Combined treatment with octreotide LAR and pegvisomant in patients with pituitary gigantism: clinical evaluation and genetic screening

Mangupli, Ruth; Rostomyan, Liliya; Castermans, Emilie; Caberg, Jean‐Hubert; Camperos, Paul; Krivoy, Jaime; Cuauro, Elvia; Daly, Adrian; Beckers, Albert

doi:10.1007/s11102-016-0732-3

Cited by 26 publications

(19 citation statements)

References 42 publications

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“…Effectiveness was confirmed in 2 of our patients in whom this medication was administrated (patients #1 and #6), a result that is similar to the one reported by Creo and Lteif (15) and Mangupli and cols. (6). A lower rate of biochemical control (50% of the patients) was observed by Rostomyan and cols.…”

Section: Discussionmentioning

confidence: 77%

“…Dopamine receptor agonists are useful in cases with associated hyperprolactinemia or as an adjunct therapy to SSAs in cases with lack of biochemical control and IGF-1 levels up to 1.5 times above the normal range (19). None of our patients presented hyperprolactinemia, in contrast to the finding by Mangupli and cols., in which 50% (4 out of 8) of the patients had hyperprolactinemia (6). Cabergoline was administered to 3 of our patients, and biochemical control was obtained in 1 (patient #3) with concomitant use of octreotide LAR.…”

Section: Discussionmentioning

confidence: 92%

“…In the cases described by Mangupli and cols. (6), none of the patients who underwent surgery obtained control. In our series, surgical reintervention was required in 33% (2 out of 6) of the patients, compared with 30% (4 out of 13) in the series by Creo and Lteif (15) and 64% in the series by Rostomyan and cols.…”

Section: Discussionmentioning

confidence: 99%

“…This finding was similar in the series by Mangupli and cols. (6), in which 100% (8 out of 8) of the patients used SSAs. Still, the use of SSA was less frequent in other studies: 50% (6 out of 12) of the patients in the series by Creo and Lteif (15), 66.7% (118 out of 208) reported by Rostomyan and cols.…”

Section: Discussionmentioning

confidence: 99%

“…The first-choice treatment for gigantism is transsphenoidal surgery (TSS) (4). However, complete remission of the disease is not usually achieved with surgical intervention alone and pharmacological therapy becomes necessary (2,(5)(6)(7), of which somatostatin analogues (SSA) is the most common. If no response is obtained with SSAs, dopamine receptor agonists (cabergoline) or GH receptor antagonists (pegvisomant) can be added (8)(9)(10).…”

Section: Introductionmentioning

confidence: 99%

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Pituitary gigantism: a case series from Hospital de San José (Bogotá, Colombia)

Rojas

Cortes

Romero

2019

Archives of Endocrinology and Metabolism

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Introduction Gigantism is a rare pediatric disease characterized by increased production of growth hormone (GH) before epiphyseal closure, that manifests clinically as tall stature, musculoskeletal abnormalities, and multiple comorbidities. Materials and methods Case series of 6 male patients with gigantism evaluated at the Endocrinology Service of Hospital de San José (Bogotá, Colombia) between 2010 and 2016. Results All patients had macroadenomas and their mean final height was 2.01 m. The mean age at diagnosis was 16 years, and the most common symptoms were headache (66%) and hyperhidrosis (66%). All patients had acral changes, and one had visual impairment secondary to compression of the optic chiasm. All patients underwent surgery, and 5 (83%) required additional therapy for biochemical control, including radiotherapy (n = 4, 66%), somatostatin analogues (n = 5, 83%), cabergoline (n = 3, 50%), and pegvisomant (n = 2, 33%). Three patients (50%) achieved complete biochemical control, while 2 patients showed IGF-1 normalization with pegvisomant. Two patients were genetically related and presented a mutation in the aryl hydrocarbon receptor-interacting protein (AIP) gene (pathogenic variant, c.504G>A in exon 4, p.Trp168*), fulfilling the diagnostic criteria of familial isolated pituitary adenoma. Conclusions This is the largest case series of patients with gigantism described to date in Colombia. Transsphenoidal surgery was the first-choice procedure, but additional pharmacological therapy was usually required. Mutations in the AIP gene should be considered in familial cases of GH-producing adenomas.

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Section: Discussionmentioning

confidence: 77%

Section: Discussionmentioning

confidence: 92%