Cystic Prolactinoma Research Articles

Audit on the Management of Cystic Prolactinomas

Audit on the Management of Cystic Prolactinomas

OR2804 Endocrine And Vision Outcomes Following 90Yttrium Therapy For Cystic Sellar Lesions—A Prospective Cohort Study

Abstract Disclosure: M.A. MacLean: None. S. Ahmad: None. A.L. Hebb: None. A. Tahir: None. R. Greene: None. D.B. Clarke: None. S.A. Imran: None. Background: Untreated cystic sellar lesions can cause vision loss, endocrine dysfunction, and obstructive hydrocephalus related to growth and mass effect. Microsurgical resection, stereotactic aspiration, and radiation treatment strategies are limited by significant complications and high risk of recurrence. Stereotactic intracavitary irradiation with the beta-emitting isotope 90Yttrium (90Yt) has been proposed as an alternative to mitigate these challenges. Long term-efficacy data is lacking. Here, our objective was to prospectively assess the effect of 90Yt on cyst volume, endocrine, and vision outcomes. Methods: We conducted a Health Canada approved, single-centre, prospective cohort study. Clinical outcome measures included cyst volume measured on magnetic resonance imaging, formal visual field (VF) testing, and hypothalamic-pituitary (HP) hormonal function. All patients provided informed consent and underwent right frontal stereotactic insertion of 90Yt colloid (200 Gy cyst wall). Results: Twenty-two patients (55% females) received 90Yt therapy. Mean age was 63.5 (SD 15.4) years with no difference between genders (p = 0.055). Mean duration of follow-up was 52 months (range 5-113). Cystic lesions included: craniopharyngioma (86%), Rathke’s cleft cyst (9%) and cystic prolactinoma (5%). Mean tumor cyst volumes were reduced by 77% at 12-month follow-up (pre-operative: 5.2, SD 4.5 cc; 12 months post-operative: 1.2, SD 1.5 cc; p = 0.002). Males had larger tumor volumes at baseline (7.7, SD 5.0 cc vs. 3.2, SD 2.8; p = 0.02) and 3 months (4.7, SD 3.4 vs. 1.8, SD 2.0; p = 0.04) but these differences had resolved by 9-month follow-up. Pre-operative clinical features included: VF deficit (36%), VF deficit plus headache (36%), headache alone (9%), hydrocephalus (5%), and 14% were asymptomatic. HP hormone dysfunction was observed in 59% of patients pre-operatively (27% complete; 73% partial) and 68% post-operatively (p = 0.50); specifically, no differences were found pre- and post-operatively for secondary hypogonadism (50% vs. 55%), hypothyroidism (45% vs. 59%), hypoadrenalism (36% vs. 27%), growth hormone deficiency (27% vs. 23%), diabetes insipidus (23% vs. 27%), and hyperprolactinemia (36% vs. 36%) (all p > 0.05). Pre-operative VF deficits were found in 68% of patients, of which 32% normalized and 36% remained stable (p = 0.180). Worsened vision was detected in 9% of patients post-operatively. Treatment failure requiring conventional surgery occurred in 18% of patients. Conclusions:90Yt therapy significantly reduced tumor cyst size and stabilized both endocrine function and vision in patients with cystic lesions of the sellar region. Presentation: Sunday, June 18, 2023

P.107 Endocrine and vision outcomes following 90Yttrium therapy for cystic sellar lesions: a prospective cohort study

Background: Due to high risk of recurrence and complications associated with microsurgical resection or aspiration of enlarging cystic sellar lesions, stereotactic intracavitary irradiation with 90Yttrium (90Yt) has been proposed as an alternative to mitigate these challenges. Long-term efficacy data for this procedure is lacking. Methods: We conducted a Health Canada approved, single-centre, prospective cohort study to assess the effect of 90Yt therapy on cyst volume, visual field (VF) testing and endocrine function. All patients underwent right frontal stereotactic insertion of 90Yt colloid (200 Gy cyst wall). Results: 22 patients [mean age = 63.5 (± 15.4) years] received 90Yt therapy. Mean follow-up was 52 (5-113) months. Cysts included: craniopharyngioma (86%), Rathke’s cleft cyst (9%) and cystic prolactinoma (5%). Mean cyst volume reduction was 77% at 12-months (5.2 ± 4.5 cc to 1.2 ± 1.5 cc; p = 0.002). Rate of pre- and post-op hormonal dysfunction was 59% and 68%, respectively (p = 0.50). Pre-operative VF deficits were found in 68% of patients, of which 32% normalized and 36% remained stable (p = 0.180). Worsened vision was detected in 9% of patients post-operatively. Conclusions: 90Yttrium therapy significantly reduces sellar cyst size without having a deleterious effect on vision and endocrine function.

Cystic Prolactinoma unmasking Meningoencephalocele: When medical treatment leads to surgery

Searchable abstracts of presentations at key conferences in endocrinology ISSN 1470-3947 (print) | ISSN 1479-6848 (online)

Operative treatment of cystic prolactinomas: a retrospective study

BackgroundThe optimal therapeutic approach for cystic prolactinomas remains unclear. This study aimed to evaluate the remission rates of prolactinoma patients after surgical treatment and the risk factors affecting postoperative remission in cystic prolactinoma patients.MethodsThe clinical data were retrospectively compiled from 141 patients with prolactinomas (including 41 cases of cystic prolactinomas, 21 cases of solid microprolactinomas and 79 cases of solid macroprolactinomas) who underwent transsphenoidal surgery (TSS) between April 2013 and October 2021 at the First Affiliated Hospital of Sun Yat-sen University.ResultsEarly postoperative remission was achieved in 65.83% (n = 27/41) of cystic prolactinomas, 80.95% (n = 17/21) of solid microprolactinomas and 40.51% (n = 32/79) of solid macroprolactinomas. The mean length of follow up in all patients was 43.95 ± 2.33 months (range: 6-105 months). The follow-up remission rates were 58.54%, 71.43% and 44.30% in cystic, solid micro- and solid macroprolactinomas, respectively. For cystic prolactinomas, the early postoperative remission rates in the patients with preoperative dopamine agonists (DA) treatment were significantly higher than those without preoperative DA treatment (p = 0.033), but the difference in the follow-up remission rates between these two groups was not significant (p = 0.209). Multivariate stepwise logistic regression analysis indicated that tumor size and preoperative prolactin (PRL) levels < 200 ng/ml were independent predictors for early postoperative remission in cystic prolactinomas.ConclusionFor cystic prolactinomas, tumor size and preoperative PRL levels were independent predictors of early postoperative remission. Preoperative DA therapy combined with TSS may be more beneficial to cystic prolactinoma patients.

Analysis of Treatment Outcomes of Patients With Cystic Prolactinomas: Report of Four Cases

Abstract Background: Prolactinomas are the most common functional pituitary adenomas. Treatment with dopamine agonists is considered as first-line therapy. Cystic prolactinoma is a variant prolactinoma. . It was assumed that since cystic area does not contain dopamine receptors, cystic prolactinomas would not respond to medical treatment. However, in recent years, it has been shown that cystic prolactinomas can also be treated with dopamine agonists, and medical treatment has become more preferred than surgical treatment. In this report, we aimed to evaluate the treatment effectiveness of patients with cystic prolactinomas who treated medically in our clinic. Materials and methods: Twenty-nine prolactinoma patients who were followed up in our hospital between 2019-2020 were evaluated retrospectively. Four patients (13%) were being followed up for cystic prolactinoma. Radiological images, laboratory findings, patients records were reviewed. Results: Four patients were being followed up for cystic prolactinoma and all were women. The average age of the patients followed up for cystic prolactinoma was 36 years. 3 patients (75%) had menstrual irregularity, 2 (50%) patients had galactorrhea, and 1 patient (25%) had headache at the time of admission. Tumor size regressed with medical treatment in two patients and the tumor disappeared completely in one patient. Although the symptomatic improvement and normalization of prolactin was achieved in one of our patients, the lesion did not shrink. We continued with medical treatment in this patient since there was no indication for surgery. Discussion: There was conflicting results in the literature regarding the management of cystic prolactinomas. Recently, dopamin agonist therapy is suggested as first- line treatment option in cystic prolactinomas. A study reported male predominance for cyst formation but we observed female predominance in our report. Faje et al. also reported female predominance in their study. Presentation of our patients were not different from solid prolactinomas. And although we evaluated very limited number of patients in our report, all of our patients respond to medical treatment which was consistent with recent literature.

SAT-260 Resolution of a Cystic Macroprolactinoma with Dopamine Agonist Therapy

Background. Prolactinomas with cystic regions occupying ≥ 50% of tumor volume are classified as cystic prolactinomas. They appear to arise from tumor necrosis or hemorrhage. Though Pituitary Society guidelines recommend surgery as first-line treatment for cystic prolactinomas, there is evidence that cystic prolactinomas are hormonally and anatomically responsive to treatment with dopamine agonists (DA). We present a case of a cystic macroprolactinoma fully responding to treatment with cabergoline. Case. A 31 year old male was referred for evaluation of gynecomastia, diminished libido, and erectile dysfunction that occurred over the course of eight months. Bilateral glandular breast tissue with a slight right asymmetry was present. Testes were 15–20 mL, soft, and without masses. Phallus and pubic hair were unremarkable. Morning (8 AM) testosterone, estradiol, and LH levels were 92 ng/dL (348–1197), 13.4 pg/mL (7.6–42.6), and 2.0 mIU/mL (1.7–8.6), respectively. Prolactin level was 331 ng/mL (4–15), and MRI with pituitary protocol revealed a 1.4 x 1.0 x 1.5 cm cystic mass with peripheral rim enhancement and extension into the suprasellar cistern. No mass effect on the optic chiasm was observed. Somatotroph, thyrotroph, and corticotroph axes were unaffected. Cystic macroprolactinoma causing hyponadotropic hypogonadism complicated by gynecomastia was diagnosed. The patient chose initial management with cabergoline 0.25 mg twice weekly. Three months after starting treatment, libido and erectile function had recovered to baseline, 8 AM testosterone was 607 ng/dL, and hyperprolactinemia was well controlled (4.2 ng/mL). At six months, anatomic resolution of the cystic macroprolactinoma was demonstrated on repeat pituitary MRI. Plastic surgery was required for management of gynecomastia. Conclusions. Surgery is recommended for management of cystic prolactinomas due to concern that a lack of dopamine receptors in the cystic components of tumors will make DA ineffective reducing tumor size. However, this case joins a growing body of evidence that DA can treat hyperprolactinemia and induce regression of cystic macroprolactinomas. For example, in a retrospective case review of patients with cystic macroprolactinomas at Massachusetts General Hospital, persistent cyst reduction occurred in 20 of 22 patients treated initially with DA, and median reduction in cyst volume exceeded 80 percent. Median time to documented cyst reduction was approximately six months, and there was no difference in degree of cyst reduction for patients treated with bromocriptine or cabergoline. The response of cystic prolactinomas to DA is similar to solid prolactinomas, and DA are reasonable initial treatment for cystic macroprolactinomas without clear indications for surgery.

An interesting case of cystic prolactinomas.

An interesting case of cystic prolactinomas.

Surgical management considerations in cystic prolactinomas-a single center case series.

The optimal treatment of prolactinomas with a predominantly cystic component remains poorly defined. The cystic tumor component is considered to respond less favorably to medical treatment, thereby advocating surgical management. The purpose of this study was to assess remission rates in surgically treated cystic prolactinomas, and to compare outcomes to similarly treated solid micro- and macroprolactinomas. Clinical and imaging data were retrospectively compiled from 56 patients who underwent transsphenoidal resection, for symptomatic prolactinomas, from 2004 to 2018, at a single academic institution. Pituitary adenomas were subdivided according to tumor size and tumor consistency: cystic prolactinomas (>50% cystic tumor component) n = 17; solid microprolactinomas (<10 mm) n = 10; and solid macroprolactinomas (≥10 mm) n = 29. Remission was defined as a prolactin level of <10 ng/dl either immediately postoperative or at a later time point. Median tumor size was 15 mm for cystic prolactinomas, 7 mm for solid microprolactinomas, and 25.5 mm for solid macroprolactinomas. Remission was achieved in 76% (n = 13/17) of surgically treated cystic prolactinomas, 100% (n = 10/10) of solid microprolactinomas, and 24% (n = 7/29) of solid macroprolactinomas. More than 44% of solid macroprolactinomas had a Knosp grade > 3, while most cystic prolactinomas (93.8%) and all solid microprolactinomas (100%) had a Knosp grade ≤ 2. Despite their large tumor size (≥10 mm), high remission rates can be expected with surgically treated cystic prolactinomas. This case series of cystic prolactinomas demonstrates the successful use of transsphenoidal surgery as a favorable, and a potentially curative alternative to dopaminergic therapy in this patient population.

SUN-443 Surgical Management of Cystic Prolactinomas

Purpose: Treatment of prolactinomas with a predominantly cystic component remains controversial. This study assessed remission rates in surgically-treated cystic prolactinomas, compared to solid micro- and macroprolactinomas. Methods: Charts were retrospectively analyzed for 57 patients who underwent transsphenoidal resection (TSS) for prolactinomas, from 2004 to 2018, by two experienced neurosurgeons. Tumors were subdivided into: cystic prolactinomas (n=17, >50% initial cystic component), microprolactinomas (n=10, <10 mm) and macroprolactinomas (n=30, ≥10 mm). Patients underwent TSS primarily for reasons of dopamine agonist (DA) intolerance, non-responders or patient preference. Remission was defined as either immediate postoperative (POD #1-3) prolactin (PRL) level of <10 ng/dl, or a normalized PRL level at a later time point. Results: 30 females and 27 males were included in the study, with a mean age of 33.5 yrs. Median tumor size for the cystic, micro- and macroprolactinomas were 15 mm, 7 mm and 22 mm, respectively. The median duration for DA treatment for the cystic, micro- and macroprolactinoma groups, prior to TSS, were 6, 24 and 12 months, respectively. Median pre-operative PRL levels were lowest with cystic prolactinomas, compared to the micro- and macroprolactinomas at 23.9 ng/ml (Standard deviation (SD)-136.7 ng/ml), 87.9 ng/ml (SD 80 ng/ml) and 229 ng/ml (SD 471.1 ng/ml), respectively (p=0.015). Immediate post-op PRL levels were significantly lower for cystic prolactinomas (n=10, 2.65 ng/ml, SD 4.72 ng/ml) than macroprolactinomas (n=19, 57 ng/ml, SD 159.4 ng/ml) (p=0.025) and for microprolactinomas (n=9.4 ng/ml, SD 3.9 ng/ml) than macroprolactinomas (p=0.030). At the last post-op f/u, no microprolactinoma patient required DA treatment, while 4 patients (24%) with cystic prolactinomas and 18 patients (60%) with macroprolactinomas still required DA therapy. Remission was ultimately achieved in 76% (n=13/17) of surgically-treated cystic prolactinomas, 100% (n=10/10) of microprolactinomas and 23% (n=7/30) of macroprolactinoma patients, at a median f/u of 6, 2.5 and 25 months, respectively. Conclusions: Surgical treatment for cystic prolactinomas, despite their macroadenoma size, resulted in high remission rates (76%), which was comparable to surgically-resected microprolactinomas and superior to macroprolactinomas. This case series supports TSS for this subset of prolactinoma patients.

Management of cystic prolactinomas: a review.

Cystic prolactinoma is a variant of prolactin-secreting pituitary adenoma. The strategies for the management of cystic prolactinoma have not been addressed thoroughly in clinical guidelines. A literature search was performed using Pubmed to review the current approaches to the treatment of cystic prolactinoma. Transsphenoidal resection is an effective and relatively safe approach for the treatment of cystic prolactinoma, however, morbidity of surgery is dependent on the skill of the surgeon. Emerging studies allude to the efficacy and safety of dopamine agonists in the management of cystic prolactinoma. Dopamine agonists are associated with considerable rates of clinical improvement and tumor shrinkage, hence reducing the need for surgical intervention. Recent studies suggest that dopamine agonist therapy may be an effective and safe treatment option in a considerable portion of patients with cystic prolactinomas. We suggest that dopamine agonists should be considered as a first-line therapy for cystic prolactinoma in the absence of indications for early surgical intervention.

A case of cystic prolactinoma responsed to medical treatment

Searchable abstracts of presentations at key conferences in endocrinology ISSN 1470-3947 (print) | ISSN 1479-6848 (online)

Significance of surgical management for cystic prolactinoma.

It is generally accepted that dopamine agonists (DA) represent the first-line treatment for most patients with prolactinoma, and patients become candidates for surgical intervention when DA is contraindicated. Surgical indication for cystic prolactinoma remains controversial. This study was performed to investigate the significance of surgery for cystic prolactinoma. A total of 28 patients that underwent transsphenoidal resection of prolactinoma between February 2004 and May 2016 were reviewed. Five consecutive patients with cystic prolactinoma were included in this study. Our surgical strategy for cystic prolactinoma was categorized as follows: first, when the purpose of surgical resection was normalization of the prolactin level, aggressive resection was performed; second, when volume reduction was essential to relieve the visual symptoms and headache, internal decompression was performed followed by DA therapy. The clinical outcomes were analyzed accordingly. All cystic prolactinoma were resected via the transsphenoidal approach without any complications, and all symptoms including visual impairment and hypogonadal activity were finally relieved combined with medication. Surgery for cystic prolactinoma could be a better option. Transsphenoidal surgery is relatively safe to remove the cystic prolactinoma, additionally it can normalize the prolactine level and achieve adequate and rapid decompression of optic chiasm. The risk of transsphenoidal surgery is highly dependent on the skill of the surgeon and treatment decision for cystic prolactinoma needs to be individualized for each patient.

Dopamine Agonists Can Reduce Cystic Prolactinomas.

Cystic prolactinomas are considered resistant to volume reduction by dopamine agonists (DAs). Although several individual case reports and small case series have suggested that DAs may reduce these lesions, larger series using standardized imaging metrics are lacking. The objectives of the study were to assess the efficacy of DAs on cyst size in patients with predominantly cystic prolactinomas and to characterize the clinical course and treatment outcomes in these patients. This study was a retrospective review. The study was conducted at a tertiary referral center. The study comprised 30 adults with cystic prolactinomas treated at Massachusetts General Hospital. The interventions included treatment with Das and transsphenoidal surgery. Cyst volume calculated by a commercial software and pituitary hormone function were measured. Median age was 31.5 years (interquartile range [IR] 24.5-39.2), and 24 of 30 were female. Median length of follow-up was 3.05 years (IR 1.04-5.28). Twenty-three of 30 patients received initial treatment with DAs. Median cyst volume for these patients was 435 mm3 (IR 255-1785). Persistent cyst reduction occurred in 20 of 22 patients after DA treatment. Median cyst volume reduction was 83.5% (IR 48.8-96.2). Median time to cyst reduction was 24.6 weeks (range 2.6-73). Chiasm compression resolved in four of five patients, and nongonadal anterior hypopituitarism improved in five of six. Transsphenoidal surgery was ultimately performed in 15 of 30 patients. Significant cyst reduction occurred in the majority of patients treated with DAs, including those with larger lesions and chiasm compression. This study is the first formal analysis of cyst reduction with DAs in patients with cystic prolactinomas, and contrary to long-held assumptions, our results suggest that medical therapy may be effective in many such patients.

Non-surgical management of cystic prolactinomas

Cystic prolactinomas are considered not amenable to dopamine agonist therapy. We present the results of dopamine agonist therapy in six patients with cystic prolactinomas. The inclusion criteria of patients were: (i) cystic macroadenomas with the cyst occupying more than 50% of the tumour volume; (ii) a serum prolactin value more than 150 ng/mL. All patients were males with a mean age of 35 years. The clinical presentations were erectile dysfunction in 66.6%, visual deficits in 50% and headache in 50% of patients. All patients were treated with bromocriptine only except one who was treated with both bromocriptine and cabergoline. The mean duration of follow up was 57.1 months. At the final follow-up 50% of patients had hormonal cure, 50% had radiological cure and 50% had reduction in the size of the tumour. Hence, it is appropriate to consider dopamine agonist therapy in patients with cystic prolactinomas before considering surgery.

Operative treatment of prolactinomas: indications and results in a current consecutive series of 212 patients

Medical therapy with dopamine agonists (DA) is the primary treatment of choice in most patients with prolactinomas. 'Classical' surgical indications are intolerance or lack of efficiency of DA therapy. Focusing on a possible shift of recent indications, we retrospectively analyzed our results of surgical treatment in prolactinomas. Between 1990 and 2005, we have operated on 212 consecutive patients with prolactinomas. Surgical indications were divided into 'classical' indications and 'modern' indications defined as cystic prolactinomas or patients with microprolactinomas who individually decided on a primary surgical treatment. Initial overall remission was accomplished in 53.2% including giant prolactinomas. However, in microadenomas, the remission rate was significantly higher with 91.3%. Overall remission at the latest follow-up was 42.7%, but 72.5% in intrasellar tumors, 80% in cystic prolactinomas, and 84.8% in microprolactinomas. The overall recurrence rate was 18.7%. Relapse of hyperprolactinemia in microprolactinomas was 7.1%. In our series, continually less patients were surgically treated for 'classical' indications. By contrast, the number of patients who individually decided on a primary surgical therapy has increased considerably. Remission rates after surgical treatment of prolactinomas remain excellent, particularly in microadenoma and intrasellar macroadenomas, whereas morbidity of transsphenoidal surgery is low in the hands of experienced pituitary surgeons. Our remission rates not only confirm the already interdisciplinarily accepted surgical indications, but also emphasize the value of primary transsphenoidal surgery as a discussion-worthy alternative to dopaminergic therapy in young patients with microprolactinomas or cystic tumors.

Cystic giant prolactinoma in childhood

In childhood and adolescence, pituitary adenomas are rare and half are prolactinomas. However, cystic giant prolactinoma in prepuberty is extremely rare. In this report, we present a 10-year-old boy with a cystic giant prolactinoma who was treated with two-stage surgery as the tumor was dumbbell shaped. To our knowledge, this is the second reported case of a cystic giant prolactinoma in a prepubertal child.