PRENATAL DIAGNOSISPrenat Diagn 2008; 28: 554–555.Published online in Wiley InterScience(www.interscience.wiley.com) DOI: 10.1002/pd.2015

RESEARCH LETTER

Prenatal two- and three-dimensional sonographic diagnosisof dacryocystocele

Arda Lembet, Harika Bodur*, Belgin Selam and Tolga ErginDepartment of Obstetrics and Gynecology, Acibadem Hospital, Istanbul, Turkey

KEY WORDS: dacryocystocele; nasolacrimal duct; prenatal diagnosis; ultrasound; midfacial anomaly

Dacryocystocele is a rare congenital midface anomalycaused by obstructions of both proximal and distal endsof the nasolacrimal duct. A bilateral lesion may haveintranasal extension and may completely obstruct thenasal passage, which may result in respiratory distress.It is the second most common cause of neonatal nasalobstruction after choanal atresia and requires prompttreatment (Lowe et al., 2000). Midface anomalies suchas dacryocystocele are not uncommon and their diagno-sis may present a challenge for the obstetricians. Prena-tal two- and three-dimensional ultrasound findings maycontribute to intrauterine differential diagnosis of thisanomaly. We report a case of prenatal detection of dacry-ocystocele at 28 weeks of gestation.

A 31-year-old gravida 1 woman had routine pre-natal care at the Acibadem Hospital. Her pregnancywas uneventful until 28 weeks of gestation. However,ultrasound examination revealed a hypoechogenic mass,situated inferomedial to the fetal right eye withoutdisplacement of the globe, at 28 weeks of gestation(Figure 1A and B). The mass was a well-circumscribedcystic lesion. Transverse view for visualization of thefetal face demonstrated that both orbits were symmetric.The interorbital distance was within the normal range.Intracranial anatomic screening was normal. We did notdetect other facial abnormalities including the facialprofile. Sonographic properties of the mass remainedunchanged during repeated scans at 32, 34, 36, and38 weeks. The couple was informed about the mostprobable etiology for differential diagnoses. The womandelivered a 2880 g female baby at 39 weeks of gestationby cesarean section (Figure 1C). The Apgar scores were9 and 10 at 1 and 5 min, respectively. Neonatal exami-nation was uneventful except a blue swelling located onthe medial site of the right orbit. Light compressive ductmassage during the first neonatal month was initiatedand regression of the dacryocystocele was observed.

Prenatal visualization and differential diagnosis offacial abnormalities such as dacryocystocele are crucialbecause bilateral cysts can have intranasal extension andthey may be part of numerous syndromes. The diagnosis

*Correspondence to: Harika Bodur, Department of Obstetrics andGynecology, Acibadem Hospital, Tekin Sok. 8 Acibadem, 34718Istanbul, Turkey. E-mail: harikabodur@yahoo.com

of dacryocystocele presents a challenge due to diverseoutcomes for the pathologies included in differentialdiagnosis. It has been sporadically reported in fetusesaround 30 weeks of gestation (Suma et al., 1999). Mostfavorable gestational age for three-dimensional scanningof the fetal face ranges between 23 and 30 weeks ofgestation (Kurjak et al., 2007). In the current case,we initially diagnosed the lesion by two- and three-dimensional ultrasound at 28 weeks.

Differential diagnosis of dacryocystocele includesnasal glioma, frontonasal/nasoethmoidal encephalocele,dermoid cyst, epidermoid cyst, facial hemangioma, andlymphangioma. The typical cystic appearance medial tothe orbit without internal vascularization on color andpower Doppler intervention as in the presented caseshould be distinguished from the other periorbital masseson axial plane. Multiplanar views are also importantduring prenatal evaluation and can help in the differ-ential diagnosis of a frontal cephalocele. Hemangiomashave greater echogenicity, and greater Doppler signals;cephalocele causes displacement of the globe and a cal-varial defect; dermoid and epidermoid cysts are usuallyhyperechoic and are situated superolaterally to the globe(Suma et al., 1999). Fetal magnetic resonance imaging(MRI) may be helpful in detecting such anomalies.

Sepulveda et al. report the outcome of ten fetuseswith prenatal diagnosis of congenital dacryocystocele(Sepulveda et al., 2005). The cystic lesions were uni-lateral in 60% of cases. In utero spontaneous resolutionwas documented in five cases out of ten. Duct mas-sage during the first few months comprises the initialtreatment for dacryocystocele. Ductal stenosis resolvesspontaneously with conservative management in 90%of infants during the first year of life (Lowe et al.,2000). Probing and balloon catheter dilation are offeredas alternative options for patients with initial treatmentfailure. Silicone intubation of lacrimal system to assureprolonged permeability or marsupialization of the nasalcyst, are possible treatment modalities in the presence ofpersistent disease (Teixeira et al., 2005; Casady et al.,2006). Endonasal endoscopic sinus surgical techniquesprovide an alternative to external dacryocystorhinostomyfor children with persistent symptomatic nasolacrimalobstruction (Cunningham, 2006). Dacryocystocele hasa tendency to become infected (Usha et al., 2006).

Copyright 2008 John Wiley & Sons, Ltd. Received: 14 December 2007Revised: 20 March 2008

Accepted: 22 March 2008

PRENATAL DIAGNOSIS OF DACRYOCYSTOCELE 555

(A)

(B) (C)

Figure 1—Prenatal two-dimensional ultrasound finding at 28 weeks(A), three-dimensional surface rendering views of a fetal face at30 weeks of gestation (B), and postnatal observation of the baby(C) presenting with dacryocystocele were demonstrated

Prophylactic antibiotics are recommended to preventdacrocystitis, periorbital cellulitis, and bacterial conjunc-tivitis during this period. Dacryocystocele was correctedwith duct massage during the postnatal second month inthe presented case.

Three-dimensional ultrasonography has remarkablyimproved the evaluation of fetal facial anomalies in clin-ical practice. Additional information provided by three-dimensional ultrasound images may impact clinical man-agement of the fetus. The data gathered by this techniquemay be useful for differential diagnosis whereby clini-cians may inform the parents about the diagnosis andits clinical consequences prenatally. The presented casewas diagnosed by two- and three-dimensional ultrasoundat 28 weeks of gestation, although the woman had anuneventful follow-up until that time. Addition of three-dimensional ultrasound examination during the follow-up in the third trimester may contribute to detection ofunusual pathologies that do not demonstrate any abnor-mal sign during fetal anatomic surveillance at secondtrimester.

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Casady DR, Meyer DR, Simon JW, Stasior GO, Zobal- Rather JL.2006. Stepwise treatment paradigm for congenital nasolacrimal ductobstruction. Ophthal Plast Reconstr Surg 22: 243–247.

Cunningham MJ. 2006. Endoscopic management of pediatric naso-lacrimal anomalies. Otolaryngol Clin North Am 39: 1059–1074.

Kurjak A, Azumendi G, Andonotopo W, Salihagic- Kadic A. 2007.Three- and four-dimensional ultrasonography for the structural andfunctional evaluation of the fetal face. Am J Obstet Gynecol 196:16–28.

Lowe LH, Booth TN, Joglar JM, Rollins NK. 2000. Midfaceanomalies in children. Radiographics 20: 907–922.

Sepulveda W, Wojakowski AB, Elias D, Otano L, Gutierrez J. 2005.Congenital dacryocystocele: prenatal 2- and 3- dimensionalsonographic findings. J Ultrasound Med 24: 225–230.

Suma V, Marini A, Bellitti F, Bucci N, Dorov D. 1999. Prenatalsonographic diagnosis of dacryocystocele. Ultrasound ObstetGynecol 14: 74.

Teixeira CC, Dias RJ, Falcao-Reis FM, Santos M. 2005. Congenitaldacryocystocele with intranasal extension. Eur J Ophthalmol 15:126–128.

Usha K, Smitha S, Shah N, Lalitha P, Kelkar R. 2006. Spectrum andthe susceptibilities of microbial isolates in cases of congenitalnasolacrimal duct obstruction. J AAPOS 10: 469–472.

Copyright 2008 John Wiley & Sons, Ltd. Prenat Diagn 2008; 28: 554–555.DOI: 10.1002/pd