Growth rates and the prevalence and progression of scoliosis in short-statured children on Australian growth hormone treatment programmes

Day, Gregory A.; McPhee, Ian; Batch, J. A.; Tomlinson, Francis H.

doi:10.1186/1748-7161-2-3

Cited by 23 publications

(25 citation statements)

References 19 publications

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“…Furthermore, studies that measure females during growth suggest that in cases of IS, abnormal prepubescent stature proportions (trunk or leg lengths) might be related to androgenic affects [4,13,26]. Endocrine factors such as growth hormone activity [27–31], estradiol levels [32,33], or testosterone levels [28, 33] have been considered as possible risk factors for curve progression in IS. Given that the androgenic and somatotrophic axes are the major hormonal systems regulating postnatal linear growth and have principal roles in regulating skeletal growth and bone mass [26], it is possible that androgenic and/or somatotrophic pathway interactions are involved in disproportionate cranial-caudal lengths associated with idiopathic-type curvature.…”

Section: Discussionmentioning

confidence: 99%

Disproportionate Body Lengths Correlate With Idiopathic-Type Curvature in the Curveback Guppy

Gorman

Breden

2010

Spine

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In both humans and guppies, idiopathic-type spinal curvature is a multi-factorial deformity that exhibits extensive phenotypic variation. This variation is likely a consequence of interactions among genetic, physiological, and environmental factors. Success at identifying the etiology of human idiopathic-type scoliosis (IS) has been limited due to trait complexity and the lack of animal models with a non-induced phenotype similar to humans [1]. Preliminary studies in otherwise healthy guppies have revealed that heritable curvature in curveback has remarkable morphological and developmental similarities to human IS (e.g., no vertebral fusion or breaks, distortion of apical vertebrae, curve onset after birth, variable rates of progression and prognoses for curve magnitude, a female bias for severe curves) [2,3]. Here we explore length variation in the curveback population, because numerous studies have associated IS with tallness and/or abnormal length proportions along the cranio-caudal axis [4][5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21].We surveyed adult curveback females for increased length, because mature females with IS have demonstrated increased cranio-caudal length [4]. We show that among curved fish, the caudal portion of the body is disproportionately longer relative to the pre-caudal body, and this disproportion is positively correlated to curve magnitude. As with anthropometric studies, these results suggest that a growth abnormality may be associated with idiopathic-type spinal curvature. Materials and Methods Study population and data collectionGuppies are live-bearing teleost fish; offspring are born with a fully ossified skeleton after 3 weeks of gestation. Curvature begins after birth and is generally stable by sexual maturity (approximately 1 month past birth) [3]. In cases of severe curvature, curves will progress into early adulthood (up to 2 months after birth). Breeding pairs were maintained in 4L plastic aquaria, and offspring were separated into individual 600 ml plastic containers after birth. All fish are kept under standardized conditions (i.e., flakes fed every afternoon, supplemented with brine shrimp nauplii; 25-26°C; pH 7-9; RO water reconstituted to 1600-1800 ppm salinity with aquarium salts; 14/10 hour light cycle). Adult females were euthanized at a minimum of 3 months past birth. Males were not measured because adult coloration obscures the spine. Euthanized females were photographed on a light table with a digital camera (Toshiba PDR-3310, NYC, USA) under 3X magnification.We measured 246 adult females with curves of varying magnitude, and 75 normal adult females, all from the same pedigree. Curvature is manifest as a sagittal anterior lordosis and posterior kyphosis. A small number of fish exhibit with curvature in the frontal or horizontal planes, and in this study these were omitted. The distribution of curve magnitude is shown in Correspondence and Reprints: Fay Gorman, Department of Biological Sciences, Simon Fraser University, Burnaby, BC V5A 1S6, phone...

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Section: Discussionmentioning

confidence: 99%

Disproportionate Body Lengths Correlate With Idiopathic-Type Curvature in the Curveback Guppy

Gorman

Breden

2010

Spine

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“…Although it is generally thought that any growth has the potential to worsen scoliosis [26, 37], there is no clear conclusion in the literature regarding the impact of hGH on the prevalence or progression of scoliosis or kyphosis. While some researchers have found a higher than expected percentage and rate of curve progression [96], others have observed little to no progression attributable to hGH therapy [2,24,71,95]. Populations with an increased baseline prevalence of scoliosis include children with Turner syndrome (TS), or Prader-Willi syndrome (PWS).…”

Section: Growth Hormonementioning

confidence: 99%

Growth patterns and the use of growth hormone in the mucopolysaccharidoses

Polgreen

Miller

2010

Journal of Pediatric Rehabilitation Medicine

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Short stature is characteristic of patients with mucopolysaccharidosis (MPS) diseases. For children with skeletal dysplasias, such as MPS, it is important to know the natural history of growth. An understanding of the natural growth pattern in each MPS disease provides a measurement to which treatments can be compared, as well as data which can help families and providers make individualized decisions about growth promoting treatments. Multiple advancements have been made in the treatment of MPS with both hematopoietic cell transplantation (HCT) and enzyme replacement therapy (ERT). The long term benefit of these treatments on growth is unknown. This article will review the published data on growth in children with MPS, and describe preliminary data on the use of human growth hormone (hGH) in children with MPS.

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“…Ninguno de nuestros pacientes recibió algun tipo de terapia con hormona del crecimiento, tratamento de uso frecuente en pacientes con esta patología y cuyo objetivo es cambiar la presentación clí-nica, en cuanto a composición de masa corporal 7,17 . Esto se basa en que este Sindrome tiene manifestaciones clínicas comunes con el Sindrome de déficit de hormona del crecimiento, como son: disminución de masa corporal, crecimiento lento, aumento de tejido graso y disminución de la densidad mineral ósea 7 .…”

Section: Materials Y Metodosunclassified

“…En muchos de estos pacientes también se ha comprobado una menor producción de dicha hormona 18 . Los efectos de la terapia hormonal en la evolución de la escoliosis son controvertidos, algunos autores reportan efectos negativos 7,12,17 , mientras otras series lo encuentran beneficioso 4,7 .…”

Section: Materials Y Metodosunclassified

Escoliosis y síndrome de Prader-Willi: a propósito de 5 casos intervenidos quirúrgicamente

Pedrals

Marcuson

Gallegos³

2012

Coluna/Columna

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RESUMENObjetivo: Analizar los resultados y las complicaciones del tratamiento quirúrgico de la escoliosis, en pacientes portadores del Síndrome de Prader-Willi, para verificar si se justifica este tipo de tratamiento. Métodos: Análisis retrospectivo de las fichas clínicas y radiografías de los cinco pacientes portadores del Síndrome de Prader-Willi que consultaron en el Departamento de Escoliosis entre los años 2005 y 2010, y fueron operados. Resultados: La edad de la consulta inicial fue en promedio 3,6 años; el promedio de escoliosis fue 61º, el que aumentó a 65º al momento de la cirugía. El promedio de operaciones realizadas fue 2,8 por paciente, lográndose una corrección promedio de 35%. Los motivos de las reintervenciones fueron pérdida de corrección en 4 casos (28,5%), aflojamiento de ganchos en 2 (14,3%) y xifosis referente a la instrumentación en uno (7,1%) No hubo complicaciones graves. Conclusiones: Aún cuando la literatura muestra autores poco proclives a indicar cirugía en este tipo de pacientes, por la elevada tasa de complicaciones graves, la ausencia de ellas en nuestro medio hace mantener válidos los mismos criterios quirúrgicos que en escoliosis idiopática.

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Growth rates and the prevalence and progression of scoliosis in short-statured children on Australian growth hormone treatment programmes

Cited by 23 publications

References 19 publications

Disproportionate Body Lengths Correlate With Idiopathic-Type Curvature in the Curveback Guppy

Disproportionate Body Lengths Correlate With Idiopathic-Type Curvature in the Curveback Guppy

Growth patterns and the use of growth hormone in the mucopolysaccharidoses

Escoliosis y síndrome de Prader-Willi: a propósito de 5 casos intervenidos quirúrgicamente

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