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Korean J Otorhinolaryngol-Head Neck Surg. 2026;.kjorl-hns.2026.00332

Publication date (electronic) : 2026 August 18

doi : https://doi.org/10.3342/kjorl-hns.2026.00332

Yeon Seok You^1,2, Heung Bum Lee^2,3, Jae Seok Jeong^2,3, Jong Seung Kim^,1,2

¹Department of Otorhinolaryngology-Head and Neck Surgery, Jeonbuk National University Medical School, Jeonju, Korea

²Research Institute of Clinical Medicine of Jeonbuk National University-Biomedical Research Institute of Jeonbuk National University Hospital, Jeonju, Korea

³Department of Internal Medicine, Research Center for Pulmonary Disorders, Jeonbuk National University Medical School, Jeonju, Korea

유연석^1,2, 이흥범^2,3, 정재석^2,3, 김종승^,1,2

¹전북대학교 의과대학 이비인후과학교실

²전북대학교 임상의학연구소-전북대학교병원 의생명연구원

³전북대학교 의과대학 내과학교실, 폐질환연구센터

Address for correspondence Jong Seung Kim, MD, PhD Department of Otorhinolaryngology-Head and Neck Surgery, Jeonbuk National University Medical School, 20 Geonji-ro, Deokjin-gu, Jeonju 54907, Korea Tel +82-63-250-2792 E-mail kjsjdk@gmail.com

Received 2026 May 3; Revised 2026 June 3; Accepted 2026 June 8.

Abstract

Introduction

Maxillary sinus fungal balls are noninvasive lesions that typically occur in immunocompetent patients. They usually remain confined to the sinus cavity, and progression to invasive fungal sinusitis or deep neck infection is rare [1,2]. However, when mucosal disruption or secondary bacterial infection is present, even a noninvasive lesion may be associated with inflammatory extension into adjacent tissues [3].

Diffuse panbronchiolitis (DPB) is a chronic airway disease associated with chronic rhinosinusitis and impaired mucociliary clearance [4]. We report a rare case of rapidly progressive deep neck infection arising from maxillary sinusitis with a noninvasive fungal ball in a patient with previously unrecognized DPB.

Case

A 72-year-old woman presented to the emergency department with progressive right facial swelling that had developed over 3 days. She had a 10-year history of asthma. She had no history of diabetes mellitus, malignancy, HIV infection, or systemic immunosuppressive treatment. Before referral, she had been evaluated at a dental clinic, but no definite odontogenic abnormality was identified.

On admission, laboratory tests revealed a white blood cell count of 20120/μL, neutrophil count of 17800/μL, and erythrocyte sedimentation rate of 93 mm/h. Paranasal sinus CT demonstrated soft-tissue attenuation with calcification in both maxillary sinuses, consistent with bilateral maxillary sinus fungal balls (Fig. 1A). On the right side, an ill-defined hypodense inflammatory lesion was identified around the anterior maxillary region, medial maxillary wall, and nasal floor, extending posteriorly toward the pterygopalatine and parapharyngeal regions (Fig. 1A). Coronal and sagittal CT images showed contiguous inflammatory extension from the right maxillary/nasal floor region posteriorly toward the pterygopalatine/parapharyngeal region and inferiorly toward the submandibular space, suggesting deep neck space involvement through contiguous spread (Fig. 1B and C). No definite CT evidence of odontogenic sinusitis was identified in the dentoalveolar region, including periapical lucency, oroantral fistula, dental root protrusion into the maxillary sinus, or focal dentoalveolar bone destruction (Fig. 1D). The presumed route of contiguous inflammatory spread from the maxillary sinus/nasal floor region to the parapharyngeal and submandibular spaces is summarized schematically in Fig. 2.

Fig. 1.

Radiologic findings of maxillary sinusitis with contiguous inflammatory extension. A: Axial paranasal sinus CT shows bilateral calcified maxillary sinus lesions and adjacent right-sided inflammatory change involving the anterior maxillary, medial maxillary, and nasal floor regions. B and C: Coronal and sagittal CT images show contiguous inflammatory extension posteriorly toward the pterygopalatine/parapharyngeal region and inferiorly toward the submandibular space. D: Sagittal bone-window CT shows no definite CT evidence of odontogenic sinusitis in the dentoalveolar region. Red arrows indicate relevant inflammatory findings.

Fig. 2.

Schematic illustration of the presumed route of contiguous inflammatory spread. A: Coronal schematic showing the suspected necrotic area involving the inferior aspect of the maxillary sinus and nasal floor region. B: Sagittal schematic showing the presumed posterior extension from the nasal floor region toward the pterygopalatine/parapharyngeal region. C: Coronal schematic showing inferior extension toward the parapharyngeal and submandibular spaces. Red arrows indicate the presumed direction of contiguous inflammatory spread. Figure 2 was created with the assistance of ChatGPT (OpenAI) using AI image-generation and editing tools (accessed May 30–June 2, 2026) and was subsequently reviewed and modified by the authors.

Chest high-resolution CT performed during the admission showed centrilobular nodules, bronchial wall thickening, and bronchiectatic changes, and pulmonary evaluation supported a diagnosis of previously unrecognized DPB (Fig. 3).

Fig. 3.

Chest high-resolution CT findings consistent with diffuse panbronchiolitis. Chest HRCT shows bilateral centrilobular nodules with tree-in-bud opacities, bronchial wall thickening, and bronchiectatic changes, consistent with diffuse panbronchiolitis. Red arrow indicates a representative tree-in-bud opacity. HRCT, high-resolution computed tomography.

Emergency surgery was performed under general anesthesia. After uncinectomy, purulent discharge was observed from the right maxillary sinus. After evacuation of the pus, a yellowish fungal ball was identified and completely removed using suction, curettage, and saline irrigation (Fig. 4A). Endoscopic inspection demonstrated necrotic-appearing mucosa and exposed necrotic tissue along the medial maxillary wall, inferior meatus, and nasal floor region (Fig. 4B and C). Subperiosteal dissection revealed mucosal necrosis involving the medial maxilla and nasal floor. Medial maxillectomy was therefore performed to remove necrotic tissue and secure adequate drainage.

Fig. 4.

Intraoperative endoscopic and transcervical findings. A: Endoscopic view shows a yellowish FB within the MS. B: Endoscopic view shows necrotic-appearing tissue at the inferior aspect of the maxillary sinus. Yellow arrows indicate necrotic-appearing areas. C: Endoscopic view after elevation of the right NM shows an irregular dark lesion at the NF, suspicious for mucosal necrosis. Yellow asterisk indicates the suspected necrotic focus. D: Transcervical drainage of the submandibular abscess was performed. FB, fungal ball; MS, maxillary sinus; NM, nasal mucosa; NF, nasal floor.

Transcervical drainage was then performed. After elevation of a subplatysmal flap, pus was drained from the submandibular space (Fig. 4D). Finger dissection confirmed continuity between the submandibular and parapharyngeal spaces. Multiple saline irrigations were performed. Because of extensive parapharyngeal and submandibular involvement and the anticipated risk of postoperative airway compromise from deep neck edema, tracheostomy was performed for airway protection.

Histopathologic examination of the sinus contents revealed septate fungal hyphae with acute-angle branching, consistent with Aspergillus species. Necrotic tissue was present around the fungal lesion, but no tissue or vascular invasion was identified. These findings supported a diagnosis of noninvasive fungal ball rather than invasive fungal sinusitis. Fungal culture from the sinus contents grew Aspergillus fumigatus, whereas pus culture from the deep neck abscess grew Pseudomonas aeruginosa. Taken together, the findings suggested contiguous spread of maxillary sinusitis with a coexisting noninvasive fungal ball, accompanied by mucosal disruption and secondary bacterial infection, rather than deep neck extension caused by invasive fungal disease.

Postoperatively, the patient’s facial swelling and inflammatory markers improved progressively with combined medical therapy. Although systemic antifungal therapy is not routinely required for noninvasive fungal balls, voriconazole was initiated empirically immediately after surgery because invasive fungal disease was initially difficult to exclude in the setting of mucosal necrosis, extensive contiguous deep neck space involvement, and a positive serum Aspergillus galactomannan antigen result obtained on the day of surgery. Although systemic antifungal therapy could have been considered preoperatively based on the imaging findings and clinical presentation suspicious for invasive fungal sinusitis, urgent surgical source control and acquisition of untreated diagnostic tissue were prioritized; therefore, voriconazole was started immediately after surgery. Final histopathology showed no tissue or vascular invasion. In addition, follow-up serum Aspergillus galactomannan antigen tests were performed weekly on three occasions and were all negative. Therefore, voriconazole was discontinued after approximately 3 weeks once secondary bacterial infection was considered the more likely mechanism. Intravenous cefpirome and ciprofloxacin were administered for the bacterial deep neck infection. The silicone drain was removed on postoperative day 10, and the tracheostomy tube was removed on postoperative day 21. At 1-year follow-up, nasal endoscopy showed no evidence of recurrence or complications.

Discussion

This case describes a rare presentation of rapidly progressive deep neck infection arising from maxillary sinusitis with a coexisting noninvasive fungal ball in an immunocompetent patient. Histopathologic examination showed fungal hyphae consistent with Aspergillus species, but no tissue or vascular invasion was identified. In addition, Aspergillus fumigatus was isolated from the sinus contents, whereas Pseudomonas aeruginosa was isolated from the deep neck abscess. These findings suggest that this case represented secondary bacterial infection and contiguous deep neck extension from maxillary sinusitis with a coexisting noninvasive fungal ball, rather than invasive fungal sinusitis [3].

Paranasal sinus fungal balls are noninvasive lesions that usually occur in immunocompetent patients. They most commonly involve the maxillary sinus and are often asymptomatic or associated with only mild, nonspecific sinonasal symptoms [1,2]. However, extrasinus inflammatory changes may occur in some patients even without histologic evidence of fungal invasion [3]. Therefore, when a fungal ball is accompanied by severe extrasinus infection, the distinction between proven invasive fungal sinusitis and secondary bacterial extension is clinically important. Invasive fungal sinusitis requires histopathologic evidence of fungal hyphae invading the sinus mucosa, submucosa, blood vessels, or bone, and may be accompanied by angioinvasion, thrombosis, tissue infarction, or necrotic eschar [5]. In contrast, secondary bacterial extension in the setting of a noninvasive fungal ball implies mucosal barrier disruption, bacterial superinfection, abscess formation, and contiguous spread beyond the sinus. In the present case, purulent discharge from the maxillary sinus, mucosal necrosis involving the medial maxilla and nasal floor, and a contiguous lesion extending to the parapharyngeal and submandibular spaces support the possibility that mucosal barrier disruption and secondary bacterial infection, rather than fungal invasion itself, contributed to the deep neck extension.

During admission, chest high-resolution CT and pulmonary evaluation revealed findings consistent with previously unrecognized DPB. DPB is closely associated with chronic rhinosinusitis and may involve impaired mucociliary clearance and mucus stasis [4]. Reduced nasal nitric oxide and aberrant MUC5B expression reported in DPB may further support impaired sinonasal defense and mucus stasis [6,7]. Therefore, DPB should be interpreted not as a direct cause of the deep neck infection, but as a possible background condition that may have contributed to impaired sinonasal defense, mucosal injury, and secondary bacterial extension.

This case suggests that although fungal balls are generally noninvasive lesions with a favorable course, they may rarely be associated with serious extrasinus complications when accompanied by conditions that can impair mucociliary clearance, such as DPB. However, because this is a single case report, a causal relationship between DPB and deep neck infection cannot be established. Secondary bacterial infection, rather than fungal invasion, was the more likely direct driver of deep neck infection.

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