Agenesia unilateral congénita de glándula lagrimal con sustitución por lipoma. Un caso raro de alacrimia congénita unilateral / Congenital unilateral absence of the lacrimal gland combined with lipoma. A rare case of unilateral congenital alacrima

El propósito de este artículo es describir un caso de agenesia lagrimal congénita unilateral en una niña de 7 años con síntomas oculares, ausencia de secreción lagrimal y una tumoración conjuntival en el ángulo superoexterno del ojo derecho. La agudeza visual era de 20/20 en ambos ojos. El ojo derecho presentaba secreción mucosa, erosiones y filamentos corneales. La anamnesis, la exploración física y las pruebas analíticas descartaron enfermedades sistémicas asociadas a la alacrimia congénita, así como enfermedades reumatológicas, inmunológicas o infecciosas. La resonancia nuclear magnética con diversas técnicas de supresión grasa mostró la ausencia de glándula lagrimal derecha y la presencia de una tumoración compatible con lipoma. Este caso, para nuestro conocimiento, representa la primera comunicación de una agenesia unilateral de glándula lagrimal combinada con un lipoma

The purpose of this report is to describe a case of a unilateral congenital absence of the lacrimal gland in a 7-year-old girl with ocular symptoms, no tear production and a conjunctival tumour at the supero-external angle of the right eye. The visual acuity was 20/20 in both the eyes. The right eye showed mucous secretion, corneal erosions and filaments. Anamnesis, physical examination and clinical tests ruled out systemic diseases associated with congenital alacrima, as well as rheumatic, immunological and infectious diseases. Nuclear magnetic resonance, with various fat suppression techniques, demonstrated the absence of the right lacrimal gland and the presence of a tumour compatible with lipoma. This case, to our knowledge, is the first report of unilateral absence of the lacrimal gland combined with lipoma

Lacrimal drainage anomalies in Tessier cleft 3 with unilateral anophthalmos.

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.

[Congenital nasolacrymal duct obstruction: pulled monocanalicular intubation (Monoka) versus pushed monocanalicular intubation (Masterka)]. / Imperméabilité lacrymonasale simple de l'enfant : comparaison entre une intubation monocanaliculaire « tirée ¼ type Monoka versus intubation monocanaliculaire « poussée ¼ type Masterka.

INTRODUCTION: The primary cause of congenital epiphora, congenital nasolacrymal duct obstruction (CNLDO) affects 5 to 20% of newborns. In children over 12 months, it is currently recommended to treat with primary probing and intubation of the nasolacrymal duct under general anesthesia. The purpose of this study is to compare "pulled" monocanalicular intubation (Monoka) versus "pushed" monocanalicular intubation (Masterka). METHODS: This retrospective study included 73 children between 2014 and 2017 who presented with CNLDO with membranous (mucosal) obstruction. All procedures were performed under general anesthesia with endotracheal intubation. Children with CNLDO underwent either monocanalicular silicone intubation (Monoka) with classic nasal recovery or pushed monocanalicular intubation (Masterka) inserted via the canthus. Treatment success was defined as complete resolution of epiphora at two months after the procedure. RESULTS: Surgical outcomes were assessed in 53 eyes with Monoka and 20 eyes with Masterka intubation. The mean age at treatment was 25 months (range, 12-69 months) for the two groups. Treatment success was achieved in 19 of 20 eyes (95.0%) in the Masterka group compared with 50 of 53 eyes (94.0%) in the Monoka group. A tube loss occured in 14% of cases in both groups. CONCLUSION: In children over 12 months of age, the Masterka "pushed" monocanalicular intubation technique is an effective treatment for congenital nasolacrymal duct obstruction.

Congenital canaliculops with punctal agenesis: is there a possibility to establish patency?

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.

Conservative management of congenital dacryocystocele: resolution and complications.

OBJECTIVE: To investigate the clinical outcomes of initial conservative management in infants with congenital dacryocystocele. DESIGN: Retrospective case series. PARTICIPANTS: A total of 30 congenital dacryocystoceles of 28 Korean infants treated between January 2006 and December 2015. METHODS: All patients were managed conservatively with lacrimal sac massage. Clinical courses and outcomes of the patients were retrospectively reviewed, and rates of resolution of dacryocystoceles and development of dacryocystitis were analyzed. RESULTS: In 27 cases of dacryocystoceles without infection, spontaneous resolution was achieved without any complication in 20 of 27 (74.1%) cases after conservative treatment. The mean duration of treatment was 27.5 days. Lacrimal probing was needed in 5 (18.5%) dacryocystoceles that persisted despite lacrimal sac massage for more than 1 month. Infectious dacryocystitis developed in 2 of 27 (7.4%) cases. Three dacryocystoceles were infected at presentation. Overall, 5 dacryocystoceles were complicated with infection and those cases received prompt systemic antibiotics treatment. External incision and drainage of the lacrimal sac was needed in 3 dacryocystoceles, and all cases were finally resolved without any additional procedures. CONCLUSIONS: In this case series, most of the uninfected dacryocystoceles could be successfully managed with conservative treatment alone. In cases with infection, systemic antibiotics were essential and external drainage was sometimes required, but these cases could be conservatively managed after the remission of infection.

Congenital Dacryocystocele: A Major Review.

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.

Congenital bilateral dacryocystocele.

INTRODUCTION: Newborns are obligatory nasal breathers. Therefore, nasal obstruction can lead to cyanosis and desaturation. In spite of being very rare, congenital bilateral dacryocystocele is a possible etiology for neonatal respiratory distress. CASE SUMMARY: Case report of a male newborn with respiratory distress caused by a bilateral polypoid and bluish lesion occupying almost the entire inferior nasal meatus. Imaging confirmed bilateral dacryocystocele. Treatment was conservative. There was spontaneous drainage, with relief of respiratory distress. Discussion The diagnosis of congenital dacryocystocele is clinical, although imaging exams may be requested to confirm it. Treatment is controversial, because the natural history is variable. An initial conservative management may be recommended, but, if there is a permanent respiratory obstruction without improvement, surgical management is mandatory.

Selection of surgical intervention for congenital dacryocystocele.

PURPOSE:: To evaluate the surgical intervention and its effect on congenital dacryocystocele. METHODS:: A total of 531 children with congenital dacryocystocele admitted to the Department of Ophthalmology of Beijing Children's Hospital, Shanghai Aier Eye Hospital, Nanjing Aier Eye Hospital, and the First Affiliated Hospital of Jinzhou Medical University between January 2007 and January 2017 were retrospectively analyzed. RESULTS:: A total of 531 patients were followed up for 3-24 months, with an average of 13.3 months. No serious intraoperative complications (such as bleeding and tissue damage) and postoperative complications (bleeding, infection, and hole atresia) were found. We classified the outcomes into three categories based on the signs (overflowing tears and empyema) and objective routine follow-up by endoscopy. Cure indicated that mass, overflow of tears, and breathing difficulties disappeared, and 81.5% cases (433/531) were postoperatively cured. Improvement indicated disappearance of mass, overflow empyema, and the presence of residual tears (due to trocar and tears puncture), and 18.5% cases (98/531) were postoperatively improved. Unhealed indicated overflowing pus and tears, and 0% cases did not heal after surgery. CONCLUSIONS:: Nasal endoscopic surgery is relatively safe, with high success rate for treatment of congenital dacryocystocele. Systematic training is required to promote the application of nasal endoscopy, so that more ophthalmologists can learn this technique.

Dacryoendoscopy-assisted nasal endoscopic marsupialization for congenital dacryocystocele.

OBJECTIVE: To examine the surgical outcome of dacryoendoscopy-assisted nasal endoscopic marsupialization for congenital dacryocystocele. METHODS: This is a retrospective, observational study that included 7 sides of 6 patients with congenital dacryocystocele. The surgical indication in 5 of the patients was acute dacryocystitis while one of the patients had no spontaneous resolution after long-term observation. Dacryoendoscopy was used for visualisation of the lacrimal cavity, direct probing of the obstruction at the common canaliculus, and localisation of a deflated intranasal cyst after an initial incision of the cyst. Surgical success was defined when the following postoperative conditions were satisfied: no epiphora, normal tear meniscus height, and complete clearance of the dye 5 min after instilling a drop of 2% fluorescein solution into the inferior conjunctional fornix. RESULTS: Out of 6 patients, 2 patients were noted to have membranous common canalicular obstruction. Postoperative follow-up ranged from 11 days to 3 months. Surgical success was achieved in all patients without any complication. CONCLUSIONS: Dacryoendoscopy-assisted nasal endoscopic marsupialization is an effective surgical management for congenital dacryocystocele.

Congenital dacryocystocele: sonographic evaluation of 11 cases.

PURPOSE: To report prenatal sonographic findings of congenital dacryocystocele. METHODS: The medical records of all cases diagnosed with congenital dacryocystocele at a tertiary care center from 2003 to 2015 were reviewed retrospectively. RESULTS: Eleven cases of congenital dacryocystocele were analyzed. No accompanying fetal anatomic anomalies where detected. Mean maternal age at evaluation was 22 years of age (range, 17-32 years). Four of 10 cases were primigravidas. The mean gestational age at evaluation was 32.6 weeks' gestational age. Ten out of 11 cases occurred in female fetuses (91%). Of the 11, 10 cases were unilateral and 1 was bilateral. The mean diameter at evaluation was 5.1 mm. Spontaneous resolution occurred in 2 cases (18%). In the remaining 9 fetuses, postnatal diagnosis of dacryocystocele were confirmed by an ophthalmologist evaluation. CONCLUSIONS: Prenatally diagnosed congenital dacryocystocele may undergo spontaneous resolution before birth. However, a better understanding of the prenatal sonographic findings can help to appropriately orient parents of potentially affected fetuses. Referral to a pediatrician and pediatric ophthalmologist may be considered for complete evaluation and postnatal management.

[Periorbital, blue tumour in newborn twins]. / Periorbitale, blauwe zwelling bij pasgeboren tweeling.

Newborn twins both had a blue, smooth tumour in the inner angle of the orbit; one of them had two such tumours. They were diagnosed with congenital dacryocystoceles. If decompression into the nose is not effective, patients should undergo probing early in life to reduce the incidence of dacryocystitis and orbital cellulitis.