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Angiomatous ethmoidochoanal polyp in an infant: case report.

Abstract

Nasal polyps are rare in children younger than 10 years. We describe the case of an infant girl who had undergone a traumatic intubation at birth that had resulted in nasal bleeding. At the age of 5 months, she was brought to us with an obstructive left nasal mass. Imaging revealed the presence o fan ethmoidochoanal polyp, as well as a fracture of the posterior cribriform plate and a small associated meningocele. Four months later, the polyp was excised, and the meningocele was corrected with endoscopic nasal surgery. Pathologic evaluation identified the lesion as an angiomatous polyp, which was probably related to the previous traumatic episode. We discuss the clinical aspects of a pathologic entity that has not been previously reported in an infant.

Introduction

Nasal obstruction is a common finding in the pediatric population as a manifestation of common conditions such as rhinitis and adenoid hypertrophy. (1) However, when we come upon an obstruction caused by an intranasal mass in an infant, the diagnostic possibilities trend toward rare conditions such as choanal malformations and congenital protrusions of an intracranial mass into the nasal cavity. (2) Other congenital masses must also be considered. Nasal polyps are rarely found in children, particularly in infants. In this article, we describe what we believe is the first reported case of an angiomatous ethmoidochoanal polyp in an infant.

Case report

A 5-month-old girl was brought to us by her parents, who reported that she exhibited some signs of nasal blockage, including runny nose, noisy breathing, and some feeding difficulties without any measurable growth impairment. The patient had been born prematurely at 30 weeks. At birth, she weighed 1,050 grams, and her Apgar score was 7/8/8. She had undergone a difficult nasogastric intubation at birth, which had resulted in left nasal bleeding.

With conservative treatment, the epistaxis diminished, but some residual bleeding persisted for another 12 days. She was admitted to the neonatology unit. Transfontanellar Doppler ultrasonography demonstrated two hyperechogenic areas; one extended from the left ventricle to the semioval center, and the other was spread over the left frontobasal area as a result of an anterior cranial fossa invasion through the posterior ethmoid sinus. There was no record of any intranasal masses, malformations, or respiratory symptoms at that time.

Examination in our ENT department revealed the presence of a nonlobulated left intranasal mass that had obliterated the choana. The surface of the mass was smooth and richly vascularized. It was attached by a pedicle and somewhat movable. Findings on the remainder of the examination were normal. No biopsy was performed.

Surgery was postponed until the patient became somewhat older (9 mo). Computed tomography (CT) at that time provided a clear picture of the expansive intranasal soft-tissue mass (figure 1, A). The lesion protruded from a fracture of the posterior cribriform plate, and it blocked an otherwise patent choana. Magnetic resonance imaging (MRI) showed that the mass was not contiguous with the intracranial contents. Intermediate heterogeneous enhancement was seen on both T1- and T2-weighted imaging. No flow-void signals were seen, and there was a break in the overhanging dura mater, with a small meningocele in the posterior ethmoid (figure 1, B). Other intracranial fibrotic lesions were noted. The mass was diagnosed as an ethmoidochoanal nasal polyp.

[FIGURE 1 OMITTED]

The polyp was excised byintranasal endoscopic surgery (figure 2). The nasal vault defect was corrected at the same time with a middle turbinate overlay technique. The patient experienced no significant bleeding, postoperative complications, or local recurrence. A definitive diagnosis was established after pathologic study identified the mass as an angiomatous polyp (figure 3).

Discussion

Because newborns are obligate nasal breathers, a complete obstruction of the nose bilaterally is a cause of acute respiratory distress soon after delivery. A unilateral blockage, on the other hand, is often diagnosed later on the basis of noisy breathing, a chronic unilateral nasal discharge, disturbed feeding with inadequate weight progression, and/or a sleep disturbance with apnea. (1) When we are confronted with a unilateral intranasal mass in an infant, the differential diagnosis is generally made up of rare entities, most of them congenital.

[FIGURE 2 OMITTED]

Obtaining a biopsy of such a mass is controversial. Biopsies are often not performed in such cases because of the possibility of inflicting an injury to a vascular tumor or neural tissue. Also, biopsy findings have little influence on therapeutic decision making when radiologic studies are available.

In our patient, the clinical picture and the absence of congenital masses during the early observations and on ultrasonography suggested an inflammatory polyp. On the other hand, the nasal trauma and the history of bleeding in a premature newborn, combined with findings of a unilateral nasal mass and evidence of a nasal vault defect on CT, strengthened the theory that the mass represented a large basal transcribriform meningoencephalocele. The MRI findings were crucial because they allowed us to conclude that both possibilities were correct to some extent: There was a small noncongenital meningocele and a moderately vascular ethmoidochoanal polyp. Both masses were addressed later during a single intranasal endoscopic surgery, and no major difficulties were encountered.

Despite being the most common masses in the nasal cavity (benign or malignant), nasal polyps are rare in children. Many times they are associated with systemic conditions such as cystic fibrosis (~30% of cases). (3) Therefore, in the presence of nasal polyps in children, one should request a sweat test. This was not done in our case because our patient was probably too young to have developed a polyp related to cystic fibrosis; moreover, she had a single unilateral polyp and no other suggestive symptoms.

[FIGURE 3 OMITTED]

Sinochoanal polyps represent only 4 to 6% of all nasal polyps in the adult population, but in the pediatric population, they account for 23 to 33% of nasal polyps. (3,4) Antrochoanal polyps are far more common than polyps that originate in the ethmoid and sphenoid sinuses. (5)

The angiomatous polyp diagnosed in our case is a very rare variant of a sinochoanal polyp. (6) This variant is characterized by marked vascular proliferation and a sinusoid appearance in an inflammatory stroma. The current consensus is that angiomatous polyps probably represent a secondary change in a preexisting inflammatory polyp. Such a change can be attributable to a deficient vascular supply (especially in the case of an antrochoanal polyp) or an episode of intranasal trauma or inflammation. (7) The second of these two possible causes is compatible with the circumstances of our case.

Sometimes surface necrosis and fibrosis impart to these polyps a less benign appearance. (8) In the current literature, it is not possible to determine the real incidence of this kind of polyp, but almost every published case has been reported in adults and teenagers. In our own review of the literature, we did not find any case report of an angiomatous polyp in an infant.

In conclusion, in the case of an infant who has a unilateral intranasal mass, the clinical history and imaging findings play a key role in therapeutic decision making. The possibility of an angiomatous polyp should be considered, especially in the setting of trauma, regardless of the patient's age.

References

(1.) Albert D. Nasal obstruction and rhinorrhea in infants and children. In: Kerr AG, ed. Scott Brown's Otolaryngology. 6th ed. London: Butterworth-Heinemann; 1997:6/17/1-6/17/16.

(2.) Cinnamond MJ. Congenital anomalies of the nose. In: Kerr AG, ed. Scott Brown's Otolaryngology. 6th ed. London: Butterworth-Heinemann; 1997:6/15/1-6/15/10.

(3.) Schramm VL Jr., Effron MZ. Nasal polyps in children. Laryngoscope 1980;90(9):1488-95.

(4.) Chen JM, Schloss MD, Azouz ME. Antro-choanal polyp: A 10-year retrospective study in the pediatric population with a review of the literature. J Otolarygol 1989;18(4):168-72.

(5.) Aydin O, Keskin G, Ustundag E, el al. Choanal polyps: An evaluation of 53 cases. Am J Rhinol 2007;21(2):164-8.

(6.) Ceylan A, Asal K, Celenk F, Uslu S. An angiomatous nasal polyp: A very rare variant of sinochoanal nasal polyps. B-ENT 2007;3 (3):145-7.

(7.) Batsakis JG, Sneige N. Choanal and angiomatous polyps of the sinonasal tract. Ann Otol Rhinol Laryngol 1992;101(7):623-5.

(8.) Sheahan P, Crotty PL, Hamilton S, et al. Infarcted angiomatous nasal polyps. Eur Arch Otorhinolaryngol2005;262(3):225-30.

Nuno Oliveira, MD; Nuno Trigueiros, MD; Delfim Duarte, MD; Manuel Rodrigues e Rodrigues, MD

From the Department of Otolaryngology, Hospital Pedro Hispano, Matosinhos, Portugal.

Corresponding author: Nuno Oliveira, MD, Av. Serpa Pinto 485, 10 Dto. Frente, 4450-282 Matosinhos, Portugal. Email: nunofilipeoliveira@ hotmail.com

Previous presentation: The information in this article has been updated from its original presentation as a poster at the Northern Nucleus Meeting of the Portuguese Society of Otolaryngology-Head and Neck Surgery; Nov. 21, 2008; Braga, Portugal.
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Title Annotation:ORIGINAL ARTICLE
Author:Oliveira, Nuno; Trigueiros, Nuno; Duarte, Delfim; Rodrigues, Manuel Rodrigues e.
Publication:Ear, Nose and Throat Journal
Article Type:Case study
Date:Nov 1, 2011
Words:1461
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