584–8. physical fitness and glycemic control on in vivo children with IDDM. Diabetes Care 7 Tsalikian E, Beck R, Chase HP, et al. Impact of insulin action in adolescents with IDDM. Diabetes 1989;12:737–40. exercise on overnight glycemic control in children Care 1990;13:9–15. 16 Raile K, Kapellen T, Schweiger A, et al. Physical with type 1 diabetes (T1DM). Diabetes 12 Sackey AH, Jefferson IG. Physical activity and activity and competitive sports in children and 2005;54(suppl 1):A64. glycaemic control in children with diabetes adolescents with type 1 diabetes. Diabetes Care 8 Massin MM, Lebrethon M-C, Rocour D, et al. mellitus. Diabet Med 1996;13:789–93. 1999;22:1904–5. Patterns of physical activity determined by heart 13 Landt KW, Campaigne BN, James FW, et al. 17 Libman IM, Pietropaolo M, Arslanian SA, et al. rate monitoring among diabetic children. Arch Effects of exercise training on insulin sensitivity in Changing prevalence of overweight children and Dis Child 2005;90:1223–6. adolescents with type I diabetes. Diabetes Care adolescents at onset of insulin-treated diabetes. 9 Dunger DB. Diabetes in puberty. Arch Dis Child 1985;8:461–5. Diabetes Care 2003;26:2871–5. 1992;67:569–70. 14 Rowland TW, Swadba LA, Biggs DE, et al. 18 Austin A, Warty V, Janosky J, et al. The 10 Campaigne BN, Gilliam TB, Spencer ML, et al. Glycemic control with physical training in insulin- relationship of physical fitness to lipid and Effects of a physical activity program on dependent diabetes mellitus. Am J Dis Child lipoprotein(a) levels in adolescents with IDDM. metabolic control and cardiovascular fitness in 1985;139:307–10. Diabetes Care 1993;16:421–5.

Management of a conscious child, 80.7%; and resus- ...... citation of an unconscious child, 71.4%. However, irrespective of these prefer- ences, Boie et al found that if a child was Inter-hospital transport for children and likely to die, most parents would want to be present. Second, we know that their parent(s) there is likely to be a conceptual gap between what physicians think is appro- R C Tasker priate for parents to see and what parents consider is their choice to ...... decide. In the survey by Boie and colleagues,9 only 6.5% of parents wanted Commentary on the paper by Davies et al (see page 1270) the attending physician to determine their presence by their child. In a similar ach year, out of a child population because of the added burden of support- emergency department study, but this of 10.5 million in England and ing relatives during transport.5 The time surveying 645 emergency staff (306 EWales, approximately 10 000 need reality, however, is that the culture has physicians and 339 nurses) on views treatment in paediatric intensive care evolved to exclude parents—we have about six scenarios, Beckman et al found units (PICU).1 Almost half of these streamlined the transport process and it that almost half of the physicians children are transported between the avoids potential parental complications, believed that they alone (44%) should referring hospital and their regional by not having them there. The report by decide whether parents should be pre- PICU by a specialist team; currently, Davies and colleagues3 reminds us that, sent.10 This difference in viewpoint— the Department of Health recommends like other areas in acute paediatric care, between parents and physicians—is not

that parents should not routinely travel it is time to hear what parents feel and altogether unexpected given the cultural http://adc.bmj.com/ with their sick child in the ambulance.2 want, and now do something about it. history of our specialty: there was a time So, should we be allowing parents to If we trace the pathway of care from when parents were excluded from many accompany their critically ill child dur- acute presentation to later transfer to aspects of hospital paediatric care (for ing inter-hospital transport—or should the PICU, we already know much about example, bedside visiting for inpatients, they make their own way? In this issue, parents. First, in accident and emer- peri-operative transfer between the the PICU team from Guy’s Hospital gency practice there has been growing ward and the operating theatre, induc- report their experience of having the interest in letting them stay by their tion of anaesthesia, etc). Now, child and child’s parent accompany them during child when procedures are performed, or parent centred care is essential to what on September 24, 2021 by guest. Protected copyright. inter-hospital transport.3 An emphatic at least giving them the choice about it. we practice—that is, good medicine in ‘‘yes’’ comes from the South Thames For example in the 1980s, Bauchner et al the context of listening to patients’ and Acute Retrieval Service (STARS) that surveyed 253 parents and found that parents’ voices, openness, good commu- covers the south of England: they still 78% would want to be present should nication, and developing a relationship ‘‘continue to provide the service’’ and their child need a blood test or insertion based on trust. In essence, what we hope that their ‘‘results may inform of an intravenous catheter.6 In follow up should learn from the studies reported other services that are considering studies, the same authors found, first, by Boie and colleagues9 and Beckman adopting a similar policy’’. that parents chose to be present in 31 of and colleagues10 are the reasons under- In many respects it has been an error 50 (62%) such procedures,7 and second, lying the gap between 6.5% and 44%, in to have not considered, before now, the as a consequence, they were less parents and physicians respectively. question of parents accompanying their anxious and more satisfied with their Third, in children who are critically ill, critically ill child. Over 10 years ago the child’s care.8 More recently, in a survey transport to a regional PICU is often the American Academy of Pediatrics stated: of 400 parents presented with five next step after presentation to the ‘‘it is sometimes beneficial when trans- emergency department scenarios, Boie emergency room.11 12 Patients may well porting the anxious and sick child to et al found that parents exhibited a have undergone resuscitation and there have a parent accompany him or her in hierarchy or order in their preference.9 could be significant risk of adverse the transport vehicle’’.4 In our defence, They were less inclined to be present outcome. In 1995, Woodward and we could cite certain hurdles to pro- with more invasive procedures, which, Fleegler (from the Transport Services gress—concerns about accident insur- in decreasing order, were: venepunc- of the Children’s Hospital of ance for passengers, shortage of space in ture, 97.5%; suturing a laceration, 94%; Philadelphia) had a unique opportunity the ambulance, and staff anxiety lumbar puncture, 86.5%; resuscitation to survey two groups of parents: a group

www.archdischild.com 1218 PERSPECTIVES Arch Dis Child: first published as 10.1136/adc.2005.077057 on 21 November 2005. Downloaded from who had been allowed to accompany Davies et al tell us that their pilot Competing interests: none declared their child in an ambulance because study—permitting parents to accom- they had made a request to do so even pany their critically ill child during REFERENCES though ‘‘this option was not actively inter-hospital transfer—was conducted encouraged’’ (n = 46), and a group who in response to the distress expressed by 1 Paediatric Intensive Care Audit Network. Annual Report 2003–2004, Universities of Leeds, did not accompany their child parents, and now, because of their Leicester and Sheffield, May 2005 (ISBN 0 85316 (n = 40).13 Eighty six per cent of these experience, it has become the estab- 254 9). parents felt accompanying their child lished practice of the STARS team.3 2 Department of Health. A framework for the future: report from the national co-ordinating was important. Prior to transfer, 74% There are at least 14 other regional group on paediatric intensive care to the chief were either worried or very worried PICU transport services in England and executive of the NHS executive. London: The about their child’s stability. Only 5% of Wales—how should we respond and Stationary Office, 1997. 3 Davies J, Tibby SM, Murdoch IA. Should parents parents accompanying their child found move on? Should we determine whether accompany critically ill children during inter- that doing so made them anxious or there is national consensus regarding hospital transport? Arch Dis Child very anxious, whereas 56% of those who involving parents in inter-hospital 2005;90:1270–3. 4 American Academy of Pediatrics Taskforce on did not accompany their child felt transport—and only act if there is? The Interhospital Transport. Guidelines for air and anxious or very anxious (p , 0.05). present standards of the United ground transport of neonatal and pediatric Finally, 94% of parents would choose Kingdom Paediatric Intensive Care patients. Elk Grove Village, IL: The Academy, 12 1993:74. to travel with their child in a future Society (1996 and 2001) do not cover 5 Hill Y. Is there a place for parents in the retrieval similar circumstance. These findings are this issue. Rather, they state that it is of critically sick children? Nursing in Critical Care not dissimilar to those found in the the referring hospital that is ‘‘obligated 1999;4:121–7. 6 Bauchner H, Waring C, Vinci R. Pediatric south of England in 2003. Colville et al to provide transport to the Lead Centre procedures: do parents want to watch? Pediatrics surveyed 233 parents, of whom only 13 (that is, regional PICU) for parents’’. 1989;84:907–8. had been allowed to travel with their Even in the United States, there did not 7 Bauchner H, Waring C, Vinci R. Parental presence during procedures in an emergency room: results child in the ambulance: in total, 70% of appear to be consensus among paedia- from 50 observations. Pediatrics 1991;87:544–8. these parents commented on the jour- tric critical care transport team man- 8 Bauchner H, Vinci R, Bak S, et al. Parents and ney they made to the PICU, often agers in 2001.15 Only 63% of teams procedures: a randomized controlled trial. Pediatrics 1996;98:861–7. travelling alone using their own trans- allowed parents to accompany their 9 Boie ET, Moore GP, Brummett C, et al. Do parents port.14 A recurring theme in what the child in the ambulance, but the authors want to be present during invasive procedures parents had to say was their sense of from Philadelphia (see also Woodward performed on their children in the emergency 13 department? A survey of 400 parents. Ann Emerg separation and the distress this journey and Fleegler ) took the view that Med 1999;34:70–4. caused them. Three quotations, relayed ‘‘awareness of this issue may help to 10 Beckman AW, Sloan BK, Moore GP, et al. Should by the authors, capture the very essence establish discussion and guidelines parents be present during emergency department procedures on children, and who should make the of what these parents are feeling, and regarding the role of parents’’. In other decision? A survey of emergency physician and we should not forget this: ‘‘the worst words, lack of unanimity should not nurse attitudes. Acad Emerg Med 2002;9:154–8. journey of our lives’’, ‘‘the worst part detract from this potential develop- 11 Britto J, Nadel S, Maconochie I, et al. Morbidity and severity of illness during interhospital was seeing the ambulance disappearing ment—it merely indicates the extent of transfer: impact of a specialized paediatric in the distance’’, and ‘‘for all I knew she change in practice that is needed if we retrieval team. BMJ 1995;311:836–9. was dying and I wasn’t allowed to be are to respond to what parents need. 12 Paediatric Intensive Care Society. Standards for paediatric intensive care, Saldatore Ltd, with her’’. In this issue, the report by There is now a literature showing that Hertfordshire, 1996 and 2001. 3 Davies and colleagues focuses on logis- the most helpful coping strategy for 13 Woodward GA, Fleegler EW. Should parents http://adc.bmj.com/ tics, staff perceptions, and adverse parents of PICU patients is to allow accompany pediatric interfacility ground ambulance transports? The parent’s perspective. events—and that is entirely right given them to stay with their child and to Pediatr Emerg Care 2000;16:383–90. the newness of this service development empower them.16–18 Inter-hospital trans- 14 Colville G, Orr F, Gracey D. ‘‘The worst journey in the south of England. However, port should be no different. However, if of our lives’’: parents’ experience of specialized paediatric retrieval service. Intensive Crit Care another—albeit less emphasised—key we cannot extrapolate the conclusion of Nurs 2003;19:103–8. message emerges from this work. The the STARS service to our own regional 15 Woodward GA, Fleegler EW. Should parents authors’ interpretation of parental feed- practices, then we should at least look at accompany pediatric interfacility ground

ambulance transports? Results of a national on September 24, 2021 by guest. Protected copyright. back (presumably compared with pre- alternative ways of better supporting survey of pediatric transport team managers. vious experience in the south of parents during the time it takes for them Pediatr Emerg Care 2001;17:22–7. England13) was that these parents were to travel from the accident and emergency 16 Miles MS, Carter MC. Sources of parental stress in pediatric intensive care units. Child Health Care less stressed as a consequence of accom- department to the regional PICU. 1983;11:65–9. panying their child. These audit data Arch Dis Child 2005;90:1217–1218. 17 Melynk BM, Alpert-Gillis LJ, Hensel PB, et al. need to be followed up with further Helping mothers cope with a critically ill child: a doi: 10.1136/adc.2005.077057 pilot test of the COPE intervention. Res Nurs clinical studies. For example, do parents Health 1997;20:3–14. who travel with their children experi- Correspondence to: Dr R C Tasker, University of 18 Melnyk BM, Alpert-Gillis L, Feinstein NF, et al. ence less acute stress disorder? Are they Cambridge School of Clinical Medicine, Creating opportunities for parent empowerment: Department of Paediatrics, Box 116, program effects on the mental health/coping at less risk of post-traumatic stress Addenbrooke’s Hospital, Hills Road, outcomes of critically ill young children and their disorder? Cambridge CB2 2QQ, UK; [email protected] mothers. Pediatrics 2004;116:e597–607.

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