KTP-laser management of neonatal respiratory distress caused by bilateral nasolacrimal mucoceles

Shetty, S.; Dawes, P.J.D.

doi:10.1016/j.pedex.2006.03.002

« Previous Next »International Journal of Pediatric Otorhinolaryngology Extra
Volume 1, Issue 2 , Pages 135-137, June 2006

KTP-laser management of neonatal respiratory distress caused by bilateral nasolacrimal mucoceles

Received 3 February 2006; received in revised form 7 March 2006; accepted 9 March 2006.

Summary

Nasolacrimal mucoceles are rare lesions of the nasolacrimal apparatus, which may cause respiratory distress in neonates and we describe such a case where early intervention resulted in complete recovery. Utility of the MRI imaging and KTP-laser assisted marsupialization of these mucoceles also described as the highlight of this case report.

Keywords: Nasolacrimal duct cyst, Nasal obstruction, Neonate, KTP-laser

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1. Introduction

Congenital dilatation of the nasolacrimal apparatus may occur because of failure to canalise parts of the nasolacrimal system. Congenital dacryocystoceles are proximal lesions of the nasolacrimal apparatus and are treated with probing and dilatation of the duct [1]. Nasolacrimal mucoceles result from distal lesions affecting the valve of Hasner and, because the neonate is an obligate nasal breather, can cause dyspnoea; especially if they are bilateral and large [2]. Large nasolacrimal mucoceles are traditionally treated using endoscopic procedures aimed at complete marsupialization [3], [4]. We present the first description of KTP-laser marsupialization of bilateral nasolacrimal mucoceles. In our case, utilization of MR for imaging and KTP-laser marsupialization has resulted in successful outcome.

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2. Case report

After an uncomplicated pregnancy with a 40-week gestation period, a 4280g male infant was born via spontaneous vaginal delivery. He was vigorous at birth and did not require resuscitation; his Apgar scores were 9 and 10 at 1 and 5min, respectively. Two to three hours after delivery he began to have respiratory grunting, nasal alar flare, subcostal recession and intercostal in drawing. On admission to NICU he had an increased respiratory effort with a respiratory rate of 80. The chest, on auscultation, was clear with no audible stridor. Percutaneous oxygen saturation monitoring showed a fall from 100 to 88–91%[in air] during feeding. A 5-F catheter could be passed with moderate difficulty through both nasal cavities. Initial blood tests and chest X-ray were normal; he was started on IV Amoxicillin, which continued for 48h until blood cultures came back negative.

Dysmorphic craniofacial features in this case prompted a geneticist's opinion and a narrow sloping forehead, down slanting palpebral fissures, hypertelorism, right auricle dysplasia, a short neck, a broad based triangular nose and micrognathia were noted. Chromosome analysis was normal. Ultrasounds scan of the renal tract showed normal kidneys but large adrenal glands. Adrenal hormone analysis was normal. Ultrasound of the head on day 1 showed slight prominence of the third and fourth ventricles and corpus callosum with a partial absence of its posterior portion. A MRI of the cranium was ordered and otolaryngology referral was made on the third day to investigate the causes of possible nasal obstruction.

The otolaryngological examination revealed constricted nasal breathing that did not improve after the instillation of decongestants. Flexible nasendoscopy showed narrowing in the midnasal passage and bilateral cystic swellings protruding in to the inferior meatus area displacing the inferior turbinates superiorly. Choanal atresia or stenosis was not seen and there were no epiphora or medial canthus fullness.

The MRI showed 1cm diameter cysts with low signal on T1-W images and intense signals on T2-W images; they appeared well encapsulated, bilobular and nonseptate. Coronal images confirmed that these were centred on the medial canthus and extended caudad into the nasal cavity occupying the inferior meatus in dumbbell fashion (Fig. 1). Sagittal images showed these structures to be independent of the cranial structures. No intracranial abnormalities were present except for the hypoplastic corpus callosum.

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Fig. 1.
MRI coronal view showing the bilateral dacryocystoceles and intranasal cysts.

Under general anaesthesia with topical vasoconstriction of the nasal mucosa endoscopic marsupialization of cysts was carried out using KTP-laser set at 2W. A portion of the mucosa forming the inferior wall of the cyst was vaporised. Stenting of the lacrimal system was not performed. Post-operatively nasal decongestant drops were used for a week, his respiratory distress completely settled immediately and oral feeding improved. Further nasendoscopy at one week showed complete absence of the mass lesion and wide nasal passages. He is currently 11 month old and there have been no respiratory problems.

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3. Discussion

Nasolacrimal cyst formation can occur without obvious symptoms, for instance: epiphora or a medial canthal mass. This is more likely to occur when the valve of Hasner is the point of obstruction. Bilateral nasal obstruction, inclusive of choanal obstruction is an important life threatening cause of respiratory distress in the neonate [2]. However, its diagnosis requires a high index of suspicion. Manifestations are protean, varying from mild respiratory distress with breast-feeding to severe airway obstruction. The newborn are obligate nasal breathers and nasal resistance can modify the magnitude of negative inspiratory pharyngeal pressure, which appears to be critical in the development of obstructive apnoea. Bilateral cysts can cause severe neonatal respiratory distress and the absence of clinically obvious lacrimal sac pathology or mass does not rule out the presence of nasolacrimal duct pathology with narrowing of the airway. Nasendoscopy allows assessment of the nasal, choanal and pharyngeal airway and in this case was diagnostic.

Endoscopic treatment of nasolacrimal cysts is well established and has a high success rate [82%] [3]. Rigid endoscopy guides marsupialization using micro debrider or forceps or CO₂ laser. We used the KTP-laser successfully to relieve the obstruction and improve the lacrimal drainage without postoperative complications such as granulation tissue, stenosis and dacryocystitis. Histological analysis may show marked inflammatory epithelial or mucosal tissue within the cyst wall and inadequate marsupialization may require temporary stenting of NLD system.

MRI accurately delineates the lesion and excludes the possibility of meningocele, encephalocele, nasal glioma, neurofibroma and lymphangioma [5]. MRI depicts these lesions as dark on T1-weighted images and very bright on T2-weighted images and they have a ‘dumbbell’ shape. A triad of CT signs has been described by Rand [6] (cystic medial canthal mass; dilatation of NL canal and a submucosal nasal cavity mass) and these can also seen on MRI. MRI offers the benefits of superior tissue contrast resolution [differentiating fluid cystic to solid lesions], multiplanar imaging capabilities and does not use ionising radiation during image acquisition, the latter being of particular concern in the paediatric age group.

The KTP-laser has practical advantages over other modalities used to marsupialise the intranasal component of these cysts. The light wavelength (532nm) is preferentially absorbed by blood so that there is enhanced haemostasis and a fiberoptic cable of 0.2mm diameter can deliver the laser light, these features enhancing both access and visibility within the narrow confines of the newborn nasal cavity.

In summary, we describe the first case of endoscopic guided KTP-laser marsupialization of bilateral nasolacrimal duct mucoceles causing respiratory distress in a neonate. The surgery was uneventful, the child had immediate relief of their symptoms, stenting of the nasolacrimal system was not required and the result has been sustained.

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References

Honavar SG, Prakash VE, Rao GN. Outcome of probing for congenital nasolacrimal duct obstruction in older children. Am. J. Ophthalmol. 2000;130:42–48

Edmond JC, Keech RV. Congenital nasolacrimal sac mucocele associated with respiratory distress. J. Pediatr. Ophthalmol. Strabismus. 1991;28:287–289
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R.G. Shashy, V.D. Durairaj, J.M. Holmes, G.G. Hohberger, D.M. Thompson, J.L. Kasperbauer, Congenital dacryocystocele associated with intranasal cysts: diagnosis and management, Laryngoscope 113 (2003), 37–40 (Erratum in: Laryngoscope 115 (2005) 759).
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