ArticleEndocrine connections2019
AIP-mutated acromegaly resistant to first-generation somatostatin analogs: long-term control with pasireotide LAR in two patients.
Article in Endocrine connections, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers, 2 of them syntheses that pooled it.
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Who cites it
28 citing papers in PubMed, 2 syntheses or guidelines pooled it, 62 citations in OpenAlex.
- Efficacy and safety of pasireotide treatment in acromegaly: A systematic review and single arm meta-analysis.Pituitary · 2024Pooled it
- Consensus guideline for the diagnosis and management of pituitary adenomas in childhood and adolescence: Part 2, specific diseases.Nature reviews. Endocrinology · 2024Guideline
- Sex differences in the clinical presentation, management and long-term outcomes of pituitary tumors in children and adolescents.Pituitary · 2026Review
- Functional analysis of AIP variants in a cohort of neuroendocrine neoplasms.Endocrine-related cancer · 2026Article
- Clinical and Genetic Spectrum of Large AIP Deletions.Human mutation · 2026Article
- Hypersomatotropism in diabetic cats in Australia.Journal of feline medicine and surgery · 2025Article
- Germline genetic variants in young-onset sporadic pituitary macroadenomas: A multigene panel analysis.Journal of clinical & translational endocrinology · 2025Article
- Use of Pasireotide in Acromegaly: Clinical Experiences From a Series of Patients in Qatar.Case reports in endocrinology · 2025Article
- Case report: Management of pediatric gigantism caused by the TADopathy, X-linked acrogigantism.Frontiers in endocrinology · 2024Article
- Management of pasireotide-induced hyperglycemia in patients with acromegaly: An experts' consensus statement.Frontiers in endocrinology · 2024Article
- Pituitary gigantism due to a novelEndocrine oncology (Bristol, England) · 2024Article
- Low frequency of AIP mutations in patients with young-onset sporadic pituitary macroadenomas.Journal of endocrinological investigation · 2023Article
- Pituitary Tumorigenesis-Implications for Management.Medicina (Kaunas, Lithuania) · 2023Review
- Clinical Management of Acromegaly: Therapeutic Frontiers and New Perspectives for Somatostatin Receptor Ligands (SRLs).Medicina (Kaunas, Lithuania) · 2022Review
- Current and Emerging Medical Therapies in Pituitary Tumors.Journal of clinical medicine · 2022Review
- Real-world experience with pasireotide-LAR in resistant acromegaly: a single center 1-year observation.Pituitary · 2022Article
- The Future of Somatostatin Receptor Ligands in Acromegaly.The Journal of clinical endocrinology and metabolism · 2022Review
- Drug resistance in pituitary tumours: from cell membrane to intracellular signalling.Nature reviews. Endocrinology · 2021Review
- Genetics, clinical features and outcomes of non-syndromic pituitary gigantism: experience of a single center from Sao Paulo, Brazil.Pituitary · 2021Article
- Genetics of Acromegaly and Gigantism.Journal of clinical medicine · 2021Review
Corrections and comments
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Authors and funding
11 authors at 4 institutions in 4 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Acromegaly is a rare disease due to chronic excess growth hormone (GH) and IGF-1. Aryl hydrocarbon receptor interacting protein (AIP) mutations are associated with an aggressive, inheritable form of acromegaly that responds poorly to SST2-specific somatostatin analogs (SSA). The role of pasireotide, an SSA with affinity for multiple SSTs, in patients with AIP mutations has not been reported. We studied two AIP mutation positive acromegaly patients with early-onset, invasive macroadenomas and inoperable residues after neurosurgery. Patient 1 came from a FIPA kindred and had uncontrolled GH/IGF-1 throughout 10 years of octreotide/lanreotide treatment. When switched to pasireotide LAR, he rapidly experienced hormonal control which was associated with marked regression of his tumor residue. Pasireotide LAR was stopped after >10 years due to low IGF-1 and he maintained hormonal control without tumor regrowth for >18 months off pasireotide LAR. Patient 2 had a pituitary adenoma diagnosed when aged 17 that was not cured by surgery. Chronic pasireotide LAR therapy produced hormonal control and marked tumor shrinkage but control was lost when switched to octreotide. Tumor immunohistochemistry showed absent AIP and SST2 staining and positive SST5. Her AIP mutation positive sister developed a 2.5 cm follicular thyroid carcinoma aged 21 with tumoral loss of heterozygosity at the AIP locus and absent AIP staining. Patients 1 and 2 required multi-modal therapy to control diabetes. On stopping pasireotide LAR after >10 years of treatment, Patient 1's glucose metabolism returned to baseline levels. Long-term pasireotide LAR therapy can be beneficial in some AIP mutation positive acromegaly patients that are resistant to first-generation SSA.
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