Recurrent nasal bleed as initial presenting complaint: a rare complication of giant prolactinoma in a 39-year-old

Abstract

Introduction and importance:

Giant prolactinoma is a rare endocrine disorder that presents with unusually large size and markedly high serum prolactin levels. The objective of this report is to show the aggressive invasive nature of giant prolactinoma with rare clinical presentation of recurrent nasal bleeds and the use of cabergoline in the successful management of patients having severe symptoms of extreme headaches and bitemporal hemianopia along with intracranial invasion of tumor.

Case presentation:

A 39-year-old married Asian male, presented with severe headaches, blurred vision in the left eye, and recurring nasal bleeding. Perimetry revealed bitemporal hemianopia. MRI showed a well-defined mass in the sellar/suprasellar region (3.7 × 4.0 × 3.8 cm). Initial prolactin levels were >4000 ng/ml and testosterone levels were 69 ng/dl (300–890 ng/dl). The patient after being diagnosed with Giant Prolactinoma started treatment with cabergoline which reduced prolactin levels to 14 ng/ml and the mass to 1.7 × 1.9 × 1.4 cm, though testosterone levels remained low at 134 ng/dl.

Clinical discussion:

Giant prolactinomas present significant diagnostic and therapeutic challenges due to their size, invasiveness, and resistance to conventional treatments. This case report demonstrates the successful management of a giant prolactinoma, treated initially with cabergoline that effectively reduced prolactin levels and tumor size, highlighting its role as a first-line therapy. Comprehensive management involved adjusting cabergoline dosage and introducing testosterone replacement therapy for hypogonadism.

Conclusion:

In conclusion, managing giant prolactinomas requires a multidisciplinary approach, with dopamine agonists as the cornerstone of treatment. Individualized strategies, adjunctive therapies, and regular monitoring ensure comprehensive care.

Introduction

Pituitary gland tumors that release prolactin are known as prolactinomas and are responsible for about 40% of all pituitary adenomas^[1^]. Giant prolactinomas are a rare form of pituitary adenoma, typically seen in men, characterized by extremely elevated prolactin levels and symptoms related to the tumor’s size^[2^]. Invasive giant prolactinomas are characterized by: (1) a tumor diameter greater than 4 cm; (2) serum prolactin levels exceeding 1000 ng/ml; and (3) symptoms resulting from mass effect or hyperprolactinemia^[3^]. Cranial nerve palsies, visual disturbances, and hypopituitarism can result due to compression of the suprasellar and cavernous sinus^[4^]. Men may report symptoms such as hypogonadism, erectile dysfunction, visual disturbances, weakness, and headaches, while galactorrhea is uncommon. These symptoms in men often go unnoticed for extended periods, sometimes even years, before diagnosis. In women, typical symptoms include primary or secondary amenorrhea, headaches, and visual impairment, with galactorrhea more commonly seen in younger women^[5^]. Nasal bleeding due to intranasal tumor extension is an uncommon complication of invasive giant prolactinomas^[6^]. This case report aims to show the aggressive invasive nature of giant prolactinoma with a rare first presentation with recurrent nasal bleeds and the use of cabergoline in successfully managing this case. This case report complies with the TITAN Guidelines 2025 on the declaration and use of artificial intelligence in research and publishing^[7^]. No AI was used in the research or manuscript preparation. The case has been written in accordance with CARE guidelines^[8^].

HIGHLIGHTS

• This case report aims to show the aggressive invasive nature of giant prolactinoma with a rare first presentation with recurrent nasal bleeds.

• Dopamine agonists like cabergoline prove to be the first-line agents in treating giant prolactinomas which significantly reduced prolactin levels to normal.

• There is a need of regular and vigilant monitoring and also informing the patient of potential complications such as CSF rhinorrhea which was managed surgically in this case.

Case presentation

A 39-year-old married Asian male, weighing 114 kg (BMI 33.67 kg/m²) and having a height of 184 cm, presented with a complex medical profile characterized by severe headaches, blurred vision from the left eye, and recurring nasal bleeding. But, he did not have other symptoms like gynecomastia, erectile dysfunction, or decreased libido. A general physical examination was performed, which came out to be unremarkable. However, perimetry was done for blurred vision, and it revealed that the patient had bitemporal hemianopia, which was more prominent in the left eye.

Upon conducting an initial MRI, a well-defined mass with lobulated contours was seen in the sellar/suprasellar region, measuring 3.7 × 4.0 × 3.8 cm (Fig. 1). This mass encased both internal carotid arteries, leading to the obliteration of the sphenoid sinus and destruction of the floor of the sella, findings that correlate with patient’s nasal bleeding. Initial prolactin levels were alarmingly elevated, exceeding 4000 ng/ml, indicating a significant endocrine disturbance. His testosterone levels were also very low, i.e., 69 ng/dl (300–890 ng/dl).

Figure 1.

Coronal view of the brain showing a well-defined mass with lobulated contours in the sellar/suprasellar region, measuring 3.7 × 4.0 × 3.8 cm (Before Treatment).

Based on clinical presentation, diagnostic imaging, and laboratory results a diagnosis of Giant Prolactinoma was made. The treatment strategy initiated included the administration of Dostinex (cabergoline), a dopamine agonist. Informed consent was obtained and potential side effects, including the risk of CSF rhinorrhea, were thoroughly discussed with the patient. However, in the event that cabergoline failed to yield a satisfactory response, surgical intervention was considered as the next step. After the initiation of treatment, there was reported a noticeable betterment in the patient’s condition. Prolactin levels dropped significantly, with a notable reduction to 176 ng/ml, indicating a positive response to the medication. A follow-up MRI scan indicated a considerable decrease in the size of the lesion, now measuring 1.7 × 1.9 × 1.4 cm (Fig. 2). Notably, the mass continued to encase the left internal carotid artery, but flow voids remained preserved. Furthermore, it closely abutted the right internal carotid artery, with maintained flow voids. To manage this case comprehensively, several additional steps were taken. The dose of cabergoline was thoughtfully adjusted, reducing the frequency from 3 to 2 tablets per week. The patient was also started on Testosterone injections for symptoms of hypogonadism. To further evaluate the patient’s endocrine status and corticosteroid requirement, a cosyntropin stimulation test was advised, with the next treatment decisions dependent on the results. The patient was instructed to schedule a follow-up visit after a month, during which repeat prolactin and relevant laboratory tests would be conducted, ensuring thorough monitoring of the patient’s progress.

Figure 2.

Coronal view of brain showing tumor size decreased to 1.7 × 1.4 × 1.9 cm (After Treatment).

In a subsequent 1-month follow-up, the patient reported some improvement but faced issues like nasal bleeding and occipital headaches. The nasal bleeding was most likely secondary to inferior tumor extension into the sphenoid sinus and erosion of its bony floor, causing mucosal irritation – a mechanism supported in literature describing similar cases. His prolactin levels had decreased to 167 ng/ml from a previous 176 ng/ml, showing a positive trend. Additionally, he developed CSF rhinorrhea, which made it necessary to consider surgical intervention alongside adjustments in his medical therapy to manage the complications.

After 2 months of follow-up, adjustments in treatment were important due to the observed changes in his condition, focusing particularly on the management of his testosterone levels and addressing the CSF rhinorrhea. Surgical intervention was considered for CSF rhinorrhea.

After the subsequent 4 months, the patient showed substantial improvement; CSF rhinorrhea had resolved and prolactin levels further decreased to an impressive 14 ng/ml, indicating excellent disease control. However, testosterone levels remained low at 134 ng/dl, highlighting ongoing hypogonadism challenges (Table 1). The suppressed LH and FSH levels pointed towards hypogonadotropic hypogonadism. This visit indicates the need for continued optimization of his hormone replacement therapy, regular monitoring of his condition, and adjustments to his treatment plan to address the low testosterone and ensure overall well-being.

Table 1.

Clinical, laboratory, and radiological parameters of the patient before and after receiving treatment.

Clinical parameters (reference range)	Before treatment	After treatment
Age	39 years	40 years
Tumor size	3.7 × 4.0 × 3.8 cm	1.7 × 1.4 × 1.9 cm
Prolactin ng/ml	>4700	14
Testosterone ng/dl	69	134
LH mIU/ml	1.17	-
FSH mIU/ml	0.76	-

Discussion

Giant prolactinomas pose significant diagnostic and therapeutic challenges due to their substantial size, invasive nature, and resistance to conventional treatments. An approach consisting of a blend of surgical, medical, and radiological methods is opted for treating this condition^[9^]. Our study highlights the successful management of a giant prolactinoma in a male presenting with severe headaches, visual disturbances, and nasal bleeding. The patient’s clinical presentation, along with elevated prolactin levels exceeding 4000 ng/ml and low testosterone levels, was consistent with a diagnosis of giant prolactinoma, as outlined by Borhan and Tan^[4^]. However, recurrent nasal bleeding is a relatively rare association with prolactinomas.

The initial treatment strategy involved administering cabergoline, the first-line therapy for prolactinomas due to its effectiveness. Cabergoline is a long-acting dopamine D2 receptor agonist that suppresses prolactin secretion from lactotroph cells, inhibits tumor cell proliferation, and promotes apoptotic pathways. This results in rapid reduction of serum prolactin levels and progressive shrinkage of prolactin-secreting tumors^[10^]. In our patient, cabergoline therapy resulted in a notable reduction in prolactin levels and a decrease in the size of the prolactinoma, as evidenced by follow-up MRI scans. This response underscores the importance of dopamine agonists as a cornerstone in the management of prolactinomas, including giant prolactinomas, in line with other studies^[11–13^].Despite the positive response to cabergoline therapy, the limitations and adverse effects due to the long-term use of dopamine agonists should be kept in mind. While cabergoline is generally well-tolerated, side effects such as nausea, dizziness, and hypotension may occur, necessitating careful monitoring and dose adjustments^[14,15^]. Long-term cabergoline therapy is typically required in giant prolactinomas. Discontinuation may lead to recurrence, especially in invasive variants. Regular MRI surveillance and prolactin monitoring are essential. Hypogonadism may persist despite biochemical improvement.

Moreover, the risk of developing cabergoline resistance or intolerance shows the importance of considering alternative treatment modalities, including surgical intervention and radiotherapy, in cases of treatment failure or intolerance^[16^].

In our case, additional measures were undertaken to optimize the patient’s management comprehensively. Adjustments in cabergoline dosage and the introduction of testosterone replacement therapy were implemented to address the patient’s hypogonadism concerns.

Surgical intervention is considered when medical therapy fails or in cases where there is evidence of involvement of surrounding structures, for example, optic chiasm compression, as seen in this case. However, given the positive response to cabergoline, surgery was not immediately required. Additionally, regular follow-ups are very important to know the prognosis of this condition as to look for any chance of reoccurrence or potential complications.

Conclusion

Our case report shows the complexity of managing giant prolactinomas and the use of medical therapy to achieve successful outcomes. While dopamine agonists such as cabergoline remain the cornerstone of therapy and often require long-term administration to maintain remission, individualized therapeutic strategies incorporating surgical intervention, radiotherapy, and adjunctive therapies may be necessary to optimize outcomes in patients with giant prolactinomas. Moreover, rare symptoms and associations such as recurrent nasal bleeding should also be taken into account while making a differential diagnosis. Regular monitoring and follow-ups are necessary for optimizing outcomes and addressing any complications in patients with giant prolactinomas.

Ethical approval

Our institution does not require ethical approval for reporting individual cases or case series.

Consent

Our institution does not require ethical approval for reporting individual cases or case series.

Conflicts of interest disclosure

The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Sources of funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

Author contributions

W.F.: conceptualization, methodology, data curation, writing original draft preparation, reviewing and editing; M.K.: methodology, writing original draft preparation, reviewing, and editing; L.S.: writing original draft preparation, reviewing, and editing; B.S.: writing original draft preparation, reviewing, and editing; M.U.I.: writing original draft preparation, reviewing, and editing; F.H.: writing original draft preparation, reviewing, and editing.

Research registration unique identifying number (UIN)

Not applicable.

Guarantor

Fatima Hajj.

Provenance and peer review

This case report was not commissioned and was externally peer reviewed.

Data availability statement

Not applicable.