scholarly journals Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case

Author(s):  
Simon Ryder ◽  
Jed Robusto ◽  
Thomas Robertson ◽  
Hamish Alexander ◽  
Emma L Duncan

Summary Although pituitary macroadenomas often cause mass effects on surrounding structures, it is extremely rare for pituitary lesions to disturb cerebrospinal fluid circulation. Sellar gangliocytoma-pituitary adenomas (SGPAs) are also extremely rare. Here we report the unique case of a man with the unusual combination of acromegaly from an SGPA, who presented with unilateral hydrocephalus. A 60-year-old man presented with rapid neurological deterioration, bitemporal hemianopia, and acromegalic features. Neuroimaging revealed a large sellar lesion extending superiorly into the left foramen of Monro, causing acute obstructive unilateral hydrocephalus. External ventricular drain placement improved consciousness immediately. Biochemical assessment confirmed acromegaly. Following trans-sphenoidal debulking, histology revealed a mixed gangliocytoma/sparsely-granulated somatotrophinoma. Despite the residual disease, his vision recovered remarkably, low-dose cabergoline controlled residual excess growth hormone (GH) secretion, and the residual tumour has remained extremely stable over 2 years. Hydrocephalus is an extremely rare complication of pituitary lesions, and unilateral hydrocephalus has never been reported previously. GH secretion in SGPAs is more common than for pituitary adenomas in general, raising questions regarding the aetiology and therapeutic approach to this rare combination tumour. Learning points Pituitary tumours most commonly present with symptoms related to endocrine disturbance or mass effects upon visual pathways (e.g. optic chiasm, nerves in the lateral cavernous sinus). However, extremely rarely, pituitary masses may disrupt cerebrospinal fluid (CSF) circulation resulting in hydrocephalus. Sellar gangliocytomas are very rare tumours and most of them are hybrid tumours with pituitary adenomas (SGPAs). SGPAs are typically indolent and may be functioning or non-functioning tumours. Growth hormone (GH)-producing SGPAs are less likely to respond to somatostatin agonists than classic somatotrophinomas. Primary surgical debulking via a trans-sphenoidal approach was effective in this individual, leading to the restoration of CSF circulation and improvement in visual disturbance, while also negating the need for permanent CSF diversion despite the residual tumour burden.

2005 ◽  
Vol 35 (2) ◽  
pp. 333-341 ◽  
Author(s):  
M C Zatelli ◽  
D Piccin ◽  
F Tagliati ◽  
A Bottoni ◽  
M R Ambrosio ◽  
...  

Dopamine (DA) and somatostatin (SRIF) receptor agonists inhibit growth hormone (GH) secretion by pituitary adenomas. We investigated DA subtype 2 receptor (DR2) and SRIF receptor (sst) subtypes 2 and 5 expression in 25 GH-secreting pituitary adenomas and tested in primary culture the effects on GH and prolactin (PRL) secretion of sst agonists selectively interacting with sst2 (BIM-23120), sst5 (BIM-23206), and sst2 and sst5 (BIM-23244). All adenomas expressed sst2; eight adenomas expressed both sst5 and DR2, eight sst5 but not DR2, and eight DR2 but not sst5. One tissue lacked expression of DR2 and sst5. GH secretion was inhibited by BIM-23120 in all samples, while it was reduced by BIM-23206 only in adenomas not expressing DR2. BIM-23120’s inhibitory effects correlated with sst2 and DR2 expression, whereas DR2 expression correlated inversely with BIM-23206 inhibitory effects on GH secretion. In seven mixed GH-/PRL-secreting pituitary adenomas, PRL secretion was inhibited in sst5-expressing tumors by BIM-23206, but not by BIM-23120. BIM-23244 reduced PRL secretion only in adenomas expressing sst2, sst5 and DR2. sst5 and DR2 expression correlated directly with BIM23206 inhibitory effects on PRL secretion. Our results suggest that adenomas expressing DR2 are less likely to respond to clinically available SRIF analogs in terms of GH secretion inhibition. Therefore, drugs interacting also with DR2 might better control secretion of pituitary adenomas.


1985 ◽  
Vol 63 (1) ◽  
pp. 35-38 ◽  
Author(s):  
Edward R. Laws ◽  
Bernd W. Scheithauer ◽  
Sandra Carpenter ◽  
Raymond V. Randall ◽  
Charles F. Abboud

✓ A series of 75 patients with acromegaly and immunocytochemically characterized pituitary adenomas has been analyzed. Tumors secreting growth hormone (GH) only were found in 21% of cases. The remainder had tumors immunoreactive for more than one pituitary hormone: GH and prolactin in 31%; GH, prolactin, and glycoprotein in 40%; and GH and glycoprotein in 8%. Microadenomas were surgically treated in 17 patients with a success rate of 82%. Overall, normalization of basal GH secretion (to ≤ 5 ng/ml) was achieved in 54% of cases. The implications of these findings for the pathogenesis and neurosurgical management of acromegaly are discussed.


Author(s):  
Laura Hamilton Adams ◽  
Derick Adams

Summary Co-secreting TSH and growth hormone pituitary adenomas are rare. We present a case of a 55-year-old woman who presented with symptoms of neck fullness. Ultrasound revealed multiple thyroid nodules and examination revealed several clinical features of acromegaly. She was found to have a co-secreting TSH and growth hormone pituitary macroadenoma. She underwent surgical resection followed by gamma knife radiation, which resulted in complete remission of her TSH and GH-secreting adenoma. Learning points: TSH-secreting pituitary adenomas are rare and about one-third co-secrete other hormones. Thyroid nodules are common in acromegaly and can be the presenting sign of a growth hormone-secreting pituitary adenoma. In the workup of acromegaly, assessment of other pituitary hormones is essential, even in the absence of symptoms of other pituitary hormone dysfunction. Complete remission of co-secreting GH and TSH pituitary macroadenomas is possible with surgery and radiation alone.


1995 ◽  
Vol 133 (6) ◽  
pp. 686-690 ◽  
Author(s):  
Shereen Ezzat ◽  
George Kontogeorgos ◽  
Donald A Redelmeier ◽  
Eva Horvath ◽  
Alan G Harris ◽  
...  

Ezzat S, Kontogeorgos G, Redelmeier DA, Horvath E, Harris AG, Kovacs K. In vivo responsiveness of morphological variants of growth hormone-producing pituitary adenomas to octreotide. Eur J Endocrinol 1995;133:686–90. ISSN 0804–4643 The somatostatin analog, octreotide, is an inhibitor of growth hormone (GH) secretion that has been used to treat patients with GH-producing pituitary tumors. In this study we investigated the in vivo responsiveness to treatment with this analog in patients harboring different morphological types of GH-producing pituitary adenomas. Both GH and insulin-like growth factor I (IGF-I) plasma levels in 30 patients treated with octreotide (300 μg/day) for 4 months preoperatively were compared with those from 30 patients who did not receive treatment preoperatively. Tissue samples were studied using ultrastructural and immunohistochemical techniques. Amongst patients harboring densely granulated (DG) adenomas, mean GH levels were reduced to 32 ± 9% by octreotide, to 30 ± 7% by surgery and to 26 ± 9% of baseline by both interventions. Surgery was equally as effective in lowering GH levels in patients with sparsely granulated (SG) adenomas as it was in those with DG adenomas; in patients with SG adenomas, GH levels were reduced by surgery alone to 37 ± 16% and to 24 ± 15% when performed following octreotide pretreatment. In contrast, treatment with octreotide alone in patients harbouring SG adenomas reduced GH levels to only 70 ± 13% of baseline (p < 0.02 compared to surgery alone, or surgery and octreotide). We conclude that the GH inhibitory effects of octreotide are significantly better in patients harboring DG somatotroph adenomas compared with those harboring SG adenomas. Shereen Ezzat, University of Toronto-Wellesley Hospital, 160 Wellesley St East, Toronto, Ontario M4Y-1J3, Canada


Author(s):  
Shervin Pejhan ◽  
Stan VanUum ◽  
Neil Duggal ◽  
Brian Rotenberg ◽  
Michael Mayich ◽  
...  

A 20-year-old male presented with evidence of gigantism/acromegaly. Endocrinological investigations identified elevated growth hormone levels and a failed glucose tolerance test. Imaging revealed a macroadenoma expanding the sella with encroachment on the optic chiasm and cavernous sinuses. Trans-sphenoidal resection was undertaken and a gross total removal was achieved. Histopathological features were typical of a densely granulated somatotroph adenoma with abundant growth hormone expression, scattered prolactin expression and sparse examples of fibrous bodies. Unexpectedly, the adenoma not only expressed PIT-1 but also SF-1 transcription factors. This finding suggests that the adenoma may have been pluripotent. The prognostic significance of this finding is uncertain although the patient is stable from an endocrinological and imaging perspective approximately one-year post-op. A pituitary adenoma of this nature has not been previously reported. The recent literature on atypical transcription factor expression patterns and revisions to the classification of pituitary adenomas will be reviewed.Learning ObjectivesAppreciate the rarity of dual transcription factor expression in pituitary adenomasRationalize the use of transcription factor characterization in the revised WHO classification of pituitary adenomas


1986 ◽  
Vol 112 (4) ◽  
pp. 473-480 ◽  
Author(s):  
Marco Losa ◽  
Roland Huss ◽  
August König ◽  
O. Albrecht Müller ◽  
Klaus von Werder

Abstract. Theophylline enhances GH-secretion in vitro, whereas in vivo a slight decrease of basal GH-levels has been observed. In the present study the effect of theophylline on the GH-responsiveness to acute and continuous administration of growth hormone releasing hormone (GHRH) was investigated. The following protocol was performed. GHRH study. Fifty μg GHRH was given as an iv bolus followed by constant GHRH-infusion (100 μg/h) over 2 h after which another GHRH bolus of 50 μg was given. GHRH plus theophylline study. GHRH was administered as in the first study and theophylline was infused at a constant rate of 3.56 mg/min over 3 h, starting one h before the GHRH bolus. Theophylline study. Only saline and theophylline were infused. GHRH alone led to a GH-rise within 30 min with a maximum of 22.8 ± 7.2 ng/ml (mean ± se) after which GH-levels decreased despite continuous GHRH-infusion to a nadir of 12.1 ± 4.4 ng/ml at 105 min. The second GHRH bolus led to a minimal GH-increase (13.3 ± 6.4 ng/ml at 135 min). Theophylline administration resulted in blunting of the GH-response to GHRH in all volunteers, with GH levels fluctuating between 4–6 ng/ml throughout GHRH-administration. Theophylline alone did not affect GH-levels in three subjects studied, whereas in the other one a GH secretory episode 90 min after administration of the drug was observed. Prl showed a minimal increase only after the second GHRH bolus (from 254.2 ± 7.7 μU/ml to 317.3 ± 96.0 μU/ml in study 1, and from 139.3 ± 26.9 μU/ml to 193.0 ± 32.6 μU/ml in study 2), TSH-levels did not change during any of the test-procedures and GHRH-levels were comparable in both study 1 and 2. Free fatty acids (FFA) rose progressively after theophylline administration (from 0.68 ± 0.10 mEq/l to 1.06 ± 0.08 mEq/l in study 2, and from 0.64 ± 0.12 mEq/l to 1.09 ± 0.05 mEq/l in study 3), but also after GHRH alone (from 0.50 ± 0.05 mEq/l to 0.78 ±0.11 mEq/l). This shows that therapeutical doses of theophylline blunt the GH-response to GHRH in normal subjects. The mechanisms involved may be a competition for the adenosine receptor at the pituitary, or an indirect effect mediated by the rise of FFA levels.


Skull Base ◽  
2011 ◽  
Vol 21 (S 01) ◽  
Author(s):  
Samuel Shin ◽  
Matthew Tormenti ◽  
Sue Challinor ◽  
Tian Wang ◽  
Juan Fernandez-Miranda ◽  
...  

1965 ◽  
Vol 49 (1) ◽  
pp. 1-16 ◽  
Author(s):  
M. Apostolakis

ABSTRACT A method for the extraction of prolactin from human pituitary glands is described. It is based on acetone drying, distilled water extraction, acetone and isoelectric precipitation. Two main products are obtained: Fraction R8 with a mean prolactin activity of 12.2 IU/mg and fraction U8 with a mean prolactin activity of 8.6 IU/mg. The former fraction does not contain any significant gonadotrophin activity and the latter contains on an average 50 HMG U/mg. In both cases contamination with ACTH and MSH is minimal. The growth hormone activity of both these fractions is low. It is postulated that in man too, prolactin and growth hormone are two distinct hormones. A total of 1250 human pituitary glands have been processed by this method. The mean prolactin content per pituitary gland has been found to be 73 IU.


1974 ◽  
Vol 75 (3) ◽  
pp. 497-502
Author(s):  
Mayer B. Davidson ◽  
Roger M. Steele

ABSTRACT Since fructose is normally metabolized in diabetics and has recently been shown to stimulate GH secretion, it was used to assess GH responses in diabetics. Fourteen diabetics (9 on insulin) and 8 controls matched for weight were studied. Fructose, infused over 10 min, was compared to arginine, infused over 30 min, both at 0.5 g/kg. Samples were collected at 0, 30, 60, 90 and 120 min and GH responses assessed as area under the curve minus the fasting area. There was no significant difference between the GH responses in diabetics and controls to either agent. Responses to arginine and fructose were significantly correlated (r = 0.60, P < 0.01) in all subjects, but not related to therapy, duration of disease or fasting glucose (75–287 mg/100 ml) in the diabetics. Oral glucose blunted the GH response to fructose in 2 controls. It is concluded that 1) fructose can stimulate GH secretion in male diabetics; 2) however, fructose-stimulated GH responses are not increased in diabetes mellitus.


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