Salivary Glands 441
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k Miscellaneous Pathologic Processes of the Salivary Glands 441 Unfortunately, the infection is often not rec- CYSTIC FIBROSIS ognized as a manifestation of a first branchial arch abnormality and is treated with drainage or The composition of saliva is changed in cystic fibro- inadequate limited exploration, which will compli- sis and the formation of viscous mucus may lead to cate subsequent surgery. In the series by Triglia, cystic dilations of the ducts and acini especially in et al. (1998), 44% of patients had undergone prior the sublingual gland. The calcium concentration in surgery, while 65% of patients had incomplete saliva is also raised and microliths of calcium com- plexes with the viscous mucous can be seen. surgery before referral in another paper (Martinez, et al. 2007). According to Maddy and Ashram (2013) 50% of their patients had prior abscess drainage and 2 of 18 cases unsuccessful previous Saliva excisions. As the fistulas and sinuses communi- cate with the external auditory canal and their SALIVA AS A DIAGNOSTIC FLUID relationship to the facial nerve is variable a wide In many ways saliva represents an ideal fluid for parotidectomy exposure with dissection of the diagnostic analysis being readily available and not nerve is essential for complete removal. In fistulas requiring invasive techniques. Currently, there is to the auditory meatus removal of the cartilage much interest in developing technologies to use surrounding the fistulous tract is recommended saliva to diagnose, monitor progress, and assess (Figure 17.4). Rarely, defects of the tympanic treatment and recurrence of oral cancer. However, membrane in association with first branchial arch its mucus nature has made it difficult to analyze. fistulas are reported (Pradhu and Ingrams 2011). If In addition there is evidence to suggest that the the fistula or sinus tract is not completely removed methods of processing saliva prior to analysis may the lesion will recur and, although the recurrence have a significant effect on the results obtained rate is small, 3–5% (Stulner, et al. 2001), this for proteins in proteome analysis (Oshiro, et al. may increase in patients with previous infection 2007). In seeking to analyze the proteome of saliva, k or inadequate surgery. Computed tomography researchers are hoping to find specific diagnostic k fistulography is best to demonstrate the com- biomarkers and develop techniques to discriminate plete course of the tract if there is a cutaneous between these biomarkers using proteomic and opening (Goff, et al. 2012). Branchial cysts will genomic technologies (Wong 2006). Several differ- usually appear as parotid masses and are usu- ent research groups have examined varying aspects ally clinically diagnosed as cystic parotid tumors of salivary composition and have demonstrated (Figure 17.5). significant differences between saliva in healthy The first branchial lesions are usually superfi- subjects and those with oral cancer. Studies have cial to the nerve and Triglia, et al. (1998) reviewed examined biochemical and immunological param- 73 cases, including their 39, and found that 63% eters (Shpitzer, et al. 2007), salivary endothelin were superficial, 29% deep, and 8% between the levels (Pickering, et al. 2007), and reactive nitrogen nerve. In the small series by Solares, et al. (2003), species and antioxidant profile (Bahar, et al. 2007). however, 7 of 10 lesions were deep to the nerve Using genomic analysis, four mRNAs (OAZ, SAT, and 1 lay between the branches. A recent review IL8, IL1b) were identified that collectively had showed 72% of cases superficial to the nerve but a discriminatory power of 91% sensitivity and sometimes adherent, deep to the nerve in 17%, and specificity for detecting oral cancer (Zimmerman, between the nerve branches in 11% (Maddy and et al. 2007). Nonetheless, despite these promising Ashram 2013) (Figure 17.6). Larger cysts deep to initial results this technique remains a research the nerve may be difficult to remove as the nerve tool at the present time. An update review on the may be adherent to them (Figure 17.7) and dissec- current status of salivary markers for interested tion can be slow and tedious. readers is provided by Yakob, et al. (2014). It is very important to recognize the first arch Another area of current interest in diagno- abnormality as its complexity and anatomical vari- sis utilizing saliva is the detection of HPV given ety necessitates wide exposure through a parotidec- itsnowprovenstatusasacausativeetiologyin tomy incision and dissection of the facial nerve to oro-pharyngeal cancer (OPC). It is not as clear minimize the chances of subsequent facial nerve whether HPV associated with oral cancer has a damage. causative role as seen in OPC. In areas of high k k 442 Chapter 17 (a) (b) k k (c) (d) (e) Figure 17.4. (a) Operative photograph of patient with a discrete mass thought to be a parotid tumor. (Patient’s ear at lower right of image). While dissecting down the external auditory meatus a fistulous tract to the cartilage was identified and the clamp points to a bead of pus from the fistulous tract. (b) The fistula was removed with a rim of the cartilage from the ear canal and a superficial parotidectomy carried out to remove the branchial cyst (arrow). (c and d) The parotidectomy specimen shows the cyst deep in the parotid but it was lying superficial to the facial nerve. (e) Following superficial parotidectomy (the ear lobe is sutured up for surgical retraction). k k Miscellaneous Pathologic Processes of the Salivary Glands 443 (a) (b) k k (c) (d) Figure 17.5. (a and b) CT scans of large branchial cyst in the parotid gland. (c and d) Histology of the branchial (lymphoep- ithelial cyst). The proteinaceous cyst contents are superior and the arrows point to the squamous epithelial lining and its associated lymphoid follicles. rates of oral cancer such as India, high levels of burden of HPV related diseases (Adamopoulou, HPV 16 and 18 have been found in women with et al. 2013). cervical cancer and oral cancer (Kulkami, et al. 2011). Studies have shown the feasibility of detect- ing HPV 16 by saliva screening in healthy patients DROOLING (Turner, et al. 2011) and those with genital HPV The term drooling is often used synomonously (Peixoto, et al. 2011). It is hard at the current time with sialorrhea; however, virtually all patients to see what a positive HPV saliva test means and who drool do not have an increase in the amount what diagnostic or therapeutic implications it may of saliva they produce. Patients with Parkinson’s have for the patient. In view of the higher risk for disease with a reduced saliva production can often asymptomatic oral infection in women with genital suffer from drooling. Drooling is the result of a lack HPV-infection some authors have postulated that of coordinated swallowing with pooling of saliva prophylactic HPV vaccination may reduce the in the anterior floor of mouth with subsequent k k 444 Chapter 17 (a) (b) k k (c) (d) (e) (f) Figure 17.6. (a and b) Axial and coronal MR showing a superficial subcutaneous cyst in the left parotid gland in a 14-year-old girl. (c and d) The extent of the cyst is outlined by dotted line: an omega incision will be used for excision. The pre-auricular limb of the proposed incision is seen in (c) and the retroauricular portion is seen in (d). (e) The cyst lies superficial to the parotid gland. (f) After further dissection the fistulous attachment of the cyst to the cartilaginous external auditory meatus is revealed. (g) The 3-cm cyst was removed intact with a portion of the cartilaginous meatus to prevent recurrence. (h) The instrument points to the area where cartilage was removed from the external auditory meatus with the fistula. (i) Closure is performed with subcuticular sutures. k k Miscellaneous Pathologic Processes of the Salivary Glands 445 (g) (h) k k (i) Figure 17.6. (Continued) drooling as exemplified in conditions such as design. Some studies were poorly designed and cerebral palsy and amyotrophic lateral sclerosis. methodological flaws identified. Conclusions were This condition can have a severe impact on the that it was not possible to assess the efficacy of patient’s quality of life. Many different methods behavioral therapy and that further research is of managing this condition have been proposed needed (Van der Burg, et al. 2007). Drug therapy and are summarized in an excellent review by may be by the use of anticholinergics given orally Meningaud, et al. (2006). These patients benefit or by botulinum toxin injections. Certain condi- from a multidisciplinary team approach, and both tions such as glaucoma preclude the use of these medical and surgical treatments are used in their oral drugs and side effects are not uncommon. In management. Medical therapy includes oral motor a systematic review of the literature, only seven therapy, orofacial regulation therapy and behav- papers were found to meet the inclusion criteria ioral modification via biofeedback. In an analysis and the authors concluded that there was some evi- of studies from 1970–2005 of behavioral treatments dence benztropine, glycopyrrolate, and benzhexol of drooling, only 17 articles with 57 patients met hydrochloride were effective in children with drool- the inclusion criteria. The evidence base found 15 ing (Jongerius, et al. 2003). Botulinum toxin A has studies that used a single participant design and been injected into the parotid glands solely or 2 that used an experimental comparison group with the submandibular glands with good results. k k 446 Chapter 17 (a) (b) k k (c) (d) (e) Figure 17.7. (a) A 27-year-old man with a large cystic lesion in his right parotid. The FNAB showed benign disease.