Endocrine Abstracts

Background: Acute invasive fungal rhinosinusitis (AIFR) is a rare but fatal infection that occurs primarily in immunocompromized individuals. It is characterized by fungal invasion into the mucosa and submucosal structures of the paranasal sinuses with extension into adjacent structures, including the paranasal soft tissues, orbit and cranial vault. We present here a case of hypopituitarism due to AIFR with intracranial lesion.

Case: A 59-year-old man, whose past medical history were total gastrectomy due to gastric carcinoma and cerebral hemorrhage, appeared general fatigue. He was diagnosed as hyponatremia due to unknown origin and transferred to our hospital for further examinations. He presented body weight los, right eyelid ptosis and ocular movement disorder. Laboratory data showed severe hyponatremia (Na 119 mEq/ml) and hypopituitarism (ACTH 8.8 pg/ml, Cortisol 4.3 μg/dl, TSH 1.1 μIU/ml, fT₄ 0.38 ng/dl) confirmed by endocrinological examinations, on the other hand, b-D-glucan was normal range (9.3 pg/ml). Brain MRI showed an intracranial mass lesion of irregular form at right cavernous sinus and sphenoidal sinus, which considered as culprit lesion of right eye symptoms. A stenosis of right internal carotid artery was also observed, and it progressed to complete obstruction later. Nasal endoscopy revealed a fungal ball at sphenoidal sinus and Aspergillus Fumigatus was detected by culture. Overall, he was diagnosed as hypopituitarism, oculomotor nerve palsy and obstruction of right internal carotid artery due to AIFR. Antifungal agents (VRCZ, MCFG) and hormone replacement therapy (hydrocortisone, LT₄) were started, and then general fatigue, hyponatremia, and eyelid ptosis were gradually improved.

Conclusion: AIFR is usually diagnosed and treated by otolaryngologist, and there are seldom reports on hypopituitarism due to AIFR as the present case. It should be considered AIFR is one of the differential diagnoses in a patient who appears hypopituitarism and/or cranial nerve disturbances, although AIFR with cranial invasion is extremely rare.