Abstract
Chromoblastomycosis is a rare chronic fungal infection typically affecting the cutaneous regions of the lower extremities, predominantly in male agricultural workers. However, its extracutaneous manifestations, particularly in the maxillary sinus, are exceedingly rare, with only three cases reported in the literature. This case report presents a 59-year-old female with a history of chronic tobacco use, who presented with an ulceroproliferative lesion of the hard palate, later diagnosed as moderately differentiated squamous cell carcinoma (SCC) invading the maxillary sinus. During surgery, necrotic debris in the maxillary sinus revealed chromoblastomycosis on histopathological examination, confirmed by the presence of characteristic sclerotic bodies.
The co-occurrence of maxillary chromoblastomycosis and SCC is unprecedented, raising important questions about their etiological interplay. The fungal infection may have been facilitated by trauma, cortical erosion, and immunosuppression induced by the invasive carcinoma. This case emphasizes the importance of careful intraoperative evaluation and highlights the need for a multidisciplinary approach to manage rare co-pathologies effectively. Postoperative treatment included antifungal therapy with oral itraconazole (400 mg/day) and adjuvant radiotherapy for SCC.
The unique combination of an aggressive malignancy with a rare fungal infection in this patient underscores the importance of detailed documentation and exploration of such atypical presentations. Further studies are required to elucidate potential causative links and to develop evidence-based guidelines for the diagnosis and management of such rare and complex clinical scenarios.
1
Introduction
Chromoblastomycosis or chromomycosis is a rare, chronic, indolent, fungal cutaneous disorder typically affecting adult males. It is an occupational-related disease with a definite preponderance for farmers and agricultural workers, especially in tropical countries of the world. The fungal infection generally involves the skin of the legs and feet following minor occupational trauma leading to subsequent inoculation by the fungi which are thought to be present in soil, vegetable debris, and wood [ ].
The lesion usually begins as a scaly papule with a pinkish hue which may insidiously progress into nodules and irregular purplish plaques and later into verrucous nodules. Very rarely has the cutaneous lesion been reported to transform into squamous cell carcinoma (SCC) [ ].
Chromoblastomycosis is classically a cutaneous fungal infection involving the lower extremities. Its involvement in the maxillary sinus is an exceedingly rare clinical event. To the best of the authors’ knowledge and after a comprehensive literature search on prestigious abstracting platforms including Medline, PubMed, Scopus and Google Scholar, chromoblastomycosis of the maxillary sinus has only been described thrice in international literature. Moreover, its co-association with oral cavity SCC has never been illustrated before.
Herein, we discuss a very intriguing clinical tale of a 59-year-old lady who underwent a wide local excision for a locally advanced squamous cell carcinoma of the left hard palate. However, during intraoperative exploration, fungal debris was identified in the left maxillary sinus, which to everyone’s surprise, turned out to be chromoblastomycosis on histopathological examination. This case study is unique owing not only to the rare site and presentation of chromoblastomycosis but also to its concurrent occurrence with an aggressive malignancy invading the bony framework of the involved maxillary sinus. It further raises an important question for everyone to ponder whether it is a mere coincidence or there is the presence of an intricate causal relationship between the two pathologies.
2
Case presentation
A 59-year-old female presented to the outpatient department with chief complaints of swelling over the left hard palate for the last 3 months. It was associated with difficulty in swallowing solid foods and liquids. She had experienced significant weight loss during this period and had intermittent low-grade fever as well. The patient gave a history of tobacco consumption for the last 30 years. However, there was no history of diabetes mellitus, immunosuppression, any other chronic ailment or any other significant drug history. The patient was a homemaker and the family history was unremarkable as well.
On examination, there was the presence of a 4 × 3.5 cm ulceroproliferative lesion which was firm to hard in consistency, had irregularly defined margins, was immobile, and non-tender. There were no clinically palpable cervical lymph nodes.
An incisional biopsy was performed on the growth, which suggested moderately differentiated keratinizing squamous cell carcinoma. The patient was advised a Contrast-Enhanced Computed Tomography (CECT), which revealed a heterogeneously enhancing soft tissue attenuation mass lesion measuring 48 × 37 × 35 mm (anteroposteriorly x transversally x craniocaudally) seen epicentre at the left posterior alveolar arch and hard palate region. Superiorly, the mass lesion was seen extending into the inferior half of the left maxillary sinus eroding the hard palate. In the maxilla, it was seen eroding the inferior parts of the anterior, medial, and posterior walls of the maxilla. Heterogeneous hypodense collections were seen in the rest of the maxilla. Medially, the mass lesion was seen projecting into the oral cavity and causing indentation over the dorsal and left lateral border of the tongue. Laterally, the mass lesion was seen extending into the gingiva-buccal sulcus and involving the buccal mucosa. Posterolaterally the growth was seen extending into the masticator space. Also, bony erosions were identified posteriorly in the medial and lateral pterygoid plates. The lesion was extending to the retromolar trigone region posteriorly. No other suspicious lesion was identified elsewhere in the body ( Fig. 1 ).
The patient underwent a wide local excision and during intraoperative exploration, necrotic debris was identified in the left maxillary sinus. The excisional specimen and the debridement material were received in 10 % neutral-buffered formalin in the histopathology department. Routine 5-μm thick Hematoxylin and Eosin-stained sections were prepared from formalin-fixed and paraffin-embedded tissue blocks. Histopathological examination of multiple sections showed the presence of an infiltrative neoplasm comprising diffuse sheets, islands and nests of atypical squamous cells. Individual tumor cells were polygonal in shape and had an abundant amount of glassy eosinophilic cytoplasm with evidence of intracellular keratinization. The nuclei were enlarged, with irregular nuclear contours, vesicular chromatin, and occasional prominent 0–1 eosinophilic nucleoli. Focal extracellular keratinization in the form of keratin pearls was noted. Areas of tumor necrosis, lymphovascular invasion, and perineural invasion were identified as well. Atypical mitoses were identified with a mitotic count of 3–4 per 10 high-power fields. All the resected margins were negative for invasive carcinoma ( Fig. 2 ).
The necrotic debris was entirely processed and the serial sections examined revealed numerous characteristic sclerotic or Medlar bodies. These were round-to-oval, pigmented (brown-coloured), thick-walled fungal cells measuring approximately 5–12 μm in diameter. The background showed the presence of numerous broad-based, aseptate fungal hyphae as well, along with dead and necrotic maxillary bone fragments ( Fig. 3 ).
