Antenatal diagnosis of a congenital nasolacrimal duct cyst by ultrasonography: A case report
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PRENATAL DIAGNOSIS, VOL.
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1 4 623-626 (1994)
ANTENATAL DIAGNOSIS OF A CONGENITAL NASOLACRIMAL DUCT CYST BY ULTRASONOGRAPHY: A CASE REPORT CUNEYT M. ALPER*, KENNY H. WAN*, LYNDON M. m L t AND PAULA CHENEVEY~
University of Pittsburgh School of Medicine, Department of Otolaryngology*, Children’s Hospital of Pittsburgh and Department of Obstetrics, Gynecology and Reproductive Sciences?, Magee Women’s Hospital, Pittsburgh, Pennsylvania, U.S.A. Received November 1993 Revised January 1994 Accepted 5 January 1994
SUMMARY With proximal and distal obstruction to the lacrimal drainage system, mucus accumulates forming a cyst. In order to diagnose a nasolacrimal duct cyst antenatally, the sonographer must image the region adjacent to the orbits. KEY woms-Antenatal sonography, nasolacrimal duct cyst. INTRODUCTION
We present a case of an antenatally diagnosed lacrimal duct cyst that resulted in neonatal respiNeonates are obligate nasal breathers. Conse- ratory distress requiring surgical intervention. quently, any obstruction to the nasal passageway can have significant respiratory consequences. BiCASE REPORT lateral total nasal obstruction generally presents as an acute airway emergency and requires immediate C.H. is a 31-year-old G4,P2. An ultrasound intervention at birth. Unilateral or partial bilateral obstruction may result in noisy breathing, in- examination at 22.3 weeks from her last menstrual creased inspiratory effort, restless sleep, poor suck- period revealed a twin gestation. Serial ultrasound ling, and, in the most severe cases, respiratory examinations confirmed normal growth of both distress. A physical examination, the passage of twins. At 34 weeks’ gestation, it was noted that nasal catheters, anterior rhinoscopy, and flexible twin B had a 9 mm right and an 8 nun left lacrimal fibreoptic rhinoscopy provide important diagnostic duct cyst (Figs 1A and 1B). At 36 weeks’ gestation, the patient had a normal information. However, imaging studies are frespontaneous vaginal delivery of both twins. Twin quently required in order to reach a definitive B weighed 2410 g and had Apgar scores of 8 and 9 diagnosis. The predominant cause of neonatal nasal at 1 and 5 min, respectively. On the second day of obstruction is unilateral or bilateral choanal atre- life, the infant developed moderate respiratory sia (incidence: 1/5000 livebirths). Other causes of distress with mouth breathing. A 6 French feeding nasal obstruction include choanal stenosis, pyri- tube was passed through each naris without difKform aperture stenosis, meningoencephalocele, culty. However, an obstruction prevented the passage of an 8.0 French catheter. A nasal CT gliomas, and dermoids (Coates, 1992). confirmed the presence of a soft tissue mass arising in the left inferior meatus (Fig. 2). Addressee for correspondence:Lyndon M. Hill, MD, Magee The patient was taken to the operating room Women’s Hospital, Ultrasound Department, 300 Halket Street, and underwent a combined ophthalmological and Pittsburgh, PA 15213, U S A . CCC 0197-385 1/94/070623-04 0 1994 by John Wiley & Sons, Ltd.
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(B) Fig. 1-Fetal ultrasound at 34 weeks’ gestation. (A) Bilateral lacrimal duct cysts (markers); (B) a 9 m m right lacrimal duct cyst (markers) inferomedial to the right globe
otolaryngological procedure. The nasal cavities were endoscoped and a large left inferior meatal cyst was noted. The left nasolacrimal cyst was marsupialized piecemeal using otological cup forceps. Once patency was established, the lacrimal
system was irrigated. The right nasolacrimal system was probed and irrigated, with no abnormalities noted. The child’s obstructive symptoms resolved post-operatively. She has been asymptomatic for the past 8 months.
CONGENITAL NASOLACRIMAL DUCT CYST
Fig. 2-Computed tomography illustrating the nasolacrimalduct cyst in the left nasal cavity in the axial view
DISCUSSION The persistence of the embryonic Hasner membrane at the vault of the inferior meatus is the presumed aetiology of nasolacrimal duct cysts. With proximal and distal obstruction to the lacrimal drainage system, mucus accumulates, forming a cyst (Jones and Wobig, 1976). Occasionally, a nasolacrimal duct cyst will present as a fulminant dacryocystitis. The management of nasal lacrimal duct cysts remains controversial. In general, an uncomplicated dacryocele can be followed for a few weeks with local massage followed by probing, if spontaneous resolution does not occur. Surgical intervention is necessary in approximately 20 per cent of cases (Mansour et al., 1991). The antenatal diagnosis of airway obstruction by ultrasound is a relatively rare event. Epignathus (Catalan0 e f al., 1992), cervical teratomas (Shoenfeld et al., 1982), and laryngeal stenosis (Richards et al., 1992) have been diagnosed antenatally and can result in acute neonatal respiratory distress. However, in these instances the sonographic findings are generally readily apparent.
Lacrimal duct cysts are considerably smaller in size and do not produce any secondary sonographic signs. As a result, the diagnosis of a lacrimal duct cyst requires not only that the fetal head be appropriately positioned, but also that the sonographer take the time to image the region adjacent to the orbits. The ophthalmological (Mansour et al., 1991), otolaryngological (Mazzara et al., 1993), and radiological (John and Boldt, 1990) literature is replete with reports of congenital nasolacrimal duct cysts causing nasal and airway obstruction. In contrast to these reports, Davis et al. (1987) reported two cases of antenatally detected lacrimal duct cysts that resolved spontaneously. Although the antenatal ultrasound examination in our case revealed bilateral nasolacrimal duct cysts, only a left-sided mass was present on the neonatal CT and at surgery. This suggests that the right-sided cyst either drained spontaneously after the initiation of neonatal respiratory efforts (Rand et al., 1989) or that the passage of the nasal catheter ruptured the cyst (Mansour et al., 1991). This case once again emphasizes the ability of sonography to define many, often subtle, fetal abnormalities and subsequent birth defects. How-
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ever, diagnosis alone is not enough. The fetal and neonatal implications of an antenatally detected congenital abnormality must be considered. While lacrimal duct cysts may be benign and resolve spontaneously, the potential for acute respiratory distress in the early neonatal period must be considered. Frequently the sonologist and obstetrician may not be familiar with the potential complications associated with what appear to be relatively benign congenital abnormalities. A multidisciplinary team approach is therefore often required in order to develop an appropriate plan of management.
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