Abstract
Treatment of congenital adrenal hyperplasia (CAH) in its salt-wasting form with appropriate
doses of glucocorticoids and mineralocorticoids should promote growth, puberty and
final height in a similar to normal pattern. However, the individual requirements
for these drugs to normalize the hormonal profile and to achieve a physiologic growth
pattern may differ. Moreover, the time of onset of puberty is also unpredictable since
the course of the disease may predispose for precocity. The aim of this study was
to explain the unexpected arrest of growth during puberty in a boy with late-onset
CAH, who had been treated with glucocorticoid from early childhood. A short course
of GnRH agonist treatment was also introduced in later years. The growth chart reflects
two periods of impaired growth velocity preceded by weight loss. The reason for the
first decline is difficult to prove retrospectively, but during the second episode
the boy became both clinically and hormonally hypogonadal. At that time the anorexia
nervosa (AN) was diagnosed according to APA DSM-IV criteria. We conclude that there
were several reasons for the discontinued growth spurt and reduced final height in
this boy with CAH: (a) early variant of puberty and subsequent late treatment with
GnRH agonist, (b) AN possibly occurring during mid-childhood and clearly during puberty
and (c) the repeated use of high doses of glucocorticoids. AN, a relatively rare disorder
in boys, appears to have had a significant negative effect on this patient's growth
and should be considered in the differential diagnosis in CAH children with impaired
growth.
Key words
growth disorder - congenital adrenal hyperplasia - anorexia nervosa - precocity
References
- 1
American Psychiatric Association
.Diagnostic and Statistical Manual of mental disorders.. 4th ed (DSM-IV) Washington,
DC: American Psychiatric Association; 1994
- 2
Andersen AE, Holman JE.
Males with eating disorders: challenges for treatment and research.
Psychopharmacol Bull.
1997;
33
391-397
- 3
Argente J, Caballo N, Barrios V. et al .
Multiple endocrine abnormalities of the growth hormone and insulin-like growth factor
axis in patients with anorexia nervosa: effect of short- and long-term weight recuperation.
J Clin Endocrinol Metab.
1997;
82
2084-2092
- 4
Barrios V, Argente J, Munoz MT. et al .
Diagnostic interest of acid-labile subunit measurement in relationship to other components
of the IGF system in pediatric patients with growth or eating disorders.
Eur J Endocrinol.
2001;
144
245-250
- 5
Barry A, Lippmann SB.
Anorexia nervosa in males.
Postgrad Med.
1990;
87
161-165
- 6
Bayley N, Pinneau SR.
Tables for predicting adult height from skeletal age: revised for use with the Greulich-Pyle
hand standards.
J Pediatr.
1952;
40
423-441
- 7
Boys IGF-I normal range
.
IGF-I normogram developed by Diagnostic Systems Laboratories, Inc., Texas, USA
http://www.dslabs.com/
2005;
- 8
Brand M, Schoof E, Partsch C-J. et al .
Anorexia nervosa in congenital adrenal hyperplasia: long-term follow-up of 4 cases.
Exp Clin Endocrinol Diabetes.
2000;
108
430-435
- 9
Caldefie-Chezet F, Moinard C, Minet-Quinard R. et al .
Dexamethasone treatment induces long-lasting hyperleptinemia and anorexia in old rats.
Metabolism.
2001;
50
1054-1058
- 10
Fosson A, Knibbs J, Bryant-Waugh R. et al .
Early onset anorexia nervosa.
Arch Dis Child.
1987;
62
114-118
- 11
Krawczynski M, Krzyzaniak A, Walkowiak J.
Normy rozwojowe wysokosci i masy ciala dzieci i mlodziezy miasta Poznania w wieku
od 3 do 18 lat (Normative developmental data of height and weight in children and
adolescents aged from 3 to 18 years in Poznan city).
Ped. Prakt. (Pediatrics in Practice).
2000;
8
341-353
(in Polish).
- 12
Nussbaum M, Baird D, Sonnenblick M. et al .
Short stature in anorexia nervosa patients.
J Adolesc Health Care.
1985;
6
453-455
- 13
Partsch CJ, Sippell WG.
Pathogenesis and epidemiology of precocious puberty. Effects of exogenous oestrogens.
Hum Reprod Update..
2001;
7
292-302
- 14
Robb AS, Dadson MJ.
Eating disorders in males.
Child Adolesc Psychiatr Clin N Am.
2002;
11
399-418
- 15
Romeo F.
Adolescent boys and anorexia nervosa.
Adolescence.
1994;
29
643-647
- 16
Scacchi M, Ida Pincelli A, Cavagnini F.
Nutritional status in the neuroendocrine control of growth hormone secretion: the
model of anorexia nervosa.
Front Neuroendocrinol.
2003;
24
200-224
- 17
Soliman AT, AlLamki M, AlSalmi I. et al .
Congenital adrenal hyperplasia complicated by central precocious puberty: linear growth
during infancy and treatment with gonadotropin-releasing hormone analog.
Metabolism.
1997;
46
513-517
- 18
Sreenivasan U.
Anorexia nervosa in boys.
Can Psychiatr Assoc J.
1978;
23
159-162
- 19
Tronche F, Opherk C, Moriggl R. et al .
Glucocorticoid receptor function in hepatocytes is essential to promote postnatal
body growth.
Genes Dev.
2004;
18
492-497
1 Presented in part at the 37th International Symposium: GH and Growth Factors in Endocrinology and Metabolism, Athens,
Greece, 4 – 5 March 2005.
Correspondence
M. NiedzielaMD, PhD
Department of Pediatric Endocrinology and Diabetes
Poznan University of Medical Sciences
Szpitalna Street 27/33
60-572 Poznan
Poland
Phone: +48/61/848 0291, 849 1481
Fax: +48/61/848 0291
Email: mniedzie@am.poznan.pl