Milind N. Naik

Orbital Cysticercosis: Clinical Manifestations, Diagnosis, Management, and Outcome

PURPOSE To describe the clinical manifestations, diagnosis, management, and outcome of orbital cysticercosis in a tertiary eye care center in Southern India. DESIGN Retrospective observational case series. PARTICIPANTS A total of 171 patients with orbital cysticercosis. METHODS Retrospective case series involving consecutive patients with orbital cysticercosis from March 1990 to December 2001. MAIN OUTCOME MEASURES Clinical resolution and significant residual deficit. RESULTS The median age at presentation was 13 years (range 2-65 years), and 93 patients (54.4%) were male. The 3 main symptoms at presentation were periocular swelling (38%), proptosis (24%), and ptosis (14%) with a median duration of 2 (range 0-24) months. The 3 main signs at presentation included ocular motility restriction (64.3%), proptosis (44.4%), and diplopia (36.8%). The cyst locations in the decreasing order of frequency were anterior orbit (69%), subconjunctival space (24.6%), posterior orbit (5.8%), and the eyelid (0.6%). In all, 80.7% of patients had cysts in relation to an extraocular muscle. The superior rectus (33.3%) was the most commonly involved extraocular muscle. Contact B-scan ultrasonography was diagnostic of cysticercosis in 84.4% of patients. Orbital cysticercosis was managed medically in 158 of 166 patients. Although 149 patients received a combination of oral albendazole and prednisolone, 1 patient received oral albendazole alone, 7 patients received oral prednisolone alone, and 1 patient received oral praziquantel. Surgery was performed in 8 patients. Clinical resolution was seen in 128 of 138 patients (92.8%) at 1 month and 81 of 85 patients (95.3%) at 3 months. A significant residual deficit was present in 29 of 138 patients (21.0%) at the final follow-up and included proptosis in 7 patients, ptosis in 6 patients, ocular motility restriction in 3 patients, diplopia in 2 patients, strabismus in 2 patients, and a combination of the above in 9 patients. CONCLUSIONS Orbital cysticercosis is a common clinical condition in the developing world. It typically affects young individuals and has a wide spectrum of clinical manifestations. Both B-scan ultrasonography and computed tomography scan are useful in confirming the diagnosis. Despite resolution of cysticercosis with medical management, a significant proportion of patients may have residual functional deficits.

Comparison of vascularization of Medpor and Medpor-Plus orbital implants: a prospective, randomized study.

Purpose: To compare vascularization of porous polyethylene (Medpor) and porous polyethylene with synthetic bone graft particulate (Medpor-Plus) orbital implants following enucleation. Methods: Prospective, randomized study involving 10 patients. A standard enucleation procedure was performed, and each patient was randomized to receive either Medpor or Medpor-Plus orbital implant with anterior scleral cap technique. Gadolinium-enhanced, 3-Tesla MRI was performed at 1.5 months, 3 months, and 4.5 months following surgery. Implant vascularization was calculated in axial, coronal, and sagittal planes with manual planimetric method using postcontrast T1-weighted Digital Imaging and Communications in Medicine images. Results: The mean area of implant vascularization at 1.5 months, 3 months, and 4.5 months for Medpor implants was 58%, 70%, and 75%, respectively, and for Medpor-Plus implants was 69%, 76%, and 85%, respectively. The mean vascularization of Medpor-Plus implants was more than Medpor implants at 1.5 months (p = 0.008), 3 months (p = 0.09), and 4.5 months (p = 0.003). The difference between the 2 groups assessed by repeated measures analysis of variance was statistically significant (p < 0.0001). During the mean follow-up of 36.7 months (range, 18–43 months), 1 patient in the Medpor group had implant exposure that responded to scleral patch graft. Conclusions: Implant vascularization is faster with Medpor-Plus implants compared with Medpor implants when assessed by a planimetric method using 3 Tesla MRI. The addition of synthetic bone graft particulate (Novabone) to porous polyethylene may enhance implant vascularization.

Rosai Dorfman Disease of the Orbit

ObjectiveTo report the clinico-histopathologic features, management and outcome of Rosai-Dorfman disease of the orbit.DesignNon-comparative case series.ResultsRosai-Dorfman disease of the orbit constituted 0.09% of all ocular specimens received at our Institute, presenting with a firm rubbery mass causing proptosis; bilateral in 4 (57%) cases. The median age at presentation was 13 years (range 5–65); median duration of symptoms was 6 (range 3–15) years. Lymphadenopathy was noted in 4 (57%); extranodal involvement in 3 (43%). After biopsy, 3 cases were treated with systemic corticosteroids, 2 cases developed local recurrence that responded to systemic corticosteroid therapy. Polymorphous population of lymphocytes, plasma cells, and characteristic S-100-positive histiocytes showing emperipolesis were pathognomonic histologic features.ConclusionRosai-Dorfman disease of the orbit, although rare, should be considered in young individuals with chronic proptosis with rubbery masses. Excision and corticosteroid therapy provide a favorable outcome.

Establishing Human Lacrimal Gland Cultures with Secretory Function

Purpose Dry eye syndrome is a multifactorial chronic disabling disease mainly caused by the functional disruptions in the lacrimal gland. The treatment involves palliation like ocular surface lubrication and rehydration. Cell therapy involving replacement of the gland is a promising alternative for providing long-term relief to patients. This study aimed to establish functionally competent lacrimal gland cultures in–vitro and explore the presence of stem cells in the native gland and the established in-vitro cultures. Methods Fresh human lacrimal gland from patients undergoing exenteration was harvested for cultures after IRB approval. The freshly isolated cells were evaluated by flow cytometry for expression of stem cell markers ABCG2, high ALDH1 levels and c-kit. Cultures were established on Matrigel, collagen and HAM and the cultured cells evaluated for the presence of stem cell markers and differentiating markers of epithelial (E-cadherin, EpCAM), mesenchymal (Vimentin, CD90) and myofibroblastic (α-SMA, S-100) origin by flow cytometry and immunocytochemistry. The conditioned media was tested for secretory proteins (scIgA, lactoferrin, lysozyme) post carbachol (100 µM) stimulation by ELISA. Results Native human lacrimal gland expressed ABCG2 (mean±SEM: 3.1±0.61%), high ALDH1 (3.8±1.26%) and c-kit (6.7±2.0%). Lacrimal gland cultures formed a monolayer, in order of preference on Matrigel, collagen and HAM within 15–20 days, containing a heterogeneous population of stem-like and differentiated cells. The epithelial cells formed ‘spherules’ with duct like connections, suggestive of ductal origin. The levels of scIgA (47.43 to 61.56 ng/ml), lysozyme (24.36 to 144.74 ng/ml) and lactoferrin (32.45 to 40.31 ng/ml) in the conditioned media were significantly higher than the negative controls (p<0.05 for all comparisons). Conclusion The study reports the novel finding of establishing functionally competent human lacrimal gland cultures in-vitro. It also provides preliminary data on the presence of stem cells and duct-like cells in the fresh and in-vitro cultured human lacrimal gland. These significant findings could pave way for cell therapy in future.

Mitomycin C in dacryocystorhinostomy: the search for the right concentration and duration--a fundamental study on human nasal mucosa fibroblasts.

Purpose: To establish primary cultures of human nasal mucosal fibroblasts (HNMFs) and to test the effect of varying concentrations of mitomycin C (MMC) and treatment durations on cellular proliferation and viability of the fibroblasts. Design: Laboratory investigation. Methods: Nasal mucosa harvested from patients undergoing a dacryocystorhinostomy was used to establish primary cultures by explant culture method. Cells were expanded and frozen at every passage, and passage 3 cells were used for further experiments. The cells were then treated with different concentrations of mitomycin C (0.1–0.5 mg/ml) for different time periods (3, 5, and 10 minutes). Cell viability was checked by MTT (3-[4,5-dimethylthiazol-2-yl]-2,5-diphenyltetrazolium bromide) assay. Cellular proliferation index was determined with bromodeoxyuridine immunostaining. Apoptotic index was measured using annexin A5 affinity assay, propidium iodide staining, and 4′,6-diamidino-2-phenylindole counterstaining. The actin cytoskeletons of fibroblasts were studied using phalloidin staining. Results: The doubling time of cultured HNMFs is approximately 24 hours. Similarly, 0.4 mg/ml beyond 5 minutes and 0.5 mg/ml concentration at all time points were lethal and caused extensive cell death when compared with controls. A concentration of 0.2 mg/ml for 3 minutes of exposure prevented cell proliferation of HNMF cells by inducing cell cycle arrest, without causing extensive apoptosis. Conclusions: The minimum effective concentration appears to be 0.2 mg/ml for 3 minutes. This in vitro study could be the starting point for further clinical and histopathologic studies to validate its clinical usefulness.

Primary canaliculitis: clinical features, microbiological profile, and management outcome.

Purpose: To describe the demographic profile, clinical presentation, microbiological profile, and management outcome of primary canaliculitis. Methods: Single-center, retrospective, interventional case series. Clinical records of all patients diagnosed with primary canaliculitis and treated at the Department of Ophthalmic Plastic Surgery, LV Prasad Eye Institute, Hyderabad, India, between 1987 and 2010 were reviewed. Retrospective data analysis included demographic profile, clinical presentation, microbiological profile, and management outcomes. The management outcome was further analyzed regarding conservative medical treatment alone, versus punctoplasty with canalicular curettage. Results: Of the 74 patients, 40 (54%) were women. Mean age at presentation was 48 years. Right eye was involved in 38 (51%) patients, left eye in 34 (46%) patients, and both eyes in 2 (3%) patients. The mean delay in diagnosis was 10 months. Lower canaliculus was involved in 48 (65%) patients, upper canaliculus in 17 (23%) patients, and both canaliculi in 9 (12%) patients. The most common presenting symptom was epiphora, noted in 63 (85%) patients, and the most common clinical sign was thickening of canalicular portion of eyelid seen in 53 (72%) patients. Microbiological workup was available in 54 patients, of whom 49 (91%) yielded positive results. The most common isolate was staphylococcus species in 19 (39%) patients. Conservative medical therapy (punctal dilatation, canalicular expression, and topical antibiotics) resulted in resolution in 35 of 51 (69%) patients, whereas punctoplasty with canalicular curettage resulted in resolution in all 39 (100%) patients. Of the 74 patients, 57 (70%) resolved completely with single intervention, 14 (19%) with 2 interventions, 6 (8%) with 3 interventions, and 2 (3%) with 4 interventions. Recurrence was noted in 2 (3%) patients that subsequently resolved with treatment. Conclusion: Primary canaliculitis is predominantly a unilateral disease with a significant delay in diagnosis. The microbiological profile of canaliculitis is evolving, with staphylococcus species emerging as the most common pathogen. Although conservative medical therapy is beneficial, punctoplasty with canalicular curettage combined with topical antibiotic therapy is the gold standard treatment for canaliculitis.

Acquired nasolacrimal duct obstructions secondary to naso-orbito-ethmoidal fractures: patterns and outcomes.

Purpose: This study reports a series of 14 consecutive patients of nasolacrimal duct obstruction secondary to naso-orbito-ethmoid fractures (NOE), their presentations, surgical interventions, and outcomes. Methods: Fourteen consecutive patients presenting with secondary acquired nasolacrimal duct obstructions following naso-orbito-ethmoid fractures were included in a retrospective, noncomparative case series. Medical records were reviewed for demographic data, mode of trauma, types of NOE fractures, type of fracture repair, symptoms at presentation, duration of symptoms, areas of lacrimal obstructions, surgical interventions, lengths of follow up, and outcomes. Success was defined as both anatomical patency of the lacrimal passages and resolution of symptoms. Results: The mean age at presentation was 26.4 years (range: 4–46). The most common mode of trauma was a road traffic accident (71.4%), and the most common type of fracture was NOE type II (64.2%). The majority of the fractures (71.4%) were repaired by open reduction and internal fixation. All patients presented with epiphora, and 50% of them also presented with a swelling below the medial canthus. The mean duration of symptoms were 19.5 months (range: 1–48). All patients underwent an external DCR with mitomycin C and silicone intubation. All stents were removed at 12 weeks following surgery. The mean follow up after stent removal was 6 months (range 3–24 months) with a minimum follow up of 3 months. At the last follow up, the success rate was 92.8%. There was one failure, which was due to cicatricial closure of the ostium. Conclusion: External dacryocystorhinostomy with Mitomycin-C and intubation is effective in treatment of acquired nasolacrimal duct obstructions following NOE fractures. A delayed dacryocystorhinostomy did not appear to influence the outcomes.

Circumostial injection of mitomycin C (COS-MMC) in external and endoscopic dacryocystorhinostomy: efficacy, safety profile, and outcomes.

Purpose: To evaluate the safety and efficacy of a new technique of circumostial mitomycin C (COS-MMC) injection in dacryocystorhinostomy (DCR) and to evaluate the outcomes. Methods: A prospective, interventional consecutive case series involving 110 DCR surgeries performed over a period of 1 year using the COS-MMC technique were evaluated. All patients were operated by a single surgeon (M.J.A.). After fashioning of the mucosal flaps, all patients underwent an intramucosal injection of 0.02% MMC at 4 points (0.1 ml at each point) along the edges of the freshly created ostium. All patients were intubated, and stent removal was done at 12 weeks following the surgery. All patients underwent regular monitoring of the ostium to look for any complications. A minimum follow up of 6 months after stent removal was considered for final analysis. The outcome measures analyzed were anatomical and functional success, complications, and ostium. Results: The mean age at presentation was 39.91 years. The diagnosis was primary acquired nasolacrimal duct obstruction in 60.9% (67/110) and persistent congenital nasolacrimal duct obstruction in 9.1% (10/110) of the patients. Twenty-six patients were known cases of failed DCR and underwent a revision DCR. Of the total patients, 19.1% (21/110) had a history of acute dacryocystitis, of which, 73% (80/110) of the surgeries were performed by an external approach and 27% (30/110) by endoscopic endonasal route. Anatomical success was noted in 97.3% (107/110) and functional success in 96.4% (106/110). All the 4 failures were due to cicatricial closure of the ostium. None of the patients were noted to have mucosal burns in the area where MMC was injected, and none of the Ostia showed any features of necrosis, infection, or any other complications. Conclusions: COS-MMC is a safe and effective adjunctive modality in DCR. No adverse effects on the ostium were noted. The technique appears to be promising for the success of cases with high risk of failures like revision DCR and traumatic secondary acquired nasolacrimal duct obstruction.

Clinical Profile and Management Outcome of Acute Dacryocystitis: Two Decades of Experience in a Tertiary Eye Care Center

Abstract Aim: To report the demographic profile, clinical presentation, and management outcome of acute dacryocystitis. Methods: Retrospective study of 347 eyes of 320 patients, who presented to the Ophthalmic Plastic Clinic of a tertiary eye care center over a period of 22 years from January 1990 to March 2012 with acute dacryocystitis, were reviewed for demographic profile, clinical presentation, and management outcome. The numbers of patients with lacrimal disorders during the same period were retrieved to assess the incidence of acute dacryocystitis. Successful anatomical outcome was defined as patency on irrigation and a successful functional outcome was defined as resolution of infection and epiphora. Results: The mean age at presentation was 37 years. The female to male ratio was 2:1. There was no difference in the laterality between the right and the left eyes. Bilateral disease was noted in 8.4% (27/320) patients. 23% (80/347) eyes presented with lacrimal abscess while 2.8% (10/347) eyes had orbital cellulitis. Intensive medical care with hospital admission was needed in 4.4% (14/320) patients. The mean time to resolution of acute symptoms was 10 days. 5.6% (18/320) patients developed a fistula, among which 83% (15/18) were following a spontaneous rupture of the lacrimal abscess. Dacryocystorhinostomy was performed in 82.5% (264/320) patients with an anatomical success of 94.5% and a functional success of 93.5%. Conclusions: Acute dacryocystitis comprises 2.4% of all patients presenting with lacrimal system disorders. Fistula formation is a sequel more commonly seen with spontaneous rupture of a lacrimal abscess. The long-term outcomes in patients presenting with acute dacryocystitis are good with a surgical success rate of 94.3%.

Incomplete punctal canalisation: the external and internal punctal membranes. Outcomes of membranotomy and adjunctive procedures

Aims To study the clinical and diagnostic profile of punctal dysgenesis with membranes, to classify and correlate the membranes clinicopathologically and study the outcomes of membranotomy. Methods A prospective interventional study involving 55 dysgenetic puncta of 22 consecutive patients seen between July 2008 ansd December 2011. Data collected include demographics, clinical presentation, laterality, age at presentation, duration of symptoms, slit lamp examination, punctal profiles, types of membranes, associated lacrimal anomalies, management and outcomes. All the patients underwent membranotomy, after which the puncta were assessed for adequacy of the opening, the canaliculi were assessed with probes and irrigation was done to assess the patency of the lacrimal system. A minimal follow-up of 6 months was taken for final analysis. Primary outcome measures included were anatomical patency on irrigation and relief from epiphora assessed subjectively and also objectively by a dye disappearance test. Results The patients included 11 males and 11 females, with a mean age of 82.4 months (range 5–264 months) at presentation. Bilateral punctal involvement was seen in 59% (13/22) and all the four puncta were affected in 31.8% (7/22) of the patients. Epiphora was the most common complaint noted in 95% (21/22), and the symptoms were noticed since birth in 68.1% (15/22). External membranes over the puncta were noted in 86.4% (19/22) and internal punctal membranes in 13.6% (3/22) of patients. All the patients underwent membranotomy, however additional procedures like probing was done in 13.6% (3/22), mini-monoka intubation in 9.1% (2/22), Crawfords bicanalicular intubation in 9.1% (2/22) and dacryocystorhinostomy in 4.5% (1/22). Uniformly, the punctal membranes on histopathological examination were fibrovascular membranes. The anatomical patency was 100% and relief from symptoms was seen in 91% (20/22) of the patients. Conclusions This study presents the largest series to date (n=55 puncta) exclusively on punctal dysgenesis with membranes and, for the first time, has shown the clinicopathological correlation of these membranes. Incomplete punctal canalisation is probably a better term and this study could be the starting point for further exploration into the etiopathogenesis and genetics of this intriguing congenital disorder.