Multidisciplinary treatment of giant invasive prolactinomas in paediatric age: long-term follow-up in two children

Fraioli, Mario Francesco; Novegno, Federica; Catena, Elisabetta; Fraioli, Chiara; Moschettoni, Laura

doi:10.1007/s00381-010-1129-7

Cited by 9 publications

(6 citation statements)

References 24 publications

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“…The giant tumors we observed looked like craniopharyngiomas (solid, cystic ± calcified) in 25%, but posterior pituitary function was deficient in only one case. In agreement with many authors,[ 28 29 30 31 32 33 34 ] we noted that bone destruction of the surrounding structures mimicking skull base tumors was relatively common. But, as total disappearance of giant prolactinomas takes years, it was difficult for us to know if those pituitary tumors destroying the skull base were eutopic ones or ectopic rising from the clivus.…”

Section: Discussionsupporting

confidence: 93%

Neurological, psychiatric, ophthalmological, and endocrine complications in giant male prolactinomas: An observational study in Algerian population

Chentli

Azzoug

Daffeur

et al. 2015

Indian J Endocr Metab

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Introduction:Prolactinomas are less frequent, but more invasive in males. Giant ones (≥4 cm) are extremely rare in literature. Their neurological, psychiatric and endocrine complications are life threatening. Our aim was to report the largest mono center series in order to analyze their frequency, their characteristics, and their complications.Subjects and Methods:All patients had clinical examination, hormonal, ophthalmological, and radiological assessment based on computed tomography scan and cerebral magnetic resonance imaging. Positive diagnosis was based on clinical symptoms, high prolactin ± immunohistochemy study. Mixed adenomas were excluded by hormonal exploration and immunohistochemy. For those who received medical treatment only, a reduction in tumor size was considered a supplementary positive point for the diagnosis.Results:Among 154 male prolactinomas seen between 1987 and 2013, we observed 44 giant tumors (28.5%). Median age = 36 years, and 38.3% were under 30. Median tumor height = 53.95 mm (40–130) and median prolactin = 15,715 ng/ml (n < 20). Solid and cystic aspect ± calcifications was observed in 25%. 42 had cavernous sinuses invasion. Other invasions were: Posterior= 65.9%, anterior= 63.6%, temporal= 15.9% and frontal = 9%. For endocrine complications: Hypogonadism = 98.4%, thyrotroph and corticotroph deficits were seen in respectively 34%, and 32%. Posterior pituitary insufficiency was observed in one case. For ophthalmological complications: Optic atrophy = 46%, Ptosis = 6.8%, diplopia/strabismus = 4.5%. Neurological complications were: Memory loss and/or unconsciousness = 18.2%, epilepsy = 15.9%, frontal syndrome = 9% and obstructive hydrocephalus = 6.8%.Conclusion:Giant prolactinomas account for 28% in our population. Severe neurological complications are frequent. But, obstructive hydrocephalus is rare, which argues for a slow progression.

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Section: Discussionsupporting

confidence: 93%

Neurological, psychiatric, ophthalmological, and endocrine complications in giant male prolactinomas: An observational study in Algerian population

Chentli

Azzoug

Daffeur

et al. 2015

Indian J Endocr Metab

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“…Among 16 cases collected from the literature and fulfilling all definition criteria of a true giant prolactinoma (55,56,72,95,105,106,107,108,109,110,111,112,113,114), there were 15 boys with a mean age of 10.8 years (range: 6-14 years) and a mean tumour diameter of 65 mm (range: 40-99 mm) and only one 14.5-year-old girl (95). These giant tumours may also lead to the same various neurologic complications as in adults, such as blindness and cranial nerve palsies (106), hydrocephalus (56,72), proptosis (56,105,108) or nasopharyngeal symptoms (71,114), and they should be considered in the differential diagnosis of every large and erosive paediatric skull base tumour.…”

Section: Giant Prolactinoma In Childrenmentioning

confidence: 99%

THERAPY OF ENDOCRINE DISEASE: The challenges in managing giant prolactinomas

Maiter

Delgrange

2014

European Journal of Endocrinology

161

166

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Giant prolactinomas are rare tumours, representing only 2-3% of all prolactin (PRL)-secreting tumours and raising special diagnostic and therapeutic challenges. Based on several considerations developed in this review, their definition should be restricted to pituitary adenomas with a diameter of 40 mm or more, significant extrasellar extension, very high PRL concentrations (usually above 1000 mg/l) and no concomitant GH or ACTH secretion. Giant prolactinomas are much more frequent in young to middle-aged men than in women, with a male to female ratio of about 9:1. Endocrine symptoms are often present but overlooked for a long period of time, and diagnosis is eventually made when neurologic complications arise from massive extension into the surrounding structures, leading to cranial nerve palsies, hydrocephalus, temporal epilepsy or exophthalmos. PRL concentrations are usually in the range of 1000-100 000 mg/l, but may be underestimated by the so-called 'high-dose hook effect'. As in every prolactinoma, dopamine agonists are the first-line treatment allowing rapid alleviation of neurologic symptoms in the majority of the cases, a significant reduction in tumour size in three-fourths of the patients and PRL normalization in 60-70%. These extensive tumours are usually not completely resectable and neurosurgery has significant morbidity and mortality. It should therefore be restricted to acute complications such as apoplexy or leakage of cerebrospinal fluid (often induced by medical treatment) or to patients with insufficient tumoural response or progression. Irradiation and temozolomide are useful adjuvant therapies in a subset of patients with aggressive/invasive tumours, which are not controlled despite combined medical and surgical treatments. Because of these various challenges, we advocate a multidisciplinary management of these giant tumours in expert centres.

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“…To the best of our knowledge, there are two reports that consider more than ten-year long-term clinical outcomes of giant prolactinomas. Fraioli et al utilized multidisciplinary treatment for IGPs in two children and finally reached satisfactory control of the disease with a follow-up period of 13 and 14 years [26]. Fernandes et al reported a giant prolactinoma with 10 years of DA treatment, whose prolactin levels decreased by 96.8% with an effective reduction in tumor size [27].…”

Section: Discussionmentioning

confidence: 99%

Long-Term Clinical Outcomes of Invasive Giant Prolactinomas after a Mean Ten-Year Followup

Cai

Lin

et al. 2016

International Journal of Endocrinology

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Objective. The aim of this study is to observe clinical outcomes after more than ten years of followup in a group of patients with invasive giant prolactinomas (IGPs) treated with dopamine agonists (DAs). Methods. Twenty-five patients met the criteria of IGPs, among which 16 patients primarily received bromocriptine (BRC) and the other nine had undergone unsuccessful microsurgery prior to BRC treatment. Results. After a mean follow-up period of 135.5 ± 4.7 months, the clinical symptoms in all patients improved by different degrees. Tumor volume was decreased by a mean of 98.6%, and the tumors of 19 patients had almost completely disappeared. The mean duration of treatment at maximal doses of BRC was 48.5 months. At the last follow-up visit, nineteen patients had normal PRL levels, and 14 of these patients had received the low-dose BRC treatment (at an average of 2.9 ± 0.3 mg/d). Younger patients < 25 years had a significantly higher rate of persistent hyperprolactinemia after long-term BRC treatment (p = 0.043). Conclusion. DAs are a first-line therapy for IGPs because they can effectively achieve long-term control in both shrinking tumor volume and normalizing the PRL level, and majority of patients need low-dose DA maintenance. Younger patients are prone to persistent hyperprolactinemia despite long-term DA treatment.

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Multidisciplinary treatment of giant invasive prolactinomas in paediatric age: long-term follow-up in two children

Cited by 9 publications

References 24 publications

Neurological, psychiatric, ophthalmological, and endocrine complications in giant male prolactinomas: An observational study in Algerian population

Neurological, psychiatric, ophthalmological, and endocrine complications in giant male prolactinomas: An observational study in Algerian population

THERAPY OF ENDOCRINE DISEASE: The challenges in managing giant prolactinomas

Long-Term Clinical Outcomes of Invasive Giant Prolactinomas after a Mean Ten-Year Followup

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