Search

Respiratory epithelial cysts are uncommon lesions that can be congenital or acquired cystic malformations. Beyond the respiratory system, the location of the respiratory epithelial cyst has been described in the central nervous system, orbits, and the maxillofacial region. The common etiology is believed to be sequestration or entrapment of the epithelial cells of the paranasal sinus. To the best of the authors' knowledge, there are no prior reports on respiratory epithelial cyst involving the lacrimal sac. The present case reports the computed tomography-dacryocystography and endoscopic features and surgical challenges in the management of one such exceptionally rare case., Competing Interests: The authors have no financial or conflicts of interest to disclose, (Copyright © 2024 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

Purpose: To explore the endoscopic and histopathologic characteristics of the lacrimal sac (LS) and the nasolacrimal duct in cases with punctal agenesis presenting with LS swellings., Methods: This is a prospective, interventional study of 13 LSs of 8 patients presenting with punctal agenesis and LS swellings over a 5-year study period (June 2018-July 2023). Complete ophthalmic examination was performed along with CT scans of the orbits. The LS was exposed with an anterior lacrimal crest incision and explored. The exterior and interior of the sac swelling were examined and LS flaps were sent for histopathological analysis., Results: The mean age of the patients was 23 years with a male:female ratio of 5:3. All patients presented with swelling in the LS region. Occasional epiphora was the presenting feature in 6 patients. All patients had both upper and lower punctal agenesis on the affected side with associated LS swelling. CT scans showed a hypodense cystic swelling with expansion of the LS fossa and bony nasolacrimal duct ending abruptly short of the inferior meatus. Intraoperatively, the LS had thin, translucent walls with mucoid secretion and an absence of common canalicular opening. Histopathological analysis showed epithelium consistent with LS with less robust development of the sac walls and poorly structured lacrimal drainage-associated lymphoid tissue with scanty stromal inflammation., Conclusions: The development of LS and the nasolacrimal duct is structurally affected in patients with punctal agenesis. Retrograde approaches for epiphora resolution are not logical in such cases but conjunctivodacryocystorhinostomy with Jones tube can be a feasible option., Competing Interests: The authors have no conflicts of interest to disclose., (Copyright © 2024 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

Purpose: To evaluate the outcomes of endoscopy-assisted modified Weber-Ferguson's approach in the management of primary lacrimal sac tumors with extension into the neighboring tissues., Methods: A retrospective interventional study was performed on all patients with lacrimal sac tumors treated with the endoscopy-assisted modified Weber-Ferguson approach between January 2010 and June 2022 at the Shanghai Ninth People's Hospital, China. Data assessed include demographics, clinical presentations, imaging features, surgical techniques, histopathology, adjuvant modalities of management, complications, and outcomes., Results: A total of 13 patients were included in the analysis. Epiphora and palpable mass lesion were the presenting complaint in 84.6% (11/13) of the patients. Nearly half of the patients (46.1%, 6/13) were misdiagnosed as lacrimal duct obstruction. All the lacrimal sac tumors in the present series showed uneven enhancement on T1-weighted MRI imaging. Postoperatively, 84.6% (11/13) patients recovered well with excellent esthetics and were disease-free after a mean follow-up of 58.6 months. Two patients who underwent additional exenteration developed recurrence and succumbed (at 41 and 96 months follow up) while they were on palliative chemoradiation., Conclusion: The endoscopic-assisted modified Weber-Fergusson surgical approach is effective in providing better visibility and accessibility to lacrimal sac tumors with extension into neighboring tissue.

Aim: To compare the demographics, clinical features, and changes in the management pattern of acute dacryocystitis at a tertiary care eye institute., Methods: A retrospective review was performed of electronic medical records of all patients diagnosed with acute dacryocystitis from January 2013 to January 2023. Data retrieved include demographics, history, presenting symptoms, duration of symptoms, surgical interventions, associated systemic conditions, management, complications, and outcomes. A successful anatomical outcome was defined as patency on lacrimal irrigation, and a successful functional outcome was defined as the resolution of infection and epiphora. The data parameters obtained were compared with the historical published data of the earlier two decades from the same Institute., Results: A total of 363 eyes of 349 patients were enrolled in this retrospective study. The median age was 45 years (range: 19-94 years). There were 216 (62%) females and 133 (38%) males. Surgery was performed in 320 (88%) patients. Needle aspiration or incision and drainage were performed in 102(32%) patients with lacrimal abscesses. Of the 320 patients, an endoscopic DCR was performed in 138 (43%) patients and an external DCR in 182 (57%). Of the 320 patients who underwent DCR surgery, 308 (96%) demonstrated anatomical and functional success at 1-year follow-up., Conclusion: There is a changing trend towards endoscopic DCR being incorporated as the primary procedure for managing acute dacryocystitis with the advantages of quicker resolution and reduced morbidity. There is a trend for choosing needle aspiration over the traditional incision and drainage in the initial management of lacrimal abscess., (© 2023. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

Purpose: To discuss the clinical spectrum and management strategies in patients with post-traumatic canalicular fistula (PTCF)., Methods: Retrospective, interventional case series of consecutive patients diagnosed with PTCF over a 6-year study period between June 2016 and June 2022. The demographics, mode of injury, location, and communication of the canalicular fistula were noted. The outcomes of several management modalities including dacryocystorhinostomy, lacrimal gland therapies, and conservative approaches were assessed., Results: Eleven cases with PTCF over the study period were included. The mean age at presentation was 23.5 years (range: 6-71 years), with male: female ratio of 8:3. The median time interval between trauma to presentation at the Dacryology clinic was 3 years (range: 1 week to 12 years). Seven had iatrogenic trauma and four had the canalicular fistula following primary trauma. Management modalities pursued include conservative approach for minimal symptoms, and dacryocystorhinostomy, dacryocystectomy, and lacrimal gland botulinum toxin injection. The mean follow-up period was 30 months (range: 3-months-6 years)., Conclusion: PTCF is a complex lacrimal condition and the management of the PTCF needs a tailored approach guided by its nature and location and patient symptomatology.

Purpose: To investigate the effect of botulinum toxin (BTX) and partial orbital lobe dacryoadenectomy (POLD) on the main lacrimal gland functions using the direct assessment of its secretory activity., Methods: A pilot study involving 14 subjects (mean age, 42.16 years) with proximal bicanalicular block ( n  = 9) or complete punctal effacement ( n  = 5) received either intraglandular 2.5 units of BTX ( n  = 7) or underwent the POLD procedure ( n  = 7). Dynamic assessment of tear secretion from the palpebral lobes was performed as per standard protocols. The parameters studied included Schirmer I, changes in the glandular outflow, and the neurosensory secretory lag., Results: Schirmer I values showed a mean reduction of 5.25 mm (range, 2-8 mm) following BTX injection, and mean tear flow decreased from 1.46 μ l / min to 1.11 μ l / min ( P  = 0.15). The POLD group showed a significant mean reduction of 0.45 μ l / min in mean tear flow at 1 month ( P  = 0.03) and 0.32 μ l / min at 3 months of follow-up ( P  = 0.20). However, there was no statistically significant change of the Schirmer values in the POLD group at 3 months., Conclusion: Direct assessment of lacrimal gland function can be used to monitor changes following the gland therapies. Although POLD shows a significant reduction in short term, significant surgical improvisation would be needed for it to be effective in long term. A larger study is ongoing to further our understanding.

Peripunctal squamous cell carcinomas with canalicular infiltration are a rare clinical entity. Although there are universal guidelines on achieving margin clearance for excision of an eyelid tumor, literature is scarce on achieving the same while dealing with the lacrimal drainage system. The present case describes the management of a rare case of squamous cell carcinoma with secondary extension into the proximal lacrimal system. The surgery was performed with intraoperative frozen section guidance.

Background: Dacryocystorhinostomy (DCR) is regarded as the gold standard for the treatment of chronic dacryocystitis (CDC). Different modifications of the surgical procedure have been developed over the years., Methods: Patients with CDC due to postsaccal lacrimal stenosis and under treatment with DCR have been included in this retrospective study. Two groups of different surgical procedures were analysed: firstly DCR without reconstruction of the ductus nasolacrimalis (DNL, group 1) and secondly patients with reopening the DNL (group 2). Criteria for success were absence of permanent epiphora, absence of inflammation of the lacrimal sac (functional success), and no recurrent surgery with free patency of the lacrimal duct (anatomical success). This was obtained by questionnaire after a follow-up of at least 12 months. The impact of gender, ectasia of the lacrimal sac, dacryoliths, and prior lacrimal surgeries was analysed., Results: Overall, 248 surgeries were enrolled in this trial. Mean age in group 1 was 68 years (range: 22 to 92 years) and gender ratio was 3.2 to 1 (female : male). In that group, 68 operations could be followed up. In group 2, 62 operations on patients of a mean age of 63 years (range: 24 to 89 years) and a gender ratio of 2.9 to 1 (female : male) were observed. Complete success occurred in 75.0% in group 1 and 75.8% in group 2. Recurrent operations were necessary in one case of group 2 (1.6%) and 4 cases of group 1 (5.9%). Gender (group 1 p = 1; group 2 p = 0.115; between groups p = 0.511), ectasia of the lacrimal sac (group 1 p = 0.877; group 2 p = 0.674; between groups p = 0.878), dacryolith (group 1 p = 1; group 2 p = 0.465; between groups p = 1), and prior lacrimal surgery (group 1 p = 0.092; group 2 p = 0.051; between groups p = 0.520) did not influence the success rates in each group or between groups. Significantly more dacryoliths were found during operations of group 2 (p = 0.010)., Conclusion: Reconstruction of the DNL during DCR is a possible and easy modification, with a slightly better success rate in curing CDC. Intraoperatively, dacryoliths might not be apparent remain in the deeper parts of the lacrimal ducts. Therefore, these segments should be inspected during surgery., Competing Interests: Die Autoren Wiebke Dettmer, Hans-Gert Struck, Andrea Schmidt-Pokrzywniak, Arne Viestenz und Jens Heichel geben an, dass keine Interessenkonflikte vorliegen. Friedrich Paulsen wurde von der Deutschen Forschungsgemeinschaft (DFG) gefördert (grant PA738/15-1). Er erhält Tantiemen von Elsevier für die 24. Edition des Anatomieatlanten „Sobotta“ und „Sobotta Textbook of Anatomy,“ in der 2. Auflage. Mohammad Javed Ali erhält Tantiemen vom Springer-Verlag für das Werk „Principles and Practice of Lacrimal Surgery“, „Atlas of Lacrimal Drainage Disorders“ und „Video Atlas of Lacrimal Surgery“./The authors Wiebke Dettmer, Hans-Gert Struck, Andrea Schmidt-Pokrzywniak, Arne Viestenz and Jens Heichel have no conficts of interest to be reported. Friedrich Paulsen has received grants from the Deutschen Forschungsgemeinschaft (DFG; PA738/15-1). He obtains royalty from Elsevier for 24th edition of Anatomy Atlas “Sobotta” and “Sobotta Textbook of Anatomy” second edition. Mohammad Javed Ali obtains royalty from Springer for “Principles and Practise of Lacrimal Surgery”, “Atlas of Lacrimal Drainage Disorders”, and “Video Atlas of Lacrimal Surgery”., (Thieme. All rights reserved.)

Epstein-Barr virus (EBV) associated dacryocystic retention presents with an acute swelling in the lacrimal sac area with epiphora and lymphadenopathy. The patient would usually have a preceding history of infectious mononucleosis with clinical features of fever, pharyngitis, preauricular and cervical lymphadenopathy, hepatosplenomegaly, subclinical hepatitis, nausea, and palatine petechiae. The present case is of a 3-year-old child who presented with a sudden painless swelling in the lacrimal sac region with epiphora. Investigations and a biopsy helped in establishing the diagnosis of EBV-associated dacryocystic retention which resolved completely with conservative management.

Competing Interests: The author has no financial or conflicts of interest to disclose.

Mucopeptide concretions, previously called dacryoliths, are macroscopic stones that commonly obstruct the lacrimal sac. The mechanism behind dacryolithiasis remains unclear; however, the involvement of various immune cells, including neutrophils, has been confirmed. These findings remain limited, and no information on neutrophil extracellular traps (NETs), essentially involved in the pathogenesis of other lithiases, is available yet. Here, we employ microcomputed tomography, magnetic resonance tomography, histochemistry, mass spectrometry, and enzyme activity analyses to investigate the role of neutrophils and NETs in dacryolithiasis. We classify mucopeptide concretions into three types, with respect to the quantity of cellular and acellular material, polysaccharides, and mucosubstances. We propose the role of neutrophils and NETs within the existing model of gradual formation and growth of mucopeptide concretions, with neutrophils contributing to the initial stages of dacryolithiasis, as they localized on the inner (older) parts of the tissue. As NETs localized on the outer (newer) parts of the tissue, we link their role to the late stages of dacryolithiasis, presumably maintaining the proinflammatory environment and preventing efficient clearance. An abundance of IgG on the surface indicates the involvement of the adaptive immune system later as well. These findings bring new perspectives on dacryolithiasis, in which the innate and adaptive immune system are essentially involved.

Purpose: To report an unusual case of an ectopic lacrimal fistula in a setting of a naso-orbito-ethmoid fracture., Case Report: The present case demonstrates a right-sided ectopic lacrimal fistula just below the glabella on the contralateral side on the outer edge of the midline. CT-Dacryocystography demonstrated a large dilated right lacrimal sac with a partly sequestered fundus traversing through the frontal process of the maxilla and frontal bone, close to the skull base and communicating with the fistula. The connection of the fistula with the sac was severed at the superior-most point, and the long fistulous tract was removed using external and endoscopic approaches, followed by endocautery and subsequent dacryocystorhinostomy. Post-operatively, the fistulous opening healed, ostium score was FICI grade 5, and the patient was asymptomatic., Conclusion: The case demonstrates multiple surgical challenges while managing an ectopic fistula with a long fistulous tract traversing several bony structures.

Purpose: To define the clinical and optical coherence tomography (OCT) morphology of the lacrimal punctum and assess the age-related changes across 8 decades of life in a normal population., Methods: A total of 1310 high-magnification slit-lamp, Fourier-domain OCT (FD-OCT), and optical coherence tomography angiography (OCTA) images were obtained from 320 puncta of normal asymptomatic individuals representing the Indian population. The punctum and peri-punctal area were defined with the help of 2 rims (inner punctal rim and outer punctal rim ) and 3 zones (white zone [Wz], separation zone [Sz], and peri-punctal vascularity). FD-OCT images were used to measure the external punctal diameters and internal lacrimal punctal diameters and the reflectivity patterns of the 3 punctal layers. OCTA was used to assess the branching and extent of vascular networks., Results: The upper puncta were narrower and more circular than the lower puncta across the decades. The elevation of the punctal papilla began in the upper puncta in the fifth decade, involved the lower puncta in the sixth decade, and gradually became exaggerated by the eighth decade. A typical punctal narrowing in previously wide puncta began to appear in the sixth decade of life, peaks in the seventh decade, and reverses gradually and spontaneously by the end of the eighth decade of life. The third and fourth decades saw a narrowing of the Sz with prominent vascularity crossing the Sz to reach the Wz. The Sz became indistinct in the fifth and sixth decades of life. The FD-OCT showed gradual thickening and dominance of the fibrous layer from the fifth to eighth decade of life. The clinical vascularity in the peri-punctal region increases from the third decade onwards, with vascular networks becoming increasingly dense, intricate, and branched as the age progresses., Conclusion: The present study defined and characterized the involutional changes in a normal population's first-eighth decades of life. Significant morphological changes were noticed across the different age groups with several clinical implications., Competing Interests: Dr. Mohammad Javed Ali receives royalties from Springer for the textbook “Principles and Practice of Lacrimal Surgery” and for the treatise “Atlas of Lacrimal Drainage Disorders.” Other authors have no conflicts of interest to disclose., (Copyright © 2023 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

Purpose: Several non-lacrimal lesions can present with lacrimal sac area swelling mimicking a dacryocystocele or mucocele with a possibility of misdiagnosis. This study investigates the clinic-radiologic characteristics of the mimicking conditions compared to true lacrimal sac distension., Design: Retrospective, comparative, interventional case series., Participants: Patients referred by primary care ophthalmologists between January 2015 and October 2021 with a misdiagnosis of dacryocystocele or lacrimal sac mucocele (n = 39) and an age-matched group of proven true lacrimal sac swelling (TLS group, n = 44)., Methods: Data collected included demographics, presenting features, investigations, management, histopathology, and outcomes. Both groups were statistically compared for several clinical and radiological variables., Results: Final diagnoses in the mimicking group were skin/subcutaneous swellings (14/39, 35.9%), vascular malformations (10/39, 25.6%), inferomedial anterior orbital cysts (7/39, 17.9%), sino-orbital masses (5/39, 12.8%). Female gender (<0.001), epiphora (p = 0.001), and discharge (p < 0.001) were significantly more frequent in the TLS group. The mimicking group was more likely to be associated with a longer swelling duration (p < 0.001), a swelling extending beyond the lacrimal sac area (p <0.001), orbital signs (p <0.001), and periorbital abnormality. Non-patent lacrimal irrigation (p < 0.001) was significantly more frequent in the TLS group. On imaging, all swellings in the mimicking group were separate from the lacrimal pathway and 94.9% (37/39) extended beyond the lacrimal sac fossa., Conclusion: Various cutaneous, subcutaneous, vascular, inferomedial orbital cystic, and sinonasal pathologies can present with lacrimal sac area swelling and mimic a TLS. A high index of suspicion, a thorough clinical evaluation and proper imaging are essential to avoid a misdiagnosis.

Iatrogenic lacrimal drainage trauma during surgery is not common. It has been reported during orbital surgeries, medial maxillectomies, post-trauma repairs, excision of adherent lesions in the vicinity, and facial reconstructive procedures. The present case reports a focal loss of the lacrimal sac wall during the excision of an intricate dermoid cyst. The defect was sealed by a nasal mucosal graft harvested from the floor of the ipsilateral nasal cavity. Postoperatively, the epiphora was resolved, and a CT-dacryocystography confirmed the anatomical integrity of the reconstructed lacrimal sac., (Copyright © 2022 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

Punctoplasty is a common procedure in the management of punctal stenosis and was first described by Sir William Bowman in 1853. Several types of punctoplasty are described, and include 1-snip, 2-snip, triangular 3-snip, rectangular 3-snip and 4-snip procedures. The understanding of the anatomy and physiology of the punctum has greatly improved in the last decade aided by electron microscopic and molecular techniques. The use of minimally invasive modalities for primary cases is on the rise with successful preservation of the physiological functions of the punctum and canaliculus. The paper reviews the current anatomical and physiological evidence to present an alternative perspective. Given our evolving understanding, it may be time to debate the practice of performing routine punctoplasty.

Competing Interests: None

Purpose: To provide a major review of the literature on diagnostic and therapeutic implications, techniques, and utility of anterior segment optical coherence tomography (OCT) for the proximal lacrimal drainage system (PLDS)., Methods: The authors performed a PubMed search of articles published in the English language on anterior segment OCT for the proximal lacrimal drainage system. Data captured include evolution, techniques, diagnostic utility, therapeutic monitoring, outcomes, and limitations. Specific emphasis was laid on addressing the existing lacunae and the current practice patterns., Results: The PLDS parameters that can be studied by OCT include punctal shape and morphology, external and internal punctum diameters, punctum depth and area, intra-punctal tear meniscus, punctal reserve, punctum-canalicular junction, canalicular diameter at various depths, canalicular depth, canalicular cross-sectional area, canalicular volume, and fluid meniscus characteristics. Normative data is now available from across the globe. Several punctal and canalicular disorders show characteristic OCT features and have adjunctive value in diagnosis. Post-operative OCT imaging can help in monitoring the outcomes of selected surgical procedures and medical therapy. OCT studies have raised doubts about the previous beliefs of vertical canalicular height and the definition of punctal stenosis. OCT dacryography (OCTD) and 3-dimensional punctal and canalicular imaging are promising modalities to further explore the anatomy, physiology, and pathophysiology of the PLDS., Conclusion: Optical coherence tomography techniques are increasingly playing a significant role in diagnosing and managing proximal lacrimal drainage disorders. Further improvements in imaging techniques, better resolution, standardized definitions, and measurements of punctal and canalicular parameters will expand its clinical usage and give impetus to minimally invasive lacrimal surgeries., (© 2021. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

Purpose : To perform a literature review on the primary malignant epithelial tumors (PMET) involving the lacrimal drainage system (LDS). Methods : The authors performed a PubMed search of all articles published in English on primary malignant epithelial tumors of the lacrimal sac and the nasolacrimal duct. Data analyzed include prevalence, demographics, clinical presentation, histopathological subtypes, management modalities, outcomes, and recurrence rates. Results : Malignant epithelial tumors of the lacrimal sac are commonly diagnosed in the fifth to seventh decades of life (mean age: 58 years) and have no gender predilection. Typical clinical presentation includes epiphora (84%) and lacrimal fossa mass lesion (78%). Pain (9%) and blood-stained epiphora (4%) were inconsistent findings. Among the PMET, squamous cell carcinomas (SCCs) were the most common (61%) followed by transitional cell carcinomas (TCC; 15%), and mucoepidermoid carcinoma (MEC; 7%). Complete surgical excision followed by radiotherapy (59%) was the preferred modality of management, and only 18% required orbital exenteration. Of the 331 tumors with available treatment details, 85% survived at a mean follow-up of 33 months. The overall recurrence rate following treatment was 20% (65/331). MECs of the lacrimal sac had the best while adenoid cystic carcinoma had the worst prognosis. Conclusion : Blood-stained epiphora was not the classical presentation for primary epithelial malignancies of the LDS. No validated staging system exists for lacrimal drainage tumors. There is a need for consistent and detailed reporting in the literature, more so on the histological subtypes and their management.

Bilateral Tessier cleft types 3 and 4 are rare and commonly involve the lacrimal drainage system owing to their anatomical location. Such clefts commonly present with associated ocular anomalies and include colobomatous eyelids, hypertelorism, microphthalmia, punctal or canalicular agenesis, and nasolacrimal duct obstruction or exstrophy. The current report presents an 18-month-old baby with bilateral Tessier cleft 3 with a unilateral anophthalmos, symmetrical eyelid colobomas, and lacrimal drainage anomalies. The lacrimal anomalies noted include small lacrimal sac with inferior canaliculus on the right side and upper and lower punctal and canalicular agenesis on the left side. Computed tomographic dacryocystography demonstrated unilateral lacrimal sac and bilateral maldevelopment of the bony nasolacrimal duct.

Purpose : To explore the utility of a novel real-time dynamic magnetic resonance dacryocystography (MRDCG) technique to assess the transit times through the lacrimal drainage system (LDS). Methods : Twenty-six patent LDS of 13 normal and asymptomatic individuals underwent MRDCG using a dynamic recalled three-dimensional spoiled gradient sequence (3DSPGR) on 1.5-Tesla magnetic resonance (1.5T MR) scanner while pre-determined diluted gadolinium drops were instilled into the eyes simultaneously. MR images of 0.8 mm thickness were captured every 9.4 s for 10 min. Evaluated parameters include the time taken for contrast to first appear in lacrimal sac, nasolacrimal duct (NLD), and inferior meatus (IM). Results : Dynamic MRDCG demonstrated contrast outflow in all cases with good structural delineation. The mean transit time for contrast to appear in lacrimal sac was 15.2 s (9.4-66 s; SD, 11.9), 50.4 s for nasolacrimal duct (18.9-151; SD, 32), and 150.5 s for inferior meatus (37.8-490; SD, 135). Contrast appeared in the IM in less than 2 min in 62% (16/26) systems, and only 19% (5/26) required more than 5 min. The maximum time taken for the contrast to reach IM was 6.2 min and all the LDS eventually showed the contrast into the nasal cavity. No statistically significant differences were noted in the transit times between the right and left sides. Conclusion : Dynamic MR-DCG using real-time 3DSPGR sequence reliably demonstrates the structural and physiological assessment of LDS and could be further explored to study the physiology and pathologies of the LDS.

Background: To review and update the syndromic and non-syndromic systemic associations of congenital lacrimal drainage anomalies., Methods: The authors performed a PubMed search of all articles published in English on congenital lacrimal drainage anomalies (CLDA). The current review provides an update from January 2017 to August 2020 on all CLDA associated with clinical syndromes or non-syndromic systemic disorders. The update intends to appraise the readers on all papers that were published in the interim 3-year period since the publication of the previous major review by authors (1933-2016). Patients with specific syndromes or systemic disorders were then reviewed. Pertinent cross-references from each of the short-listed articles were also included. Data reviewed include syndromic descriptions, systemic details, demographics, lacrimal presentations, management, and outcomes., Results: There have been significant new updates. Eleven new syndromes have been added to the list of syndromic associations, of which three were suspects. Among the new syndromic associations, three (PHACE, Williams-Beuren, and Peter's plus syndromes) described CLDA details in depth. Several new non-syndromic systemic conditions with associated CLDA also came to light, the foremost amongst them being proboscis lateralis, diprosopus dirrhinus, cerebral palsy, and NGLY-1 related disorders. Although familial presentations have been reported, the inheritance patterns are unclear for most anomalies. Surgical challenges in these patients are distinct, and a thorough pre-operative assessment, including detailed imaging when indicated, may facilitate good outcomes. Two updated tables reflecting the summary of syndromic and non-syndromic systemic associations are provided to capture the details at a glance., Conclusion: It is not very uncommon to find CLDA in syndromic or non-syndromic systemic disorders. Diagnosis of a craniofacial syndrome should prompt the physician to look out for CLDA. Similarly, a diagnosis of multiple CLDA should alert the examiner for the possible presence of associated systemic anomalies., (Copyright © 2020 Elsevier GmbH. All rights reserved.)

Canaliculops is a non-inflammatory dilatation of the canaliculus which can be secondary to trauma, punctal agenesis, herpetic infections or chronic drug use. This condition can affect either canaliculi and has shown female preponderance. Treatment described so far has consisted of cyst marsupialization with no literature on the post-operative epiphora in these cases. We describe two cases of canaliculops with punctal agenesis where we have tried to establish lacrimal patency.

Primary adenocarcinoma of the lacrimal drainage system is a rare malignancy. Poorly differentiated subtypes of this tumor demonstrate aggressive behaviors, and metastasis is not uncommon. We present a patient with poorly differentiated adenocarcinoma of the lacrimal sac and nasolacrimal duct with an orbital extension. The case was managed by a combined external and endoscopic approach excision followed by adjuvant radiotherapy.

Purpose: To provide a literature review on lymphoproliferative lesions involving the lacrimal drainage system., Methods: The authors performed a pubmed search of all articles published in English on lymphoma/leukemia of lacrimal sac and the nasolacrimal duct. Data analyzed include prevalence, demographics, clinical presentations, treatment outcomes of primary versus secondary lacrimal involvement, and recurrence rates., Results: Lymphoma/leukemia of lacrimal sac presented at a mean age of 55 years. The majority of the tumors (63%) were primary involvement of lacrimal sac, bilateral involvement being more common in secondary than primary lacrimal sac lymphoma. Epiphora (96%, 98/102), swelling in the lacrimal sac region (75%, 77/102), and acute dacryocystitis (31%) were the most frequent presenting features. Thirty-six percent of the lesions were diagnosed incidentally while performing a dacryocystorhinostomy (DCR). Among primary sac lymphomas, diffuse large B-cell lymphoma (DLBCL) (43%) was the most common sub-type followed by MALToma (24%), unclassified B-cell lymphoma (21%), lymphoid hyperplasia (5%) and 3% each small lymphocytic lymphoma (SLL) and NK/T cell lymphoma. Management usually involves chemotherapy and/ or radiotherapy with or without surgical resection. Successful outcomes in terms of local disease control could be achieved in all the cases; however, 15% died of the systemic disease after a mean 18 months from the time of sac involvement. Aggressive lymphomas like NK/T-cell have the worst prognosis., Conclusion: Lymphoproliferative involvement of lacrimal sac has a high incidence of acute dacryocystitis with a good response to chemotherapy. Epiphora in patients with leukemia/lymphoma should arouse suspicion of a lacrimal drainage involvement.

Purpose: To report techniques and outcomes of a combined external and endoscopic surgical approach in the management of solitary fibrous tumors of the lacrimal drainage system with variable orbital and sinonasal extensions., Methods: Biopsy proven cases of solitary fibrous tumors of the lacrimal drainage system involving the lacrimal sac and the nasolacrimal ducts with an orbital, sinonasal, or intranasal extension were studied. Parameters assessed include patient demographics, clinical presentation and course, operative techniques, complications, postoperative adjuvant therapies and outcomes., Results: The technique is described in 3 patients. Two patients had massive orbital extension and one had gross sinonasal extension. Nasolacrimal duct extension was involved in all the patients with intranasal extension in 2 patients. External approach was used to access the lacrimal sac and the orbital components while the endoscopic approach was used to assess the orbital clearance and manage the sinonasal extension and the nasolacrimal duct excision up to the inferior meatus. Following complete excision, additional biopsies were performed from the adjacent bones and sinonasal tissues. A detailed histopathology with immunohistochemistry is essential for not only the diagnosis but also to assess the margin clearance. There were no major postoperative complications. All the patients were advised for postoperative adjuvant external beam radiotherapy. At the last follow-up, none of the patients had any clinical or endoscopic evidence of a recurrence., Conclusions: A combined external and endoscopic approach en-block excision is most suitable management modality for solitary fibrous tumors involving the lacrimal sac and the nasolacrimal ducts with orbital and sinonasal extension.

The aim of this review was to propose multi-pronged resumption strategies for lacrimal practice in an effort to plan a sustainable recommencement of elective surgeries after we emerge from the peak of COVID-19 pandemic. The strategies for lacrimal practice were classified into 7 subtypes, and each of the blueprints were reassessed based on existing information on resumption strategies of elective surgeries from other specialties in COVID-19 era. The specific needs of lacrimal practice were then added to construct algorithms summarizing the resumption strategies. The basic principle of 'primum non nocere' needs to be followed. The overall proposed plan advocates the transition to a more sustainable health care reality in a world where we would still co-exist with COVID-19. A comprehensive effort involving screening, laboratory testing, appropriate triage, effective personal protection and specific precautionary measures for lacrimal clinics and operating room are needed to be able to safely resume elective surgery when the pandemic peak declines. To predict the timing of the resumption of elective surgeries is quite complex and influenced by several geographic, political and economic factors. It is equally important to remember that COVID-19 crisis is a dynamic situation and constantly evolving, hence the strategies provided are subject to change. Strict adherence to standard COVID-19 guidelines combined with effective testing and personal protection strategies can ensure slow yet smooth and safe return to full lacrimal practice after the COVID-19 pandemic calms down. The local government directives, individual and institutional discretion are advised., Competing Interests: None

Oculoplastic surgeries encompass both emergency surgeries for traumatic conditions and infectious disorders as well as elective aesthetic procedures. The COVID-19 pandemic has brought about a drastic change in this practice. Given the highly infectious nature of the disease as well as the global scarcity of medical resources; it is only prudent to treat only emergent conditions during the pandemic as we incorporate evidence-based screening and protective measures into our practices. This manuscript is a compilation of evidence-based guidelines for surgical procedures that oculoplastic surgeons can employ during the COVID-19 pandemic. These guidelines also serve as the basic framework upon which further recommendations may be based on in the future, as elective surgeries start being performed on a regular basis., Competing Interests: None

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.

Purpose: To provide a systematic review of the literature on diagnostic and therapeutic indications, techniques, utility, and complications of dacryoendoscopy (DEN)., Methods: The authors performed a PubMed search of articles published in English/German/Japanese/Chinese on DEN. Data captured include evolution and techniques, diagnostic utility, therapeutic interventions, outcomes, and limitations. Specific emphasis was laid on addressing the current practice patterns and existing lacunae., Results: Diagnostic DEN has enabled the exact localization and differentiation of mucosal edema, stenosis, strictures, and obstructions throughout the lacrimal drainage system (LDS). Guided expansion of the stenosis, laser dacryoplasty (LDP) for strictures and microdrill removal of dacryoliths (microdrill dacryoplasty [MDP]) are some of the therapeutic uses of DEN. Successful outcomes of up to 60%-70% were achieved with stenosis anywhere along the LDS with a restenosis rate of 20%. Longer segment of stenosis (>2 mm), presence of enlarged lacrimal sac, history of dacryocystitis, postsaccal stenosis, and complete obstruction were associated with poorer outcomes. Microendoscopic procedures like LDP or MDP achieve canalicular and NLD recanalization with optimal results. DEN guided-probing demonstrated high success rates in cases with an earlier failed probing. Available dacryoendoscopes allow high-quality image capture (15,000 pixels) with multiple ports for additional instrumentation and irrigation., Conclusions: Dacryoendoscopic examination of canaliculus and nasolacrimal duct plays a significant role in the diagnosis and management of lacrimal disorders. Further improvements in microendoscopic techniques and instrumentation will expand its clinical usage and give impetus to minimally invasive lacrimal surgeries.Dacryoendoscopy examinations are increasingly playing a significant role in the diagnosis and therapies of lacrimal drainage disorders.

Purpose: To provide a systematic review of the literature on the disorders and management of the lacrimal drainage system (LDS) in few of the species of the animal kingdom., Methods: The authors performed a PubMed search of all articles that were published in English with specific reference to lacrimal drainage disorders in animals. Data captured include demographics, presentations, investigation, diagnoses and management modalities. Emphasis was also on anatomical differences, evolutionary perspectives and addressing the lacunae and potential directions for future research., Results: The lacrimal drainage system is a terrestrial adaptation in vertebrates. Evolutionary development of the LDS is closely linked to the Harderian gland and the vomeronasal organ. Variable differences in the clinical presentations and management of lacrimal drainage disorders (LDD) are noted in comparison to humans. These are secondary to unique structural and pathophysiological differences. Uniformity in usage and reporting of disease terminologies is required. Diagnostic challenges in clinical examination can be met with the development of customized lacrimal instruments. Contrast dacryocystorhinography is a very useful investigation in the diagnosis of LDD. Multiple bypass procedures like conjunctivorhinostomy, conjunctivobuccostomy and conjunctivomaxillosinosotomy have been described for nasolacrimal duct obstruction (NLDO). Advances of endoscopy and radiological techniques are paralleled by minimally invasive lacrimal interventions. The search for an ideal animal model for human LDS is far from over and the choice of the animal needs to be customized based on the research objectives., Conclusion: The lacrimal drainage system is an ancestral feature in tetrapod vertebrates and it is present, in some form or another, in most descendant species. The lacrimal drainage system has evolved considerably, adapting to the needs of the species. It is essential to understand the lacrimal drainage disorders of domestic animals so that the animal and human lacrimal sciences contribute more meaningfully to each other., (Copyright © 2019 Elsevier GmbH. All rights reserved.)

Purpose: To provide a systematic review of the literature on congenital dacryocystoceles (CDCs) and summarize their presentations, investigations, management, and outcomes., Methods: The authors performed a PubMed search of all articles published in English on CDCs. Data captured include demographics, clinical presentations, investigations, management modalities, complications, and outcomes. Fourteen major series (10 or more than 10 cases) and 89 isolated case reports/series on CDCs with a collective patient pool of 1,063 were studied in detail. Specific emphasis was laid on addressing the controversial issues including initial conservative versus surgical management and the role of endoscopic evaluation., Results: Numerous terminologies have been used to describe CDC. Congenital dacryocystoceles are rare variants of congenital nasolacrimal duct obstructions and comprise of 0.1% to 0.3% of all such cases. There is a female predilection (64.2%, 683/1,063) and the mean age at presentation is at 7 days of birth. Initial conservative treatment can be a viable option in the absence of an acute dacryocystitis or a respiratory distress. Endoscopy-assisted probing appears to have better outcomes as compared with the in-office probing. Congenital dacryocystoceles with acute dacryocystitis are preferably managed with intravenous antibiotics and an early probing under endoscopy guidance to avoid missing intranasal cysts. Marsupialization is the preferred technique in the management of intranasal cysts. Silicone intubation was rarely used and has no definitive indications. Dacryocystorhinostomy is very rarely needed in the management of CDC., Conclusions: Congenital dacryocystocele is a commonly accepted term and its use should be advocated to enhance uniformity in reporting. Endoscopic evaluation of CDC is useful in the diagnosis and treatment of associated intranasal cysts and enhances the rates of successful outcomes.

Canaliculops is a noninflammatory ectasia of the canaliculus and so far only 7 histopathologically confirmed cases have been reported worldwide. The authors present a middle aged female with right-sided bicanalicular cystic swellings associated with punctal agenesis. Ultrasound biomicroscopy showed a cystic dilatation of the canaliculus and dacryoendoscopy showed a cavity lined by whitish canalicular mucosa, thrown into numerous irregular folds with a blind medial end. Histopathological examination confirmed the diagnosis of canaliculops. To the best of authors' knowledge, this is the first description of a bicanalicular canaliculops and its dacryoendoscopic findings.

Canaliculops is a noninflammatory and noninfectious ectasia of the canaliculus with serous fluid accumulation. Currently, the etiology is uncertain. To the best of the authors' knowledge only 6 confirmed cases have been published earlier; however, the imaging features were not described. The authors report the ultrasound biomicroscopic and ocular coherence tomography features of a histopathologically proven canaliculops.