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Clinical Features and Longterm Prognosis of Trochlear Headaches

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Clinical Features and Longterm Prognosis of Trochlear Headaches European Journal of Neurology 2013 doi:10.1111/ene.12312 Clinical features and long-term prognosis of trochlear headaches J. H. Smitha, J. A. Garrityb and C. J. Boesc aDepartment of Neurology, University of Kentucky, Lexington, KY; bDepartment of Ophthalmology, Mayo Clinic, Rochester, MN; and cDepartment of Neurology, Mayo Clinic, Rochester, MN, USA Keywords: Background and purpose: Trochlear headaches are a recently recognized cause of chronic daily headache, headache, of which both primary and inflammatory subtypes are recognized. The migraine, neuro- clinical features, long-term prognosis and optimal treatment strategy have not been ophthalmology, ocular well defined. movements, secondary Methods: A cohort of 25 patients with trochlear headache seen at the Mayo Clinic headache disorders, between 10 July 2007 and 28 June 2012 were identified. trochlea, trochleitis Results: The diagnosis of trochlear headache was not recognized by the referring neurologist or ophthalmologist in any case. Patients most often presented with a new Received 13 September 2013 daily from onset headache (n = 22, 88%). The most characteristic headache syndrome Accepted 21 October 2013 was reported as continuous, achy, periorbital pain associated with photophobia and aggravation by eye movement, especially reading. Individuals with a prior history of migraine were likely to have associated nausea and experience trochlear migraine. Amongst individuals with trochleitis, 5/12 (41.6%) had an identified secondary mecha- nism. Treatment responses were generally, but not invariably, favorable to dexameth- asone/lidocaine injections near the trochlea. At a median follow-up of 34 months (range 0–68), 10/25 (40%) of the cohort had experienced complete remission. Conclusions: Trochlear headaches are poorly recognized, have characteristic clinical features, and often require serial injections to optimize the treatment outcome. The identification of trochleitis should prompt neuroimaging to look for a secondary cause. Introduction generally thought to be associated with a positive response. The trochlea is a saddle-like cartilaginous structure Unfortunately, only a very limited number of cases located in the superomedial orbit, which contains the have been reported in the literature [3–5]. Therefore, tendon of the superior oblique muscle. Histologically, information regarding the clinical features, treatment the trochlea is surrounded by a synovial membrane, and prognosis is limited. To help clarify these issues, analogous to the structure of a joint [1]. The trochlea our experience with trochlear headache in 25 previ- is innervated by an ophthalmic nerve branch and is ously unreported cases is reported. capable of generating pain in the setting of trochleitis, which is most often idiopathic, but may develop Methods secondary to autoimmune connective tissue disorders [2]. Trochleitis is characterized by swelling and tender- Prior to the initiation of the study, Mayo Clinic Insti- ness of the trochlea, and aggravation by vertical tutional Review Board approval was obtained. The ductions. Swelling may be observed clinically or docu- procedural records of one of the investigators (JAG) mented on orbital imaging studies [2]. A non-inflam- was searched from 10 July 2007 to 28 June 2012 to matory condition, termed primary trochlear headache identify all adult (age ≥ 18) patients who had received (PTRH), has also been described [3]. Treatment of all a trochlear injection. The electronic medical records trochlear headaches is presumed to require local were manually abstracted to identify individuals where steroid injection in the vicinity of the trochlea, and is a diagnosis of a trochlear headache could be defini- tively assigned and adequate clinical documentation Correspondence: J. H. Smith, Department of Neurology, University was available. of Kentucky, 740 S. Limestone, L445, Lexington, KY 40503, USA (tel.: 859-323-5661; fax: 859-323-5943; e-mail: jonathan.smith@uky. In our paper, the general term trochlear headache is edu). used to describe any headache referable to the troch- © 2013 The Author(s) European Journal of Neurology © 2013 EFNS 1 2 J. H. Smith, J. A. Garrity and C. J. Boes lear apparatus. Given the lack of validated criteria, sheath meningioma. One individual developed the specific diagnoses of PTRH and trochlear migraine headache 6 months following removal of an orbital were assigned based on previously suggested dermoid tumor (case 5), 1 month following orbital definitions [3,5]. Trochlear migraine refers to a troch- decompression for Graves ophthalmopathy (case 6), lear headache which then triggers a secondary 3 months post-partum (case 11) and 1 month follow- migraine attack [2,5]. The diagnostic criteria for head- ing a Roux-en-Y gastric bypass (case 13). No enlarge- ache secondary to trochleitis were used as suggested ment of the superior oblique muscle had been by the newly published International Classification of radiographically noted in case 6. There were no com- Headache Disorders, 3rd edition (online beta version) plications reported in review of any of the above men- [6]. A diagnosis of secondary trochleitis was assigned tioned procedures. if there was clinical, radiographic and/or histopatho- Trochlear headaches had an acute, daily from logical evidence for orbital localization of an inflam- onset, presentation in the majority of cases (n = 22, matory or neoplastic process. The diagnosis of either 88%), being subacute, daily within 1 month, in the trochleitis or PTRH could be supported by aggrava- remainder. A final diagnosis of PTRH (n = 13, 52%) tion and/or reproduction of pain by action of the was made slightly more often than trochleitis (n = 12, superior oblique muscle. 48%). Bilateral involvement was seen in 8/13 (61.5%) Patients were excluded if an alternative diagnosis cases of PTRH and in 4/12 (33.3%) cases of trochle- for the headache was established, such as a periorbital itis. In all bilateral cases this always occurred sequen- cranial neuralgia, a trigeminal autonomic cephalalgia tially, and within 1 year of onset. Amongst patients or carotid-cavernous fistula. with trochleitis, five (41.6%) had an identified second- Acuity of onset was summarized as acute if the ary mechanism. These included incomplete Behcet’s headache was daily from onset, subacute if daily (n = 1), idiopathic TolosaÀHunt (n = 1), granuloma- within 1 month of onset, and insidious if daily within tosis polyangiitis (GPA) (n = 2) and orbital lymphoma 1 year of onset. Due to the retrospective nature of the (n = 1). All five cases of secondary trochleitis under- study, treatment efficacy was summarized as complete, went biopsy of an orbital mass located near the troch- partial or ineffective based on the information avail- lea. In case 11, the trochlear headache had been the able in the medical record. If the patient reported a presenting symptom of GPA. Secondary trochleitis quantitative indicator of pain relief, this information was most often bilateral, but was observed unilaterally was specifically recorded. in one individual (case 11). The location of the trochlear headache was most Results often focused at the medial eyebrow, orbit or forehead (n = 9, 36%), with radiation into the forehead, tem- Over a 5-year period of chart review, 25 individuals ple, periorbit or retro-orbitally. The pain was reported were identified with sufficient clinical characterization as continuous (n = 25, 100%), and achy, dull or pres- for study inclusion (Table 1). Patients were generally sure-like (n = 19, 76%). The average intensity as rated female (n = 20, 80%), with a median age at diagnosis on a numerical rating scale was severe (7–10) in seven of 46 (range 18–77). Patients had a median time from (35%), moderate (4–6) in 10 (50%) and mild (0–3) in symptom onset to diagnosis of 6.7 months (range three (15%) cases. The most common associated 2 weeks to 10 years). The diagnosis of a trochlear symptoms were photophobia (n = 15, 68.1%) and bin- headache had not been made in any patient prior to ocular diplopia (n = 10, 45.4%). Four out of the five referral, despite all patients having been seen previ- patients who reported associated nausea also had co- ously by either a neurologist or ophthalmologist prior morbid migraine. The headache was characteristically to presentation. The most common mis-diagnoses aggravated by eye movements (especially reading) by prior to presentation were chronic migraine (n = 13), 18 (75%) patients. new daily persistent headache (n = 5), no diagnosis Amongst individuals with a prior diagnosis of (n = 4), hemicrania continua (n = 2) and atypical migraine (n = 7), six were considered to have attacks facial pain (n = 1). of trochlear migraine following the onset of the new All patients with a prior headache diagnosis self- headache syndrome. These headaches were reported reported a new superimposed pain syndrome, which to be ipsilateral to the trochlear pain. was very apparent to all patients as distinct. Anteced- Diagnostic evaluation consisted of either a magnetic ent events were only occasionally noted by patients. resonance imaging (MRI) of the head or a computed The most concrete association was in case 3, where a tomography (CT) study of the orbits in all patients new daily trochlear headache began immediately fol- except one (case 13). Three patients were found to lowing orbital surgery for removal of an optic nerve have an imaging abnormality directly involving the © 2013 The Author(s) European Journal of Neurology © 2013 EFNS European Journal of© Neurology Table
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