Sustained Clinical Inactivity and Stabilization of GH/IGF-1 Levels in an Acromegalic Patient after Discontinuation of Somatostatin Analogue Treatment

Avramidis, Athanassios N.; Polyzos, Stergios A.; Efstathiadou, Zoe; Kita, Marina

doi:10.1507/endocrj.k07e-055

Cited by 4 publications

(4 citation statements)

References 42 publications

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“…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). …”

Section: Discussionmentioning

confidence: 99%

Active postoperative acromegaly: sustained remission after discontinuation of somatostatin analogues

Álvarez-Escolá

Cárdenas-Salas

2016

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryIn patients with active acromegaly after pituitary surgery, somatostatin analogues are effective in controlling the disease and can even be curative in some cases. After treatment discontinuation, the likelihood of disease recurrence is high. However, a small subset of patients remains symptom-free after discontinuation, with normalized growth hormone (GH) and insulin-like growth factor (IGF1) levels. The characteristics of patients most likely to achieve sustained remission after treatment discontinuation are not well understood, although limited evidence suggests that sustained remission is more likely in patients with lower GH and IGF1 levels before treatment withdrawal, in those who respond well to low-dose treatment, in those without evidence of adenoma on an MRI scan and/or in patients who receive long-term treatment. In this report, we describe the case of a 56-year-old female patient treated with lanreotide Autogel for 11 years. Treatment was successfully discontinued, and the patient is currently disease-free on all relevant parameters (clinical, biochemical and tumour status). The successful outcome in this case adds to the small body of literature suggesting that some well-selected patients who receive long-term treatment with somatostatin analogues may achieve sustained remission.Learning points:The probability of disease recurrence is high after discontinuation of treatment with somatostatin analogues.Current data indicate that remission after treatment discontinuation may be more likely in patients with low GH and IGF1 levels before treatment withdrawal, in those who respond well to low-dose treatment, in those without evidence of adenoma on MRI, and/or in patients receiving prolonged treatment.This case report suggests that prolonged treatment with somatostatin analogues can be curative in carefully selected patients.

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Section: Discussionmentioning

confidence: 99%

Active postoperative acromegaly: sustained remission after discontinuation of somatostatin analogues

Álvarez-Escolá

Cárdenas-Salas

2016

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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“…In the paper by Livadas (29), tumor disappearance has been obtained after long-term first-line therapy with lanreotide-SR, administered at the dosage of 30 mg every 2 weeks; after the achievement of disease remission, medical treatment was withdrawn. Avramidis (30) has documented the complete biochemical remission of acromegaly induced by therapy with octreotide-LAR in a patient with a stable residual of a pituitary macroadenoma previously treated with firstline neurosurgery, followed by the recurrence of the adenoma 6 months after therapy discontinuation (30). Thus, the present paper is the first demonstrating the complete clinical and biochemical remission simultaneously with the complete disappearance of an invasive GH-secreting pituitary macroadenoma after treatment with ATG.…”

Section: Discussionmentioning

confidence: 53%

“…Particularly, the longacting SA lanreotide has been found to be well tolerated and effective in reducing GH and IGF1 levels as well as the tumor mass in a high percentage of patients previously either untreated or treated by unsuccessful neurosurgery for acromegaly . Only three cases of total biochemical and/or radiological remission of a GH-secreting pituitary adenoma after medical therapy with long-acting SA have been reported (28)(29)(30). Moreover, few data on the effects of SA therapy discontinuation on disease control are presently available, and the majority of the authors reported the disease recurrence during a short follow-up.…”

Section: Introductionmentioning

confidence: 99%

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

Auriemma¹,

Galdiero²,

Grasso³

et al. 2010

European Journal of Endocrinology

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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.

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“…Because of its insidious onset and slow progression, the diagnosis is usually made after 4-10 years of disease progression [1]. Biochemical evidence of the disease can be found in most patients, and strict criteria for its diagnosis have been developed [2]. However, some patients may exhibit physical stigmata of acromegaly without apparent biochemical evidence, which is called fugitive acromegaly [3].…”

Section: Introductionmentioning

confidence: 99%

A case of fugitive acromegaly, initially presented as invasive prolactinoma

Lim

Lee

et al. 2010

Endocr

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Fugitive acromegaly is most commonly caused by pituitary acidophil stem cell adenomas, and is characterized by a relatively short clinical history, a large and locally invasive tumor, and relatively low hormonal activity. Here, we report an unusual case of fugitive acromegaly that initially presented as invasive prolactinoma. A 48-year-old man with a huge pituitary mass extending to the suprasellar area was referred to our hospital in December 2007. He had undergone transsphenoidal surgery in November 1999 because of a large invasive prolactinoma. The tumor had grown progressively, despite therapy with dopamine agonists. Subtle features of acromegaly were noted and serum IGF-1 levels were high (733 ng/ml). An oral glucose tolerance test revealed that basal and nadir levels of growth hormone (GH) were 1.56 and 1 ng/ml, respectively. As a therapeutic trial, long-acting octreotide (20 mg IM, monthly) was added, and the tumor size markedly reduced within 6 months on magnetic resonance imaging examination. Immunohistochemical staining of the tumor tissue obtained at the surgery in 1999 showed positive staining for GH and prolactin (PRL). Double immunofluorescence staining showed a mixed positivity for GH and PRL in the majority of tumor cells; however, the two hormones colocalized in a minority of tumor cells, indicating that the tumor was composed of three different cell types (GH, PRL, and GH/PRL). The diagnosis of fugitive acromegaly was initially overlooked in this patient because of normal serum GH levels and a lack of acromegalic features, although histological evidence for GH production was present. IGF-1 determinations would be helpful for the diagnosis of fugitive acromegaly.

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Sustained Clinical Inactivity and Stabilization of GH/IGF-1 Levels in an Acromegalic Patient after Discontinuation of Somatostatin Analogue Treatment

Cited by 4 publications

References 42 publications

Active postoperative acromegaly: sustained remission after discontinuation of somatostatin analogues

Active postoperative acromegaly: sustained remission after discontinuation of somatostatin analogues

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

A case of fugitive acromegaly, initially presented as invasive prolactinoma

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