Neurologia i neurochirurgia polska · 2025
Cerebrospinal fluid fistula following dopamine agonist therapy in giant prolactinomas: A case series and review of the literature.
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Altunyuva Oguz, Ozmarasali Ali Imran, Eker Oguzhan, Eser Pinar, Oz Gul Ozen, Yilmazlar Selcuk
Аннотация
AIM OF THE STUDY: We aimed to analyze cases of cerebrospinal fluid (CSF) rhinorrhea/fistula emerging after dopamine agonist (DA) therapy in patients with giant prolactinomas, and to identify clinical patterns and treatment strategies. We also sought to contextualize our findings through a systematic review of published cases.
CLINICAL RATIONALE FOR THE STUDY: Dopamine agonist therapy is first line for prolactinomas; however, in invasive macroadenomas, rapid shrinkage may unmask skull base defects, leading to CSF leaks and infections like meningitis. These rare but critical events lack standard treatment protocols.
MATERIAL AND METHODS: We reviewed six prolactinoma cases with post-cabergoline CSF rhinorrhea (2010-2024) and conducted a PubMed-based review (1980-2024) to compare clinical and treatment features.
RESULTS: In our series (mean age: 49.3 years) CSF rhinorrhea developed between 14 and 420 days after therapy initiation. The sellar floor was the most frequent defect site (5 of 6 patients). All patients required surgical or CSF diversion procedures, including transsphenoidal or transcranial repair, external ventricular drainage, lumbar drainage, or ventriculoperitoneal shunting. One case resulted in fatal meningitis. The literature review included 68 cases across 33 studies. The median time to CSF leak onset was 90 days (range: 7-1460 days). Surgical repair, particularly transsphenoidal, was the most common treatment. Recurrence occurred in 25/55 cases (45.5%) and meningitis in 6/59 cases (10.2%); outcome data were not available for all patients.
CONCLUSIONS AND CLINICAL IMPLICATIONS: Skull base erosion and rapid tumor shrinkage are major contributors. Our findings support early recognition, dose titration of DA therapy, close radiological follow-up, and timely surgical intervention. Further studies are needed to define risk stratification and evidence-based treatment protocols.
CLINICAL RATIONALE FOR THE STUDY: Dopamine agonist therapy is first line for prolactinomas; however, in invasive macroadenomas, rapid shrinkage may unmask skull base defects, leading to CSF leaks and infections like meningitis. These rare but critical events lack standard treatment protocols.
MATERIAL AND METHODS: We reviewed six prolactinoma cases with post-cabergoline CSF rhinorrhea (2010-2024) and conducted a PubMed-based review (1980-2024) to compare clinical and treatment features.
RESULTS: In our series (mean age: 49.3 years) CSF rhinorrhea developed between 14 and 420 days after therapy initiation. The sellar floor was the most frequent defect site (5 of 6 patients). All patients required surgical or CSF diversion procedures, including transsphenoidal or transcranial repair, external ventricular drainage, lumbar drainage, or ventriculoperitoneal shunting. One case resulted in fatal meningitis. The literature review included 68 cases across 33 studies. The median time to CSF leak onset was 90 days (range: 7-1460 days). Surgical repair, particularly transsphenoidal, was the most common treatment. Recurrence occurred in 25/55 cases (45.5%) and meningitis in 6/59 cases (10.2%); outcome data were not available for all patients.
CONCLUSIONS AND CLINICAL IMPLICATIONS: Skull base erosion and rapid tumor shrinkage are major contributors. Our findings support early recognition, dose titration of DA therapy, close radiological follow-up, and timely surgical intervention. Further studies are needed to define risk stratification and evidence-based treatment protocols.