The ectopic accessory parotid system with congenital cheek fistula: An overview and current update.

OBJECTIVE: This review attempts to provide a comprehensive, updated overview of the ectopic accessory parotid system (EAPS) from clinical and developmental perspectives and investigates its eligibility to be included in the oculo-auriculo-vertebral spectrum (OAVS). REVIEW METHODS: Results of the keyword-based search in the PubMed/MEDLINE, Google Scholar, LILACS, and Cochrane Library were subjected to the given inclusion and exclusion criteria that corroborated with the definition of EAPS. Eleven records were shortlisted, their full texts studied and references cross-checked. Finally, 10 articles collectively describing 16 patients were considered for review. RESULTS: The average age of presentation was 8.33 years, with 44% of the patients aged below 5 years. Boys were affected 1.67 times more. The cheek fistula was always unilateral, with marginal left predilection. About 94% of the patients were of East Asian descent, chiefly from China and India. Thirteen children had preauricular appendages, mostly ipsilateral, with occasional ipsilateral mandibular hypoplasia (25%). The ectopic parotid was anterolateral to the masseter, lateral to buccinator, and inferior to the Stensen duct. Complete surgical removal of the EAPS (intraoral approach) or transposing the fistula into the oral cavity formed the mainstay of treatment. CONCLUSION: The EAPS is a rare craniofacial anomaly presenting with a saliva-draining cheek fistula, associated with ipsilateral preauricular appendages (microtia) and occasional mandibular hypoplasia. Speculative developmental theories suggest dysmorphogenesis of the first two pharyngeal arches, clinically and embryologically relating it with OAVS. This review summarizes the clinical aspects, and establishes isolated EAPS as a mild but discrete phenotype of OAVS irrespective of the presence of other congenital stigmata. Laryngoscope, 127:1351-1360, 2017.

Congenital ectopic fistula of a minor salivary gland.

A rare case of congenital salivary gland fistula is reported. A 3-year-old girl presented with clear discharge from a dimple on the left cheek. An ectopic salivary gland fistula was unexpectedly diagnosed during operation. This is the first case of congenital salivary fistula draining from a minor salivary gland to a cutaneous pit. We completely excised the lesion, and the patient remains complication free.

[Congenital anterior cervical fistula originating from the Wharton duct].

A 14-year-old boy presented with a spontaneous anterior cervical fistula, which exuded a saliva-like fluid during eating. Cervical examination revealed a hole 7 mm posterior to the hyoid bone, 5mm left of the median. Magnetic resonance imaging showed a low-intensity spindly mass with high-intensity margins. The mass was connected to the left submandibular gland. Contrast radiography of the fistula using Gastrografin demonstrated an ascending fistula that extended to the submandibular area. Intraoperatively, the fistula was shown to have ascended superficial to the level of the anterior cervical muscles, and it formed a cyst anterior to the hyoid bone. It then continued to the left submandibular gland. The fistula and submandibular gland were removed together. Wharton's duct was not found, and the orifice of the duct was unclear. Pathological examination of the fistula showed the lining epithelium at the side of the submandibular gland to be composed of columnar epithelial cells with stratified squamous cells at the epidermal side. Several acinar systems were found along the orifice of the fistula. There have been no previous reports of the Wharton duct running from the submandibular gland to the anterior cervical skin.

Salivary fistulae.

Three cases of unusual cervical fistulae are presented with a review of the literature to caution against labelling all lateral cervical fistulae as simple branchial fistulae or midline ones as dermoid cysts.

Parotid disease and human immunodeficiency virus infection in Zambia.

Among the salivary glands the parotid is unusual in that it contains lymphoid tissue within its capsule. The focus of infection with human immunodeficiency virus (HIV) is the lymphatic system and this results in a specific HIV-related pathology in the parotid. This 2-year surgical audit of parotid disease in HIV-infected patients in Lusaka shows patients presenting with parotid lymphadenopathy, bilateral diffuse parotid enlargement and parotid lymphoepithelial cysts. Clinical presentation and management are discussed.

PIP: The focus of infection with HIV is the lymphatic system, resulting in a specific HIV-related pathology in the parotid. Of the salivary glands, the parotid gland is unusual in including lymphoid tissue within its capsule. Infection by HIV is accompanied by a characteristic follicular hyperplasia of lymphoid tissue which can be recognized histologically as primary HIV lymphadenopathy and presents clinically as persistent generalized lymphadenopathy. Subsequent opportunistic infections and HIV-related neoplasia can result in secondary HIV lymphadenopathy. Parotid lymph nodes reflect these HIV-related changes. Diffuse enlargement of the parotid glands are further manifestations of HIV infection. All patients who presented to a general surgical unit of the University Teaching Hospital, Lusaka, Zambia, over the 2-year period of 1989-1991 were studied in a prospective clinicopathological study of lymphadenopathy. The diagnosis of HIV infection was made clinically and with a single serological test. Parotid lymphadenopathy was present in 69 of 261 HIV-seropositive patients with generalized lymphadenopathy who underwent lymph node biopsy during the 2-year period. In all but one patient, biopsy of a node other than the parotid provided the histological diagnosis. 9 patients presented with bilateral parotid enlargement. 8 of the patients had generalized lymphadenopathy caused by primary HIV lymphadenopathy in 5, Kaposi's disease in 2 and tuberculous lymphadenitis in 1. 4 patients presented with multiple cystic parotid lesions of between 1 month and 4 years duration. Unilateral extraparotid lymphoepithelial cysts of a diameter of 2 and 3 cm were removed from the jugulodigastric area of 2 patients with generalized lymphadenopathy. Parotid disease not related to HIV included: 1 case each of papillary carcinoma and pleomorphic adenoma; 7 patients with parotid lymph nodes, and 3 patients with diffuse bilateral parotid enlargement.

[Cutaneous parotid fistula of calculous origin]. / Fistule cutanée parotidienne d'origine lithiasique.

A case of cutaneous salivary fistula is presented. A 42-year-old man had a recurrent inflammatory parotid disease which disappeared with the expulsion of the calculus. The authors note the rarity of this evolution in the literature and suggest a protocol of treatment.

Congenital cervical salivary fistula.

A study of 35 lesions diagnosed as branchial cleft sinuses or fistulas identified a homogeneous subgroup characterized by ectopic salivary tissue, draining serous or mucoid fluid through a short sinus tract to the base of the neck anteriorly. These lesions should more properly be called congenital cervical salivary fistulas, since they may not arise from the branchial apparatus but rather from the embryonic placodal duct.

Sublingual gland salivary fistula and sialocele.

A case of cutaneous salivary fistula and sialocele in the submental area, arising from the sublingual gland, is reported and its etiology, development, and management are discussed. The entity of plunging ranula is compared to the lesion the case presented, and the literature regarding this lesion is reviewed. The successful surgical treatment of this case supports the theory that the sublingual gland is the source of the disorder. The effect of the altered salivary function on the adjacent submandibular gland and duct presented an operative complication.

[Effects of neurectomy of the tympanic plexus on secretion of the parotid gland in man. Histopathological, histochemical and clinical study (author's transl)]. / Effets de la neurectomie du plexus tympanique sur la sécrétion de la glande parotide chez l'homme.

This study involved five patients suffering from bilateral chronic parotiditis and then with a persistent unilateral fistula of the parotid gland. Two types of parotid biopsy were examined: the first taken during surgery and the second six months later. All of the specimens underwent histopathological and histochemical investigations. Definitive degeneration and atrophic type changes were seen in all the parotid fragments removed six months after selective neurectomy of the tympanic plexus. Six months after surgery, all of the patients were cured.

Effects of tympanic neurectomy on experimentally induced parotid fistulae.

The present work was designed to determine whether or not tympanic neurectomy alone could cure parotid fistulae. In six adult dogs parotid fistulae were induced and the tympanic nerve was transected on the right, while the nerve on the left side was left intact as a control. Complete healing of the fistulae on the right side was noticed, while those on the left side persisted. The findings and their significance are discussed.

Effect of tympanic neurectomy on human parotid salivary gland. Histopathologic, Histochemical, and Clinical Study.

Five patients with bilateral chronic recurrent parotiditis and ten patients with unilateral persistent paratoid gland fistulas underwent tympanic neurectomies. Two biopsy specimens from the diseased gland were examined. The first biopsy specimen was taken at the time of operation, while the second one was taken six months later. All specimens were subjected to both histopathologic and histochemical investigations. Definite degenerative and atrophic changes occured in all parotid gland specimens taken six months after selective tympanic neurectomy. All patients were completely cured at about that time. This supported the practical indication of the selective tympanic neurectomy for treating certain benign disorders or diseases of the parotid gland that require persistent arrest of the function of its secretory units.