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Review Respiratory and Otolaryngologic Manifestations Of Review Respiratory and otolaryngologic manifestations of giant cell arteritis T.F. Imran1, S. Helfgott2 1Rutgers University, New Jersey Medical ABSTRACT disease and “acute, sharp, catarrhal af- School, Newark, USA; Objective. The classical presentation fections, including those showing heat 2 Division of Rheumatology, Brigham and of giant cell arteritis (GCA) includes in and inflammation of the temporal Women’s Hospital and Harvard Medical the new onset of headache, scalp ten- muscles” and that these conditions may School, Boston, MA USA. derness, facial pain or jaw claudica- lead to vision loss (1). In the English Tasnim F. Imran, MD tion in an older patient. Many patients literature, one of the earliest descrip- Simon Helfgott, MD with GCA have features consistent with tions of GCA is attributed to Sir Jona- Please address correspondence to: the diagnosis of polymyalgia rheumat- Simon Helfgott, MD, than Hutchinson, who, in 1890, de- Division of Rheumatology, ic (PMR) and nearly all have elevated scribed a patient who had “red streaks Brigham and Women’s Hospital, markers of inflammation such as the on his head” which were painful and 45 Francis Street B-3, erythrocyte sedimentation rate (ESR) prevented him from wearing his hat. Boston MA 02115, USA. or the serum C-reactive protein (CRP) These red streaks were thought to be E-mail: [email protected] Respiratory and ear-nose-throat (ENT) swollen temporal arteries (2). Horton et Received on September 30, 2014; accepted signs and symptoms such as cough, al. characterised GCA as being a sys- in revised form on February 10, 2015. tongue infarction, trismus, hearing loss temic illness (3). They observed ten- Clin Exp Rheumatol 2015; 33 (Suppl. 89): and facial swelling are less commonly derness over the scalp, painful nodules S164-S170. described, yet they may be the initial along the temporal arteries, headaches, © Copyright CLINICAL AND presentation of GCA and weight loss and jaw stiffness in EXPERIMENTAL RHEUMATOLOGY 2015. Our aim was to review the published two patients. In 1938, the observation literature on the topic of respiratory of an increased erythrocyte sedimen- Key words: giant cell arteritis, and otologic manifestations of GCA. tation rate was first noted (4) and a temporal arteritis, respiratory Methods. A literature search was per- decade later striking improvement of manifestations, otologic disease formed on PUBMED and MEDLINE symptoms after steroid treatment was using the following keywords: GCA, first reported (5). temporal arteritis, pulmonary, respira- Headache is the most common symp- tory, ENT, cough, tongue necrosis. tom, occurring in about 70% of pa- Results. The upper and lower airways tients with GCA. The headache may be manifestations of GCA include a wide variable in location, intensity and qual- variety of conditions that could be caused ity. However, it is often of new onset. by ischaemia due to the vasculitis. Jaw claudication occurs due to ischae- Conclusions. It is important to recog- mia of the masseter muscles, leading to nise these atypical presentations be- pain when chewing tough foods. Visual cause they may be the sole initial mani- symptoms such as vision loss and di- festation of the disease. Early suspicion plopia may develop in about a third of and confirmation of the diagnosis of patients. The most concerning compli- GCA can help to prevent more cata- cation is blindness due to its irrevers- strophic consequences of unrecognised ible nature. Other manifestations entail disease, including stroke and blindness. large vessel involvement including aortic aneurysm and dissection, aortic Introduction arch syndrome, and limb artery steno- Giant cell arteritis (GCA) is an ancient sis (6, 7). disease. One of the first descriptions Respiratory symptoms involving the of GCA can be found in Ali Ibn Isa’s upper and lower airways are occa- Tazkiratul-Kahhaleen, Notebook of sionally observed. These include dry the Oculist, published in the tenth cen- cough, throat and tongue pain and tury AD. Ibn Isa describes migraines, hoarseness. Patients may present with Competing interests: none declared. headaches in patients with chronic eye otolaryngologic symptoms such as S-164 Respiratory manifestations of GCA / T.F. Imran et al. REVIEW tongue necrosis, dysphagia, and sub- category, one may observe non-specific view of the histopathology of the mass mandibular swelling. The presence of facial swelling (9, 12). In one prospec- (17). Other less commonly reported these symptoms may be overlooked in tive study of 345 patients with GCA, symptoms include necrosis at the tip of some patients, further delaying the di- the frequency of facial oedema was the nose, lip necrosis, paresthesias felt agnosis. Given the risk for permanent 12% (13). Similarly, a chart and litera- over the chin, glossitis, and dental pain visual loss and other major morbidities, ture review of 260 patients with GCA (19). identifying the link between the respir- found 37 cases (14%) of head and neck atory tract, otolaryngologic manifesta- swelling that lasted at least one day Audiovestibular manifestations tions and other more classical features (13). Head and neck swelling appeared One of the earliest links of deafness of GCA can help to expedite the diag- to be a transient but heralding feature, with GCA was provided by Cook et nosis and initiate appropriate therapy. preceding the development of GCA. al., who described seven patients who Glucocorticoids are a highly effective The swelling mainly involved the orbit- developed unilateral, and subsequent- therapy for GCA and should be initi- al region, the lower part of the cheeks ly bilateral deafness which improved ated early and utilised for the shortest and maxillae. Periorbital pain has also spontaneously (20). A later study found period of time, to avoid long-term ad- been described as a presenting feature that 5 out of 68 patients with GCA had verse effects (8). This review is intend- of GCA (14). Less commonly, oedema bilateral sensorineural deafness, which ed to increase clinicians’ awareness of was noted in the lateral and anterior as- did not improve after steroid therapy the less commonly seen respiratory and pects of the neck, the tongue and the su- (21). Progressive hearing loss as an ini- otolaryngologic symptoms of GCA. praclavicular space. The swelling typi- tial manifestation of GCA has also been cally resolves following with therapy reported in a handful of cases, with Methods and in some patients it may spontane- improvement of hearing loss noted in A literature review using the PUB- ously remit over time (13). Tongue pain some patients (21). Whether or not the MED and MEDLINE databases was may be tested by assessing for repetitive hearing loss is reversible and to what performed with the following key- rapid protrusion of the tongue, which extent, may depend on how early the words: giant cell arteritis, temporal may produce pain, burning or fatigue corticosteroid treatment is initiated. arteritis, pulmonary, respiratory, ear- (10). The tongue has a rich blood sup- Audiovestibular manifestations may nose-throat, cough, and tongue necro- ply; thus necrosis is rare and indicates occur prior to other symptoms of GCA sis. Case reports, controlled studies, extensive vascular involvement. The (22). Thus, early recognition of these retrospective studies and review arti- presence of tongue necrosis portends a signs is important since the treatment of cles were included. Studies published worse prognosis as these patients tend this condition with corticosteroids may in languages other than English were to be older and also have a higher inci- be quite different than the more watch- excluded. Table I lists these sources. dence of vision loss. However, the non- ful approach that is often used in the specific swelling may be mistaken for management of patients with these is- Clinical manifestations other diagnoses including sinusitis and sues. In addition, missing the diagnosis Orofacial manifestations angioedema. of GCA and the opportunity to initiate Since GCA has a predilection for in- As noted earlier, jaw claudication, is appropriate therapy may lead to even volvement of cranial arteries, it should the most specific non-neurological sign more dire consequences.Thus, in a pa- not be surprising to observe orofacial of GCA and may be misdiagnosed as tient with PMR and new audiovestibu- signs and symptoms in some patients. a temporomandibular joint disorder lar symptoms, the clinical suspicion of These can be divided into two distinct (15).However, trismus or reduction in underlying GCA should be considered groups: those that are associated with jaw opening, is much less recognised. and higher doses of corticosteroids may ischaemia and those that demonstrate One retrospective analysis of 88 pa- need to be instituted (4). features of localised inflammation. In tients, noted six to have reduction in Spontaneous ear pain has been de- the former category, one can observe jaw opening. These patients had a more scribed as an initial manifestation of temporal and scalp pain, jaw pain and acute presentation with shorter time to GCA (23). In addition, a recent pro- complaints of claudication. Jaw clau- diagnosis (average of four weeks ver- spective study with 44 patients with dication is elicited in about half of sus twelve weeks), and there was a high GCA showed that audiovestibular dys- patients. Tongue or scalp necrosis has correlation with eye involvement in function was significantly more com- been rarely observed, yet GCA is the about half of the patients (16). mon in patients with GCA than among most common cause of tongue necrosis Submandibular swelling has also been age-matched controls (4). A patient was (9). It is often localised to small areas noted to be an initial manifestation of defined as having abnormal vestibu- of the tongue mucosa due to an exten- giant cell arteritis (14, 17, 18). Ruiz- lar tests if any of these three findings sive collateral blood supply network in Masera et al. reported a case of a 75 were noted: spontaneous nystagmus, the tongue (10).
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