International Journal of Pediatric Otorhinolaryngology 78 (2014) 1993–1995
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case Report
A child with silent sinus syndrome and spontaneous improvement after sinus surgery David T. Chang a, Mai Thy Truong a,b,* a b
Department of Otolaryngology – Head and Neck Surgery, Stanford University, 801 Welch Road, Stanford 94305, United States Department of ENT – Head and Neck Surgery, Kaiser Santa Clara, 710 Lawrence Expressway, Department 296, Santa Clara, CA 95051, United States
A R T I C L E I N F O
A B S T R A C T
Article history: Received 13 January 2014 Received in revised form 30 July 2014 Accepted 1 August 2014 Available online 10 August 2014
Silent sinus syndrome is characterized by an asymptomatic hypoplastic maxillary sinus with progressive enophthalmos and hypoglobus. This is a disease rarely affecting children with the majority of reported cases involving adult patients. Treatment is primarily surgical with endoscopic sinus surgery to restore aeration of the sinus along with orbital reconstruction to restore facial symmetry. In this report, we describe a 7 year old child with facial asymmetry and no sinonasal symptoms. CT showed an opacified hypoplastic right maxillary sinus. One year after endoscopic sinus surgery, there was spontaneous improvement of facial asymmetry and relative maxillary sinus size. ß 2014 Elsevier Ireland Ltd. All rights reserved.
Keywords: Silent sinus syndrome Child Enophthalmos Hypoglobus Endoscopic sinus surgery
1. Introduction Silent sinus syndrome describes an uncommon disease process characterized by progressive enophthalmos and hypoglobus associated with a hypoplastic maxillary sinus, and an absence of sinonasal complaints. Hypoventilation resulting from obstruction of the maxillary sinus ostiomeatal complex leads to negative pressure within the maxillary sinus and subsequent atelectasis and wall collapse [1,2]. The majority of reported cases in the literature describe this occurrence in adults [1–5]. Occurrences in children are even more rare but have been reported [5]. Babar-Craig et al. reported a case series with age of patients as young as 14 and up to 79 years, with a mean age of 40.6 years [5]. Wise et al. reported a case series of patients ranging from 11 to 70 years old, with a mean of 43.1 years [6]. The treatment of silent sinus syndrome is surgical, with the primary goal of restoring aeration and drainage of the maxillary sinus to prevent progression of the disease process. Spontaneous improvement of enophthalmos has been described [5,7–9].
* Corresponding author at: Department of ENT – Head and Neck Surgery, Kaiser Santa Clara, 710 Lawrence Expressway, Department 296, Santa Clara, CA 95051, United States. Tel.: +1 650 721 6962; fax: +1 650 721 2163. E-mail address: [email protected] (M.T. Truong). http://dx.doi.org/10.1016/j.ijporl.2014.08.002 0165-5876/ß 2014 Elsevier Ireland Ltd. All rights reserved.
However, these reports are limited in number and describe only adult patients. In this report, we describe a 7 year old child with silent sinus syndrome who experienced spontaneous improvement after surgical management. 2. Case report A 7 year old healthy girl presented to our clinic with complaints of headache. She reported no nasal congestion, nasal drainage, or facial pressure. History was significant for symptoms of obstructive sleep apnea including poor sleep, night time wakening, snoring, daytime tiredness, and mouth breathing. Physical examination showed facial asymmetry with hypoglobus and enophthalmos. She was found to have 3+ tonsillar hypertrophy. An MRI done for the work up of her headaches by primary care incidentally found a hypoplastic maxillary sinus. Computed tomography (CT) showed a hypoplastic right maxillary sinus with complete opacification (Fig. 1A). There was also bowing of the medial wall of the right maxillary sinus. The rest of the paranasal sinuses were well developed and well aerated. After discussion of the risks, benefits, and alternatives to right functional endoscopic sinus surgery, tonsillectomy, and adenoidectomy, the family elected to proceed. A right maxillary antrostomy was performed and intraoperative findings showed a hypoplastic right maxillary sinus with inspissated mucus.
D.T. Chang, M.T. Truong / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1993–1995
1994
Fig. 1. (A) Preoperative and (B) postoperative coronal CT illustrating the improved aeration and size after endoscopic right maxillary antrostomy.
Postoperatively, the patient did well. After 1 year, the facial asymmetry had improved. A CT obtained 1 year after surgery showed a patent right maxillary antrostomy and well aerated right maxillary sinus. Although the right maxillary sinus continued to be hypoplastic, the size appeared to have improved (Fig. 1B). Otherwise, paranasal sinuses were normally aerated. In order to determine whether the endoscopic right maxillary antrostomy had any effect on the postoperative size of the hypoplastic right maxillary sinus, the height and width of bilateral maxillary sinuses was measured and analyzed. The maximum sizes are listed in Table 1. Preoperatively, the greatest height and width of the normal left maxillary sinus was 21.5 mm and 22.5 cm, respectively, while the right maxillary sinus was 13.2 mm and 10.7 mm, respectively. The maximum height and width of the right hypoplastic maxillary sinus was 61% and 48% of the left normal maxillary sinus, respectively. Postoperatively, the greatest height and width of the left maxillary sinus was 21.9 mm and 22.3 mm, respectively, while the right maxillary sinus was 16.8 mm and 12.9 mm, respectively. The height of the right hypoplastic maxillary sinus had increased 3.6 mm and the width had increased 2.2 mm. This corresponded to 77% and 58% of the height and width of the left normal maxillary sinus, which is an improvement in relative size. In contrast, the maximum dimensions of the nondiseased left maxillary sinus remained the same over the same 1 year time period.
3. Discussion Silent sinus syndrome is an uncommon disease process generally considered to affect adults. Patients usually present in their third to fifth decade of life [1–5]. Although rare, younger patients can also be affected. Babar-Craig et al. reported a case series including a 14 year old patient affected; while Wise et al. reported a case series of patients with an 11 year old with silent sinus syndrome [5,6]. We describe in this report a 7 year old with silent sinus syndrome.
The maxillary sinus begins its development prenatally after the 10th week of gestation [10]. Initially, the sinus is fluid-filled, but becomes pneumatized at birth. Further pneumatization laterally and inferiorly occurs into early adulthood in a biphasic growth pattern [11,12]. Occlusion of the ostiomeatal complex during development may interfere in this natural process. Perhaps adult silent sinus syndrome actually begins in childhood but is not identified until the process has increased in severity. This young patient was a case in which it was fortunately found early. The treatment for silent sinus syndrome is surgical and involves restoring the aeration and drainage pathway for the affected sinus. In the pediatric population, treatment of silent sinus syndrome may be more challenging depending on the child’s age and the overall pneumatization of the sinuses. Because of the lateral growth of the maxillary sinus, uncinectomy in children may be more difficult as the uncinate process may be more medial to the lamina papyracea compared to adults [13]. In the preoperative period, there was extensive discussion with the parents in regards to surgical management with endoscopic antrostomy, including the risks, benefits, and alternatives. One of the primary questions the family had was whether the facial asymmetry and the hypoplastic right maxillary sinus would improve after surgery. This was difficult to answer given the paucity of evidence in the literature. For years, the management of silent sinus syndrome involved endoscopic antrostomy with concurrent orbital reconstruction of the orbital floor to address the enophthalmos. Although limited in the number of patients, there have been reports showing spontaneous improvement in enophthalmos and hypoglobus after only the endoscopic antrostomy with no need for orbital floor reconstruction [7,8]. Sivasubramaniam et al. performed a restrospective review of 23 patient with silent sinus syndrome and described postoperative changes in the orbital floor via nasal endoscopy and improvement in enophthalmos over time after endoscopic uncinectomy and antrostomy [9]. However, these have all been reported in patients older than 18 years of age. Our 7 year old girl with right silent sinus syndrome underwent endoscopic sinus surgery for a right maxillary antrostomy. She
Table 1 Measurements of maximum maxillary sinus dimensions. Maxillary sinus
Maximum height (mm)
Maximum width (mm)
Ratio of maximum right to left sinus height
Ratio of maximum right to left sinus width
Preoperative Left Right
21.5 13.2
22.5 10.7
0.61
0.48
Postoperative Left Right
21.9 16.8
22.3 12.9
0.77
0.58
D.T. Chang, M.T. Truong / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1993–1995
showed spontaneous clinical improvement postoperatively in her facial asymmetry. Her CT one year after surgery showed 3.6 mm increase in height and 2.2 mm increase in width of her hypoplastic maxillary sinus, corresponding to an improvement in the asymmetry in the height and width of the maxillary sinus. This spontaneous improvement illustrated in this patient can help with future discussions of silent sinus syndrome and allow better education of patients and families. 4. Conclusions Silent sinus syndrome generally affects adults. As we have shown, young children can also be affected. Restoring aeration of the maxillary sinus can allow for spontaneous improvement of maxillary sinus size and improvement in facial asymmetry. Conflict of interest None. Financial disclosure None. Acknowledgement We thank Dr. Peter Hwang for helpful productive discussions.
1995
References [1] D.J. Annino Jr., L.A. Goguen, Silent sinus syndrome, Curr. Opin. Otolaryngol. Head Neck Surg. 16 (February (1)) (2008) 22–25. [2] 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 (September (6)) (2012) e81–e85. [3] C.N. Soparkar, J.R. Patrinely, M.J. Cuaycong, R.A. Dailey, R.C. Kersten, P.A. Rubin, et al., The silent sinus syndrome. A cause of spontaneous enophthalmos, Ophthalmology 101 (April (4)) (1994) 772–778. [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 (January– February (1)) (2008) 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 (August (3)) (2011) 315–317. [6] S.K. Wise, T.H. Wojno, J.M. DelGaudio, Silent sinus syndrome: lack of orbital findings in early presentation, Am. J. Rhinol. 21 (July–August (4)) (2007) 489–494. [7] M.K. Wan, I.C. Francis, P.R. Carter, R. Griffits, M.L. van Rooijen, M.T. Coroneo, The spectrum of presentation of silent sinus syndrome, J. Neuroophthalmol. 20 (September (3)) (2000) 207–212. [8] R.D. Thomas, S.M. Graham, K.D. Carter, J.A. Nerad, Management of the orbital floor in silent sinus syndrome, Am. J. Rhinol. 17 (March–April (2)) (2003) 97–100. [9] R. Sivasubramaniam, R. Sacks, M. Thornton, Silent sinus syndrome: dynamic changes in the position of the orbital floor after restoration of normal sinus pressure, J. Laryngol. Otol. 125 (December (12)) (2011) 1239–1243. [10] A. Nunez-Castruita, N. Lopez-Serna, S. Guzman-Lopez, Prenatal development of the maxillary sinus: a perspective for paranasal sinus surgery, Otolaryngology 146 (June (6)) (2012) 997–1003. [11] J.M. Sanchez Fernandez, J.A. Anta Escuredo, A. Sanchez Del Rey, F. Santaolalla Montoya, Morphometric study of the paranasal sinuses in normal and pathological conditions, Acta Otolaryngol. (Stockh.) 120 (March (2)) (2000) 273–278. [12] W. Lawson, Z.M. Patel, F.Y. Lin, The development and pathologic processes that influence maxillary sinus pneumatization, Anat. Rec. 291 (November (11)) (2008) 1554–1563. [13] A.J. Scuderi, H.R. Harnsberger, R.S. Boyer, Pneumatization of the paranasal sinuses: normal features of importance to the accurate interpretation of CT scans and MR images, AJR Am. J. Roentgenol. 160 (May (5)) (1993) 1101–1104.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case Report
A child with silent sinus syndrome and spontaneous improvement after sinus surgery David T. Chang a, Mai Thy Truong a,b,* a b
Department of Otolaryngology – Head and Neck Surgery, Stanford University, 801 Welch Road, Stanford 94305, United States Department of ENT – Head and Neck Surgery, Kaiser Santa Clara, 710 Lawrence Expressway, Department 296, Santa Clara, CA 95051, United States
A R T I C L E I N F O
A B S T R A C T
Article history: Received 13 January 2014 Received in revised form 30 July 2014 Accepted 1 August 2014 Available online 10 August 2014
Silent sinus syndrome is characterized by an asymptomatic hypoplastic maxillary sinus with progressive enophthalmos and hypoglobus. This is a disease rarely affecting children with the majority of reported cases involving adult patients. Treatment is primarily surgical with endoscopic sinus surgery to restore aeration of the sinus along with orbital reconstruction to restore facial symmetry. In this report, we describe a 7 year old child with facial asymmetry and no sinonasal symptoms. CT showed an opacified hypoplastic right maxillary sinus. One year after endoscopic sinus surgery, there was spontaneous improvement of facial asymmetry and relative maxillary sinus size. ß 2014 Elsevier Ireland Ltd. All rights reserved.
Keywords: Silent sinus syndrome Child Enophthalmos Hypoglobus Endoscopic sinus surgery
1. Introduction Silent sinus syndrome describes an uncommon disease process characterized by progressive enophthalmos and hypoglobus associated with a hypoplastic maxillary sinus, and an absence of sinonasal complaints. Hypoventilation resulting from obstruction of the maxillary sinus ostiomeatal complex leads to negative pressure within the maxillary sinus and subsequent atelectasis and wall collapse [1,2]. The majority of reported cases in the literature describe this occurrence in adults [1–5]. Occurrences in children are even more rare but have been reported [5]. Babar-Craig et al. reported a case series with age of patients as young as 14 and up to 79 years, with a mean age of 40.6 years [5]. Wise et al. reported a case series of patients ranging from 11 to 70 years old, with a mean of 43.1 years [6]. The treatment of silent sinus syndrome is surgical, with the primary goal of restoring aeration and drainage of the maxillary sinus to prevent progression of the disease process. Spontaneous improvement of enophthalmos has been described [5,7–9].
* Corresponding author at: Department of ENT – Head and Neck Surgery, Kaiser Santa Clara, 710 Lawrence Expressway, Department 296, Santa Clara, CA 95051, United States. Tel.: +1 650 721 6962; fax: +1 650 721 2163. E-mail address: [email protected] (M.T. Truong). http://dx.doi.org/10.1016/j.ijporl.2014.08.002 0165-5876/ß 2014 Elsevier Ireland Ltd. All rights reserved.
However, these reports are limited in number and describe only adult patients. In this report, we describe a 7 year old child with silent sinus syndrome who experienced spontaneous improvement after surgical management. 2. Case report A 7 year old healthy girl presented to our clinic with complaints of headache. She reported no nasal congestion, nasal drainage, or facial pressure. History was significant for symptoms of obstructive sleep apnea including poor sleep, night time wakening, snoring, daytime tiredness, and mouth breathing. Physical examination showed facial asymmetry with hypoglobus and enophthalmos. She was found to have 3+ tonsillar hypertrophy. An MRI done for the work up of her headaches by primary care incidentally found a hypoplastic maxillary sinus. Computed tomography (CT) showed a hypoplastic right maxillary sinus with complete opacification (Fig. 1A). There was also bowing of the medial wall of the right maxillary sinus. The rest of the paranasal sinuses were well developed and well aerated. After discussion of the risks, benefits, and alternatives to right functional endoscopic sinus surgery, tonsillectomy, and adenoidectomy, the family elected to proceed. A right maxillary antrostomy was performed and intraoperative findings showed a hypoplastic right maxillary sinus with inspissated mucus.
D.T. Chang, M.T. Truong / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1993–1995
1994
Fig. 1. (A) Preoperative and (B) postoperative coronal CT illustrating the improved aeration and size after endoscopic right maxillary antrostomy.
Postoperatively, the patient did well. After 1 year, the facial asymmetry had improved. A CT obtained 1 year after surgery showed a patent right maxillary antrostomy and well aerated right maxillary sinus. Although the right maxillary sinus continued to be hypoplastic, the size appeared to have improved (Fig. 1B). Otherwise, paranasal sinuses were normally aerated. In order to determine whether the endoscopic right maxillary antrostomy had any effect on the postoperative size of the hypoplastic right maxillary sinus, the height and width of bilateral maxillary sinuses was measured and analyzed. The maximum sizes are listed in Table 1. Preoperatively, the greatest height and width of the normal left maxillary sinus was 21.5 mm and 22.5 cm, respectively, while the right maxillary sinus was 13.2 mm and 10.7 mm, respectively. The maximum height and width of the right hypoplastic maxillary sinus was 61% and 48% of the left normal maxillary sinus, respectively. Postoperatively, the greatest height and width of the left maxillary sinus was 21.9 mm and 22.3 mm, respectively, while the right maxillary sinus was 16.8 mm and 12.9 mm, respectively. The height of the right hypoplastic maxillary sinus had increased 3.6 mm and the width had increased 2.2 mm. This corresponded to 77% and 58% of the height and width of the left normal maxillary sinus, which is an improvement in relative size. In contrast, the maximum dimensions of the nondiseased left maxillary sinus remained the same over the same 1 year time period.
3. Discussion Silent sinus syndrome is an uncommon disease process generally considered to affect adults. Patients usually present in their third to fifth decade of life [1–5]. Although rare, younger patients can also be affected. Babar-Craig et al. reported a case series including a 14 year old patient affected; while Wise et al. reported a case series of patients with an 11 year old with silent sinus syndrome [5,6]. We describe in this report a 7 year old with silent sinus syndrome.
The maxillary sinus begins its development prenatally after the 10th week of gestation [10]. Initially, the sinus is fluid-filled, but becomes pneumatized at birth. Further pneumatization laterally and inferiorly occurs into early adulthood in a biphasic growth pattern [11,12]. Occlusion of the ostiomeatal complex during development may interfere in this natural process. Perhaps adult silent sinus syndrome actually begins in childhood but is not identified until the process has increased in severity. This young patient was a case in which it was fortunately found early. The treatment for silent sinus syndrome is surgical and involves restoring the aeration and drainage pathway for the affected sinus. In the pediatric population, treatment of silent sinus syndrome may be more challenging depending on the child’s age and the overall pneumatization of the sinuses. Because of the lateral growth of the maxillary sinus, uncinectomy in children may be more difficult as the uncinate process may be more medial to the lamina papyracea compared to adults [13]. In the preoperative period, there was extensive discussion with the parents in regards to surgical management with endoscopic antrostomy, including the risks, benefits, and alternatives. One of the primary questions the family had was whether the facial asymmetry and the hypoplastic right maxillary sinus would improve after surgery. This was difficult to answer given the paucity of evidence in the literature. For years, the management of silent sinus syndrome involved endoscopic antrostomy with concurrent orbital reconstruction of the orbital floor to address the enophthalmos. Although limited in the number of patients, there have been reports showing spontaneous improvement in enophthalmos and hypoglobus after only the endoscopic antrostomy with no need for orbital floor reconstruction [7,8]. Sivasubramaniam et al. performed a restrospective review of 23 patient with silent sinus syndrome and described postoperative changes in the orbital floor via nasal endoscopy and improvement in enophthalmos over time after endoscopic uncinectomy and antrostomy [9]. However, these have all been reported in patients older than 18 years of age. Our 7 year old girl with right silent sinus syndrome underwent endoscopic sinus surgery for a right maxillary antrostomy. She
Table 1 Measurements of maximum maxillary sinus dimensions. Maxillary sinus
Maximum height (mm)
Maximum width (mm)
Ratio of maximum right to left sinus height
Ratio of maximum right to left sinus width
Preoperative Left Right
21.5 13.2
22.5 10.7
0.61
0.48
Postoperative Left Right
21.9 16.8
22.3 12.9
0.77
0.58
D.T. Chang, M.T. Truong / International Journal of Pediatric Otorhinolaryngology 78 (2014) 1993–1995
showed spontaneous clinical improvement postoperatively in her facial asymmetry. Her CT one year after surgery showed 3.6 mm increase in height and 2.2 mm increase in width of her hypoplastic maxillary sinus, corresponding to an improvement in the asymmetry in the height and width of the maxillary sinus. This spontaneous improvement illustrated in this patient can help with future discussions of silent sinus syndrome and allow better education of patients and families. 4. Conclusions Silent sinus syndrome generally affects adults. As we have shown, young children can also be affected. Restoring aeration of the maxillary sinus can allow for spontaneous improvement of maxillary sinus size and improvement in facial asymmetry. Conflict of interest None. Financial disclosure None. Acknowledgement We thank Dr. Peter Hwang for helpful productive discussions.
1995
References [1] D.J. Annino Jr., L.A. Goguen, Silent sinus syndrome, Curr. Opin. Otolaryngol. Head Neck Surg. 16 (February (1)) (2008) 22–25. [2] 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 (September (6)) (2012) e81–e85. [3] C.N. Soparkar, J.R. Patrinely, M.J. Cuaycong, R.A. Dailey, R.C. Kersten, P.A. Rubin, et al., The silent sinus syndrome. A cause of spontaneous enophthalmos, Ophthalmology 101 (April (4)) (1994) 772–778. [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 (January– February (1)) (2008) 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 (August (3)) (2011) 315–317. [6] S.K. Wise, T.H. Wojno, J.M. DelGaudio, Silent sinus syndrome: lack of orbital findings in early presentation, Am. J. Rhinol. 21 (July–August (4)) (2007) 489–494. [7] M.K. Wan, I.C. Francis, P.R. Carter, R. Griffits, M.L. van Rooijen, M.T. Coroneo, The spectrum of presentation of silent sinus syndrome, J. Neuroophthalmol. 20 (September (3)) (2000) 207–212. [8] R.D. Thomas, S.M. Graham, K.D. Carter, J.A. Nerad, Management of the orbital floor in silent sinus syndrome, Am. J. Rhinol. 17 (March–April (2)) (2003) 97–100. [9] R. Sivasubramaniam, R. Sacks, M. Thornton, Silent sinus syndrome: dynamic changes in the position of the orbital floor after restoration of normal sinus pressure, J. Laryngol. Otol. 125 (December (12)) (2011) 1239–1243. [10] A. Nunez-Castruita, N. Lopez-Serna, S. Guzman-Lopez, Prenatal development of the maxillary sinus: a perspective for paranasal sinus surgery, Otolaryngology 146 (June (6)) (2012) 997–1003. [11] J.M. Sanchez Fernandez, J.A. Anta Escuredo, A. Sanchez Del Rey, F. Santaolalla Montoya, Morphometric study of the paranasal sinuses in normal and pathological conditions, Acta Otolaryngol. (Stockh.) 120 (March (2)) (2000) 273–278. [12] W. Lawson, Z.M. Patel, F.Y. Lin, The development and pathologic processes that influence maxillary sinus pneumatization, Anat. Rec. 291 (November (11)) (2008) 1554–1563. [13] A.J. Scuderi, H.R. Harnsberger, R.S. Boyer, Pneumatization of the paranasal sinuses: normal features of importance to the accurate interpretation of CT scans and MR images, AJR Am. J. Roentgenol. 160 (May (5)) (1993) 1101–1104.
