Challenging Diagnosis of Invasive Sinus Aspergillosis Mimicking Gradenigo's Syndrome in an Elderly Patient with T-Cell Lymphoma

Abstract

(1) Background: Gradenigo's Syndrome (GS) is a rare complication of acute otitis media characterized by the triad of diplopia, otitis, and facial pain. The widespread use of antibiotics has significantly reduced its occurrence. (2) Case summary: We present the case of an elderly patient with T-cell lymphoma who developed neurological deficits resembling GS. The patient was ultimately diagnosed with invasive sinus aspergillosis. The diagnostic process was challenging due to the atypical clinical presentation and the lack of specific imaging findings. A biopsy was the most important test for clarifying the diagnosis. (3) Conclusions: The prognosis for this complication is extremely poor without surgery, and the patient died despite adequate antifungal coverage. Therefore, maintaining high clinical suspicion is paramount to avoid adverse outcomes in similar cases, particularly in the geriatric population, wherein this syndrome's occurrence may not be expected.

Keywords: T-cell lymphoma; elderly; fungal infection; immunosuppression; invasive aspergillosis.

Figure 1

Gantt chart showing the timeline of the clinical, diagnostic, and treatment evolution of the case, as well as the evolution of the laboratory parameters C-Reactive Protein and neutrophil count. ¹ Cyclophosphamide (400 mg/m²) on day 1; Doxorubicin (25 mg/m²) on day 1; Vincristine (1 mg) on day 1; Prednisone (40 mg/m²) on days 1, 2, 3, 4, and 5. ² Meropenem (1 g) administered every 8 h IV. ³ Treatment with meropenem was combined with linezolid at a dose of 600 mg administered every 12 h IV. ⁴ Treatment was initiated with liposomal preparation of amphotericin-B at a dose of 3 mg/kg administered IV. ⁵ Amphotericin-B dose was increased to 5 mg/kg IV. ⁶ Treatment was associated with Acyclovir at a dose of 500 mg administered every 8 h orally. ⁷ Meropenem dose was increased to 2 g administered every 8 h IV. ^a Cranial CT: inflammation of the maxillar and sphenoid-ethmoidal sinuses. ^b Serum GM (galactomannan antigen test): 0.34 (negative) ^c Brain MRI: consistent with CT findings, without identifying PA. ^d Lumbar puncture: clear and sterile cerebrospinal fluid. Normal lymphocyte immunophenotype performed via FCM. ^e Paranasal sinuses CT: worsening inflammatory changes, with no evidence of bone erosions. ^f Biopsy of the middle turbinate: septate hyphae compatible with Aspergillus spp. infection.

Figure 2

(A–D) CT and MRI scans show an inflammation of the paranasal sinuses without erosion of the petrous bone (note that bone destruction in these cases can be extensive, subtle, or even inapparent). (A) Axial CT: Moderate to severe mucoperiosteal thickening predominantly in the posterior ethmoidal cells, with subtotal cell occupancy (horizontal arrows). (B) Coronal CT: Subtotal occupation with mucoperiosteal thickening and soft tissue attenuation in ethmoidal and maxillary sinuses as well as in the left nasal cavity (vertical arrow). (C) Coronal T2 WI MRI: Mild hyperintense mucosal peripheral thickening of the sphenoid sinus (diagonal arrow). Note central hypointensity, which might be due to either air or fungal elements. (D) Axial T2 WI MRI: hyperintense peripheral soft tissue in the left paranasal sinuses with central hypointensity within the posterior ethmoidal cells, consistent with fungal elements (diagonal arrow). No fat stranding indicating invasion outside the sinus.

Figure 3

Histopathology of the middle turbinate biopsy showing septate hyphae (Hematoxylin-eosin stain, ×400).