Complete disappearance of a GH-secreting pituitary macroadenoma in a patient with acromegaly: effect of treatment with lanreotide Autogel and consequence of treatment withdrawal

Abstract

Background: Somatostatin analogs (SA) are the cornerstone in the medical treatment of acromegaly, used as either primary or adjunctive therapy. In particular, SA are effective in inducing the biochemical remission of the disease and tumor shrinkage, although only few cases of complete disappearance of the pituitary tumor in patients treated with SA as long-acting formulations have been reported. SA withdrawal has been demonstrated to keep safe levels of GH and IGF1 at least in a small subset of patients well responsive to SA, although it is generally followed by disease recurrence after several months. Case report: A 61-year-old female patient bearing a very large GH-secreting pituitary macroadenoma was treated with 12-month lanreotide Autogel (ATG), at the initial dose of 120 mg/28 days. After 3 months, GH and IGF1 levels were fully normalized, to prolong the administration interval from 28 to 56 days. After 6 months of treatment, a significant tumor shrinkage (90% of baseline size) was observed, whereas GH and IGF1 excess was still well controlled. After 12-month therapy, a complete disappearance of the pituitary tumor was observed, and the hormonal evaluation confirmed the complete biochemical remission of acromegaly. Lanreotide ATG treatment was withdrawn. The clinical, biochemical, and radiological remission of acromegaly was maintained 24 months after lanreotide ATG treatment discontinuation, without evidence of disease recurrence. Conclusions: This report represents an exemplary case of the potentiality of treatment with lanreotide ATG in inducing a complete remission of acromegalic disease, persistent after a long period of time from treatment withdrawal.

Active postoperative acromegaly: sustained remission after discontinuation of somatostatin analogues

“…In the PRIMARYS trial, a reduction of >20% tumour volume was achieved in 56% of treatment-naïve acromegalic patients after 6 months of treatment with lanreotide Autogel, and nearly one in four (23.9%) patients achieved a GH level ≤1.0 ng/mL with IGF1 normalization (15). In most cases, recurrence is common after discontinuation of somatostatin analogue treatment (12, 14), although numerous cases of prolonged remission and even complete tumour disappearance and/or biochemical control of the acromegaly have been reported (5, 6, 7, 13, 16, 17). …”

“…These drugs are effective in inducing biochemical remission and shrinking the tumour, thereby reducing the clinical influence of the disease (3). In rare cases, complete disappearance of the pituitary tumour has been reported (4, 5, 6, 7). Despite the well-documented effectiveness of somatostatin analogue therapy, discontinuation—when feasible—is desirable to eliminate the possible treatment-related adverse effects (cholelithiasis, blood glucose alterations, injection site pain, gastrointestinal cramps, among others) (3).…”

Expert consensus document: A consensus on the medical treatment of acromegaly

“…If IGF-I levels remain normal with this regimen, drug withdrawal may be considered in rare cases of persistent optimum control despite progressive dose reduction (DR), but lifelong monitoring of IGF-I levels should be maintained in these patients (SR). [68][69][70][71] Novel agents New pharmacological approaches to the treatment of acromegaly in advanced stages of clinical development include new SRLs with different somatostatin receptor binding profiles (such as pasireotide) 72 and oral octreotide, which uses a transient permeability enhancer to enable gut absorption. 30 Novel therapeutic approaches in early stages of clinical development include an antisense oligonucleotide of 20 bases that binds to the GH receptor mRNA and inhibits translation of the receptor protein, and a targeted secretion inhibitor, comprising a botulinum toxin-GH-releasing hormone (GHRH) chimera molecule that binds to cells expressing GHRH receptors, internalizes botulinum toxin and inhibits GH secretion.…”

Autozhel' v kontrole biokhimicheskikh markerovaktivnosti akromegalii: predstavlenieklinicheskogo sluchaya

“…В литературе описан случай полного исчезновения макроаденомы гипофиза у 61-летней пациентки с ак-ромегалией на фоне лечения Соматулином ® Аутожель ® 120 мг каждые 4 недели [5]. Целевые значения СТГ и ИФР-1 были достигнуты через 3 месяца терапии, после чего промежутки между инъекциями были уве-личены до 8 недель.…”