Abstract
Salivary tissue can be present in the head and neck outside the usual locations of the major and minor salivary glands. This can be in the form of accessory salivary glands, in association with branchial cleft anomalies, or, less commonly, as heterotopic salivary gland tissue (HSGT). Heterotopic salivary gland tissue is defined as salivary tissue outside of the expected locations of major, minor, and accessory salivary glands with absence of clinical or histologic features of branchial cleft anomalies. Here we present the case of a 13-year-old girl who presented with a draining sinus of the lower neck, which was excised and, on histologic analysis, was consistent with HSGT. We include photographs and histologic images. A review of the literature on heterotopic salivary tissue in the neck is then presented including discussion of the presentation, clinical features, important considerations, and recommendations for management.
1
Case report
An otherwise healthy 13-year-old girl was referred to the pediatric otolaryngology clinic at Children’s Hospital of Wisconsin for drainage from a small lesion in her right lower neck. She and her parents reported that the lesion had been present since birth and was productive of clear fluid intermittently. The frequency had been increasing over the past year. There had never been purulence, edema, or erythema present to suggest acute infection, and there was no reported change with upper respiratory infections. Physical examination revealed the presence of a small dimple in the right lower neck, approximately 1 cm above the clavicle and anterior to the sternocleidomastoid muscle ( Fig. 1 ). There was no active drainage and no palpable cyst in association with the lesion. There were no neck masses or adenopathy present. To further characterize and define the extent of a suspected branchial cleft anomaly, a computed tomographic scan of the neck was performed. This revealed a 2-mm subcutaneous cyst at the site of the lesion but no further extension or association with the deeper structures of the neck.
Excision was recommended to the patient and her parents who wished to proceed. The patient was subsequently taken to the operating room. Under general anesthesia, the opening was able to be probed with a size 0000 lacrimal probe for a distance of approximately 1 cm. Next, a small elliptical incision was made in a skin crease around the lesion, and blunt dissection was carried down along the extent of the probed tract. This ended blindly in the subcutaneous tissue with no evidence of further extension. The base was tied off, and the specimen was excised and sent for histologic analysis. The wound was irrigated and closed, and the patient wakened from anesthesia without difficulty. She had an unremarkable postoperative course with no complications.
2
Histologic evaluation
On microscopic analysis, the lesion proved to be a minor salivary gland, located approximately 1 cm beneath the epidermal surface in the subcutaneous tissue, complete with a glandular duct leading toward the skin surface with feeding and draining vessels in the salivary gland hilum ( Fig. 2 ). The well-defined seromucinous salivary gland lobules showed a minor limited reactive lymphoplasmacytic infiltrate. Salivary gland ductules, the main draining duct, and feeding and draining vessels in the hilum were demonstrable in favorable orientation ( Fig. 3 ). The skin surface overlying the gland was intact in sections available for examination, with a central epidermal dimple and salivary duct-type cross-sections in the underlying dermis, 1.5 mm beneath the epidermis ( Fig. 4 ).
There were several histologic features present to suggest that this gland drained onto the skin surface as opposed to other mucosal surfaces not represented in this specimen. First, the salivary gland was completely excised, with a good amount of underlying subcutaneous tissue free of other histologic structures or inflammation. Second, the hilum of the salivary gland, the draining glandular ductule, and accompanying feeding vessels were all oriented toward the skin surface, and similar ductular cross-sections were seen beneath the skin surface. Third, the collagenous tissue in the 1 cm between the skin and the minor salivary gland were moderately dense and histologically uniform, uninterrupted by other structures. Fourth, there was a dimple on the skin surface in the region overlying the gland and ductular cross-sections. Fifth, the gland is normally formed, of appropriate size. There was no ductular ectasia or other evidence of obstruction or rupture, consistent with unobstructed drainage of produced saliva. Finally, no other types of structures or developmental remnants were identified; several deeper levels have been examined. Although midsegments of the draining duct and actual connection to the surface were not represented on sections available for examination, the above histologic features are supportive of the clinical interpretation that this minor salivary gland drained onto the epidermal surface—also correlating with the clinical history.
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