Chronic maxillary atelectasis (CMA) is characterized by a progressive decrease in maxillary sinus volume. The factors that promote the stage progression of CMA remain poorly understood. Here, we describe the time course of anatomical changes in a 40-year-old woman with stage II CMA that progressed to stage III disease. She did not show stage progression until she started to develop repetitive sinus-related symptoms. The stage progression was characterized by ocular symptoms. The repetitive inflammatory episodes may have increased the negative pressure in the affected sinus and weakened the bone walls, thereby promoting stage progression. Thus, a history of repetitive sinus-related symptoms may be a risk factor for stage progression in CMA.
Chronic maxillary atelectasis (CMA) is characterized by a persistent and progressive decrease in the maxillary sinus volume and occlusion of the infundibulum as a result of inward bowing of the antral walls [
A 40-year-old Hungarian woman suddenly noticed upgaze diplopia and right cheek compression when she woke up in the morning. Shortly thereafter, she consulted an ophthalmologist and then an otolaryngologist in another hospital. The investigations suggested that she had a carcinoma in the pterygopalatine fossa on the right side. Therefore, the otolaryngologist referred the patient to our institution for further examination. Examination of the facial appearance of the patient indicated more deepening of the right upper eyelid sulcus than the left eyelid sulcus (Figure
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.
Three years before the onset of the upgaze diplopia and right cheek compression, the patient had undergone brain MRI screening for a lacunar infarction. She had not previously presented with symptoms related to the nose or sinuses. When we reviewed these MRI images, we observed mucosal hyperplasia of the maxillary sinus and deviation of the medial wall of the maxillary sinus on the right side (as compared to the contralateral unaffected side). Thus, we retrospectively diagnosed the patient with stage II CMA. As shown by Figure
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.
Two years before the clinical onset of CMA, the patient underwent follow-up MR imaging. As shown by Figure
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
To treat the ocular symptoms, we used endoscopic sinus surgery (ESS) to eliminate the negative pressure within the maxillary sinus. We removed the laterally drifted uncinate process that was in close contact with the floor of the orbit and observed mucosal hypertrophy of the maxillary sinus. Pathology of the mucosae in the maxillary sinus revealed infiltration with inflammatory cells. After the treatment, the ocular symptoms of the patients disappeared rapidly.
After surgery, we regularly followed the patient by endoscopy and CT imaging for more than 1 year. Six months after the ESS, right enophthalmos seemed to be equivalent to that on the affected side (Figure
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.
We were able to observe the anatomical changes in our CMA patient as she progressed from stage II to stage III CMA over a period of 3 years. The progression of the condition appeared to be associated with the development of repeated sinus-related symptoms over 2 years. These symptoms preceded the development of the ocular symptoms that led to the diagnosis of stage III CMA. These observations suggest that a history of repetitive sinus-related symptoms may indicate progression of the anatomical deformity in CMA. As described below, these symptoms may be indicative of processes that promote CMA progression.
Occluded maxillary infundibulum produces an enclosed hypoventilated environment in the maxillary sinus [
It has been reported that some patients in the advanced stage have persistent ocular complaints after the negative pressure is removed by sinus surgery [
Our CMA patient had a history of repetitive sinus-related symptoms for 2 years before clinical and MRI evidence indicated that the CMA had progressed from stage II to stage III. Thus, a history of repetitive severe inflammation that is accompanied with sinus-related symptoms such as cheek pain or pressure and purulent nasal discharge may associate with stage progression of CMA. Early sinus surgery may prevent CMA stage progression and could be required if the patient frequently experiences sinus-related symptoms.
This study was reviewed and approved by the Institutional Review Board of the University of Tokyo Hospital (#2487). The requirement for informed consent was waived. The investigation of this case was conducted according to the principles of the Declaration of Helsinki and its revisions.
The authors report no conflicts of interest.
Shu Kikuta and Kyohei Horikiri contributed equally to the work.