Pituitary adenoma surgeries are common in neurosurgical setup. Majority are tackled
by a transsphenoidal route either by endoscopic or microscopic aid. Complications
such as cerebrospinal fluid (CSF) leak, meningitis, diabetes insipidus, hematoma,
and loss of vision are known, but midbrain infarct is rarely reported. We report and
discuss the possible mechanism of this rare and unusual complication in transsphenoidal
surgery. A 36-year-old nulliparous female with no comorbidities suffering from primary
infertility presented with intermittent headache for 9 months with bitemporal vision
disturbances for 3 months, pigmentation around the neck and nape, oligomenorrhea with
an established diagnosis of plurihormonal secreting pituitary tumor (predominantly
adrenocorticotropic hormone and prolactin), hyperparathyroidism, and diabetes. The
whole symptom fitted into the diagnosis of multiple endocrine neoplasia 1 (MEN-1)
syndrome. Molecular genetic testing was done with full gene sequencing analysis of
MEN-1 gene using polymerase chain reaction. Furthermore, Sanger DNA sequencing was
done, and two novel variations, namely IVS 9c.1364 + 99C>G and EXON 10 c.1813 C>T
(p. L605 L), were detected. Radiology detected a microadenoma in the right lobe of
the pituitary with mild deviation of the pituitary stalk on dynamic contrast-enhancing
magnetic resonance imaging. Gross total excision of the tumor was done through transsphenoidal
approach. The surgery was uneventful other than some blood-mixed CSF leak. Post excision,
fat and tissue glue was packed. The patient did not wake up from anesthesia and had
bilateral dilated pupil with no oculocephalic reflex. Investigations revealed bilateral
thalamic and midbrain infarct. The patient subsequently expired. This case is reported
in view of its unusual complication and to create awareness for such a fatal complication
following transsphenoidal surgery for pituitary microadenoma and the importance of
diligent approach to transsphenoidal surgery and to report novel genetic mutation
of MEN-I gene.
Key-words:
Adenoma - hemorrhage - infarction - multiple endocrine neoplasia 1 - pituitary - subarachnoid
- thalamic - transsphenoidal