1. Giant Infantile Nasal Minor Salivary Gland Lipomatosis: Case Report and Review of the Literature Basel Jabarin, MD, Tal Marom, MD, Ephraim Eviatar, MD, Jacob Pitaro, MD, MSc. Department of Otolaryngology-Head and Neck Surgery, Assaf Harofeh Medical Center, Tel Aviv University Sackler School of Medicine, Zerifin, Israel
INTRODUCTION
PATHOLOGY
Nasal obstruction in newborns may present at birth or develop over time.
Nasal obstruction at birth may lead to the development of early respiratory distress and failure to thrive (FTT).
Prenatal screening and tests, nasal endoscopy, and post-natal imaging studies, preferably magnetic resonance imaging (MRI), are helpful in defining and characterizing the obstructing mass. However, only histopathological examination can ultimately make the final diagnosis.
Minor salivary gland lipomatosis of the nasal cavity is a rare tumor that may cause nasal obstruction. In this report, we describe this pathology in an infant and review the literature.
Figure 3 shows H&E stained sections. The nasal polypoid masses were shown to be mainly composed of maturing adipose tissue, covered by respiratory mucosa, while the nasopharyngeal mass by squamous mucosa with an area of minor salivary glands embedded within the adipose tissue in the nasal part, and a well circumscribed mature nodule of cartilage in the nasopharyngeal part.
The source for the cartilage tissue could be from the vomeronasal organ, or more likely from the inferior turbinate since the two nasal masses were closely related to the lateral nasal wall.
DISCUSSION
Minor salivary gland lipomatosis occurring in the nasal cavity is extremely rare.
Lipomatosis described as “nontumoral deposition of adipose tissue throughout the gland resulting in its diffuse enlargement” (Saleh et al. 1998).
Since adipose tissue is rarely present in the nasal cavity, nasopharynx, and paranasal sinuses, adipose tissue tumors are not common in these regions (Fu and Perzin 1977).
This Table summarizes reported cases of minor salivary gland lipoma occurring in the head and neck region. As seen, most cases occur in the oral cavity where minor salivary glands are more abundant.
The present case stresses the importance of nasal endoscopy and imaging in a newborn presenting with dyspnea.
In our case the mass was seen on the second endoscopy only after it was identified on the MRI. This is due to the slowly growing characteristic of mass that was not apparent early. Therefore, a repeated nasal endoscopy should be considered when a dyspneic baby does not improve over time.
In conclusion, although minor salivary gland lipomatosis is rare in the nasal cavity, it should be considered as part of the differential diagnosis in a newborn with obstructing nasal mass.
Fig 1. Pre-operative MRI imaging study (T1 gadolinium-enhanced).
(A) Axial view: a well-defined homogenous fat-rich mass filling the left nasal cavity (white arrow).
(B) Fat suppressed sequence, axial view. The mass in the left nasal cavity is dark, confirming its fat content.
(C) Sagittal view, showing two separate masses in the anterior nose (arrow head) and the posterior nose/nasopharynx (white arrow).
(D) Fat suppressed sagittal view: the masses are clearly seen separated (arrow head). Note the hypointense area in the posterior lesion, which may represent bony or cartilaginous tissue (white arrow).
CASE PRESENTATION
A full-term female neonate presented with dyspnea that had developed few hours after birth.
Prenatal screening included normal ultrasound scans and fetal echocardiography. Pregnancy and delivery were normal.
Immediate intervention included oxygen supplementation and nasogastric tube insertion for feeding, with successful attempts of nasal passage from both nostrils.
Trans-nasal endoscopy demonstrated mild laryngomalacia, and flexible bronchoscopy was normal.
Since symptoms worsened, brain MRI was performed in order to exclude congenital malformations.
MRI showed normal brain and brainstem structures however, there were two separate masses in the left nasal cavity and nasopharynx, none had intra-cranial extension (Figure 1).
Repeated flexible nasal endoscopy demonstrated two smooth polypoid lesions obstructing the left nasal cavity and nasopharynx (Figure 2A).
Under general anesthesia, both masses were resected by endoscopic approach (Figure 2B, 2C).
The two masses were found to be connected to the floor of the nose and to the superior aspect of the soft palate. A small fibrous tissue band connected the two masses.
Post-operative course was remarkable for a significant improvement in the baby's condition, oral feeding was initiated few days after surgery, and the baby started to breathe normally and gained weight.
Fig 2. Intra-operative Findings.
(A) Endoscopic view of the anterior nasal mass.
(B) Excision of the anterior nasal mass.
(C) Gross surgical pathology of the anterior nasal mass, measuring 2.2*1*0.5 cm.
Fig 3. Histopathological Examination, H&E Staining (X100).
lobulated adipose proliferation covered by pseudostratified respiratory mucosa - nasal cavity part of the lesion.
Adipose tissue in the nasopharyngeal part of the lesion, covered by squamous mucosa.
Serous mucinous minor salivary glands embedded in the adipose tissue (X20).
Cartilage nodule in proximity to the adipose tissue.
CONTACT INFORMATION
Basel Jabarin, MD