case_images: PMC5512096_01
case_images: Summary of the clinical examination at the time the patient presented at our hospital. (a) Facial appearance: the arrows in the right and left pictures indicate the superior sulcus. The right superior sulcus appears to be deeper than the left superior sulcus. (b) Endoscopic findings: the right middle meatus is more enlarged than the left middle meatus. (c) Axial and coronal views on computed tomography. (d) T1 and T2 weighted images on magnetic resonance imaging. The T1 axial image shows that the right medial wall of the maxillary sinus (shown by the arrow) deviates laterally. The T2 coronal image indicates inferior bowing of the inferior wall of the orbit (shown by the arrow). The T2 sagittal images show prominent deviation of the posterior wall in the right maxillary sinus (shown by the arrow in the right image) relative to the structure in the left maxillary sinus (left image). The red and blue lines in the T2 axial image indicate the distance from the center line to the most deviated medial wall of the maxillary sinus.
case_images: CRIOT2017-4296195.001.jpg
case_images: PMC5512096_01_CRIOT2017-4296195.001.jpg
case_images: fig1
case_images: Examination of the facial appearance of the patient indicated more deepening of the right upper eyelid sulcus than the left eyelid sulcus (Figure 1(a)). An endoscopic examination showed that the right uncinate process could not be clearly detected and seemed to adhere to the medial wall of the maxillary sinus (Figure 1(b)). Computed tomography (CT) imaging then revealed inferior bowing of the floor of the orbit into the right maxillary sinus, lateral drifting of the right uncinate process into close contact with the floor of the orbit, and partial opacification of the maxillary sinus and anterior ethmoidal cells (Figure 1(c)). Magnetic resonance imaging (MRI) findings confirmed prolapse of the inferior inward retraction of the posterior, lateral, and medial walls of the maxillary sinus on the right side (Figure 1(d)).
case_images: Magnetic resonance images taken 3 and 2 years before the development of ocular manifestations. (a) Magnetic resonance image (MRI) findings: (A) the T2 axial view of MRI 3 years before the ocular presentation; (B) the T2 axial view of MRI 2 years before the ocular presentation. Both images show that the medial wall of the right maxillary sinus is deviated compared to the medial wall of the left maxillary sinus. Similar anatomical changes are observed. Thus, 3 and 2 years before ocular manifestations appeared, the distances from the center line to the most deviated medial wall of the right maxillary sinus were 19 and 19 mm (red lines), respectively. By contrast, the distances from the center line to the most deviated medial wall of the left (unaffected) maxillary sinus were 11.5 and 11 mm (blue lines), respectively. Both situations indicate stage II disease. (b) Time course of the CMA patient. The first MRI was performed 3 years before ocular symptom presentation (A). The second MRI was performed 2 years before ocular symptom presentation (B). The patient did not present with any sinusitis-related symptoms before or at the first (A) and the second (B) MRI. However, in the 2 years following the second MRI, the patient frequently presented with sinus-related symptoms. At the end of that period, the patient was diagnosed with stage III disease.
case_images: CRIOT2017-4296195.002.jpg
case_images: PMC5512096_01_CRIOT2017-4296195.002.jpg
case_images: fig2
case_images: As shown by Figure 2(a), at that time point, the distance from the center line (defined as the line from the center of the midbrain to the nose tip) to the most deviated medial wall of the maxillary sinus was 19 mm on the right side (red line in (A)) and 11.5 mm on the left side (blue line in (A)). As shown by Figure 2(a), retrieval and retrospective examination of these images indicated that the anatomical deformity in the right maxillary sinus had not progressed (the red and blue lines in (B) indicated distances from the center line of 19 and 11 mm, resp.). In the 2 years after the second MRI, the patient started complaining of repeated sinus-related symptoms such as cheek pain or pressure on both sides and anterior purulent nasal discharge once every 1 or 2 months (Figure 2(b)). Two years after the second MRI, the patient developed the ophthalmological symptoms, was referred to our hospital, and was diagnosed with stage III CMA (Figure 2(b)).
case_images: Summary of the clinical examination after the endoscopic sinus surgery. (a) Facial appearance 6 months after the endoscopic sinus surgery (ESS). The arrows in the right and left pictures show the superior sulcus. The deepening of the right superior sulcus that was observed before the sinus surgery appeared to have been eliminated by the ESS. (b) Endoscopic findings 6 months after the ESS. The right uncinate process was removed and recurrence of the deformity was not observed. (c) Computed tomography findings 10 months after the ESS. The deviation of the posterior wall in the right maxillary sinus (shown by the arrow) seems to have been eliminated by the ESS.
case_images: CRIOT2017-4296195.003.jpg
case_images: PMC5512096_01_CRIOT2017-4296195.003.jpg
case_images: fig3
case_images: Six months after the ESS, right enophthalmos seemed to be equivalent to that on the affected side (Figure 3(a), arrow) and endoscopy showed that there was no recurrence of the nasal deformity (Figure 3(b)). Ten months after the ESS, CT imaging showed that the anatomical deformity of the posterior wall in the maxillary sinus had been repaired (Figure 3(c), arrow).