Oral Maxillofac Surg (2010) 14:187–191 DOI 10.1007/s10006-009-0200-6
CASE REPORT
Maxillary sinus aplasia Bruno Ramos Chrcanovic & Belini Freire-Maia
Published online: 22 January 2010 # Springer-Verlag 2010
Abstract Purpose A case of unilateral aplasia is reported. Associated anatomical anomalies and review of the literature is described, and a comparison with maxillary sinus hypoplasia is made. Patient A 40-year-old male was complaining of an esthetic “defect” in his face. There was an antero-posterior depression in the left infraorbital region. Computed tomography showed absence of the left maxillary antrum, uncinate process, and maxillary infundibulum. The ipsilateral orbit was increased in volume inferiorly, and the inferior margin was immediately adjacent to the root of the inferior turbinate. The left inferior and middle turbinate was hyperplastic. The nasal septum was a little dislocated to the ipsilateral side. There was no history of antecedent sinusitis or surgery, and no evidence of systemic disease. An appositional en bloc autogenous bone graft was placed on the left infraorbital depression. Conclusions With precise computed tomography assessments, a maxillary sinus hypoplasia or aplasia can be diagnosed and distinguished from other maxillary sinus anomalies, and may help the surgeon to plan his surgical
B. R. Chrcanovic (*) Av. Raja Gabaglia, 1000/1209, Gutierrez, Belo Horizonte, MG CEP 30441-070, Brazil e-mail:
[email protected]
procedure and identify the limits of dissection accordingly. The condition can also “cause” an esthetical deficit in the infraorbital/zygomatic region, which can be resolved with an appositional graft. Keywords Maxillary sinus . Aplasia . Hypoplasia . Computed tomography
Introduction Maxillary sinus hypoplasia (MSH) is an anomaly of the paranasal sinuses occasionally encountered. Maxillary sinus aplasia (MSA) however is a rarely encountered condition [1]. These clinical entities can arise as a result of the developmental defects, trauma, surgery, systemic illness, or neoplasm [2]. The correct diagnosis is important, because the apparent opacification of the maxillary antrum on sinus radiographs can lead to an incorrect diagnosis of chronic sinusitis of maxillary antral tumor and enlargement of the ipsilateral orbital cavity can misdiagnosed as orbital masses [3], and the patient are often treated inappropriately. A case of unilateral aplasia is reported. Associated anatomical anomalies and review of the literature is described, and a comparison with maxillary sinus hypoplasia is made.
Case report B. Freire-Maia Av. do Contorno 4747/1010, Serra, Belo Horizonte, MG CEP 30110-921, Brazil e-mail:
[email protected] B. Freire-Maia Department of Oral and Maxillofacial Surgery, School of Dentistry, Pontifícia Universidade Católica De Minas Gerais, Belo Horizonte, Brazil
A 40-year-old male presented to the Oral and Maxillofacial Surgery Department at the Pontifícia Universidade Católica de Minas Gerais, Belo Horizonte, Brazil complaining of an esthetic “defect” in his face. There was an antero-posterior depression in the left infraorbital region (Fig. 1). The patient related no headaches and no occurrences of previous sinusitis.
188
Fig. 1 Right (a) and left (b) lateral view of the patient, showing the infraorbital depression in the left side
Coronal computed tomography (CT; Fig. 2) showed absence of the left maxillary antrum, uncinate process, and maxillary infundibulum. The ipsilateral orbit was increased in volume inferiorly, and the inferior margin was immediately adjacent to the root of the inferior turbinate. The left inferior and middle turbinate was hyperplastic. The nasal septum (NS) was a little dislocated to the ipsilateral side (Figs. 2 and 3). The anterior view of the tridimensional CT is shown in Fig. 4. The vertical red line compares the inferior orbital rim of both orbits, and the vertical blue line compares the most cranial position of the infrazygomatic arch. The distance between these two anatomical landmarks was
Oral Maxillofac Surg (2010) 14:187–191
Fig. 3 Axial CT. RMS right maxillary sinus, NS nasal septum, RIT right inferior turbinate, LIT right inferior turbinate, NF nasopharynge
shorter in the left side (see vertical black lines in Fig. 4). The green vertical line demarks the facial skeletal midline; there was a small left (ipsilateral) dislocation of the nasal septum. It was decided to place an appositional en bloc autogenous bone graft on the left infraorbital depression. The mentum was the harvest donor site. A stereolithographic model was made (Fig. 5), and an aluminum foil template was used (Fig. 6) in order to better dimension the donor graft. The graft was rigid fixed with titanium screws (Fig. 7). One can see in the stereolithographic model the lateral bulging of the left lateral wall of the nose (Fig. 5), as in coronal and axial CT (Figs. 2 and 3). Figure 8 shows the 4-week follow-up result (compare with Fig. 1b).
Discussion The maxillary sinus begins as an outgrowth in the lateral wall of the ethmoid area of the nasal capsule in the third
Fig. 2 Coronal CT. RMS right maxillary sinus, NS nasal septum, IT inferior turbinate, MT middle turbinate, EB ethmoid bulla, dotted line hiatus semilunaris, SBR subrabullar recess (connects with retrobullar recess posteriorly), IOC infraorbital canal, * maxillary sinus ostium, × ethmoid infundibulum ends in a blind pocket known as recessus terminalis
Fig. 4 Tridimensional CT. R right, L left (description of lines in text)
Oral Maxillofac Surg (2010) 14:187–191
189
Fig. 5 Upper view of the stereolithography model Fig. 7 Appositional en bloc autogenous bone graft
month of fetal life. It is the first sinus to appear. Resorption of surrounding tissue and growth of the maxillary pouch occur simultaneously. The maxillary cavity is identifiable at the 16th week of gestation [4]. It is unknown whether factors influencing frontal sinus development (i.e., racial differences, the degree of environmental coldness, and various systemic factors) [5] also affect maxillary sinus development. Clerico and Grabo [3] proposed that development of the maxillary sinus is disrupted between the third and fifth fetal months. In their patient, endoscopic exploration showed a small indentation in place of the maxillary sinus. Because the frontal and sphenoid sinuses of their patient were well developed, they concluded that either there had been a temporary intrauterine insult or the disruption was site-specific to the maxillary sinus. Clerico and Grabo [3] and Aydinlioğlu and Erdem [6] observed a depression in place of the maxillary sinus, the same occurring in our case (Fig. 1). Because of this esthetic defect caused by the depression, the patient decided to undergo this surgery for bone graft. The developmental basis of MSH/MSA remains unclear. It has been suggested that an abnormal formation of the
uncinate process may lead to impaired development of the maxillary sinus [7]. Other observers, noting that both the maxillary sinus and uncinate process have a common origin from the cartilaginous nasal capsule, postulate an abnormality of the cartilaginous capsule. This results in abnormal development of the entire lateral nasal wall, accounting for the high frequency of associated lateral nasal wall structures, such as the uncinate, middle turbinate, and ethmoid infundibulum abnormalities seen with MSH [8]. The proposed mechanisms include activation of osteogenesis within the maxilla and the removal of pneumatization centers, caused by sinus inflammation, combined with the effect of surgical trauma. It can be hypothesized, however, that any local or systemic disease that causes MSH (Treacher-Collins syndrome, Crouzon’s syndrome, Apert’s syndrome, thalassemia, Paget’s disease, and cretinism) may also cause MSA [2]. In the case described in this report, there was no history of antecedent sinusitis or surgery, and no evidence of systemic disease. It would seem, therefore, Fig. 8 Final esthetic result
Fig. 6 Anterior view of the stereolithography model, with the aluminum foil template
190
that in our case a primary developmental anomaly is the most likely etiology. MSA is a very rare congenital anomaly. MSA was first reported in 1723 by Morgagni [9]. Only a few cases have been reported in the literature [1, 3, 6, 10–17]. In the present case, the main diagnostic obstacle was to differentiate sinus aplasia from severe hypoplasia. Although maxillary hypoplasia is relatively common, thin slices may be obtained to rule out a severely hypoplastic sinus cavity and a rudimentary infundibular passage into the nasal cavity [18], which were not evident in the present case. In a case of MSH, thin slices also help in better identification of the uncinate process abnormality. Geraghty and Dolan [19] have described diagnostic criteria for the MSH and they are: (1) enlargement of vertical orbit; (2) lateral position of infraorbital neurovascular canal; (3) elevated canine fossa; (4) enlargement of superior orbital fissure; and (5) enlargement of pterygopalatine fissure. Some coexisting anatomic variations were observed in another study [20]: middle conchal pneumatization was the most common coexisting anatomic anomaly, followed by agger nasi cell; secondary middle concha, paradoxical middle concha, and upper conchal pneumatization; and rudimentary frontal sinus and sphenoidal sinus hypoplasia. The diameters of the infraorbital fissures were similar on both sides. An enlargement was not encountered in the pterygopalatine or infratemporal fossa. In all cases of uncinate process hypoplasia or aplasia, MSH was present. Uncinate process hypoplasia is closely associated with MSH [2, 7, 20]. As might be expected, the uncinate process is also completely absent in cases of MSA [3, 6]. The left uncinate process was not identified in any coronal or axial CT slice in the present case, which confirmed the left MSA situation. In the event of MSH without an uncinate process, the maxillary sinus loses its aeration and shows soft tissue opacification. The maxillary sinus is ventilated normally during the expiratory phase of respiration. For this to occur, an uncinate process may be necessary in order to direct the expired air toward the sinus cavity. Possibly, in the absence of the uncinate process, this mechanism becomes disrupted which will lead to a maxillary sinus filled with soft tissue rather than air [20]. A silent sinus syndrome that presents with enophthalmos and hypoglobus should also be considered in the case of MSH. Therefore, not only the otolaryngologist or radiologist, but also the ophthalmologist, should keep in mind the differential diagnosis of MSH in a patient presenting with enophtalmos [20]. An association between enophtalmos and MSH has been reported [21]. Moreover, an association between MSH and orbital enlargement was also previously shown. As the maxillary sinus becomes more hypoplastic, the orbital cavity size increases and displaces inferiorly
Oral Maxillofac Surg (2010) 14:187–191
[20]. This can be easily noted in the present case (Figs. 2 and 4). MSA may also cause the lateral wall of the nose to bulge laterally [22], which also occurred in the present case (Figs. 2, 3, and 5). Another variation noted was the ipsilateral dislocation of the nasal septum (Fig. 4). An incorrect diagnosis of MSH should be avoided when there is an ethmomaxillary sinus due to over-expansion of a posterior ethmoidal air-cell into the maxillary sinus, and is characterized by drainage into an enlarged superior meatus, reduction in the size of the maxillary sinus, and a normal bony orbital cavity [23]. It is also important to distinguish MSH from a chronic maxillary atelectasis (CMA), which is characterized by a persistent decrease in maxillary sinus volume due to inward bowing of one or more antral walls. A laterally malpositioned medial infundibular wall can exist, but there is no evidence that untreated CMA will lead to orbital deformity [24]. A hypoplastic maxillary sinus is more prone to infection [18]. This might be related to the aberrant anatomy of the ipsilateral uncinate process, which might impede mucociliary clearance of the sinuses. As MSA has no antral space, it cannot become infected [7]. In MSH-associated sinusitis, the membranous lateral nasal wall of the posterior fontanelle is actually displaced laterally, as if sucked into the maxillary sinus [7] and should alert the clinician to the possibility that there may be underlying MSH. Furthermore, the laterally bowed posterior fontanelle serves as a landmark at operation, as it marks the preferred site of antrostomy formation. In the case reported here, the uncinate process could not be identified and the inferomedial wall of the orbit was virtually touching the inferior turbinate. A posterior fontanelle antrostomy is not normally recommended in sinus surgery, as it may lead to futile mucus cycling between the surgical antrostomy opening and the normal, more anteriorly placed, ostium [25]. However, in MSH-associated sinusitis, there is no natural anterior ostium and this complication cannot occur. Associated anomaly of the uncinate process, a critical landmark in functional endoscopic sinus surgery, can impact on dissection at the initial phase of surgery, which may subsequently lead to navigation problems within the ostiomeatal complex. Additionally, there may be difficulty in finding the ostium of the maxillary sinus. Preoperative knowledge of the anatomic variations helps the surgeon to plan his procedure and identify the limits of dissection accordingly [17, 20]. Unilateral opacified maxillary antrum on plain films may be due to mucosal thickening in infectious diseases and tumoral involvement, atelectasis of the sinus due to chronic sinusitis, and severe hypoplasia or aplasia of the sinus [3]. Routine clinical use of paranasal sinus CT scans have lead to a better understanding of the anatomy and variations of
Oral Maxillofac Surg (2010) 14:187–191
the paranasal sinuses, which, in turn, facilitated the precise diagnosis and treatment of sinus disease and decreased the complication rates. It was not easy to diagnose the anomaly prior to the use of paranasal CT scans.
Conclusions Knowledge of an association between the unilateral absence of the maxillary sinus and an ipsilateral lower infraorbital margin is important during endoscopic surgery of the sinus. Great care must be taken to avoid injury to the orbital floor [18, 20]. Recognition of associated anomalies of the uncinate process in patients with MSH or its absence in patients with MSA undergoing sinus surgery is of the utmost clinical significance because the uncinate process serves as a key landmark during functional endoscopic sinus surgery [7]. Failure to recognize hypoplasia or absence of the uncinate process could lead to inadvertent intraoperative damage to the adjacent medial orbital wall [7]. With precise CT assessments, a MSH or MSA can be diagnosed and distinguished from other maxillary sinus anomalies, and may help the surgeon to plan his surgical procedure and identify the limits of dissection accordingly [17]. MSH/MSA can also “cause” an esthetical deficit in the infraorbital/zygomatic region, which can be resolved with an appositional graft.
References 1. Baykara M, Erdoğan N, Öztürk M, Erkan M (2002) Maxillary sinus aplasia. Turk J Med Sci 32:273–275 2. Erdem T, Aktas D, Erdem G, Miman MC, Ozturan O (2002) Maxillary sinus hypoplasia. Rhinology 40:150–153 3. Clerico DM, Grabo DJ (1997) Maxillary sinus aplasia. Ann Otol Rhinol Laryngol 106:799–800 4. Kosko JR, Hall BE, Tunkel DE (1996) Acquired maxillary sinus hypoplasia. A consequence of endoscopic sinus surgery? Laryngoscope 106:1210–1213 5. Aydinlioğlu A, Kavakli A, Erdem S (2003) Absence of frontal sinus in Turkish individuals. Yonsei Med J 44:215–218 6. Aydinlioğlu A, Erdem S (2004) Maxillary and sphenoid sinus aplasia in Turkish individuals: a retrospective review using computed tomography. Clin Anat 17:618–622
191 7. Bolger WE, Woodruff WW, Morehead J, Parsons DS (1990) Maxillary sinus hypoplasia: classification and description of associated uncinate process hypoplasia. Otolaryngol Head Neck Surg 103:759–765 8. Wang RG, Jiang SC, Gu R (1994) The cartilaginous nasal capsule and embryonic development of human paranasal sinuses. J Otolaryngol 23:239–243 9. Blanton PL, Biggs NL (1969) Eighteen hundred years of controversy: the paranasal sinuses. Am J Anat 124:135–148 10. Traserra J (1967) Maxillary sinus aplasia. Acta Otorinolaringol Iberam 18:458–471 11. Gouzy J, Voilque G, Jacubowicz B (1968) Two cases of agenesis of the maxillary sinus. J Fr Otorhinolaryngol Audiophonol Chir Maxillofac 17:759–764 12. Mocellin L (1968) Panagenesis of the paranasal sinuses: report of a case. Arch Otolayngol 83:311 13. Kuźniar A, Wolczyk C (1971) Case of maxillary sinus agenesis. Vestn Otorinolaringol 25:463–465 14. Khechinashvili SN, Kitovani LG (1979) Case of the absence of the maxillary sinus. Vestn Otorinolaringol 4:71–72 15. Rydzewski B, Lukowiak G (1996) A case of congenital unilateral absence of maxillary and frontal sinuses. Otolaryngol Pol 50:445–450 16. Tsue TT, Bailet JW, Barlow DW, Makielski KH (1997) Bilateral sinonasal papillomas in aplastic maxillary sinuses. Am J Otolaryngol 18:263–268 17. Tasar M, Cankal F, Bozlar U, Hidir Y, Saglam M, Ors F (2007) Bilateral maxillary sinus hypoplasia and aplasia: radiological and clinical findings. Dentomaxillofac Radiol 36:412–415 18. Milczuk HA, Dalley RW, Wessbacher FW, Richardson MA (1993) Nasal and paranasal sinus anomalies in children with chronic sinusitis. Laryngoscope 103:247–252 19. Geraghty JJ, Dolan KD (1989) Computed tomography of the hypoplastic maxillary sinus. Ann Otol Rhinol Laryngol 98:916– 918 20. Sirikçi A, Bayazit Y, Gümüşburun E, Bayram M, Kanlikana M (2000) A new approach to the classification of maxillary sinus aplasia with relevant clinical implications. Surg Radiol Anat 22:243–247 21. Michielsens A, Herzeel R, Gordts F (1999) Acquired enophthalmos associated with hypoplasia of the maxillary sinus and asymptomatic chronic maxillary sinusitis. J Fr Ophtalmol 22:451–455 22. Kapoor PK, Kumar BN, Watson SD (2002) Maxillary sinus hypoplasia. J Laryngol Otol 116:135–137 23. Khanobthamchai K, Shankar L, Hawke M, Bingham B (1991) Ethmomaxillary sinus and hypoplasia of maxillary sinus. J Otolaryngol 20:425–427 24. Kass ES, Salman S (1999) Developmental changes in the antrum of a child with chronic maxillary atelectasis. Ann Otol Rhinol Laryngol 108:1091–1094 25. Parsons DS, Stivers FE, Talbot AR (1996) The missed ostium sequence and the surgical approach in functional endoscopic sinus surgery. Otolaryngol Clin North Am 29:169–183
