Case Report: Lipoma of the Tuber Cinereum Mimicking a Pituitary Gland Abnormality in a Girl With Central Precocious Puberty

IntroductionMagnetic Resonance Imaging (MRI) is the best approach to investigate the hypothalamic-pituitary region in children with central precocious puberty (CPP). Routine scanning is controversial in girls aged 6-8 year, due to the overwhelming prevalence of idiopathic forms and unrelated incidentalomas. Cerebral lipomas are rare and accidental findings, not usually expected in CPP. We report a girl with CPP and an unusually shaped posterior pituitary gland on SE-T1w sequences.Case DescriptionA 7.3-year-old female was referred for breast development started at age 7. Her past medical history and physical examination were unremarkable, apart from the Tanner stage 2 breast. X-ray of the left-hand revealed a bone age 2-years ahead of her chronological age, projecting her adult height prognosis below the mid parental height. LHRH test and pelvic ultrasound were suggestive for CPP. Routine brain MRI sequences, SE T1w and TSE T2w, showed the posterior pituitary bright spot increased in size and stretched upward. The finding was considered as an anatomical variant, in an otherwise normal brain imaging. Patient was started on treatment with GnRH analogue. At a thorough revaluation, imaging overlap with adipose tissue was suspected and a new MRI scan with 3D-fat-suppression T1w-VIBE sequences demonstrated a lipoma of the tuber cinereum, bordering a perfectly normal neurohypophysis. 3D-T2w-SPACE sequences, acquired at first MRI scan, would have provided a more correct interpretation if rightly considered.ConclusionThis is the first evidence, to our knowledge, of a cerebral lipoma mimicking pituitary gland abnormalities. Our experience highlights the importance of considering suprasellar lipomas in the MRI investigation of children with CPP, despite their rarity, should the T1w sequences show an unexpected pituitary shape. 3D-T2w SPACE sequences could be integrated into standard ones, especially when performing MRI routinely, to avoid potential misinterpretations.

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Development of Prediction Models Using Machine Learning Algorithms for Girls with Suspected Central Precocious Puberty: Retrospective Study (Preprint)

10.2196/preprints.11728 ◽

2018 ◽

Author(s):

Liyan Pan ◽

Guangjian Liu ◽

Xiaojian Mao ◽

Huixian Li ◽

Jiexin Zhang ◽

...

Keyword(s):

Machine Learning ◽

Retrospective Study ◽

Random Forest ◽

Precocious Puberty ◽

Prediction Models ◽

Central Precocious Puberty ◽

Machine Learning Algorithms ◽

Stimulation Test ◽

Gnrh Analogue ◽

Prediction Probability

BACKGROUND Central precocious puberty (CPP) in girls seriously affects their physical and mental development in childhood. The method of diagnosis—gonadotropin-releasing hormone (GnRH)–stimulation test or GnRH analogue (GnRHa)–stimulation test—is expensive and makes patients uncomfortable due to the need for repeated blood sampling. OBJECTIVE We aimed to combine multiple CPP–related features and construct machine learning models to predict response to the GnRHa-stimulation test. METHODS In this retrospective study, we analyzed clinical and laboratory data of 1757 girls who underwent a GnRHa test in order to develop XGBoost and random forest classifiers for prediction of response to the GnRHa test. The local interpretable model-agnostic explanations (LIME) algorithm was used with the black-box classifiers to increase their interpretability. We measured sensitivity, specificity, and area under receiver operating characteristic (AUC) of the models. RESULTS Both the XGBoost and random forest models achieved good performance in distinguishing between positive and negative responses, with the AUC ranging from 0.88 to 0.90, sensitivity ranging from 77.91% to 77.94%, and specificity ranging from 84.32% to 87.66%. Basal serum luteinizing hormone, follicle-stimulating hormone, and insulin-like growth factor-I levels were found to be the three most important factors. In the interpretable models of LIME, the abovementioned variables made high contributions to the prediction probability. CONCLUSIONS The prediction models we developed can help diagnose CPP and may be used as a prescreening tool before the GnRHa-stimulation test.

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Long-Term Results of GnRH Analogue (Buserelin) Treatment in Girls with Central Precocious Puberty

Acta Paediatrica ◽

10.1111/j.1651-2227.1985.tb10062.x ◽

1985 ◽

Vol 74 (6) ◽

pp. 945-949 ◽

Cited By ~ 10

Author(s):

R. BRAUNER ◽

E. THIBAUD ◽

P. BISCHOF ◽

PC. SIZONENKO ◽

R. RAPPAPORT

Keyword(s):

Precocious Puberty ◽

Central Precocious Puberty ◽

Long Term Results ◽

Gnrh Analogue ◽

Buserelin Treatment

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PDG10 Cost-Effectiveness Analysis of Gnrh Analogue ALONE Versus Combined Growth Hormone for Girls with Central Precocious Puberty

Value in Health ◽

10.1016/j.jval.2021.04.458 ◽

2021 ◽

Vol 24 ◽

pp. S88-S89

Author(s):

J. Zhu ◽

Q. Zhang ◽

Y. Fang ◽

Q. Li

Keyword(s):

Growth Hormone ◽

Cost Effectiveness ◽

Precocious Puberty ◽

Central Precocious Puberty ◽

Cost Effectiveness Analysis ◽

Gnrh Analogue ◽

Effectiveness Analysis

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Insulin-like growth factor-I and IGF binding protein-3 remain high after GnRH analogue therapy in girls with central precocious puberty

Clinical Endocrinology ◽

10.1111/j.1365-2265.1996.tb02053.x ◽

1996 ◽

Vol 45 (1) ◽

pp. 7-12 ◽

Cited By ~ 14

Author(s):

Hannah Kanety ◽

Avraham Karasik ◽

Clara Pariente ◽

Arieh Kauschansky

Keyword(s):

Growth Factor ◽

Precocious Puberty ◽

Binding Protein ◽

Central Precocious Puberty ◽

Gnrh Analogue ◽

Insulin Like Growth Factor ◽

Factor I ◽

Igf Binding ◽

Growth Factor I ◽

Igf Binding Protein

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MR imaging of the pituitary gland in central precocious puberty

Pediatric Radiology ◽

10.1007/bf02012988 ◽

1992 ◽

Vol 22 (7) ◽

pp. 481-484 ◽

Cited By ~ 15

Author(s):

S. C. S. Kao ◽

J. S. Cook ◽

J. R. Hansen ◽

T. M. Simonson

Keyword(s):

Mr Imaging ◽

Pituitary Gland ◽

Precocious Puberty ◽

Central Precocious Puberty

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Should Skeletal Maturation Be Manipulated for Extra Height Gain?

Frontiers in Endocrinology ◽

10.3389/fendo.2021.812196 ◽

2021 ◽

Vol 12 ◽

Author(s):

Jan M. Wit

Keyword(s):

Growth Hormone ◽

Growth Hormone Deficiency ◽

Short Stature ◽

Precocious Puberty ◽

Adult Height ◽

Central Precocious Puberty ◽

Idiopathic Short Stature ◽

Hormone Deficiency ◽

Skeletal Maturation ◽

Gnrh Analogue

Skeletal maturation can be delayed by reducing the exposure to estrogens, either by halting pubertal development through administering a GnRH analogue (GnRHa), or by blocking the conversion of androgens to estrogens through an aromatase inhibitor (AI). These agents have been investigated in children with growth disorders (off-label), either alone or in combination with recombinant human growth hormone (rhGH). GnRHa is effective in attaining a normal adult height (AH) in the treatment of children with central precocious puberty, but its effect in short children with normal timing of puberty is equivocal. If rhGH-treated children with growth hormone deficiency or those who were born small-for-gestational age are still short at pubertal onset, co-treatment with a GnRHa for 2-3 years increases AH. A similar effect was seen by adding rhGH to GnRHa treatment of children with central precocious puberty with a poor AH prediction and by adding rhGH plus GnRHa to children with congenital adrenal hyperplasia with a poor predicted adult height on conventional treatment with gluco- and mineralocorticoids. In girls with idiopathic short stature and relatively early puberty, rhGH plus GnRHa increases AH. Administration of letrozole to boys with constitutional delay of growth puberty may increase AH, and rhGH plus anastrozole may increase AH in boys with growth hormone deficiency or idiopathic short stature, but the lack of data on attained AH and potential selective loss-of-follow-up in several studies precludes firm conclusions. GnRHas appear to have a good overall safety profile, while for aromatase inhibitors conflicting data have been reported.

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SUN-094 Long-Term Safety and Efficacy of Leuprorelin in Treating Central Precocious Puberty: A Large, Open-Label, Multicenter, Phase IV Study in China

Journal of the Endocrine Society ◽

10.1210/jendso/bvaa046.571 ◽

2020 ◽

Vol 4 (Supplement_1) ◽

Author(s):

Xiaoping Luo ◽

Ling Hou ◽

Yan Zhong ◽

Yu Yang ◽

Pin Li ◽

...

Keyword(s):

Adverse Events ◽

Treatment Period ◽

Precocious Puberty ◽

Tanner Stage ◽

Central Precocious Puberty ◽

Chinese Patients ◽

Gnrh Analogue ◽

Open Label ◽

Safety And Efficacy

Abstract BACKGROUND: Leuprorelin (Enantone®) is a gonadotropin-releasing hormone (GnRH) analogue used worldwide to treat central precocious puberty (CPP). This clinical trial aimed to evaluate the long-term safety and efficacy of leuprorelin in treating Chinese CPP children. Methods: This is the first, prospective, open-label, and multicenter study conducted from 2015 to 2018, in China. As a large interventional study, it included a four-week screening period, a 96-week treatment period, and a four-week safety follow-up period. Eligible subjects were treated with leuprorelin subcutaneously once every four weeks for 96 weeks. At the beginning of the study, subjects whose body weight ≥20 kg received a dose of 3.75 mg and those <20 kg received a dose of 1.88 mg and then the dose was allowed to be adjusted during the study based on subject’s condition and investigator’s judgment. The primary endpoint was the incidence of adverse events during treatment, and the secondary endpoint was the percentage of subjects who had regression or no progression in Tanner stage at Week 96 compared with baseline. Results: A total of 307 CPP patients from 11 Chinese medical centers received leuprorelin, of which 305 (99.3%) were girls and 2 were boys (0.7%), with a mean (±SD) age of 7.95±0.982 years and a mean height of 133.68±7.108 cm. Two hundred eighty-three (92.2%) patients completed the 96-week treatment period. Two hundred fifty-two patients (82.1%) reported treatment-emergent adverse events (TEAEs)—most of which (79.5%) were mild to moderate. Only 33 (10.7%) patients experienced TEAEs that were considered related to leuprorelin. The most frequent (>2%) drug-related TEAEs were injection site induration (4.6%, 14/307) and vaginal bleeding (2.3%, 7/305). After the 96-week treatment period, 83.5% female subjects had regression or no progression in Tanner stage compared with baseline (95% CI: 78.68%, 87.62%) and the 2 male subjects had progression of 1 point in Tanner stage genital score occurred at Week 12 and then remained stable throughout the study. By the end of the treatment period, the majority of subjects had decreased GnRH stimulated peak LH and FSH, as well as reduced sex hormone levels and bone age/chronological age ratio compared with baseline. The subjects also had increased predicted adult height and BMI after treatment. Conclusions: This Chinese study demonstrated that CPP was effectively treated in most patients who received leuprorelin (Enantone®) for nearly two years. Any drug-related adverse events were reported with low incidence (<5%) and were consistent with the known safety profile of leuprorelin. Leuprorelin was shown to be well tolerated and effective in the management of CPP in Chinese patients.

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