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.

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.

Bilateral congenital mucous extravasation phenomenon: A rare case with literature review.

The purpose of this paper is to discuss a rare case of bilateral congenital mucous extravasation phenomenon on buccal mucosa near commissure of mouth. The lesions were noted at birth, subsequently enlarged to interfere with feeding. The lesion frequently ruptured, regressed, and again enlarged to interfere with normal function. The lesions were surgically removed under general anesthesia at 11 months of age and the diagnosis was confirmed by histopathological examination. Postoperative follow-up after 14 months showed no recurrence.

Simultaneous, congenital, mucocoele of the Blandin-Nühn glands and teratoma of the tongue: early management and follow up at one year.

Mucocoele of the Blandin-Nühn glands and teratomas of the tongue are rare in newborn babies. We present what is to our knowledge the first documented case of both at the same time, and describe management and follow up over 12 months after the birth. Prenatal diagnosis defined the treatment plan during delivery to establish an airway for the baby promptly. A normal delivery is possible if the cyst is anterior, and can be punctured during delivery. Delayed excision reduces the possibility of oedema and obstruction of the airway, ensures complete resection, avoids recurrence, and confirms the diagnosis. Mucocoeles are classified as extravasation or retention types; our observation supports the hypothesis of a "malformative type". A long follow-up is essential to monitor macroglossia, open bite, and the development of the mandible.

Bilateral dacryocystocele with an intranasal cyst as the cause of respiratory distress in a newborn.

A congenital dacryocystocele with an intranasal cyst is an uncommon lesion that is usually treated by ophthalmologists, although sometimes an otorhinolaryngologist is consulted first because of nasal obstruction. The nasal cavity is narrow in newborns and can easily be obstructed, even by small lesions. Prolapse or expansion of the cyst into the nose may lead to respiratory distress and difficulty in feeding, since newborns are obligate nose breathers. Here we report a case of bilatera dacryocystocele with intranasal extension in a 3-day-old female infant. The infant presented with respiratory distress and episodic desaturation and was managed successfully by bilateral endoscopic marsupialization of the intranasal cysts. This case report discusses the diagnosis and management and reviews the relevant literature. These finding suggest tha congenital dacryocystocele with an intranasal cyst must be considered in the differential diagnosis of newborns suffering from nasal respiratory difficulty.

Management of congenital dacryocystocele: report of 3 clinical cases.

This case study describes three newborns referred to our otolaryngologic service for investigating and treating a cystic dilatation of the lacrimal duct. These dilatations corresponded to unilateral or bilateral dacryocystoceles, with or without complications. The first newborn exhibited respiratory distress at birth and received early surgery and endoscopic marsupialization of intranasal and bilateral cysts. The second newborn did not show any signs of complications, and after conservative treatment for a week, the cyst spontaneously resolved. The third newborn was diagnosed in utero with ultrasonography, and the cyst resolved spontaneously during childbirth. These cases provided an opportunity to review the pathophysiology of this rare congenital lacrimal anomaly and to note responses to different therapeutic approaches. Indeed, these three cases illustrated three different management approaches, and allowed us to address the issue of prenatal diagnosis.

Congenital labial mucocele: rare presentation of a common disease.

Oral mucoceles are common lesions characterized by accumulation of mucus following rupture of a minor salivary gland duct. However, congenital mucoceles are a rare and distinctive oral condition observed in newborns. This case report details the features of a congenital labial nodule diagnosed as congenital mucocele. These lesions are rare in the oral cavity and should be diagnosed in the delivery room, but many cases are referred for further evaluation. Management is simple, and recurrence is not expected.

[Congenital mucocele of the ventral face of the tongue]. / Mucocèle congénitale de la face ventrale de la langue.

BACKGROUND: Congenital cystic lesions of the oral cavity are an extremely rare occurrence. Their prenatal diagnosis is essential since they can impede respiratory and swallowing functions. We describe a case that was detected prenatally and discuss its management. CASE REPORT: A 21-year-old primigravida patient who was 23 weeks pregnant was referred to our obstetrics and gynecology center after fetal ultrasonography showed a cystic lesion of the oral cavity. She had no family history of any congenital anomalies. Ultrasonography showed a male fetus with an anechoic mass measuring 21×11 mm encompassing the entire oral cavity, evoking either a mucocele or a cystic hygroma. Magnetic resonance imaging (MRI) showed a fetus with a wide-open mouth, due to a well-demarcated protruding cystic mass with no solid component, suggestive of a mucocele. A prenatal sonographically guided percutaneous needle aspiration of mucous fluid was performed at 33 gestational weeks. Although the mucocele decreased significantly in size, it nevertheless continued to expand progressively. After an uncomplicated pregnancy, the patient had spontaneous onset of labor at 39 weeks of gestation. An iterative aspiration was performed in the same manner in utero, resulting in a complete collapse of the mucocele. If needed, intubation could be considered. A 3030-g male was born by vaginal delivery, without respiratory distress. Clinical examination showed the extremely opened mouth and confirmed the presence of a large cystic mass approximately 4 cm in diameter, of sublingual origin and encompassing the entire oral cavity. The continuous protrusion of the tongue was responsible for the infant's inability to close the mouth and be breastfed. After insertion of a feeding tube, the newborn had maxillofacial surgery consisting in marsupialization of the cyst at 2 days of age. The mucocele decreased in size and the postoperative course was uneventful. No recurrence was observed at 6 months' follow-up. DISCUSSION AND CONCLUSION: Congenital mucoceles of the tongue are very rare benign lesions of the oral cavity, resulting from extravasation or retention of mucus from minor salivary glands. Their prevalence is unknown and, to our knowledge, less than ten cases of prenatal diagnosis have been previously reported. Such cystic lesions can cause respiratory distress and swallowing disorders in newborns. They are usually suspected on ultrasonography. MRI highlights the nature of the lesion and its locoregional connections with muscles and blood vessels. It provides a good analysis of the soft tissues and can distinguish between the muscles of the tongue and the pathologic mass. However, the use of CT has been reported when the diagnosis was made after childbirth or in adulthood. Given the risks of interference of the lesion with respiratory and swallowing functions, intrauterine decompression of the mucocele can be an option to prevent respiratory distress at birth and the need for neonatal intubation. Mucoceles provide somewhat confusing and disturbing ultrasonographic appearances, which can be stressful for the medical team and parents. Prenatal diagnosis and early surgical intervention (marsupialization, complete excision of the cyst or the salivary gland) can prevent risks of breathing distress and breastfeeding problems. Therefore, this strategy is essential to offer fast and satisfactory management of this rare but anxiety-producing congenital situation.

[Congenital dacryocystocele: Surgical treatment or routine follow-up?]. / Dacryocystocèles congénitales : traitement chirurgical ou simple surveillance ?

INTRODUCTION: Congenital dacryocystoceles are rare and often misunderstood pathologies. Their treatment varies and consists in simple follow-up, lacrimal catheterization or endoscopic surgical drainage, depending on medical teams. The aim of our study was to discuss the place of endoscopic drainage in the treatment of congenital dacryocystocele. METHODS: We conducted a retrospective review on 18 cases of congenital dacryocystoceles taken in charge in a tertiary care center between 2009 and 2012. RESULTS: Thirteen newborns, including five bilateral cases, were taken in charge. The average age was 14.6 days. Six newborns presented with an acute dacryocystitis at the time of diagnosis. No respiratory complications were observed. Spontaneous drainage of the dacryocystocele was observed in 38.8% of the cases, occurring at 22 days of life on average. Endonasal endoscopic drainage was performed in 66.6% of the cases. No recurrence or complication was observed after surgery. After spontaneous drainage, one recurrence was observed. The mean follow-up period of these patients was 8.8 months. DISCUSSION: Spontaneous drainage is common. Conservative management may therefore be considered in absence of infection. In case of infection and/or persistence of dacryocystocele after 4 weeks of life, endonasal surgical drainage should be considered. Imaging of the facial structure should be performed before any surgical treatment.

Congenital laryngomucocele: a rare cause of airway obstruction in a newborn.

Congenital laryngoceles are defined as cystic dilatation of laryngeal saccules and are an extremely rare cause of newborn respiratory distress. A laryngomucocele occurs when the neck of the laryngocele gets obstructed and fills with the mucoid secretions of the saccule. It may cause stridor, respiratory distress, and severe airway obstruction in the narrow airway of a newborn and necessitates urgent surgical intervention. There is only 1 case of congenital laryngomucocele reported in an autopsy examination in the English literature, and here we report the first living congenital laryngomucocele case and discuss the clinical approach.

Congenital mucocele of the tongue: a case report and review of the literature.

Mucoceles are benign lesions of the oral cavity that develop as a result of retention or extravasation of mucous material from minor salivary glands. Congenital mucoceles are very rare. These lesions in newborns may interfere with breastfeeding and may even compromise respiratory function. A patient with a congenital mucocele diagnosed by prenatal ultrasound screening showing a cystic lesion of the tongue is presented herein. The physical examination, lesion evolution and imaging are described, together with the surgical management, histopathology and two-year follow-up. Early clinical assessment, differential diagnosis and magnetic resonance imaging allow clinicians to diagnose and treat this rare congenital condition with surgery in early infancy.

A congenital mucocele of the anterior dorsal tongue.

We report on a new-born with a congenital mucocele on the anterior dorsal side of the tongue. The presentation as well as the differential diagnosis of congenital oral swellings is discussed. Because of breastfeeding problems the mucinous swelling was incised and drained two days after birth. Immediately after drainage the swelling disappeared. Congenital oral swellings are rare. Most of them are mucoceles. Post-partum treatment is surgically, but spontaneous remission has been described. High incidence of recurrence should be taken into account when (micro-)marsupialization or incision as sole treatment is performed.

Congenital dacryocystocele: comparative findings of dacryoendoscopy and histopathology in a patient.

A full-term baby girl showed a bluish mass at birth in the right medial canthal area. She later demonstrated dacryocystitis and cellulitis. Although probing was performed, it was unsuccessful, and the dacryocystitis and cellulitis worsened. Computed tomography showed a dilated lacrimal sac, an enlarged nasolacrimal canal and a nasal cyst. Topical and intravenous systemic antibiotics were then started, and the symptoms partially subsided. Surgery was performed under general anesthesia with endoscopic assistance, under which cystic distention was visualized beneath the inferior turbinate. A dacryoendoscope showed injection and hemorrhages on the inner wall of the cyst. Marsupialization of the cyst was performed. Histopathologic examination showed submucosal tissue including fibrous thickening with infiltration of mononuclear leukocytes. The patient remained symptom free 6 months thereafter. Dacryoendoscopy for a congenital dacryocystocele demonstrated injection with hemorrhages, and this inflammatory sign corresponded to the histopathologic findings.