Effects of Octreotide Infusion, Surgery and Estrogen on Suppression of Height Increase and 20K Growth Hormone Ratio in a Girl with Gigantism due to a Growth Hormone-Secreting Macroadenoma

Minagawa, Masahiro; Yasuda, Toshiyuki; Someya, Tomohiro; Kohno, Yoichi; Saeki, Naokatsu; Hashimoto, Yoshihide

doi:10.1159/000023556

Cited by 3 publications

(1 citation statement)

References 16 publications

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“…Both cases, however, continued to show persistent GH hypersecretion and overgrowth and required long-term postoperative octreotide therapy for hormonal control and growth suppression 1 ' 19 . Both cases, however, continued to show persistent GH hypersecretion and overgrowth and required long-term postoperative octreotide therapy for hormonal control and growth suppression 1 ' 19 .…”

Section: Discussionmentioning

confidence: 99%

Successful Treatment of an Invasive Growth Hormone-Secreting Pituitary Macroadenoma in an 8 Year-old Boy

Bowden¹,

Sotos²,

Stratakis³

et al. 2007

Journal of Pediatric Endocrinology and Metabolism

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Section: Discussionmentioning

confidence: 99%

Successful Treatment of an Invasive Growth Hormone-Secreting Pituitary Macroadenoma in an 8 Year-old Boy

Bowden¹,

Sotos²,

Stratakis³

et al. 2007

Journal of Pediatric Endocrinology and Metabolism

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Clinical Features and Treatment of Pediatric Somatotropinoma: Case Study of an Aggressive Tumor due to a New <i>AIP</i> Mutation and Extensive Literature Review

Personnier¹,

Cazabat²,

Bertherat³

et al. 2011

Horm Res Paediatr

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Context: Pediatric somatotropinoma is uncommon but usually more aggressive than in adults, creating therapeutic challenges. No treatment guidelines are available. Objectives: To describe the features of pediatric somatotropinomas and to assess therapeutic strategies based on an extensive literature review. Design: We describe a pediatric case of aggressive somatotropinoma with an AIP mutation. We identified 137 pediatric somatotropinoma cases published between 1981 and 2010, and found 41 cases with AIP mutations in the main review. Results: We found a slight male preponderance (59%). Median age was 9 years at symptom onset and 14 years at diagnosis. Macroadenomas accounted for 90% of the tumors; 2/3 of the children had hyperprolactinemia at diagnosis. The first-line treatment was pharmacotherapy in one third and surgery in 2/3 of the patients. Pegvisomant was used in 7 patients and produced significant improvement in 4. The male preponderance was higher in the subgroup with AIP mutations. Mutations leading to severe protein abnormalities were more common than reported in adults. Conclusion: Higher invasiveness and tumor volume in pediatric somatotropinomas require complex treatment combinations, which produce variable results. Pegvisomant is an effective drug whose usefulness in children remains to be determined. Genetic screening, particularly for AIP mutations, should be performed routinely.

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Pegvisomant therapy in pituitary gigantism: successful treatment in a 12-year-old girl

Rix¹,

Laurberg²,

Hoejberg³

et al. 2005

eur j endocrinol

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Objective: The use of a growth hormone (GH) receptor antagonist, pegvisomant has shown great promise in adults with acromegaly, but experience in paediatric patients is lacking. We aimed to describe the results of pegvisomant therapy in a 12-year-old girl with an aggressive GH-secreting pituitary tumour. Design: To evaluate the ability of pegvisomant therapy to control the effects of peripheral GH excess in a case of pituitary gigantism. Methods: Pegvisomant was introduced at 10 mg/day, given subcutaneously, and gradually increased to 20 mg/day until serum IGF-I was normal for age. Results: A large pituitary adenoma with suprasellar extension was diagnosed in a 12-year-old girl with progressive tall stature (178 cm), GH hypersecretion without suppression during oral glucose loading (nadir serum GH, 90 mU/l), high serum IGF-I and serum prolactin levels. Surgical extirpation was not possible because tumour tissue was fibrous and adherent to the optical nerves. Histological examination showed a mixed GH-and prolactin-secreting adenoma with lymphocytic infiltration of B and T cells. Treatment with a dopamine agonist, cabergoline, normalized serum prolactin, but GH secretion was resistant to both somatostatin analogue, octreotide and cabergoline. Radiation followed by pegvisomant therapy titrated up in dose to 20 mg/day led to a marked reduction in GH secretion and normalization of IGF-I, and to growth arrest and improvement of well-being. Conclusions: We suggest that treatment in pituitary gigantism with pegvisomant is safe and may normalize IGF-I levels and effectively stop growing.

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Effects of Octreotide Infusion, Surgery and Estrogen on Suppression of Height Increase and 20K Growth Hormone Ratio in a Girl with Gigantism due to a Growth Hormone-Secreting Macroadenoma

Cited by 3 publications

References 16 publications

Successful Treatment of an Invasive Growth Hormone-Secreting Pituitary Macroadenoma in an 8 Year-old Boy

Successful Treatment of an Invasive Growth Hormone-Secreting Pituitary Macroadenoma in an 8 Year-old Boy

Clinical Features and Treatment of Pediatric Somatotropinoma: Case Study of an Aggressive Tumor due to a New <i>AIP</i> Mutation and Extensive Literature Review

Pegvisomant therapy in pituitary gigantism: successful treatment in a 12-year-old girl

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