Pediatric trochleitis associated with paranasal sinusitis: a case report
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Trochleitis is trochlear pain with evidence of inflammation in the trochlear area on radiologic examination. The etiology of trochleitis is mostly idiopathic. Secondary trochleitis is rare, and trochleitis associated with paranasal sinusitis in children has not yet been reported.
An 8-year-old boy presented with left periorbital pain for a week. His visual acuity and eye movement were normal. There was point tenderness on palpitation over the left trochlear region without swelling or redness. Orbital magnetic resonance imaging showed focal enhancement on the left trochlea and paranasal sinusitis on the left side. The patient’s symptoms and signs were completely resolved after empirical treatment for sinusitis. There was no need to inject a local steroid.
Although rare, sinusitis should be considered when diagnosing and treating trochleitis in children with periorbital pain.
KeywordsTrochleitis Trochlear pain Sinusitis Children
magnetic resonance imaging
non-steroidal anti-inflammatory drugs
Trochleitis is the local inflammation of the superior oblique tendon trochlea. The diagnosis of trochleitis is based on clinical findings and radiologic images. It is characterized by superomedial orbital pain and point tenderness on palpitation over the trochlear region.
Trochleitis is mostly idiopathic. Secondary trochleitis is rare and is associated with systemic autoimmune disease or lymphoma [1, 2]. Herein, we report the first case of a pediatric patient with trochleitis associated with sinusitis.
Discussion and conclusions
Trochlear pain is subclassified into primary trochlear headache, trochlear migraine, trochleitis, and inflammatory Brown syndrome . Primary trochlear headache is a specific headache localized in the trochlear region without evidence of inflammation . Trochlear migraine is diagnosed when exacerbation of the trochlear pain triggers a migraine attack . Trochleitis is trochlear pain with evidence of inflammation in the trochlear area on radiologic examination . Finally, inflammatory Brown syndrome is trochleitis accompanied by the limitation of the upward gaze of the affected eye in adduction . Trochleitis is one of the differential diagnoses of headache and orbital pain. However, it is poorly recognized and often misdiagnosed as many clinicians are unfamiliar with this condition. Trochleitis is characterized by pain and tenderness in the involved trochlea, and this condition is often worsened by elevation. Swelling and inflammatory changes are demonstrated on radiologic evaluations such as computerized tomography (CT), MRI, and ultrasonography .
Most cases of trochleitis are idiopathic and usually occur unilaterally . Continuous friction of the superior oblique tendon through a trochlea generates chronic microtrauma, which results in stenosing tenosynovitis . Secondary trochleitis associated with rheumatologic and immunologic disorders, including systemic lupus erythematous and rheumatoid arthritis, can develop [1, 7]. Because the majority of cases involve non-infectious inflammation, oral non-steroidal anti-inflammatory drugs (NSAIDs), systemic corticosteroids, and local steroid injections show favorable outcomes [3, 4, 5]. To the best of our knowledge, trochleitis associated with paranasal sinusitis, however, has not been reported, despite the anatomical proximity between the paranasal sinus and trochlear nerve. Only one case of inflammatory Brown syndrome after frontal sinus surgery has been reported . Trochleitis in children has never been reported. However, orbital infections including orbital cellulitis and orbital abscesses are well known and common complications of sinusitis in children [9, 10]. Incomplete paranasal sinus development and thinner bony barriers in this age are thought to be the main reason .
In our case, the child with paranasal sinusitis presented with focal isolated inflammation in the trochlea area. His symptoms resolved after 2 weeks of empirical treatments for sinusitis and rhinitis. There was no need for local steroid injection or oral corticosteroid treatment, which are considered as the treatment for general trochleitis. Ruling out paranasal sinusitis is recommended before initiating oral or local corticosteroid treatment because the immunosuppressive effects of steroids may worsen infections.
To the best of our knowledge, this is the first report of isolated trochleitis associated with paranasal sinusitis in a pediatric patient. Although rare, sinusitis should be considered when diagnosing and treating trochleitis in children with periorbital pain.
We thank Ji-Sun Kim, Department of Otorhinolaryngology, Eulji University School of Medicine, Eulji Medical Center, for treating the patient’s sinusitis.
This work was supported the National Research Foundation of Korea (NRF) grant funded by the Korean government (MSIP; no. 2017R1C1B5017079).
Availability of data and materials
All data generated or analyzed during this study are included in this published article.
IJL was responsible for the design of the study, cared for the patient, analyzed the data, wrote the manuscript, and revised the manuscript. HH participated in the collecting and analyzing data, manuscript writing, and revised the manuscript. All authors read and approved the final manuscript.
Ethics approval and consent to participate
This study was performed in accordance with the tenets of the Declaration of Helsinki. Approval to conduct this study was obtained from the Institutional Review Board of Nowon Eulji Medical Center (Seoul, Republic of Korea).
Consent for publication
Written informed consent was obtained from the parents for publication of this case report. A copy of the written consent is available for review by the Editor of BMC Ophthalmology.
The authors declare that they have no competing interests.
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