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Isolated sphenoid sinus fungal mucoceles: A rare entity with a high propensity for causing neurological complications

Ibrahim M, Alqunaee M, Najibullah M, Shabbir Z, Azab WA

Surg Neurol Int, 2024;15:444.
PMID: 39640329 DOI: 10.25259/SNI_849_2024

BACKGROUND: Isolated sphenoid sinus fungal mucoceles are extremely rare and potentially associated with visual disturbances, cranial nerve (CN) deficits, or pituitary dysfunction. Their initial symptoms are often absent or nonspecific, and routine examination offers little information, resulting in diagnostic and therapeutic delays. A high index of suspicion and a thorough understanding of their clinical presentation, neuroradiological features, microbiological implications, and complication profile are crucial for early diagnosis and prompt management. We, herein, analyze a series of consecutive cases of isolated sphenoid sinus fungal mucoceles whom we treated, add to the currently existing published cases, and review the pertinent literature.
METHODS: From the databases of endoscopic endonasal skull base and rhinological surgical procedures maintained by our groups, all cases with isolated sphenoid sinus fungal mucoceles were retrieved and included in the study. Clinical and radiological findings, histopathologic evidence of fungal rhinosinusitis, culture results, clinicopathological designation, treatment details, and outcome of CN neuropathies were analyzed.
RESULTS: Headache was the most common symptom (seven cases). Oculomotor (three cases) and abducens (two cases) nerve palsies were encountered in five out of eight patients. Visual loss was seen in two cases. Hypopituitarism was seen in one case. All patients underwent endoscopic endonasal wide bilateral sphenoidectomy. CN palsies improved in four out of five cases.
CONCLUSION: Endoscopic endonasal wide sphenoidectomy is the surgical treatment of choice and should be performed in a timely manner to prevent permanent sequelae. Histopathological and microbiological examination findings should both be obtained as they dictate the next steps of therapeutic intervention.
Fulltext A case report of a patient with recurrent and severe infections highlighting the importance of considering inborn errors of immunity

Altammar F, Alshamali M, Alqunaee M, Alali AJ, Elshafie RM, Al-Herz W

Front Pediatr, 2024;12:1340367.
PMID: 38487470 DOI: 10.3389/fped.2024.1340367

Inborn errors of immunity (IEI) can often be misdiagnosed early in life due to their heterogenous clinical presentations. Interleukin-1 receptor-associated kinase 4 (IRAK-4) deficiency is one of the rare innate immunodeficiency disorders. We present the case of a patient who presented at the age of 15 days with meningitis and septic shock that responded to antibiotics. She was admitted again at the age of 45 days with pseudomonas aeruginosa bacteremia that was associated with increased inflammatory markers. Her third admission was at the age of 2.5 months due to left sided peri-orbital cellulitis that was again associated with elevated inflammatory markers. At 3.5 months, she experienced left orbital cellulitis, which was complicated by extensive sinus involvement, erosion, and abscess formation in the pterygopalatine fossa. Her condition progressed to septic shock and required multiple antibiotics and surgical interventions for drainage and control of the infection source. Both abscess and blood culture were positive for pseudomonas aeruginosa. An IEI was suspected but basic immunology testing was normal. Whole Exome Sequencing was performed and a novel mutation in IRAK4 was detected. In conclusion, we highlight the importance of raising awareness among pediatricians about the potentially lethal IEI and the need to consult specialists when these diseases are suspected. Among them is IRAK-4 deficiency which can be diagnosed by sophisticated functional assays and/or genetic testing.

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