Tongue lesions in the pediatric population are rare. The differential diagnosis of these lesions is broad, and rapid growth of the lesion is concerning for a neoplastic process. We present a rare case of a fungal lesion mimicking a neoplastic growth in a 22-month-old girl. She underwent complete excision successfully. Full evaluation for benign and malignant neoplasms was negative. Tissue culture demonstrated growth of a rare Candida species to be the cause of the lesion. Postoperatively, she continues to do well, without regrowth 6 months later. This case reinforces the role of tissue culture when histology fails to demonstrate a diagnosis and emphasizes the need for efficient communication between the pediatrician, otolaryngologist, and pathologist for timely excision.
Tongue lesions in the pediatric population are uncommon. Although a majority of tongue lesions in this population are neoplastic, there is a broad range of potential diagnoses. When neoplastic lesions are present, the pathology is often benign, such as teratoma or epulis.1 Traumatic causes of tongue lesions can occur in the form of a rare disease process named Riga-Fede, after its discoverers.2 Pathologic infection of the tongue is often not considered in immunocompetent patients. Yet, evaluation of the prevalence of fungal species colonizing the oral cavity in the pediatric population demonstrated that Candida was identified in 66% of the study population.3 Colonization does not indicate disease state, however, and a fungal infection presenting as a rapidly growing tongue mass in a child has not been described in the literature before to our knowledge.
We present a case of an otherwise normal 22-month-old girl with a rapidly growing dorsal tongue mass. Efficient communication between the pediatrician and otolaryngologist and prompt referral to the otolaryngologist facilitated early excisional biopsy due to concern for a neoplastic process. Full pathologic evaluation for malignancy was negative. Tissue culture demonstrated growth of an unusual fungal species, Candida lusitaniae. Pharmacologic antifungal treatment was deferred and the patient has not demonstrated regrowth after 6 months. This case was submitted to our institution’s institutional review board and was exempt from review.
A 22-month-old, nonsyndromic girl presented to her pediatrician with reports of a rapidly growing tongue lesion over the previous 2 days. There were no associated symptoms of respiratory or feeding difficulties, sick contacts, or change in behavior. The patient’s medical, surgical, and prenatal history was unremarkable. Her exam showed a 2-cm firm, circular lesion on the dorsal surface of the left tongue with overlying fibrinous exudates (Fig 1). The tongue was mobile, and there was no stridor. She was prescribed antibiotics and promptly referred to the otolaryngologist. To evaluate a potential response to antibiotics, no immediate intervention was taken and 1-week follow-up was recommended. She returned to the clinic after 3 days, reporting increasing size of the tongue mass and increased fussiness but no airway concerns. The lesion now appeared to be 2.5 to 3 cm in size without change in character.
Due to the rapid growth of the tongue mass, the family elected to go to the operating room for excisional biopsy 5 days after initial presentation. In the operating room, the patient was orotracheally intubated without issue. The lesion was completely excised with primary closure and did not appear to be infiltrative to adjacent tissues (Fig 2). The gross specimen was a 3.5 × 2.6 × 1.5–cm soft-tissue mass. Histology showed normal squamous mucosa with ulceration and mixed inflammatory infiltrates on pathologic examination. Stains for Langerhans cell histiocytosis, sarcoma, lymphoma, and granular cell tumors were negative. Fungal cultures demonstrated the lesion to be colonized with C. lusitaniae. Postoperatively, the patient was extubated and spent 1 evening in the PICU before discharge. Pharmacologic antifungal treatment was offered, but the family deferred treatment due to the fact that there was complete excision with negative margins. Six-months postoperatively, she has no new growths and has excellent tongue mobility without difficulty swallowing or phonating.
The differential diagnosis for rapidly growing tongue lesions is broad and should include neoplastic growths and nonneoplastic growths. Horn et al1 performed one of the largest reviews on pediatric tongue lesions and demonstrated all pathologies to be benign. Although malignant tumors, such as lymphoma and rhabdomyosarcoma, were considered, these are exceedingly rare in this location.4 Benign tumors, such as teratoma and epulis, are more likely in this location and can present as rapidly growing tongue lesions, but these entities are also rare.5,6 Although, grossly, the lesion appeared to be neoplastic, a thorough evaluation with multiple tumor-specific stains did not demonstrate a neoplastic lesion.
The nonneoplastic, inflammatory infiltrate seen on pathology can be seen in the setting of a tongue lesion with rare diseases, such as Riga-Fede disease.7 Riga-Fede is a disease process whereby erupted mandibular incisors create repetitive persistent trauma to the ventral surface of the tongue mucosa leading to a fibrous, ulcerative growth. The tongue lesion seen in our patient, however, was not in the appropriate location to fit this disease description, and there was no history of repetitive trauma.
With a negative neoplastic evaluation and history inconsistent with trauma, cultures of the tissue were ordered to evaluate for potential infectious causes. Culture of the specimen for bacterial and fungal species demonstrated an unusual Candida species, C. lusitaniae, without bacterial growth. It is unclear whether this represents a contaminant that simply incited an inflammatory reaction that caused growth of a new tongue lesion, or if this represents a pathologic state of overgrowth. Nonetheless, pharmacologic antifungal agents were considered after the excision. However, because the patient demonstrated no signs of regrowth in the early postoperative period and the specimen had normal margins, antifungal treatment was deferred. At the 6-month follow-up, she continued to do well without signs of regrowth.
It is important to emphasize that the diagnosis of a fungal tongue lesion was only made after biopsy. Thus, we stress the concept that all rapidly growing lesions warrant histopathologic evaluation by incisional or excisional biopsy. This allows diagnosis and relief of a potential source of airway obstruction and feeding difficulties, which may be of concern in young children.
We present a rare case of a fungal lesion of the tongue presenting as a rapidly growing tongue mass. This case demonstrates the importance of early identification and facile communication between the pediatrician, the surgical specialist, and the pathologist, which allowed early excisional biopsy and diagnosis of a tongue mass that was concerning for a malignant process. Although it is unclear why a fungal lesion presented as a rapidly growing tongue mass, it is an unusual diagnosis that should remind the treating physician that culture of tissue should be part of the evaluation if there is uncertainty in diagnosis.
- Accepted November 15, 2016.
- Address correspondence to James Naples, MD, Department of Otolaryngology, University of Connecticut Health Center, 263 Farmington Ave, Farmington, CT 06030. E-mail:
FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose.
FUNDING: No external funding.
POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose.
- Copyright © 2017 by the American Academy of Pediatrics