Periocular Mass Due to Persistent Secretion Within a Transplanted Parotid Duct.

A male patient underwent microvascular transplantation of an autologous submandibular gland and its associated (Wharton's) duct for management of very severe right keratoconjunctivitis sicca due to Stevens-Johnson syndrome; due to hypersecretion, the gland was later removed in toto. Twenty-two years later, he presented with a nontender, sausage-like mass lying subcutaneously in the right temple. Intraoperatively a fluctuant tubular mass passing from the temporalis fossa to the superotemporal conjunctiva was identified and excised intact. Histology showed a slightly proteinaceous fluid within a duct lined with cuboidal epithelium, this being compatible with Wharton's duct. This case highlights that iatrogenic causes should be considered with any history of periocular injury or surgery.

Microvascular Submandibular Gland Transplantation for Severe Keratoconjunctivitis Sicca: A Single-Institution Experience of 61 Grafts.

PURPOSE: Keratoconjunctivitis sicca (KCS) is a relatively common disease that results in discomfort, tear film instability, visual impairment, and ocular surface damage. Artificial tear substitutes may be suitable for the treatment of mild KCS, but no effective treatment currently exists for severe KCS. Therefore, this study evaluated the effectiveness of autologous microvascular submandibular gland transplantation in the treatment of severe KCS. PATIENTS AND METHODS: A total of 61 eyes (56 patients) with severe KCS were treated with autologous submandibular gland transplantation from June 2002 to June 2017. The cephalic vein or the great saphenous vein was applied to solve the problem of unmatched veins. RESULTS: In 53 cases (53 of 56, 94.6%), 58 glands (58 of 61, 95.1%) were transplanted successfully. The mean Schirmer I test value improved from 0.78 ± 0.84 mm preoperatively to 18.83 ± 5.72 mm in the stable period after transplantation. Epiphora (14 of 58, 24.14%) was the most common complication of this procedure. Other postoperative complications included venous thrombosis (6 of 61, 9.84%), local infection (2 of 58, 3.45%), xerostomia (2 of 53, 3.77%), duct fistula (1 of 58, 1.72%), sialolithiasis (1 of 58, 1.72%), and ranula (1 of 58, 1.72%). CONCLUSIONS: Autologous microvascular submandibular gland transplantation is a credible and effective solution for severe KCS.

Canine oral mucosa evaluation as a potential autograft tissue for the treatment of unresponsive keratoconjunctivitis sicca.

PURPOSE: Labial mucosa transplantation for the treatment of canine keratoconjunctivitis sicca (KCS) has been reported recently. Postoperative alleviation of clinical signs was noted and assumed to be the result of labial salivary glands providing lubrication to the ocular tissue. The aim of this study was to evaluate the presence of minor salivary glands (MSG) in the canine oral mucosa. METHODS: Oral mucosal biopsies were collected from six dogs that died (n = 1) or were euthanized (n = 5) for reasons unrelated to this study. The breeds included were two Doberman Pinschers, one Labrador Retriever, one Portuguese Water Dog, one German Shepherd Dog, and one mixed canine. Three were spayed females, and three were castrated males with the median age of 9 years (range, 6-13 years). Samples were obtained by an 8-mm punch biopsy at the following locations of the canine oral cavity: upper rostral labial mucosa at midline, lower rostral labial mucosa at midline, upper labial mucosa near the commissure, lower labial mucosa near the commissure, and buccal mucosa approximately 1 cm caudal to the commissure. Samples were routinely processed with hematoxylin and eosin, and periodic acid-Schiff stains. Samples were evaluated by light microscopy. RESULTS: At the selected locations, no MSG or other secreting cells were detected. CONCLUSIONS: Minor salivary glands are not associated with alleviation of canine KCS symptoms following labial mucosa transplantation. Further studies are needed to determine the mechanism leading to the transient improvement of KCS symptoms in canine patients following labial mucosa transplantation.

A novel treatment for keratitis sicca (Dry eye): Anatomical feasibility study.

Chronic dry eye (keratitis sicca) is a significant problem that in certain populations can result in corneal desiccation and the potential for blindness. Therefore, novel treatments for such disorders might decrease patient morbidity. The present study aimed to investigate a potential treatment for chronic dry eye via a cadaveric feasibility study. On 10 cadaveric sides, the parotid gland branch of the auriculotemporal nerve (ATN) was identified and anastomosed to an anterior superficial temporal branch (STb) of this same nerve. The STb was then transposed anteriorly and sutured to the lacrimal gland. The parotid branch of the ATN was easily identified on all sides. The STb of the ATN was easily identified and mobilized on all sides. This latter nerve had adequate length to be moved to the ipsilateral lacrimal gland on all sides. Rerouting parotid gland secretomotor fibers to the superficial branch of the ATN and then moving this branch to the lacrimal gland is a feasible surgical maneuver based on our cadaveric study. Clinical studies are now necessary to show utility of this procedure in patients with chronic dry eye. Clin. Anat. 30:839-843, 2017. © 2017Wiley Periodicals, Inc.

Bilateral parotid duct transposition for keratoconjunctivitis sicca in a Connemara stallion.

A 7-year-old Connemara stallion was presented with a 4 month history of blepharospasm, recurrent corneal ulcerations, mucopurulent ocular discharge, and keratoconjunctivitis sicca (KCS) in both eyes unresponsive to medical therapy. Ophthalmic examination revealed lackluster corneas, axial corneal scarring and pigmentation with associated neovascularization, and absolute KCS in both eyes. Computed tomography scan and endoscopic evaluation of the upper airway and guttural pouches revealed no structural abnormalities to indicate neurogenic KCS. The stallion was diagnosed with immune-mediated dacryoadenitis as all other causes of KCS were excluded. Parotid duct transposition (PDT) was performed in the right eye followed by PDT in the left eye 4 weeks later. The right PDT was functional 2 years post-operatively with significant improvement in ocular comfort and reduced corneal fibrosis and neovascularization. The left PDT developed a salivary-cutaneous fistula over the left masseter muscle post-operatively due to avascular necrosis of the distal parotid duct (PD). Surgical reconstruction of the PDT using an expanded-polytetrafluoroethylene (e-PTFE) tube graft, an e-PTFE tube graft to autogenous caudal auricular vein graft, and an autogenous saphenous vein graft were all unsuccessful. Tear production in the left eye improved at 1 year post-surgery as a result of long term lacrostimulant therapy, and a permanent PD-cutaneous fistula was performed on the left PD at the level of the ventral mandible. Bilateral PDT in the horse is effective in resolving clinical signs associated with KCS; however, morbidity associated with avascular necrosis of the transposed PD may be significant and can result in surgical failure.

Parotid duct transposition in dogs: a retrospective review of 92 eyes from 1999 to 2009.

OBJECTIVE: The aim of this study was to perform a retrospective review of parotid duct transposition (PDT) in the dog to determine the rate and nature of complications, the success and failure rates and to evaluate owner satisfaction. METHODS: Medical records of 56 dogs (92 eyes) that underwent PDT and subsequent follow-up by a veterinary ophthalmologist were reviewed. Forty owners (40 dogs/66 eyes) were contacted by telephone and 37 owners (37 dogs/60 eyes) also completed a visual analog scale questionnaire. Statistical evaluation included Wilcoxon Signed Rank tests, one-way analysis of variance and Kaplan-Meier survival analysis with Wilcoxon and Log-rank tests. RESULTS: The mean follow-up was 38.7 months (range 1-109 months). The surgical success rate was 92% (85/92 eyes). Total failures (8%, 7/92 eyes) were because of severe saliva intolerance (n = 5 eyes) and PDT failure (n = 2 eyes). The complication rate was 50% (46/92 eyes) of which 61% (28/46 eyes) were managed medically and 39% (18/46 eyes) required further surgery. Ninety percent (36/40) of owners indicated that they would proceed with surgery again. Statistically significant improvements in owner perception of ocular comfort, number of daily topical treatments, ocular wetness, and postoperative vision were identified. CONCLUSIONS: This study shows that PDT is a successful procedure based on clinical findings and in terms of owner perception. It has also demonstrated that PDT improves ocular comfort and vision in medically refractive cases of keratoconjunctivitis sicca, and that a low level of on-going management is required in 33% of cases.

Partial ligation of the transposed parotid duct at the level of the parotid gland for excessive salivary secretions.

OBJECTIVE: To determine the outcome and effect of a partial ligation of the transposed parotid duct at the level of the parotid gland in four dogs with excessive salivation and ocular irritation. METHODS: Four dogs were previously diagnosed with absolute keratoconjunctivitis sicca. After a parotid duct transposition (PDT) surgery, these dogs experienced excessive saliva production and abundant salivary precipitates, which resulted in epiphora, moist dermatitis, blepharospasm, and keratitis. In an effort to decrease saliva production, a partial ligation of the transposed duct at the level of the parotid gland was performed. Two or three accessory branches to the primary parotid duct were ligated at the level of the salivary gland. RESULTS: The four cases were three Yorkshire terriers and a Chihuahua. The average age of the four patients was 2.5 years. Partial ligation of accessory branches of the parotid duct at the level of the parotid gland after a PDT in this study demonstrated improved ocular comfort, decreased salivary precipitates, and adequate Schirmer tear test results without marked epiphora in three of the four animals. The male Yorkshire had epiphora after the initial partial ligations of two accessory branches were placed at the level of the parotid gland. To correct the excessive salivary flow, two additional ligatures were placed at a later date, which resolved the epiphora. CONCLUSION: Partial ligation of the parotid duct at the level of the parotid gland proved to be an effective technique in moderating the salivation in these four patients with excessive salivary secretions after PDT.

Changes in tear production in a dog with keratoconjunctivitis sicca for a year after inevitable nictitating membrane gland resection due to suspicion of a malignant tumour.

Since the removal of the NM causes KCS in dogs, it was contraindicated to remove the NM unless unavoidable such as in a malignant tumour. However, to the best of author's knowledge, there are no reports of conjunctivitis and keratitis owing to decreased tear production following removal of the NM gland. This case study demonstrates the tear production changes in a dog for a year after removal of the nictitating membrane (NM) due to suspicion of a malignant tumour. A 13-year-old spayed female English Cocker Spaniel who had suffered from severe ocular discharge, discomfort, keratoconjunctivitis sicca (KCS), and NM enlargement in the right eye was brought to our hospital. The dog could not tolerate treatment with topical 0.2% cyclosporine or corticosteroids. The dog's right eye had NM gland prolapse, severe follicular conjunctivitis and a very low Schirmer tear test-1 (STT-1) value of 3 mm/min. Furthermore, the result of fine needle aspiration of the enlarged NM gland suggested a risk of malignancy. Despite the risk of KCS, complete NM resection was performed to diagnose the tumour. Fortunately, the final histopathological evaluation revealed chronic inflammation without any evidence of malignancy. Contrary to concerns that the STT-1 value would further decrease after the removal of the NM gland, the STT-1 value remained elevated compared to that before surgery, and the clinical symptoms improved for a year. It is generally known that NM gland resection is not recommended due to the risk of developing iatrogenic KCS unless a malignant tumour is suspected. In this case, surgical removal of the inflammatory NM gland that was not responsive to medications had a positive effect on KCS. Since the inflammatory and structural disease of the NM was strengthening KCS, the outcome was thought to be different from that when the normal third eyelid was removed.

Microanatomic study of the vascular and duct system of the submandibular gland.

PURPOSE: Autologous microvascular submandibular gland (SMG) transfer is an effective treatment for severe keratoconjunctivitis sicca (KCS). However, epiphora (excessive tear secretion) may occur after the successful transfer of whole submandibular gland because tear secretion level is closely related to the size of the transferred gland. The aim of this study was to investigate the microanatomy of SMG to explore the possibility of partial SMG transfer to prevent postoperative epiphora. MATERIALS AND METHODS: Sixty intact and histologically normal human SMGs from patients with benign tumor of the mandible who underwent vascularized mandibular reconstruction and removal of the SMG for anastomosis of the blood vessels were included in the study. SMGs were perfused with methacrylate to form resin casts of blood vessels and ducts. The length and diameter of the blood vessels and ducts in the casts were measured using a sliding caliper. The numbers of lobules, distribution of arteries, veins, and ducts, as well as the relationship among them, were analyzed. RESULTS: The resin cast of the gland showed a treelike structure, with the vessels gradually dividing into multiple branches. The arteries, veins, and ducts run in parallel and were roughly divided into 3 levels: from the stem extending into the main branches (level I), into the narrower secondary branches (level II), and then the secondary branches subsequently divided into terminal branches (level III). The structures of the blood vessels and ducts were similar at each level in the lobules. In the vein casts, communicating vessels were found between the anterior facial vein and the concomitant vein of the facial artery. CONCLUSION: The characteristic treelike structure of the SMG vascular and ductal system may provide useful information for partial gland transfers.

Clinical and anatomic study on the ducts of the submandibular and sublingual glands.

PURPOSE: To investigate the relationship between the ducts of the submandibular gland (SMG) and sublingual gland (SLG) and discuss its clinical application relating to SMG radiologic examinations and transfer. MATERIALS AND METHODS: The microanatomy of the SMG and SLG was investigated by use of 30 adult cadavers through anatomic dissection by use of a microscope. The relationship between the SMG and SLG ducts was observed and recorded during operations of microvascular autologous SMG transfer in 63 cases of severe keratoconjunctivitis sicca. RESULTS: There were 3 patterns of SLG and SMG duct anatomic variation: 1) The SMG and SLG have their own respective ducts that secrete separately at the orifices of the ducts in the floor of the mouth. 2) The SLG has a major duct that joins the duct of the SMG. 3) The SLG only has many fine ducts (7-15) that secrete in the floor of the mouth. CONCLUSIONS: The anatomy of the ducts of the SMG and SLG is quite complicated. More attention should be paid to the anatomy of the ducts during surgery or imaging procedures related to the SMG.

[Post-operation nursing for patients with autologous transplantation of the microvascular submandibular gland].

OBJECTIVE: To summarize the complications' prevention and nursing experience for patients after autologous transplantation of the microvascular submandibular gland for severe keratoconjunctivis sicca. METHODS: Retrospective analysis of 141 patients(151 sides) after autologous microvascular submandibular gland transfer operation was undertaken to record the timing and incidence of major complications and to summarize the nursing experience. RESULTS: The main complications after the transplantation were: vascular crisis, catheter obstruction, salivary fistula and temporary paralysis of hypoglossal nerve. Related nursing measures in response to these complications were discussed. CONCLUSION: Targeted nursing interventions for patients with autologous transplantation the submandibular gland have a positive effect on the prevention of the surgical complications.

Factors influencing the long-term results of autologous microvascular submandibular gland transplantation for severe dry eye disease.

We assessed long-term outcomes of autologous microvascular submandibular gland (SMG) transplantation for severe dry eye disease and investigated factors influencing long-term results. From August 1999 to January 2015, 185 patients (200 eyes) with severe dry eye received SMG transplantation. Subjective assessments and ophthalmologic evaluations were performed before and after transplantation. Follow-up results showed successful transplantation in 180 of 200 eyes (success rate: 90%), resulting in marked symptomatic relief of xerophthalmia. Surgery failed due to vascular thrombosis (15 glands) and duct obstruction (5 glands). Follow-up data were available for 163 eyes. Epiphora occurred in 98 (60.1%) eyes and was effectively managed by surgical reduction of graft, topical atropine gel and botulinum toxin injection. Wharton's duct obstruction occurred in 16 (10.6%) eyes and was treated by duct reconstruction. Subjective satisfaction was achieved in 143 (87.7%) eyes. Mean score of fluorescent staining reduced from 11.25±1.42 to 7.25±3.37. Postoperative best-corrected visual acuity improved in 85 (56.3%) eyes. Our clinical experience proved that SMG transplantation is effective and grants long-term improvement in severe dry eye. Secretory function of transplanted SMGs remains active and stable. Blood vessel thrombosis, Wharton's duct obstruction, and epiphora are primary factors influencing results.

Long-term results of autologous submandibular gland transfer for the surgical treatment of severe keratoconjunctivitis sicca.

AIM: The aim of this study was to assess the long-term results of autologous submandibular gland transfer for surgical correction of severe keratoconjunctivitis sicca. PATIENTS AND METHODS: A survey was undertaken of 32 patients who had undergone submandibular gland transfer (42 glands) and by following up 11 patients (15 glands) for 5-10 years. Subjective benefit was evaluated as well as clinical findings at the ocular surface. The biochemical consistency of the secreted "saliva-tear" was analysed and compared with natural submandibular saliva of a matched control-group. The vitality and function of the transplants was tested by means of sialoscintigraphy. Immunohistochemical investigations were carried out in specimens of submandibular tissue, gained during reduction procedures of the transplants to correct secretory excess. RESULTS: Patient evaluation and clinical assessment revealed a long-lasting subjective benefit in 2/3 of the patients and a stabilisation at the ocular surface in all cases. The secretion remained as highly concentrated submandibular saliva. Glandular vitality and function was shown scintgraphically. Immunohistochemical investigations revealed no progressive atrophy after transplantation, the ability of cell division remained intact and there was still neuronal tissue in all transplants, even several years after transfer. As all transplants responded well to parasympathomimetic drugs, this might be an indication of re-innervation of the gland.

Boston Type I Keratoprosthesis Implantation Following Autologous Submandibular Gland Transplantation for End Stage Ocular Surface Disorders.

PURPOSE: To describe the results of Boston keratoprosthesis implantation in a patient with Stevens-Johnson syndrome following autologous submandibular gland transplantation. DESIGN: Observational case report. METHODS: We report the case of a man diagnosed with Stevens-Johnson syndrome. Autologous submandibular gland transplantation was performed in the right eye in 2008 to ameliorate tear film deficiency. Due to the improvement in the microenvironment of the ocular surface, Boston keratoprosthesis implantation was performed in the right eye in 2011. RESULTS: He maintained a VA of 20/100 for 12 months. At the last follow-up visit (54 months), his VA had declined to finger count level due to de novo glaucoma, which was treated with glaucoma drainage device implantation, cyclophotocoagulation and topical anti-glaucoma medications. CONCLUSIONS: The procedures described provide patients with end-stage ocular surface disease an option to lessen the severity of keratoconjunctivitis sicca and to regain their VA.

Autonomic reinnervation and functional regeneration in autologous transplanted submandibular glands in patients with severe keratoconjunctivitis sicca.

Autologous submandibular gland (SMG) transplantation has been proved to ameliorate the discomforts in patients with severe keratoconjunctivitis sicca. The transplanted glands underwent a hypofunctional period and then restored secretion spontaneously. This study aims to investigate whether autonomic nerves reinnervate the grafts and contribute to the functional recovery, and further determine the origin of these nerves. Parts of the transplanted SMGs were collected from the epiphora patients, and a rabbit SMG transplantation model was established to fulfill the serial observation on the transplanted glands with time. The results showed that autonomic nerves distributed in the transplanted SMGs and parasympathetic ganglionic cells were observed in the stroma of the glands. Low-dense and unevenly distributed cholinergic axons, severe acinar atrophy and fibrosis were visible in the patients' glands 4-6 months post-transplantation, whereas the cholinergic axon density and acinar area were increased with time. The acinar area or the secretory flow rate of the transplanted glands was statistically correlated with the cholinergic axon density in the rabbit model, respectively. Meanwhile, large cholinergic nerve trunks were found to locate in the temporal fascia lower to the gland, and sympathetic plexus concomitant with the arteries was observed both in the adjacent fascia and in the stroma of the glands. In summary, the transplanted SMGs are reinnervated by autonomic nerves and the cholinergic nerves play a role in the morphological and functional restoration of the glands. Moreover, these autonomic nerves might originate from the auriculotemporal nerve and the sympathetic plexus around the supplying arteries.

Application of 99mTc-pertechnetate scintigraphy to microvascular autologous transplantation of the submandibular gland in patients with severe keratoconjunctivitis sicca.

UNLABELLED: Our objective was to evaluate the role of (99m)Tc-pertechnetate scintigraphy on microvascular autologous transplantation of the submandibular gland in patients with severe keratoconjunctivitis sicca (KCS). METHODS: (99m)Tc-Pertechnetate scintigraphy was performed on 106 patients with severe KCS. The patients were examined before surgery and at 1 wk and 3 mo afterward using a standardized protocol that included static scintigrams, time-activity curves, and delayed scintigrams to check the function and secretion of the major salivary glands. The scintigraphic findings were assessed visually. When possible, the scintigraphic findings were compared with the clinical appearance of the transplanted gland. RESULTS: The function of all 4 major salivary glands was almost completely lost in 10 patients, indicating that these patients were not suitable for transplantation. The other 96 patients were treated by autologous transplantation of the submandibular gland. In 14 patients, the function of the major salivary glands was below normal. One patient's scintigram, obtained on the second day after surgery, showed no uptake of (99m)Tc-pertechnetate in the transplanted gland. Surgical exploration showed embolism of the artery of the transplanted gland. Scintigraphy was performed 1 wk after surgery in 90 patients. There was no uptake of (99m)Tc-pertechnetate in the temporal region in 8 patients, indicating that the glands were not revascularized. Scintigraphy showed obvious uptake of (99m)Tc-pertechnetate in the temporal region of the other 82 patients, indicating that the transplanted glands were viable. At more than 3 mo after surgery, scintigraphy was again performed on 30 patients. Scintigraphy after a 120-min delay showed that (99m)Tc-pertechnetate drained into the orbit through the duct of the transplanted gland in 26 patients. However, no secretion into the orbit was found in 4 patients, indicating obstruction of the duct. CONCLUSION: Scintigraphy plays an important role in microvascular autologous transplantation of the submandibular gland in patients with severe KCS. Scintigraphy can be used to select patients and donor glands, to evaluate the viability of the graft, and to check the patency of Wharton's duct of the transplanted gland.

Lacrimal punctum occlusion in the treatment of severe keratoconjunctivitis Sicca caused by Sjögren syndrome: a uniocular evaluation.

PURPOSE: A controlled uniocular study to evaluate the short-term efficacy of lacrimal punctum occlusion in the treatment of severe dry eye caused by Sjögren syndrome. METHODS: Uniocular punctum occlusion by punctum plug in the upper and lower puncta in 1 eye was performed in 20 patients with severe keratoconjunctivitis Sicca caused by Sjögren syndrome. To overcome possible interindividual variability between patients, the other eye, in the same patient, was not occluded and served as a control eye. The eye to be occluded was randomly selected. The patients were instructed to continue using their dry eye medications. Tear function tests (Schirmer test, rose Bengal test, and debris in de cul-de-sac) were performed in both eyes. Subjective complaints (discomfort) were registered for both eyes. All the above-mentioned data were collected before starting the treatment and at least 6 weeks later. The above-mentioned parameters were compared and statistically analyzed in both eyes. RESULTS: Of the 20 patients, 7 patients dropped out. The remaining 13 patients completed the final analysis. In the occluded eye, we found a significant improvement in both the subjective complaints and the rose Bengal score, but the Schirmer test and the tear mucus score did not change. CONCLUSION: Punctum occlusion therapy in a short-term study improved the rose Bengal score and discomfort score in our patients and thus may be helpful in the treatment of severe dry eye caused by Sjögren syndrome.

Spontaneous Canalicular Plug Erosion After Punctal Plug Placement.

PURPOSE: To describe a case of keratoconjunctivitis sicca treated with punctal plug placement with the first reported complication of subsequent plug erosion through the canaliculus and extrusion through the lid. METHODS: Punctal plugs were placed in a patient with Sjogren syndrome for treatment of keratoconjunctivitis sicca. More than 12 years later, the plug was noted to have eroded through the canaliculus and extruded through the conjunctiva and lid. RESULTS: The extruded plug was removed, and punctal cautery was performed on 2 other puncta with improved symptom control. CONCLUSIONS: Plug extrusion through the conjunctiva and lid is a possible consequence of punctal plug placement.

The Use of Conjunctival Pedicle Flaps to Prevent Corneal Perforation in Graft-Versus-Host Disease.

PURPOSE: We report the clinical outcomes of two chronic graft-versus-host disease (cGVHD) corneal thinning cases that were successfully treated with conjunctival pedicle flaps in an effort to prevent impending corneal perforation. METHODS: The patients were treated with topical and systemic corticosteroids, therapeutic contact lenses, lubricants, and punctual plugs. Conjunctival pedicle flaps were used because conventional treatments had failed, and corneal perforation was imminent. RESULTS: Both patients were successfully treated with conjunctival pedicle flaps. Following conjunctival flap removal, one patient had corneal clarity restored following penetrating keratoplasty. CONCLUSIONS: Conjunctival pedicle flaps provided a strong barrier for the corneal surface and stabilized anterior chamber in cGVHD patients with impending corneal perforation following allogeneic hematopoietic stem cell transplant.