Incidence of Growth Hormone Deficiency in Pediatric-Onset Langerhans Cell Histiocytosis: Efficacy and Safety of Growth Hormone Treatment

Donadieu, Jean; Rolon, Maria-Alejandra; Pion, Isabelle; Thomas, Caroline; Doz, François; Barkaoui, Mohamed; Robert, Alain; Deville, Anne; Mazingue, Françoise; Dávid, M; Brauner, Raja; Cabrol, Sylvie; Garel, Catherine; Polak, M.

doi:10.1210/jc.2003-030907

Cited by 87 publications

(48 citation statements)

References 21 publications

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“…The unexpected observation of ADH as the sole additional deficiency in nine patients may relate to the association of ADH deficiency and histiocytosis (21), and MRI may potentially identify the structural lesion in such cases (22). Patients with additional pituitary deficiencies diagnosed during follow-up had a higher incidence of breech presentation, perinatal asphyxia, and neonatal complications, and a trend toward greater delivery complications.…”

Section: European Journal Of Endocrinologymentioning

confidence: 94%

Development of additional pituitary hormone deficiencies in pediatric patients originally diagnosed with idiopathic isolated GH deficiency

Blum

Deal

Zimmermann

et al. 2014

European Journal of Endocrinology

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Objective: We assessed the characteristics of children initially diagnosed with idiopathic isolated GH deficiency (IGHD) who later developed additional (multiple) pituitary hormone deficiencies (MPHD). Design: Data were analyzed for 5805 pediatric patients with idiopathic IGHD, who were GH-naïve at baseline and GH-treated in the multinational, observational Genetics and Neuroendocrinology of Short Stature International Study. Methods: Development of MPHD was assessed from investigator diagnoses, adverse events, and concomitant medications. Analyses were performed for all patients and for those who developed MPHD within 4.5 years or had R3.5 years, follow-up and continued to have IGHD (4-year cohort). Results: MPHD developed in 118/5805 (2.0%) children overall, and in 96/1757 (5.5%) in the 4-year cohort. Patients who developed MPHD had more profound GHD, with decreased height SDS, IGF1 SDS and peak stimulated GH, and greater height decrement vs target, compared with children who continued to have IGHD (P!0.001 for each variable). Delivery complications, congenital anomalies, and perinatal/neonatal adverse events occurred more frequently in patients who developed MPHD. The most frequent additional deficiency was TSH (82 patients overall); four patients developed two pituitary hormone deficiencies and one developed three deficiencies. Multivariable logistic regression indicated that years of follow-up (odds ratio 1.55), baseline age (1.17), baseline height SDS (0.69), and peak stimulated GH (0.64) were associated with the development of MPHD. Conclusions: MPHD is more likely to develop in patients with more severe idiopathic IGHD. Older baseline age, lower baseline height SDS, and longer follow-up duration are associated with increased risk of development of MPHD.

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Section: European Journal Of Endocrinologymentioning

confidence: 94%

Development of additional pituitary hormone deficiencies in pediatric patients originally diagnosed with idiopathic isolated GH deficiency

Blum

Deal

Zimmermann

et al. 2014

European Journal of Endocrinology

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“…Growth hormone deficiency is the most frequent additional deficit, accounting for 42% of cases with CDI and LCH. The 10-year cumulative incidence of growth hormone deficiency among patients with CDI in the French nationwide LCH survey was approximately 54% [38]. The identification of circulating AVPc-Abs in LCH patients and their tendency toward spontaneous clearance [24,25] suggest that these autoantibodies might be an LCH-related immune epiphenomenon.…”

Section: Acquired Forms Of CDImentioning

confidence: 99%

Diabetes Insipidus – Diagnosis and Management

et al. 2012

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Central diabetes insipidus (CDI) is the end result of a number of conditions that affect the hypothalamic-neurohypophyseal system. The known causes include germinoma/craniopharyngioma, Langerhans cell histiocytosis (LCH), local inflammatory, autoimmune or vascular diseases, trauma resulting from surgery or an accident, sarcoidosis, metastases and midline cerebral and cranial malformations. In rare cases, the underlying cause can be genetic defects in vasopressin synthesis that are inherited as autosomal dominant, autosomal recessive or X-linked recessive traits. The diagnosis of the underlying condition is challenging and raises several concerns for patients and parents as it requires long-term follow-up. Proper etiological diagnosis can be achieved via a series of steps that start with clinical observations and then progress to more sophisticated tools. Specifically, MRI identification of pituitary hyperintensity in the posterior part of the sella, now considered a clear marker of neurohypophyseal functional integrity, together with the careful analysis of pituitary stalk shape and size, have provided the most striking findings contributing to the diagnosis and understanding of some forms of ‘idiopathic’ CDI. MRI STIR (short-inversion-time inversion recovery sequencing) is a promising technology for the early identification of LCH-dependent CDI.

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“…Compared with historical controls, the subjects on GH did not appear to have an increased number of recurrences. Most recently, Donadieu et al [27]reported on 61 subjects with LCH treated with GH from the French LCH Study Group. Among those with pituitary involvement, the number of recurrences were similar between those treated with GH and those who were not.…”

Section: Cancer Risk and Gh Therapymentioning

confidence: 99%

Growth Hormone Treatment: Cancer Risk

Sklar¹

2004

Horm Res Paediatr

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There have been concerns that growth hormone (GH) therapy may be associated with an increased risk of cancer. Although data are limited and conflicting, one recent report on cancer risk in individuals with no cancer history or risk factors for cancer who were treated with pituitary GH demonstrated a small increased risk of colon cancer and deaths from colon cancer and Hodgkin disease. The data from cancer survivors have consistently shown no increased risk of recurrence of the primary tumor in survivors of all tumor types who are treated with GH. One recent study did show a small increased risk of second solid tumors in survivors previously treated with GH. Limited data suggest that GH therapy is not associated with excess cancer risk in individuals with Langerhans cell histiocytosis and neurofibromatosis type 1. Overall, the clinical data are reassuring, but continued surveillance is mandatory.

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Incidence of Growth Hormone Deficiency in Pediatric-Onset Langerhans Cell Histiocytosis: Efficacy and Safety of Growth Hormone Treatment

Cited by 87 publications

References 21 publications

Development of additional pituitary hormone deficiencies in pediatric patients originally diagnosed with idiopathic isolated GH deficiency

Development of additional pituitary hormone deficiencies in pediatric patients originally diagnosed with idiopathic isolated GH deficiency

Diabetes Insipidus – Diagnosis and Management

Growth Hormone Treatment: Cancer Risk

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