Dr. pragya saini
Dr.Nandini Bothra, Dr. MOHD. JAVED ALI
Abstract
Purpose:To study clinical profile,associated lacrimal disorders,long-term outcomes following membranotomy,in patients with IPC.Methods:Retrospective interventional study was performed of all the patients diagnosed with IPC from Jan 15-Dec 20.Diagnosis of IPC was made.Nettleship’s punctal dilators were used for effective membranotomy.Results:98 puncta of 62 eyes of 46 patients with IPC were examined. External membrane variant was seen in 62% and internal membrane variant in 38%.78 puncta underwent membranotomy Associated lacrimal drainage pathway deformities seen in 31%.3 puncta had minimonoka insertion for associated canalicular stenosis and canalicular obstruction.5 patients with associated cNLDO underwent probing, of which 3 patients needed DCR.Associated punctal agenesis was noted in 3 patients involving the other punctum of the same eye.Conclusions:Longterm outcomes of membranotomy for IPC are excellent.Associated congenital lacrimal drainage anomalies are common.
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INTRODUCTION
Incomplete punctal canalization (IPC) is a congenital lacrimal drainage disorder characterized by the presence of fibrovascular membranes over the puncta, which are believed to be due to failure of canalization of the most proximal lacrimal drainage pathway. This pathology was initially referred to using a plethora of terms ranging from punctal membranes to punctal atresia to punctal dysgenesis or, incorrectly, even agenesis. The term “‘incomplete punctal canalization” or IPC was introduced in 2013, with further classification into incomplete punctal canalization-external membrane variant and incomplete punctal canalization-internal membrane variety. The incomplete punctal canalization-internal membrane variety gives an impression of blurred punctal margins with complete obscuration of the opening by the membrane, which is present just within the margins. The pars lacrimalis portion of the eyelid is normal, unlike that of punctal agenesis.
The external membrane variant presents as a smooth, translucent membrane with or without a vessel traversing over the membrane’s surface, giving a false impression of punctal agenesis. Rarely, there could also be a balloon variant of the incomplete punctal canalization external membrane variant. A balloon variant of IPC is a form of external membrane where a dome-shaped elevation is seen in the punctal area, translucent, and relatively avascular. Anterior segment optical coherence tomography scan of the IPC demonstrates a hyperreflective membrane covering the punctal area in a table-top configuration with visible patent vertical canaliculus underneath. About 27% had associated lacrimal drainage pathway disorders like canalicular stenosis and congenital nasolacrimal duct obstruction in the earlier study. The management of IPC is a simple membranotomy using a Nettleship’s punctal dilator. Any other associated lacrimal drainage pathway disorder is dealt with as per standard protocols. The authors have earlier shown a 91% functional success rates with this simple procedure over a 6-month followup period. This study aims to assess the long-term outcomes of membranotomy and adjunctive measures in patients with IPC.
METHODS
A retrospective interventional study was performed of patients diagnosed with IPC during the period January 2015 to June 2020. Institutional ethics committee approval was obtained, and the study adhered to the tenets of the Declaration of Helsinki. Data collected on chart reviews include demographics, clinical presentation, laterality, type of IPC, associated lacrimal anomalies, management modalities, and long-term outcomes. Clinical diagnosis was made on observation of the puncta under indirect illumination of a single slit beam focused just adjacent to the puncta. A high degree of suspicion is needed to diagnose this condition. On slit-lamp examination, a slight dimpling or greyish translucency is visible at the puncta site, medial to the meibomian gland orifices. The external membrane variant completely covers the punctal area, with the membrane giving a false appearance of punctal agenesis.
There may or may not be a vessel traversing over the surface of the membrane. The internal membrane variant appears as blurred punctal margins with the vessel bayonetting over the membrane surface. The vertical canaliculus is not visualized. The rest of the eyelid architecture, especially the pars lacrimalis portion of the eyelid, appears normal. All patients who opted for surgery underwent membranotomy using Nettleship’s punctal dilator as per earlier published protocols. Following the membranotomy, the canaliculi, and the distal lacrimal drainage pathways were assessed by probing and irrigation. Associated lacrimal drainage pathologies were also assessed by the clinical examination and treated appropriately as per earlier described protocols. Anatomical outcomes were assessed by the punctal patency confirmed by the morphological features of the punctum and lacrimal irrigation. The functional outcomes were assessed by the fluorescein dye disappearance test and subjective resolution of epiphora.
RESULTS
Ninety-eight puncta of 62 eyes of 46 patients with IPC were examined in the clinic. The mean age was 12.5 years (range: 2–35 years) with a male to female ratio of 1:1. Epiphora was the presenting complaint in all the patients. However, those presenting with clear history of epiphora since birth amounted to 32 patients (70%), and the rest noticed it within the first 3 years after birth. Bilateral involvement was seen in 35% of patients (16/46). All 4 puncta were involved in 16% of patients (7/46), and both upper and lower puncta of the same eye were involved in 34 eyes (55%). Lower puncta were marginally more involved (54%, 53/98) than the upper puncta (46%, 45/98). Incomplete punctal canalization- external membrane variants were noted in 62% (61/98) and the internal membrane variant in 38% (37/98). Seventy-eight puncta of 39 patients (79.5%, 78/98) underwent membranotomy using the Nettleship’s punctal dilator.
Associated lacrimal drainage pathway deformities were seen in 31% of patients (12/39). Three puncta (4%, 3/78) in addition required mini-monoka stent insertion for partial canalicular obstruction (2.5%, 2/78) and canalicular stenosis (1%, 1/78). Five patients (13%, 5/39) had associated congenital nasolacrimal duct obstruction for which probing was performed under endoscopic guidance. Of these 5 patients, 3 patients required dacryocystorhinostomy for bony obstruction of the nasolacrimal duct (complex congenital nasolacrimal duct obstruction). A single patient had 3-wall canalicular hypoplasia (2.5%, 1/39). Associated punctal agenesis was noted in 3 patients involving the other punctum of the same eye (8%, 3/39). The mean follow-up duration was 28 months (12–60 months). Anatomical and functional outcomes were noted in 100% (39/39) and 97.4% (38/39) of the patients, respectively.
DISCUSSION
Punctal membranes in children have been described earlier; however, the terminologies differ, and so also the management protocols. The paucity of knowledge of this condition is reflected in the lack of its understanding and subsequent management. In 2014, Ali et al. coined the term IPC to introduce uniformity and described the likely pathophysiology of IPC along with the associated lacrimal disorders and management protocols. The 6-monthly outcomes with membranotomy alone were very encouraging; however, the long-term outcomes of the procedure were not known. The present study described the anatomical and functional outcomes over a mean follow up of 28 months to be 100% and 97.4%, respectively, validating the earlier described management protocols.
Associated lacrimal drainage disorders like congenital nasolacrimal duct obstruction (5/39, 13%) and canalicular pathologies (3/39, 8%) were also noted in almost similar proportions as mentioned in the study earlier.1 However, 2.5% (1/39) had canalicular wall hypoplasia, which was not seen in the earlier study. Three patients (8%, 3/39) also showed punctal agenesis involving the other puncta in the same eye. Familial associations of this condition have been described earlier. Although, the condition is rare, the authors could find one such familial association involving the mother and her 2 children in the series. However, the majority were sporadic with no familial association. Systemic associations, as seen in certain cases of punctal agenesis, were absent in IPC, although other lacrimal drainage pathologies have been found associated as described earlier.
Other than these newer findings in the present study, the experience of nearly 7 years with IPC has provided newer insights. First, the membranotomy works best if the first puncture into the membrane is performed with a fine dilator (one that rapidly tapers to a needlepoint), followed by a slow taper dilator (broad one). It is important to make sure the membranotomy is complete without any residual hanging membranes from anywhere circumferentially. The second meaningful learning was that IPC, unlike punctal agenesis, was not associated with any systemic disorders. The only exception was its rare occurrence in patients with congenital rubella syndrome.
Third, following restoration of the anatomical patency in IPC, it is rare to encounter a functional failure. It could be possible that functional epiphora may represent accompanying incomplete maturation of the lacrimal pump or distal causes of a functional failure. However, this needs to be further studied. In conclusion, the present study validates the previously described protocols over a long-term follow up. The study also emphasizes that more invasive approaches are not warranted for isolated cases of IPC. Patients with IPC demonstrate other lacrimal drainage anomalies associations. As shown in an earlier publication, familial association can exist; however, further studies with detailed genetic analysis would be required for demonstrating a genetic basis.



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