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Inicio Acta Otorrinolaringológica Española Silent sinus syndrome in children
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Vol. 69. Núm. 1.
Páginas 1-60 (Enero - Febrero 2018)
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Vol. 69. Núm. 1.
Páginas 1-60 (Enero - Febrero 2018)
Case study
DOI: 10.1016/j.otorri.2016.11.007
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Silent sinus syndrome in children
Síndrome del Seno Silente en Niños
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Francisco Rosa
Autor para correspondencia
franciscorosa97@gmail.com

Corresponding author.
, Rosário Figueirinhas, Jorge Oliveira, Cecília Almeida e Sousa
Department of Otolaryngology, Head and Neck Surgery, Centro Hospitalar do Porto, Portugal
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Table 1. Measurements of maximum maxillary sinus dimensions.
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Case report

A 10-year-old girl, with no relevant medical history, presented to our Hospital with complaints of headache. The patient reported no nasal congestion, nasal drainage, or facial pressure. Physical examination showed a slight facial asymmetry with hypoglobus and enophthalmos of the right eye. A Computed Tomography (CT) scan of the sinuses showed a hypoplastic right maxillary sinus with complete opacification (Fig. 1A). Downward bowing was evident in the right orbital floor. The remainder of the scan was unremarkable. A right endoscopic maxillary antrostomy was performed and intraoperative findings showed a hypoplastic right maxillary sinus with thick mucus.

Figure 1.

Coronal CT (A) preoperative and (B) 6 months after surgery. CT showed a permeable right maxillary antrostomy and well aerated right maxillary sinus.

(0,17MB).

Postoperative recovery progressed uneventfully. Six months after surgery, the facial asymmetry had improved and the CT showed a permeable right maxillary antrostomy and well aerated right maxillary sinus. The right maxillary sinus size has improved, despite continuing to be hypoplastic (Fig. 1B and Table 1).

Table 1.

Measurements of maximum maxillary sinus dimensions.

Maxillary sinus  RightRatio  LeftRatio 
  Preoperative  Postoperative    Preoperative  Postoperative   
Height (mm)  13.8  17.1  0.81  33.8  34.2  0.99 
Width (mm)  17.9  22.2  0.81  27.2  27.7  0.98 
Discussion

The silent sinus syndrome is an uncommon disease process, described as the progressive development of painless facial asymmetry, unilateral enophthalmos and hypoglobus secondary to the occupation and chronic atelectasis of the maxillary sinus without nasal or sinus symptoms.1–3 The majority of reported cases in literature describe this occurrence in adults, usually in their third to fifth decade of life.4–6 Although rare, children can also be affected.7,8

Maxillary sinuses are present at birth but do not fully developed until 15–18 years of age. The final pneumatization occurs in the inferior direction with the eruption of the upper teeth.9 The pathogeny of the syndrome is uncertain. According to the current hypotheses, the silent sinus syndrome is caused by maxillary sinus hypoventilation due to the obstruction of the osteomeatal complex.3 Progressive gas reabsorption produces negative pressure with subsequent thinning an bowing of the maxillary sinus walls, sinus volume retraction an reduction. The syndrome appears to affect males and females equally.4 It is thought that adult silent sinus begins in childhood but is only identified when the process increases in severity.7,8

The diagnosis of silent sinus syndrome was based on the clinical and radiological findings. These clinical signs are not pathognomonic and a differential diagnosis must be carried out with chronic sinusitis, osteomyelitis, malignancies, orbit traumatism, Wegener's granulomatosis and systemic disease (sclerodermia).5 Even though the suspicion is clinical, diagnosis is achieved by means of radiology. CT is the reference test and usually shows internal retraction of the sinus walls, infundibulum occupation and maxillary sinus opacification.6 The orbital floor is thinned or completely absent, and orbit contents are caudally displaced due to orbit floor depression.10

The treatment for silent sinus syndrome is surgical and involves restoration of maxillary sinus ventilation and repair of orbital floor defect.4 Endoscopic maxillary antrostomy and uncinectomy has replaced the Caldwell–Luc procedure as the treatment approach of choice.5 The orbital floor reconstruction is controversial. Little has been published about the management of silent sinus syndrome in children.6 The reduction in enophthalmos observed in adults patients suggests that children through maxillary sinus aeration could benefit from a two-stage approach that delays orbital floor augmentation. In children, due to the lateral growth of the maxillary sinus, uncinectomy may be more demanding as the uncinated process may be more medial to the lamina payracea compared to adults.7,8

We describe in this report a 10-year-old girl with silent sinus syndrome who underwent endoscopic sinus surgery for maxillary antrostomy. Before surgery, one of the main doubts of the family, was about the recovery of facial asymmetry and hypoplastic maxillary sinus after surgery. These clinical reports show that restoring aeration of the maxillary sinus allows spontaneous improvement of maxillary sinus size and improvement in facial asymmetry.

Financial support

This research received no specific grant from any funding agency, commercial or not-for-profit sectors.

Conflicts of interest

None.

References
[1]
W.W. Montgomery.
Mucocele of the maxillary sinus causing enophthalmos.
Eye Ear Nose Throat Mon, 43 (1964), pp. 41-44
[2]
C.N. Soparkar, J.R. Patrinely, M.J. Cauycong, R.A. Dailey, R.C. Kersten, P.A. Rubin, et al.
The silent sinus syndrome. A cause of spontaneous enophthalmos.
Ophthalmology, 101 (1994), pp. 772-778
[3]
A.R. Cobb, R. Murthy, G.C. Cousin, A. El-Rasheed, A. Toma, J. Uddin, et al.
Silent sinus syndrome.
Br J Oral Maxillofac Surg, 50 (2012), pp. e81-e85
[4]
M.G. Brandt, E.D. Wright.
The silent sinus syndrome is a form of chronic maxillary atelectasis: a systematic review of all reported cases.
Am J Rhinol, 22 (2008), pp. 68-73
[5]
H. Babar-Craig, H. Kayhanian, D.J. De Silva, G.E. Rose, V.J. Lund.
Spontaneous silent sinus syndrome (imploding antrum syndrome): case series of 16 patients.
Rhinology, 49 (2011), pp. 315
[6]
S.K. Wise, T.H. Wojno, J.M. DelGaudio.
Silent sinus syndrome: lack of orbital findings in early presentation.
Am J Rhinol, 21 (2007), pp. 489-494
[7]
D.L. Price, O. Friedman.
Facial asymmetry in maxillary sinus hypoplasia.
Int J Pediatr Otorhinolaryngol, 71 (2007), pp. 1627-1630
[8]
D.T. Chang, M.T. Truong.
A child with silent sinus syndrome and spontaneous improvement after sinus surgery.
Int J Pediatr Otorhinolaryngol, 78 (2014), pp. 1993-1995
[9]
W. Lawson, Z.M. Patel, F.Y. Lin.
The development and pathologic processes that influence maxillary sinus pneumatization.
Anat Rec, 291 (2008), pp. 1554-1563
[10]
L. Gómez, E. Fontán, J.C. León, J. Garrido.
Silent sinus syndrome. Clinical case.
Arch Soc Esp Oftalmol (English Ed), 89 (2014), pp. 121-123
Copyright © 2016. Elsevier España, S.L.U. and Sociedad Española de Otorrinolaringología y Cirugía de Cabeza y Cuello
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