Mohammad Javed Ali, Dilip Kumar Mishra & Milind N. Naik
Urbach-Weithe syndrome is an exceedingly rare multisystem disorder characterized by pathognomonic clinical findings of multiple beaded papules along the eyelid margins and peri-ocular areas and hoarseness of voice secondary to intercellular deposition of periodic acid Schiff (PAS)-positive hyaline material. Lacrimal drainage anomalies are not well defined in this syndrome; however, punctal involvement and acquired nasolacrimal duct obstructions have been reported. We present a patient of Urbach-Weithe syndrome with bilateral punctal and peri-punctal involvement.
Rijul S. Kshirsagar, Priscilla Q. Vu & Jonathan Liang
Purpose: Endoscopic surgeries, such as dacryocystorhinostomy (DCR), are increasingly performed for orbital and lacrimal conditions. This study describes and compares recent trends in endoscopic DCR with open, or external, DCR in the United States (US).
Methods: Medicare-Part-B National Summary data files were analyzed from 2000 to 2015 for temporal and geographic trends in endoscopic and external DCR. Medicare Physician and Other Supplier public use files detailing provider information were collected and analyzed from 2012 to 2015.
Results: Between 2000 and 2015, the number of external DCRs remained relatively unchanged (8008 to 7086, −0.7% average annual growth), while the number of endoscopic DCRs steadily increased (881 to 1674, 4.6% average annual growth). The greatest number of endoscopic DCRs were performed in the South Atlantic region, whereas the Mountain region had the greatest number per capita. From 2000 to 2015, the average payment per procedure for external DCR was $526.63, compared with $512.45 for endoscopic DCR. Of endoscopic DCRs performed from 2012 to 2015, 831 (79%) were performed by Ophthalmology, 184 (18%) were performed by Otolaryngology, and the remainder by other subspecialties.
Conclusions: The number of endoscopic DCR surgeries increased over the last 15 years while the number of external DCR surgeries remained stable and continued to surpass endoscopic procedures. While ophthalmologists perform the overwhelming majority of endoscopic DCR, otolaryngologists are performing a growing number.
Everett, Lesley A.; Copperman, Thomas S.; Kersten, Robert C.
Ductal carcinoma of the lacrimal gland is a very rare and aggressive neoplasm, with clinical and histopathologic similarities to salivary ductal carcinoma. Of the 25 previously reported cases, 2 patients had metastases to local lymph nodes confirmed on pathologic examination. The authors now report the clinical presentation, histopathologic and immunohistochemical features, and the treatment of a third patient with lacrimal gland ductal carcinoma with spread to local lymph nodes. In contrast to ductal carcinoma, lymph node involvement has not been reported in the largest series of adenoid cystic carcinoma, a much more common lacrimal gland malignancy. This case highlights the need for possible lymph node surveillance in patients with lacrimal gland ductal carcinoma.
Kwan, Changyow C.; Prager, Alisa J.; Huang, Russell M.; Bryar, Paul J.; Thyparampil, Preeti J.
This is the first case of histopathologically proven blastomycosis involving the lacrimal gland. A 51-year-old woman with a history of disseminated blastomycosis involving her lungs and skin, on oral itraconazole, presented with 3 days of right upper eyelid swelling, erythema, and pain concerning for recurrent dacryoadenitis. MRI showed enlargement of the right lacrimal gland with a cystic lesion at the anterior aspect of the gland with a radiographic differential diagnosis of abscess versus cyst. After no improvement with intravenous antibiotics, orbitotomy with lacrimal gland biopsy and incision and drainage of the cystic lesion were performed. Culture and pathology of the drained fluid demonstrated an abscess with both viable and nonviable broad-based budding yeast consistent with partially treated blastomycosis. The patient’s symptoms improved after the surgery and continued itraconazole therapy.
Alsulaiman, Naif; Alsuhaibani, Adel H.
Purpose: To evaluate the use of bicanalicular silicone intubation for the management of punctal stenosis and obstruction in patients with allergic conjunctivitis.
Methods: A retrospective interventional case series of patients with acquired epiphora due to stenotic or obstructed puncta as a result of allergic conjunctivitis was performed. Punctal dilation and bicanalicular silicone intubation were performed in all patients. Munk Scale for grading of epiphora along with grading of fluorescein dye disappearance test was used to evaluate the functional improvement. Grading of punctal stenosis using Kashkouli’s grading system was applied to evaluate the anatomical improvement. Patients having canalicular or nasolacrimal duct obstruction, punctal stenosis, and obstruction due to other causes were excluded.
Results: Fifty-one patients met the inclusion criteria with male-female ratio of 1:1.55 and an average age of 46 years at presentation. High patient tolerances without complications with the use of tubes were reported. Significant improvement 6 months after tube removal in comparison to preoperative period was found with anatomical and functional success rate of 91.83% and 87.75%, respectively. Two patients had a recurrence of the punctal stenosis many months after stent removal because of the early tube prolapse and exacerbation of the allergic conjunctivitis.
Conclusions: Bicanalicular silicone intubation seems to be a well-tolerated and effective tool in the management of acquired punctal stenosis or obstruction secondary to allergic conjunctivitis. Bicanalicular silicone intubation appears to be a good option as patients with allergic conjunctivitis typically present with bilateral involvement of both puncti.
Ali, Mohammad Javed; Paulsen, Friedrich
Purpose: To provide a systematic review of the literature on the etiopathogenesis of primary acquired nasolacrimal duct obstruction (PANDO).
Methods: The authors performed a PubMed search of all articles published in English with specific reference to etiopathogenesis of PANDO or associations of PANDO. Data captured include demographics, study techniques, hypothesis, presumed or confirmed interpretations with regards to pathogenesis, mechanisms, or pathways. Specific emphasis was laid on addressing the lacunae and potential directions for future research.
Results: Numerous factors are believed to contribute to the etiopathogenesis of PANDO. The basic pathogenesis involves inflammation, vascular congestion, mucosal edema, fibrosis, obstruction, and stasis. Bony nasolacrimal duct diameter does not appear to play a significant role. There is no convincing data to substantiate nose as the site of disease origin and nasal factors appear to be comorbidities. Hormonal mechanisms are more evidence-based but can only partly explain the pathogenesis. Vascular theories are based on the behavior of perilacrimal cavernous bodies, their autonomic control, and additional structural changes in the helical patterns of connective tissue fiber arrangements. Repeated vascular malfunction leading to structural epithelial and subepithelial changes currently appears to be the most evidence-based and accepted theory. Tear proteomics holds a promise in decoding the etiopathogenesis of PANDO, at least in part.
Conclusions: The etiopathogenesis of PANDO appears to be multifactorial. Hormonal microenvironments, vascular factors, and tear proteomics are promising candidates for further work. There is a need for focused work by Clinician-Scientists and the answers can have far reaching clinical implications.
Edward J. Wladis, Vinay K. Aakalu, Jeremiah P. Tao, Rachel K. Sobel, Suzanne K. Freitag, Jill A. Foster, Louise A. Mawn
To determine the efficacy and complication rates of monocanalicular stents in the setting of canalicular lacerations.
A literature search was performed in May 2018 in the PubMed database to identify all English-language reports of monocanalicular stenting to address canalicular lacerations. Studies that did not include at least 10 patients with at least 3 months of follow-up evaluation after surgery were excluded. Ninety-nine articles were identified, and 15 of these met criteria for data abstraction and were included in this assessment. The panel methodologist (V.K.A.) evaluated the quality of evidence and assigned a level-of-evidence rating to each of these studies.
All 15 studies were rated as level III evidence. Anatomic and functional success rates after surgery ranged from 68% to 100% and 79% to 100%, respectively. Stents were generally well tolerated, although extrusion rates varied from 0% to 29%.
Only level III evidence was available, and studies were not powered to detect differences between groups for rare complications or failure. Monocanalicular stents seem to be efficacious and well tolerated in the management of canalicular lacerations. Potential complications include extrusion (most commonly), tube displacement, granuloma, ectropion, slit punctum, fistula, and infection. Further comparative studies would help to identify the optimal time for device removal and to directly compare monocanalicular with bicanalicular stents.
Tom Kornhauser, Avichai Segal, Eyal Walter, Tova Lifshitz, Morris Hartstein, Erez Tsumi
To describe a previously unreported phenomenon of idiopathic edematous punctal stenosis (IEPS) with chronic epiphora, presenting almost exclusively in young female patients.
A review of patients who presented with chronic epiphora and edematous punctal stenosis of unknown cause (IEPS) at the outpatient clinic of Soroka Medical Center between August 2011 and August 2015. Associated findings from clinical examination were recorded.
A total of 32 patients with IEPS were documented. There were 30 female and 2 male patients. Average age at diagnosis was 37.6 ± 13.4 years, range 19–63 years, median 35 years. Temporary alleviation of symptoms was reported in subjects treated with topical steroids (43.8%), tacrolimus ointment (15.6%), and matrix metalloproteinase inhibitors (3.1%). No improvement in symptoms was reported in 37.5% of patients. Symptoms were bilateral in 78.1% of the patients. Spontaneous resolution was achieved in only 6.3% (2/32).
IEPS accompanied by chronic epiphora has not been characterized to date. Our data show a clear predominance of females, most of them in their fertile years.
Kashkouli, Mohsen B.; Karimi, Nasser; Khademi, Behzad
Purpose of review After failed conservative management, most clinicians treat congenital nasolacrimal duct obstruction (CNLDO) in a conventional stepwise fashion: starting with probing, then repeat probing, next silicone intubation and/or balloon dilatation, and finally dacryocystorhinostomy. This approach is based on the patient’s age and previously failed procedures and recruits ‘one procedure for all’ CNLDO. A newly introduced approach is based on the type of obstruction and recruits ‘all procedures for one’ CNLDO. The aim of this review is to examine the best available evidence regarding CNLDO management.
Recent findings Recent articles support the concept that through intraoperative evaluation of obstruction in CNLDO, clinicians may predict probing failure and instantaneously employ more appropriate treatment modalities. This review addresses whether an age-based approach should be changed into a one-stage obstruction-based approach.
Summary An age-based approach treats CNLDO as a homogeneous disease and thus treats all patients with one predetermined procedure. A one-stage obstruction-based approach, however, considers CNLDO to be a heterogeneous disease, and therefore allows recruitment of all procedures simultaneously and selects the best intraoperatively. It may allow replacement of the conventional stepwise approach to CNLDO treatment provided that randomized trials verify its efficacy, safety, and cost-effectiveness.
Ilse Mombaerts, Elodie Witters
Background/aims Although a Jones tube is considered the mainstay for epiphora in patients with total blockage of the canalicular system, it has been discouraged in children for reasons of inadequate self-care and maintenance. The purpose of this study is to compare the long-term outcome of Jones tube surgery in paediatric versus adult patients.
Methods Retrospective, interventional case series of a single academic institution. The medical records of all children (≤16 years old) and adults (>16 years old) who underwent conjunctivorhinostomy with placement of a 130° angled extended Jones tube were reviewed. The outcome measures were patency and anatomical position of the tube, type and frequency of complications and subjective relief of epiphora.
Results The study included 10 children (11 eyes) (range, 5.1–16.0 years old) and 102 adults (127 eyes) (range, 19.7–82.4 years old). The success and complication rate did not differ between the two age groups. Tube dislodgement and obstruction occurred in 4 (36.4%) of the paediatric tubes with an incidence rate of 6.1%/year and in 47 (37.0%) of the adult tubes with an incidence rate of 9.3%/year (p=0.3867). Two adults required routine self-irrigation of the tube. The median follow-up was 6.7 years for the children and 8.7 years for the adults (p=0.3430).
Conclusion With a similar outcome profile and minimal self-care, young age is not a prognostic nor limiting factor for surgery with angled Jones tubes. Exchange with tubes of a longer length is not required during growth.