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Journal of Clinical Endocrinology & Metabolism , doi:10.1210/jc.2004-1395
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The Journal of Clinical Endocrinology & Metabolism Vol. 90, No. 4 1992-1997
Copyright © 2005 by The Endocrine Society

Quality of Life Determinants in Young Women with Turner’s Syndrome after Growth Hormone Treatment: Results of the StaTur Population-Based Cohort Study

Jean-Claude Carel, Emmanuel Ecosse, Irène Bastie-Sigeac, Sylvie Cabrol, Maïté Tauber, Juliane Léger, Marc Nicolino, Raja Brauner, Jean-Louis Chaussain and Joël Coste

Department of Pediatric Endocrinology and Institut National de la Santé et de la Recherche Médicale Unité 561 (J.-C.C., J.-L.C., I.B.-S.) and Department of Biostatistics (E.E., J.C.), Groupe Hospitalier Cochin-Saint Vincent de Paul and Faculté de Médecine René Descartes Paris 5, 75674 Paris, France; Department of Pediatric Endocrinology (S.C.), Hôpital Armand Trousseau, 75571 Paris, France; Department of Pediatric Endocrinology (M.T.), Hôpital des Enfants, 31059 Toulouse, France; Department of Pediatric Endocrinology (J.L.), Hôpital Robert Debré, 75935 Paris, France; Department of Pediatric Endocrinology (M.N.), Hôpital Debrousse, 69322 Lyon, France; and Department of Pediatric Endocrinology (R.B.), Hôpital de Bicêtre, 94275 Le Kremlin Bicêtre, France

Address all correspondence and requests for reprints to: Professor Jean-Claude Carel, Pediatric Endocrinology and Institut National de la Santé et de la Recherche Médicale Unité 561, Groupe Hospitalier Cochin-Saint Vincent de Paul, 82 av Denfert Rochereau, 75014 Paris, France. E-mail: carel{at}paris5.inserm.fr.


   Abstract
Top
 Abstract
Introduction
 Subjects and Methods
 Results
 Discussion
 References
 
GH is used to increase adult height in children with Turner’s syndrome with little knowledge of the impact on quality of life. We carried out a population-based cohort study of quality-of-life determinants in young women with Turner’s syndrome, all previously treated with GH. Of 891 eligible women aged over 18 yr and recorded in the French Growth Hormone Register, 818 were available and 568 participated (69%). They were assessed for demographic characteristics, health status, sexual life, treatment expectations, scores for Medical Outcome Study Short Form 36 (SF-36), and General Health Questionnaire 12. Participants were 22.6 ± 2.6 yr old (mean ± SD), measured 150.9 ± 5.6 cm, and had received GH for 4.8 ± 2.2 yr. SF-36 scores were similar in participants and French women of the general population. Cardiac (12% of participants) or otological (26% of participants) involvement or induction of puberty after 15 yr of age was associated with lower scores for at least one of the SF-36 dimensions. Height and estimated height gain from treatment were not associated with quality-of-life scores. Higher expectations from treatment were associated with lower quality of life. We conclude that quality of life is normal and unaffected by height in young adults with Turner’s syndrome treated with GH. These data emphasize the need to give appropriate attention to general health and otological care rather than focus on stature in the care of children with Turner’s syndrome.


   Introduction
Top
 Abstract
 Introduction
Subjects and Methods
 Results
 Discussion
 References
 
TURNER’S SYNDROME AFFECTS 1 in 2500 females and results from the total or partial absence of one of the X chromosomes (1, 2). The principal features of Turner’s syndrome are short stature (adult height about 20 cm below the mean for women of the corresponding ethnic group) and dysfunctional gonads resulting in a lack of, or incomplete, pubertal development and infertility. Other features include cardiac and renal malformations and otological problems leading to hearing impairment and various degrees of dysmorphic features. Females with Turner’s syndrome are short at birth and display growth retardation in all phases of growth, particularly late childhood and adolescence, but have no GH deficiency. Several studies have reported increases in adult heights after GH treatment (3, 4, 5, 6), although there is some debate as to the magnitude of these increases (7, 8, 9). GH is approved for use in children with Turner’s syndrome in most industrialized countries and is recommended in recently issued guidelines (9).

Treatments to promote growth in girls with Turner’s syndrome aim to reduce the impact of short stature on psychosocial functioning and quality of life. However, these aspects have not been evaluated in young adults after treatment completion. Several small studies have reported changes in psychosocial functioning and quality of life in adolescents and women with Turner’s syndrome (reviewed in Refs. 2 and 10). However, the effects on psychosocial functioning of height or height gain from GH treatment remain unclear (11, 12).

In this study, we aimed to evaluate the determinants of quality of life, by means of standardized questionnaires, in all young women with Turner’s syndrome who had been treated with GH in France. These women were treated during the 1990s and received GH regimens similar to those currently recommended for Turner’s syndrome.


   Subjects and Methods
Top
 Abstract
 Introduction
 Subjects and Methods
Results
 Discussion
 References
 
We included all patients with a diagnosis of Turner’s syndrome, based on karyotype analysis, who had been treated with GH in France during the study period and who were obligatorily registered in the Association France-Hypophyse database (13). We then selected patients who were over the age of 18 yr on March 1, 2001. Of the 891 eligible patients, 73 were lost to follow-up or unable to answer the questionnaire, and 818 received the questionnaire (Fig. 1Go). A total of 568 (69%) responded (participants); the remaining 250 did not complete the questionnaire, despite several contacts (nonparticipants). The study was approved by the Departement de la Recherche Clinique et du Développement of Assistance Publique-Hôpitaux de Paris and was considered by their judicial department as requiring no informed consent because on-treatment data had been collected as part of a national mandatory program and patients were free to respond to the questionnaire.



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  		FIG. 1. Distribution of the patients.

 
Data collection

Data relating to growth and features associated with Turner’s syndrome were collected before GH treatment and at follow-up visits (every 3–6 months) by the treating physicians. Karyotype, dysmorphic features, cardiac and renal malformations, associated morbidity, height, weight, age, bone age, pubertal stage, GH dose, frequency of injections, and associated treatments were analyzed (13). Height and weight were expressed as SD scores (SDSs), based on normative general population data (13) and Turner’s syndrome data (14). We estimated height gain from GH treatment by comparing attained and projected height, using Turner’s syndrome normative data (14).

In 2001 a postal survey was sent to all eligible patients. This survey included the validated French version of Medical Outcome Study Short Form 36 (SF-36) (15) the General Health Questionnaire 12 (GHQ-12) (16) and dealt with demographic characteristics, current health status (morbidity, medication), sexual intercourse experience, and expectations from GH treatment (patients were asked to give the minimum height gain they considered worthwhile for the further use of GH in Turner’s syndrome). The SF-36 contains eight subscales: general health (five items), physical functioning (10 items), limitations on usual role-related activities due to physical health problems (four items), bodily pain (two items), energy and fatigue (vitality, four items), limitations on usual role-related activities due to emotional or mental problems (three items), social functioning (two items), and emotional or mental health (five items). Scores for all eight subscales range from 0 to 100, with higher scores indicating better health or function. All items refer to the subject’s functioning/status during the past 4 wk, except those for physical functioning and general health, which relate to the time at which the form is completed. We used the 12-item version of the general health questionnaire as a measure of psychological distress. Scores on this questionnaire range from 0 to 12, with a score of 3 or more identifying a probable case of minor psychiatric disorder such as anxiety or depression (16).

GH treatment

Treatment was generally initiated on the diagnosis of Turner’s syndrome. However, for the older patients, treatment was initiated when GH became widely available, in the early 1990s. The criteria for treatment discontinuation were a growth rate less than 3 cm/yr or a bone age of 13 yr or more.

Analysis of quality of life and statistical methods

The scores for the SF-36 scales are presented as absolute scores and SDSs, calculated by dividing the difference between the patient’s score and the mean of the general population of the same sex and age group by the SD of the general population group. French general population reference values for women aged 18–24 yr were used (17). Pearson correlation coefficient was used to correlate GHQ-12 score (12 point scale) with the eight SF-36 scores (100-point scales).

Multiple regression models were constructed to identify factors associated with SF-36 scale (absolute) scores. These models included paternal socioprofessional class, educational level, and GHQ-12 scores (classified as high if the score was ≥ 3 or lower). Some of the scores for SF-36 scales were skewed, and residual plots from the corresponding regression analyses were checked for departure from normality. The {chi}2 test (simple goodness of fit test) was used to compare the proportion of cases with GHQ-12 score of 3 or more with that for the general population. Calculations were performed with SAS software (18).


   Results
Top
 Abstract
 Introduction
 Subjects and Methods
 Results
Discussion
 References
 
Characteristics of the subjects and treatments (Table 1Go)

Eligible patients (participants and nonparticipants) had a mean age of 12 ± 2.5 yr at the start of treatment and used GH for a mean of 4.7 yr, at a mean dose of 0.8 IU/kg·wk (0.27 mg/kg·wk), administered as six or seven injections per week. They stopped treatment at the age of 16.7 ± 1.6 yr and received the questionnaire a mean of 6 yr later, at the age of 22.6 ± 2.6 yr. In the 72% of patients who had no spontaneous pubertal development, estrogen treatment was started at a mean age of 15 yr. Most of the remaining patients required estrogen treatment at a later stage to complete their pubertal development. The mean adult height for the whole group (participants and nonparticipants) was 150 ± 6.1 cm (range 127–167 cm). Participants were a mean of 2.7 cm taller than nonparticipants (P < 0.005). Participants and nonparticipants were similar in all other characteristics.


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  		TABLE 1. Clinical characteristics of patients with Turner’s syndrome included in this study

 
Quality of life and its determinants

SF-36 scores in the 568 women with Turner’s syndrome were similar to those of women of the same age from the general population (Fig. 2Go). If expressed as SDSs, these scores did not differ significantly from zero. The proportion of women with Turner’s syndrome who had a high GHQ-12 score (≥3) was lower than that for the general population (24 vs. 31%, respectively, P < 0.001).



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  		FIG. 2. Quality-of-life measurements in Turner’s syndrome patients. Results are shown as absolute scores for patients with Turner’s syndrome (hashed bars) or for French women from the general population aged 18–24 yr (dotted bars) (A) or as SDS using data obtained for French women from the general population aged 18–24 yr (B); means ± SD are shown.

 
We assessed the effect of several of the patients’ characteristics on SF-36 scores (Table 2Go). The father’s socioprofessional background (manual vs. nonmanual) and the patient’s level of education (secondary education completed or not) were associated with several quality-of-life dimensions. GHQ-12 scores were correlated with SF-36 scores, in particular those reflecting psychosocial dimensions with the following correlation coefficients: –0.22 (physical functioning), –0.29 (general health), –0.37 (bodily pain), –0.39 (role limitation, physical), –0.43 (vitality), –0.59 (role limitation, emotional), –0.62 (social functioning), and –0.66 (mental health). Further analyses were performed after adjustment for these three potentially confounding variables (paternal socioprofessional status, participant’s educational level, and GHQ-12 score). Adult height was not associated with quality of life, regardless of whether height was treated as a continuous variable or broken down into categories, as in Table 2Go. Similarly, duration of treatment, age at treatment initiation, total GH dose, and estimated height gain were not associated with quality of life. The patients who had the highest expectations regarding GH treatment had the lowest quality-of-life scores. Cardiac involvement was strongly associated with low physical scores, whereas kidney and genital malformations associated with the presence of Y chromosome material were not. Unexpectedly, otological involvement, present in 26% of the patients and either detected during childhood care or declared by the patients at the time of the survey, was strongly associated with perceived health-related quality of life in all but one dimension. Otological conditions resulted in a loss of 4–13 score points or 0.2–0.7 SDS units. Patients whose puberty had been induced after the age of 15 yr had significantly lower general health perception scores. Other factors were analyzed and found not to be associated with quality of life as assessed by the SF-36 questionnaire: karyotype (either in two categories as in Table 2Go or using subgroups as in Table 1Go), dysmorphic features of Turner’s syndrome, sexual intercourse experience, presence of thyroid dysfunction, or self-reported visual problems.


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  		TABLE 2. Determinants of quality of life in patients with Turner’s syndrome

 

   Discussion
Top
 Abstract
 Introduction
 Subjects and Methods
 Results
 Discussion
References
 
Our main findings, in young women with Turner’s syndrome treated with GH who had a mean gain of 8.9 cm, compared with projected height were: 1) perceived health-related quality of life, as measured by the SF-36 questionnaire, was not statistically different from the reference values obtained for young French women of the general population; 2) height and other variables associated with GH treatment did not affect perceived health-related quality of life, with the exception of unrealistic expectations about GH; and 3) other features associated with Turner’s syndrome, in particular otological conditions, had a profound impact on perceived health. These findings strongly suggest that priorities should be redefined in the care of these patients during childhood.

This is, to our knowledge, the first study addressing the question of quality of life in patients with Turner’s syndrome using standardized questionnaires in a register-based population. The use of a standardized questionnaire made it possible to compare patients’ scores with those obtained recently for French women of the same age from the general population (17). Several limitations of our study should be pointed out. First, the 31% of patients who did not participate might have different quality-of-life characteristics from those who did and being on average 2.7 cm shorter might be of greater concern to them than to those who participated. Although this cannot be dismissed, we believe it is unlikely, given the lack of influence of height on quality of life in the rest of the population. Moreover, the response rate of 69% to an unsolicited questionnaire assessing personally intimate information is high and well within acceptable standards for similar surveys. Second, it could be argued that the SF-36 scale used was not sensitive enough to detect small changes in quality of life and that we might have missed important factors, such as height. Although we cannot exclude this possibility, we were able to detect several effects, including those of paternal socioprofessional background, educational level, and the presence of heart malformations or otological conditions. However, the SF-36 scales were not designed to detect the influence of height and the use of syndrome-specific quality-of-life measures addressing the relation between height and ability for daily life activities might have given different results. Altogether, the SF-36 has been used successfully in a variety of similar studies (19, 20). Third, the design of our study, in GH-treated patients, focused on young adults with this condition, and our conclusions may not be applicable to other age groups, in particular adolescents or older adults. It would have been interesting to study untreated women with Turner’s syndrome of a similar age. However, since the early 1990s, almost all children diagnosed with Turner’s syndrome are treated with GH in accordance with current international recommendations (9). Fourth, all data were obtained from routine examination in daily practice, and their reliability may be questioned. Last, whether our findings are generalizable to cultural contexts other than France remains to be established.

Several studies have assessed quality-of-life or psychosocial parameters in adults or youths with Turner’s syndrome (reviewed in Refs. 2 and 10). Most have concluded that self-image or social functioning is low (10, 21). Depression scores were high (22, 23) or low (24). However, the recruitment of these patients from medical clinics or patient support organizations certainly introduced biases. In children and adolescent girls with Turner’s syndrome, studies assessing the psychological benefits of increasing height by GH therapy have given inconsistent results (11, 12).

Our results have important implications for the care of children with Turner’s syndrome and, more generally, with conditions that limit growth. In Turner’s syndrome, height and the use of GH has been a major focus of health providers during the past 15 yr. Indeed, the patients studied here had a mean height of 150 cm, 6–8 cm greater than historical data on French Turner’s syndrome patients (14). A long-term untreated contemporary control would have been needed for an optimal assessment of the influence of GH on health-related quality of life. However, the variance of height of our patients (95% between 139 and 161 cm) was three times greater than the estimate of height gain by GH. Another possibility is that the intervention itself (GH treatment) rather than its result (height increment) might have improved perceived health-related quality of life, therefore obscuring the influence of height itself. However, given the limitation indicated above, we found no evidence suggesting that interventions that increase heights by 5–10 cm have an effect on quality of life. In addition, the lower quality of life of patients with higher expectations from GH treatment indicates a long-term negative impact of unduly high expectations, as previously suggested (25). Finally, we found that quality of life was significantly lower in patients with otological disorders. Otological disorders are common in Turner’s syndrome, affecting up to 70% of patients, and lead to hearing impairment in up to 90% of adult patients (2, 26, 27). In a placebo-controlled study (6) and in the Canadian long-term randomized, controlled trial (28), GH use was associated with an increase in frequency of otitis media by a factor of two or more. Our results therefore indicate that higher priority should be given to the care of otological problems in childhood and the prevention of their long-term consequences in adulthood.

In young adults with short stature, height has no detectable influence on quality of life (29). GH is increasingly used in GH-sufficient individuals with short stature, but quality of life has not been measured as an end point of these pharmacological interventions. Current estimates indicate adult height gains of 3.7–6 cm (30, 31, 32) that are unlikely to influence the quality of life in such individuals with short stature.

We conclude that quality of life, as assessed by SF-36, is normal and unaffected by height in young adults with Turner’s syndrome treated with GH. However, in the absence of a contemporaneous group of untreated patients with Turner’s syndrome, a beneficial effect of GH treatment irrespective of adult height attained cannot be ruled out. In that regard, our data raise the question as to whether GH treatment is justified in this patient population, given its constraints, costs, and potential side effects (33, 34, 35). Moreover, our results clearly indicate that greater attention should be paid to otological care and other specific needs in these patients, given the negative impact of otological problems on daily life. These findings also call into question the use of GH to treat other conditions involving short stature, associated with a specific diagnosis or idiopathic, for which GH is currently used without evaluation of the psychosocial impact of these demanding and costly treatments. Above all, our findings suggest that for current and future indications for GH treatment, research into the overall impact of pharmacological intervention is necessary.


   Acknowledgments
 
We are grateful for the invaluable contributions of Vean Eng Ly and Sabine Ximenes. Drs. Noel Cabet, Mireille Castanet, Catherine Fernando, Nadia Lounis, and Caroline Thalassinos also participated in the collection of the data. In addition, we thank all the physicians involved in the follow-up of patients and the review process at Association France-Hypophyse. The following clinicians were involved in the follow-up of a significant number of children in the study: Pascal Barat, Sabine Baron, Pascale Berlier, Philippe Bernard, Anne-Marie Bertrand, Hélène Bony-Trifunovic, Michel Bost, Bernard Boudailliez, Raja Brauner, Maryvonne Bouchard, Sylvie Cabrol, Jean Caron, Jean-Pierre Charvet, Pierre Chatelain, Jean-Louis Chaussain, Maurice Chupin, Michel Colle, Jean-Benoît Cotton, Paul Czernichow, Albert David, Michel David, Marc De Kerdanet, Francois Despert, Pierre Doyard, Christine Fedou, Anne Fjellestad-Paulsen, Patrick Garandeau, Philippe Garnier, Frédéric Huet, Jean-Claude Job, Michel Lecornu, Juliane Léger, Bruno Leheup, Anne Lienhardt, Jean-Marie Limal, Guy-André Loeuille, Eric Mallet, Georges Malpuech, Roger Mariani, Chantal Metz, Catherine Naud-Saudreau, Jean-Louis Nivelon, Sylvie Nivot, Christian Pauwels, Marc Pétrus, Catherine Pienkowski, Olivier Puel, Odile Richard, Pierre Rochiccioli, Gilbert Simonin, Sylvie Soskin, Véronique Sulmont, Charles Sultan, Maïté Tauber, Jean-Edmond Toublanc, Kathy Wagner, Jacques Weill, and Catherine Wémeau.


   Footnotes
 
This work was supported by a grant from Ministère de la Santé, Programe Hospitalier de Recherche Clinique (AOM98009.

First Published Online January 11, 2005

Abbreviations: GHQ-12, General Health Questionnaire 12; SDS, SD score; SF-36, Medical Outcome Study Short Form 36.

Received July 16, 2004.

Accepted January 4, 2005.


   References
Top
 Abstract
 Introduction
 Subjects and Methods
 Results
 Discussion
 References
 

  1. Sybert VP, McCauley E 2004 Turner’s syndrome. N Engl J Med 351:1227–1238[Free Full Text]
  2. Elsheikh M, Dunger DB, Conway GS, Wass JA 2002 Turner’s syndrome in adulthood. Endocr Rev 23:120–140[Abstract/Free Full Text]
  3. Ranke MB, Saenger P 2001 Turner’s syndrome. Lancet 358:309–314[CrossRef][Medline]
  4. Carel JC, Mathivon L, Gendrel C, Ducret JP, Chaussain JL 1998 Near normalisation of final height with adapted doses of growth hormone in Turner’s syndrome. J Clin Endocrinol Metab 83:1462–1466[Abstract/Free Full Text]
  5. van Pareren YK, de Muinck Keizer-Schrama SM, Stijnen T, Sas TC, Jansen M, Otten BJ, Hoorweg-Nijman JJ, Vulsma T, Stokvis-Brantsma WH, Rouwe CW, Reeser HM, Gerver WJ, Gosen JJ, Rongen-Westerlaken C, Drop SL 2003 Final height in girls with Turner syndrome after long-term growth hormone treatment in three dosages and low dose estrogens. J Clin Endocrinol Metab 88:1119–1125[Abstract/Free Full Text]
  6. Quigley CA, Crowe BJ, Anglin DG, Chipman JJ 2002 Growth hormone and low dose estrogen in Turner syndrome: results of a United States multi-center trial to near-final height. J Clin Endocrinol Metab 87:2033–2041[Abstract/Free Full Text]
  7. Taback SP, Collu R, Deal CL, Guyda HJ, Salisbury S, Dean HJ, Van Vliet G 1996 Does growth-hormone supplementation affect adult height in Turner’s syndrome? Lancet 348:25–27[CrossRef][Medline]
  8. Donaldson MD 1997 Growth hormone therapy in Turner syndrome—current uncertainties and future strategies. Horm Res 48(Suppl 5):35–44
  9. Saenger P, Wikland KA, Conway GS, Davenport M, Gravholt CH, Hintz R, Hovatta O, Hultcrantz M, Landin-Wilhelmsen K, Lin A, Lippe B, Pasquino AM, Ranke MB, Rosenfeld R, Silberbach M 2001 Recommendations for the diagnosis and management of Turner syndrome. J Clin Endocrinol Metab 86:3061–3069[Abstract/Free Full Text]
  10. Boman UW, Moller A, Albertsson-Wikland K 1998 Psychological aspects of Turner syndrome. J Psychosom Obstet Gynaecol 19:1–18[Medline]
  11. Lagrou K, Xhrouet-Heinrichs D, Heinrichs C, Craen M, Chanoine JP, Malvaux P, Bourguignon JP 1998 Age-related perception of stature, acceptance of therapy, and psychosocial functioning in human growth hormone-treated girls with Turner’s syndrome. J Clin Endocrinol Metab 83:1494–1501[Abstract/Free Full Text]
  12. Siegel PT, Clopper R, Stabler B 1998 The psychological consequences of Turner syndrome and review of the National Cooperative Growth Study psychological substudy. Pediatrics 102:488–491[Abstract/Free Full Text]
  13. Carel JC, Ecosse E, Nicolino M, Tauber M, Leger J, Cabrol S, Bastié-Sigeac I, Chaussain JL, Coste J 2002 Adult height after long-term recombinant growth hormone treatment for idiopathic isolated growth hormone deficiency: observational follow-up study of the French population-based registry. BMJ 325:70–73[Abstract/Free Full Text]
  14. Sempé M 1997 Dysgénésie gonadique féminine non traitée (syndrome de Turner et syndromes apparentés). Références françaises de 0 à 22 ans. Ann Pediatr (Paris) 44:529–537
  15. Leplege A, Ecosse E, Verdier A, Perneger TV 1998 The French SF-36 Health Survey: translation, cultural adaptation and preliminary psychometric evaluation. J Clin Epidemiol 51:1013–1023[CrossRef][Medline]
  16. Goldberg DP 1972 The detection of psychiatric illness by questionnaire. London: Oxford University Press
  17. Leplege A, Ecosse E, Coste J, Pouchot J, Perneger TV 2001 Le questionnaire MOS SF-36. Manuel de l’utilisateur et guide d’interprétation des scores. Paris: ESTEM
  18. 1990 SAS user’s guide statistics. 6th ed. Cary, NC: SAS Institute Inc.
  19. Thomas KS, Muir KR, Doherty M, Jones AC, O’Reilly SC, Bassey EJ 2002 Home based exercise programme for knee pain and knee osteoarthritis: randomised controlled trial. BMJ 325:752[Abstract/Free Full Text]
  20. Hays J, Ockene JK, Brunner RL, Kotchen JM, Manson JE, Patterson RE, Aragaki AK, Shumaker SA, Brzyski RG, LaCroix AZ, Granek IA, Valanis BG 2003 Effects of estrogen plus progestin on health-related quality of life. N Engl J Med 348:1839–1854[Abstract/Free Full Text]
  21. McCauley E, Feuillan P, Kushner H, Ross JL 2001 Psychosocial development in adolescents with Turner syndrome. J Dev Behav Pediatr 22:360–365[Medline]
  22. McCauley E, Sybert VP, Ehrhardt AA 1986 Psychosocial adjustment of adult women with Turner syndrome. Clin Genet 29:284–290[Medline]
  23. Pavlidis K, McCauley E, Sybert VP 1995 Psychosocial and sexual functioning in women with Turner syndrome. Clin Genet 47:85–89[Medline]
  24. Downey J, Ehrhardt AA, Gruen R, Bell JJ, Morishima A 1989 Psychopathology and social functioning in women with Turner syndrome. J Nerv Ment Dis 177:191–201[Medline]
  25. Rotnem D, Cohen DJ, Hintz R, Genel M 1979 Psychological sequelae of relative "treatment failure" for children receiving human growth hormone replacement. J Am Acad Child Psychiatry 18:505–520[Medline]
  26. Hultcrantz M, Sylven L 1997 Turner’s syndrome and hearing disorders in women aged 16–34. Hear Res 103:69–74[CrossRef][Medline]
  27. Gravholt CH, Juul S, Naeraa RW, Hansen J 1998 Morbidity in Turner syndrome. J Clin Epidemiol 51:147–158[CrossRef][Medline]
  28. Canadian Growth Hormone Advisory Committee 1998 Growth hormone treatment to final height in Turner syndrome: a randomized controlled trial. Horm Res 50(Suppl 3):25
  29. Sandberg DE, Voss LD 2002 The psychosocial consequences of short stature: a review of the evidence. Best Pract Res Clin Endocrinol Metab 16:449–463[CrossRef][Medline]
  30. Finkelstein BS, Imperiale TF, Speroff T, Marrero U, Radcliffe DJ, Cuttler L 2002 Effect of growth hormone therapy on height in children with idiopathic short stature: a meta-analysis. Arch Pediatr Adolesc Med 156:230–240[Abstract/Free Full Text]
  31. Carel JC, Chatelain P, Rochiccioli P, Chaussain JL 2003 Improvement in adult height after growth hormone treatment in adolescents with short stature born small for gestational age: results of a randomized controlled study. J Clin Endocrinol Metab 88:1587–1593[Abstract/Free Full Text]
  32. Leschek EW, Rose SR, Yanovski JA, Troendle JF, Quigley CA, Chipman JJ, Crowe BJ, Ross JL, Cassorla FG, Blum WF, Cutler Jr GB, Baron J 2004 Effect of growth hormone treatment on adult height in peripubertal children with idiopathic short stature: a randomized, double-blind, placebo-controlled trial. J Clin Endocrinol Metab 89:3140–3148[Abstract/Free Full Text]
  33. Sperling MA, Saenger PH, Ray H, Tom W, Rose SR 2002 Growth hormone treatment and neoplasia-coincidence or consequence? J Clin Endocrinol Metab 87:5351–5352[Free Full Text]
  34. Juul A, Bernasconi S, Carel JC, Clayton PE, Kiess W, DeMuinck-Keizer Schrama S 2003 Growth hormone treatment and risk of solid tumours. A statement from the Drugs and Therapeutics Committee of the European Society for Paediatric Endocrinology (ESPE). Horm Res 60:103–104
  35. Wilson TA, Rose SR, Cohen P, Rogol AD, Backeljauw P, Brown R, Hardin DS, Kemp SF, Lawson M, Radovick S, Rosenthal SM, Silverman L, Speiser P 2003 Update of guidelines for the use of growth hormone in children: the Lawson Wilkins Pediatric Endocrinology Society Drug and Therapeutics Committee. J Pediatr 143:415–421[CrossRef][Medline]



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Growth Hormone in Turner Syndrome: Twenty Years after, What Can We Tell our Patients?
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