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INTRODUCTION

Congenital nasolacrimal duct obstruction typically involves a blockage at the valve of Hasner. It is frequently present at birth.1,2 Infrequently, it may occur in association with a blockage at the point where the canaliculi join the tear sac, giving rise to a distended tear sac.3 The most popular term used to describe this entity is congenital dacryocele, but it has also been referred to as amniotocele, aminocele, mucocele, or dacryocystocele. Treatment of this disorder has been controversial. Some authors advocate a trial of medical management,4-8 while others advocate immediate probing to prevent infection.9-12 We undertook a prospective trial of at least 2 weeks of medical care in the treatment of congenital dacryoceles.

MATERIALS AND METHODS

AD patients younger than 4 weeks who presented to the authors with dacryocele were included in this study. On the initial evaluation, the age at which the dacryocele was first noted by the family and referring physician was elicited. An attempt was made to reduce the dacryocele on the initial visit with moderate pressure over the dacryocele. A cycloplegic refraction was performed when possible.

If the dacryocele was not infected, the parents were instructed to treat the dacryocele with antibiotic drops, warm compresses, and massage, three times a day. The length of time to resolution or onset of infection was noted. If the dacryocele did not resolve after at least 2 weeks of medical management, a probing was performed. Medical management was continued beyond 2 weeks if the parents and ophthalmologist desired. Probing could be performed with the patient awake or under general anesthesia, at the surgeon's preference.

If the dacryocele was infected at time of presentation, intravenous (IV) antibiotics were given in addition to warm compresses and massage. Length of time to resolution or length of infection was noted. These dacryoceles were to be probed once the infection had resolved. A dacryocele that became infected during the course of medical management was treated similarly.

Subsequent to the resolution of the dacryocele, these patients were reexamined on an ongoing basis to look for the presence of nasolacrimal duct obstruction in addition to having cycloplegic refractions to look for evidence of astigmatism in the involved eye.

TABLESummary of Results

Any patient with respiratory distress or suspected of having intranasal cyst with congenital dacryocele was urgently evaluated by an ear, nose, and throat specialist or imaging study. Neonates identified as having intranasal cysts were not to be included in this trial of medical management.

RESULTS

Over a 3½-year period, 17 patients with 21 dacryoceles were enrolled. Follow-up ranged from 6 weeks to 45 months with a mean follow-up of 15 months. There were six boys (35%) and 11 girls (65%). The right nasolacrimal system was affected in five cases (29%), the left system in eight cases (47%). Bilateral involvement occurred in four cases (24%). Three of the 21 dacryoceles were infected at the time of initial evaluation.

All 21 dacryoceles were first noted by the parents or referring physician by the tenth day of life. Eleven of the dacryoceles were noted on the first day of life. Three dacryoceles were detected by the primary care physician only after becoming infected. This occurred in two cases on the second day of life prior to discharge from the hospital and in one case on the sixth day of life. A review of the nursing assessment after delivery and birth pictures indicate that a bluish swelling was present in the medial aspect of the lower lid of these patients on the first day of life; however, these bluish masses were interpreted as bruising.

Eighteen dacryoceles were not infected at the time of their initial evaluation. During the course of the medical management, one dacryocele that was seen by the ophthalmologist on the second day of life became infected on the fourth day of life. This patient was readmitted to the hospital and treated with IV antibiotics. Of the 17 dacryoceles that did not become infected, 12 resolved with conservative care within 1 to 6 days (Fig). Of the 12 dacryoceles that did not become infected and resolved with conservative care, four (33%) were noted to have a typical nasolacrimal duct obstruction after resolution of the dacryocele, indicating that in these patients only the obstruction proximal to the lacrimal sac resolved. The nasolacrimal duct obstruction in these four patients resolved in 4 weeks to 4 months with conservative management.

A total of four dacryoceles were infected (Table). Three dacryoceles were infected at the time of their initial evaluation. Two of these patients presented on the second day of life. One patient presented at 6 days with bilateral dacryoceles, with the right dacryocele being infected. One additional patient developed dacryocystitis and an overlying cellulitis during the course of her conservative treatment. All four of these patients were treated as inpatients with IV antibiotics, warm compresses, and massage. All four of these infected dacryoceles resolved within 1 day. The nurses were instructed to suction the nose as soon as they noted disappearance of the dacryocele. Typically, the nurses recovered a large amount of purulent material from the nose upon resolution of the dacryocele. None of these patients had a return of symptoms or subsequent nasolacrimal duct obstruction.

Five dacryoceles were probed after failing to resolve with 2 to 4 weeks of medical management. Three were probed while awake, two were probed under general anesthesia. Four of these dacryoceles resolved after the initial probing. In one case in which only topical anesthesia was used, the probe was passed into the sac, but could not be passed down the nasolacrimal duct. Subsequently, the material could be expressed from the tear sac with digital pressure. The dacryocele recurred and was probed a second time 8 weeks later. This time, the probe could be passed into the nose. The swelling of the tear sac resolved.

Refractive data were available on eight patients. Cycloplegic refractions were performed after resolution of the dacryocele. None of the patients was found to have significant anisometropia (1.00 D or greater) in their astigmatism. None of the patients was noted to have permanent distortion of the medial canthal area as a result of the dacryocele. One patient did develop rotary nystagmus, but this was felt to be unrelated to her unilateral dacryocele.

DISCUSSION

Obstruction of the distal aspect of the nasolacrimal duct at birth is common, occurring in 35%2 to 73% J of newborns according to anatomic studies. The tear system usually becomes patent in the majority of these newborns by 1 month of age, with only 6% of infants developing symptoms of a nasolacrimal duct obstruction.13

Congenital dacryocele is a distention of the lacrimal sac that results from an obstruction of the distal portion of the nasolacrimal duct as well as a proximal obstruction at the site where the canaliculi join the nasolacrimal sac.4 It may be present at birth or develop in the first few days of life.4 Clinically, it appears as a deep blue or pink grey medial canthal swelling.8 It causes an upward slanting of the palpebral fissure nasally.7 It is firm, but with digital pressure, material may reflux through the canalicular system.7 Its clinical appearance can be mimicked by hemangioma and meningoencephalocele.6

Some authors9-12 advocate early probing of congenital dacryoceles to prevent possible complications which include dacryocystitis,7,10 celluhtis,7 atony of the tear sac,12 astigmatism,7 and permanent distortion of the canthal tissues.6 Probing of a dacryocele can be technically difficult. The distended tear sac distorts the surrounding tissue. There is tense obstruction at the point where the canaliculi join the tear sac through which the probe must be passed. Probing of a dacryocele must relieve both the proximal as well as the distal obstruction to prevent its recurrence. Petersen and Robb10 probed three dacryoceles into the sac only. In two of these cases the swollen sac was initially decompressed, however these two patients had a recurrence of their dacryoceles and were eventually probed into the nose with resolution of their symptoms.

Mansour et al7 noted that 10 of the 45 dacryoceles probed in their series recurred. These unsuccessful probings were characterized by difficulty in passing the probe and irrigating. With probing, there is the risk of anesthesia as well as the creation of a false passage. One of the authors of this study did probe three dacryoceles while the patients were awake. This author has subsequently changed to probing congenital dacryoceles with the patient under general endotracheal anesthesia, recognizing the risk of drainage of the contents of the dacryocele into the nose with the potential of aspiration of this material by the neonate.

Since a congenital nasolacrimal duct obstruction is necessary to have a dacryocele, one would expect congenital dacryoceles to have a high rate of spontaneous resolution, just as congenital nasolacrimal duct obstruction. Petersen and Robb,10 while advocating immediate probing had two dacryoceles in their seven-patient series resolve before they had the opportunity to probe. Ffooks,9 who advocated early probing, had one dacryocele in his seven-patient series resolve before a probing could be carried out. Many authors,4-8 therefore, advocate a trial of medical management if the dacryocele is not infected.

In this series, 16 of 21 dacryoceles (76%) resolved with medical management. Four of these 21 dacryoceles were infected during their clinical course. This success rate is similar to what has been reported in the literature. In 1991, Mansour et al7 reported on 54 cases collected from seven medical centers. Of these 54 cases, five consecutive uncomplicated dacryoceles were treated with medical management in one center. Four (80%) resolved with medical care; one developed dacryocystitis and eventually required probing.

Harris and DiClementi,6 in 1992, reported on four consecutive cases managed conservatively. Three (75%) of these four cases resolved with medical management by the first to eighth week of life. One dacryocele in this series which had become infected was treated with probing and silicone intubation after not resolving by 12 weeks of age.

Sullivan et al,8 in 1992, reported on five patients treated conservatively. Three of the five dacryoceles resolved by 21 days of age. Two of the dacryoceles became infected, requiring systemic antibiotics. One case resolved with use of the antibiotics, while the second infected dacryocele required two probings and silastic intubation. Overall, four (80%) of these five dacryoceles resolved with medical management.

The protocol was for a minimum of 2 weeks of medical management. Four weeks was the maximum any patient was treated with medical management. It is possible that if medical management would have been continued for longer than 2 to 4 weeks, more dacryoceles would have resolved without probing. There are reports in the literature of dacryoceles resolving at 10 weeks of age with medical management.46

Fig: Graph illustrating the time required for resolution with medical management in 17 noninfected dacryoceles. Five dacryoceles did not resolve with 2 weeks of medicai management.

No patient in our series appeared to develop astigmatism or distortion of the medial canthal area as a result of their dacryocele. However, one patient who was being treated conservatively became infected and required admission to the hospital. All of the dacryoceles in our series that resolved with medical management did so within 6 days. Based upon our data, 1 week appears to be an adequate trial of medical management.

Four patients had an infected dacryocele with overlying cellulitis. The protocol called for these patients to be probed once their infection had quieted. However, all of these dacryoceles resolved within 24 hours with the use of IV antibiotics. None of these patients had a return of the dacryocele, and therefore none required a probing. It is likely that the membrane obstructing outflow into the nose was weakened in these patients by an inflammatory cell infiltrate,7 leading to spontaneous rupture of this membrane and release of the lacrimal sac contents into the nose. While the four infected dacryoceles in our study resolved quickly with medical management, there are reports of persistence of infected dacryoceles after medical treatment, requiring multiple probings or silicone intubation.6,9

Intranasal cysts have been reported to result in respiratory distress in association with congenital dacryocele.7,14 Our protocol called for any neonate with dacryocele and respiratory distress to be investigated for intranasal cyst with CT scan or nasal endoscopy. None of the neonates in our study had respiratory distress and therefore no patient was investigated for intranasal cyst. Therefore it is possible that some of the neonates in our study did have undetected intranasal cyst.

Several of the dacryoceles in this series were not discovered by the pediatrician until several days of age, or when they became infected. Review of the birth records and photographs indicated that the three dacryoceles that were infected at the time of presentation were present on the first day of life, but were not detected by the pediatrician. A dacryocele is an uncommon finding in a neonate that a pediatrician may not be aware of. In addition, the newborns often have swelling and bruising as a result of the birth process, which may confuse the clinical picture. All of this makes it likely that some dacryoceles are occurring and resolving spontaneously without coming to the attention of an ophthalmologist, unless they persist or become infected.

Treatment of congenital dacryoceles must be individualized. In cases complicated by respiratory distress, urgent evaluation and treatment of an associated intranasal cyst may be required.14 Infected dacryoceles require hospital admission and systemic antibiotics. Uncomplicated dacryoceles can be treated with a trial of medical management for 1 to 2 weeks. If not resolved the dacryocele should be probed into the nose confirmed by inspection15,16 or irrigation.16 The inability to irrigate after probing may indicate the presence of a nasal cyst best treated with excision via nasal endoscopy.16 The caregivers of these patients need to be informed of the signs of infection and instructed to contact the physician promptly if infection occurs.