Predictor Variables of Developing Anterior Pituitary Deficiencies in a Group of Paediatric Patients with Central Diabetes Insipidus and Langerhans Cell Histiocytosis

Vaiani, Elisa; Malossetti, Carmen; Vega, Lina Margarita; Zubizarreta, Pedro; Braier, Jorge; Belgorosky, Alicia

doi:10.1159/000452996

Cited by 6 publications

(8 citation statements)

References 37 publications

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“…APD is almost never associated with DI. Older age at the diagnosis of LCH or the presence of low risk clinical forms and fewer reactivation episodes seem to be the main predictors of a higher risk of developing APD when DI is already diagnosed (43).…”

Section: Discussionmentioning

confidence: 97%

Endocrine manifestations in a cohort of 63 adulthood and childhood onset patients with Langerhans cell histiocytosis

Sagna

Courtillot

Drabo³

et al. 2019

European Journal of Endocrinology

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Objective Langerhans cell histiocytosis (LCH) is a rare inflammatory myeloid neoplasm which can infiltrate any organ or tissue. Endocrine involvement has mostly been described in case reports and small retrospective studies. We aimed to describe endocrine manifestations in a large cohort of adulthood onset (AO) and childhood onset (CO) patients with LCH. Design Single-center observational study conducted between January 2002 and December 2017 at Pitié-Salpêtrière University Hospital (Paris, France), a tertiary care hospital. Method Clinical, biological and morphological evaluations of pituitary, gonadal, adrenal and thyroid function evaluations performed in 63 consecutive patients with LCH (AO patients: 40, CO patients: 23). Fifty-eight patients underwent follow-up assessments. Results Complete pituitary evaluation was performed in 38/63 patients (60.3%); at least one anterior pituitary dysfunction (APD) was found in 63.2% of them. In this subgroup of patients, the most prevalent deficiencies were diabetes insipidus (DI) and GHD (55.3% each), followed by gonadotropin deficiency (34.2%) and thyrotropin deficiency (23.7%). In the subgroup of the 25 incompletely evaluated patients, we found DI in 44%, GHD in 50%, gonadotropin deficiency in 30.4% and thyrotropin deficiency in 16%. APD was more common in CO patients (P = 0.003) but was not systematically associated with DI regardless of the age of onset. Endocrine dysfunction was most often permanent; moreover, occurrence of new deficiencies has been described during follow-up. Conclusion The spectrum of endocrine disorders appears to be large in LCH (both in AO and CO patients) and should be evaluated carefully at diagnosis and during follow-up. APD was not always associated with DI.

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Section: Discussionmentioning

confidence: 97%

Endocrine manifestations in a cohort of 63 adulthood and childhood onset patients with Langerhans cell histiocytosis

Sagna

Courtillot

Drabo³

et al. 2019

European Journal of Endocrinology

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“…However, the role of copeptin in the differential diagnosis of DI in children is still lacking. CDI could be diagnosed concomitantly or during follow-up especially within the first 5 years from LCH diagnosis [29, 33, 34, 38, 48, 56, 57].…”

Section: Lch: Hypothalamus-pituitary Involvementmentioning

confidence: 99%

“…Otherwise, CDI may occur as an isolated and inaugural manifestation of LCH. The reported incidence is variable, and according to the different cohorts reported, the percentage ranged between 11 and 50% [28, 29, 43, 48, 56, 57]. The variability in incidence may be due to the fact that in some studies the definition of CDI as the first isolated symptom is not clearly addressed.…”

Section: Lch: Hypothalamus-pituitary Involvementmentioning

confidence: 99%

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Paediatric Langerhans Cell Histiocytosis Disease: Long-Term Sequelae in the Hypothalamic Endocrine System

et al. 2021

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Langerhans cell histiocytosis (LCH) is a disorder of the mononuclear phagocyte system that can affect almost any organ and system. The most common central nervous system (CNS) manifestation in LCH is the infiltration of the hypothalamic-pituitary region leading to destruction and neurodegeneration of CNS tissue. The latter causes the most frequent endocrinological manifestation, that is, central diabetes insipidus (CDI), and less often anterior pituitary hormone deficiency (APD). The reported incidence of CDI is estimated between 11.5 and 24% and is considered a risk factor for neurodegenerative disease and APD. Three risk factors for development of CDI are recognized in the majority of the studies: (1) multisystem disease, (2) the occurrence of reactivations or active disease for a prolonged period, and (3) the presence of craniofacial bone lesions. Since CDI may occur as the first manifestation of LCH, differential diagnosis of malignant diseases like germ cell tumours must be made. APD is almost always associated with CDI and can appear several years after the diagnosis of CDI. Growth hormone is the most commonly affected anterior pituitary hormone. Despite significant advances in the knowledge of LCH in recent years, little progress has been made in preventing long-term sequelae such as those affecting the hypothalamic-pituitary system.

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“…Such findings indicate the importance and the role of pituitary and hypothalamus MRI scan in determining the cause. Langerhans Cell Histocytosis (LCH) is a rare and heterogeneous disease, characterized by accumulation and cloned proliferation of immature dendritic cells in different organs, present in 3 (1.5%) of patients [34,35].…”

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confidence: 99%

Childhood Hypopituitarism: A Three Decades of Experience from a Major Teaching Hospital in Central Region (Riyadh) Saudi Arabia

Khalifah

Jurayyan

et al. 2018

BJSTR

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children with acquired forms are discovered relatively early in life [2][3][4][5][6][7][8]. This article reports on the clinical experience of childhood hypopituitarism from a major teaching hospital, King Khalid University Hospital (KKUH) in the central region of Saudi Arabia over more than 25 years, January 1989 to December 2017. KKUH is affiliated to King Saud University and provides primary, secondary and tertiary health care service to the local population and receives patients' referral from all over the country. Materials and MethodsThe medical records of patients who were diagnosed to have hypopituitarism were retrospectively reviewed from January 1989 to December 2017. Data included were age, sex, clinical presentation and results of the relevant laboratory investigations and radiological images. Magnetic Resonance Imaging (MRI) was done when appropriate. The diagnosis of hypopituitarism was based on clinical suspicion suggested by the appropriate hormonal testing. The various hormonal testing to assess both the anterior and posterior pituitary gland functions, were performed following

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Predictor Variables of Developing Anterior Pituitary Deficiencies in a Group of Paediatric Patients with Central Diabetes Insipidus and Langerhans Cell Histiocytosis

Cited by 6 publications

References 37 publications

Endocrine manifestations in a cohort of 63 adulthood and childhood onset patients with Langerhans cell histiocytosis

Endocrine manifestations in a cohort of 63 adulthood and childhood onset patients with Langerhans cell histiocytosis

Paediatric Langerhans Cell Histiocytosis Disease: Long-Term Sequelae in the Hypothalamic Endocrine System

Childhood Hypopituitarism: A Three Decades of Experience from a Major Teaching Hospital in Central Region (Riyadh) Saudi Arabia

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