Abstract
Sinus pain is commonly diagnosed, and patients with headache often name maxillary sinus as the cause of their discomfort. In 98%, it would be classified as migraine or migraine-like headache in the diagnostic criteria set by the International Headache Society (IHS) (1, 2).
These criteria are very strict, and most sinus pathologies are not considered to be a major cause of headache or facial pain (e.g. chronic sinusitis) (2).
In this classification system, the role of non-obstructive foreign bodies in the maxillary sinus with regard to generating pain is still subject to discussion (3). A difficult problem in dealing with patients with sino-nasal headaches is the identification of the primary source of pain.
We report a case of an exceptional cause of orbito-facial pain and discuss the therapeutic approach taken.
Case report
A 41-year-old woman, with an unremarkable medical history, complained of facial pain located in the area of the nose, cheek and the right orbit, which had been present for 7 months. The pain was excruciating, with conjunctival injection, lacrimation, homolateral photophobia, nasal congestion and systemic autonomic symptoms such as nausea and vomiting (once).
The patient had experienced multiple crises, from once a month to two or three attacks a week. No precipitating mechanism had been identified. Each episode had for lasted for 24–72 h. Previous treatment with paracetamol and acetylsalicylic acid had had a partial effect on the pain severity. Others drugs had had no effect. Neither familiar history of headache nor personal problems of algia (before this episode) had been noted.
The results of general physical and neurological examination were normal. A cerebral computed tomography (CT) scan with iodine injection and brain magnetic resonance imaging, including a study of the cerbello-pontine angle, were also normal.
The patient had consulted an otorhinolaryngologist and an intranasal endoscopy had been performed. Inflammation of all the nasal mucosa (oedema, rhinorrhoea) was found, without pus or polyps.
A paranasal sinus CT scan was requested and revealed a well-defined radio-opaque mass in the right maxillary sinus (without sinusitis or inflammatory reaction), close to the posterior part of the infundibulum (Fig. 1). The osteomeatal complex was free, and the foreign body was not obstructive in nature. The CT scan also revealed a bilateral concha bullosa that made no contact with the septum.
Paranasal sinus computed tomography scan without injection, in coronal view. Radio-opaque mass (1) in the upper part of the right maxillary infundibulum. There is no right maxillary sinusitis or other sinus disease. There is a bilateral concha bullosa with no contact point with the septum.
The Otorhinolaryngologic Department had prescribed amoxicillin (1 week) for presumed sinusitis 1 month previously, with no effect.
A new CT scan of the paranasal sinus had been performed 3 months later, when symptoms were the same, with no reduction in the severity of attacks.
Surgery was finally proposed to the patient, and she was informed that the foreign body might be inducing her headache.
Functional endoscopic sinus surgery as a right middle meatotomy with a middle turbinectomy was performed. A metallic foreign body was found and sent to the anatomopathologist. No sinus mucosal abnormalities were noted. The foreign body proved to be a dental amalgam without mycosis.
Her recovery was uncomplicated, and she was discharged on the first postoperative day. She noted the resolution of her headache almost immediately after the general anaesthesia had worn off. Three years later she was pain free.
Discussion
We report a case of a metallic infundibulum mass responsible for orbito-facial pain without sinus mucosa involvement. The concept of referred pain is common, but referred cephalalgia from non-inflamed, non-diseased nasal structures has not been well recognized or described (4, 5). In this case, the severe unilateral pain in the orbital and supraorbital regions, the autonomic symptoms (lacrimation, conjunctival injection and nasal congestion) and the frequency and duration of episodes were phenotypically similar to trigeminal autonomic cephalalgia (TAC) (6–8), even if the description does not meet exactly the IHS criteria (2). The presence of nausea, vomiting and photophobia can also characterize migraine-like attacks without aura. Unilateral cranial autonomic symptoms may also occur together with systemic autonomic symptoms in some migraineurs during attacks (8).
However, the close relationship between the associated disease, the onset of pain, side concordance, medical treatment failure, total response after surgical treatment (with disappearance of pain) and prolonged post-therapeutic follow-up strongly suggest a cause and effect relationship between the foreign body and the headache.
The pathophysiology (6, 9) of these symptoms is likely to involve the connections between the trigeminal nerve and the nucleus tractus solitarus. Activation of the trigemino-autonomic reflex could explain the symptomatology of lesions located in the facial region. The anatomical basis of this reflex would consist of painful afferences through the V cranial nerve toward the spinal nucleus of the trigeminal nerve, leading to the release of factors that produce pain. Activation of the superior salivary nucleus would also occur through its connection with the spinal nucleus of the V pair, releasing neuropeptides such as the vasoactive intestinal polypeptide and the calcitonin gene-related peptide (10). The most commonly affected sinus, the maxillary sinus, transmits sensory input via branches of the maxillary nerve: the posterolateral nerve and the superior alveolar branches of the infraorbital nerve (4, 5).
Sinus pain has been further studied by Wolff and colleagues (11) with a series of experiments triggering headache by directly manipulating various areas of the sinonasal mucosa and they found that the sinus ostia were much more sensitive than the sinus cavities (10).
By the same mechanism, patients with headaches (without any clear aetiology) must be also evaluated for contact point headache (between intranasal mucosa and others structures: deviation, conch bullosa …). Such areas may result in pressure that results in local trauma and the release mediator of pain in peripheral nerve terminals (12, 13).
Diagnostic criteria relating to migraine and TACs developed by the IHS are useful, but not yet ideal. The origin of headache is sometimes a diagnostic dilemma for specialists. To best facilitate diagnosis and treatment, physicians in all involved specialities need to be proactive in their questioning, to elicit as much information as possible from patients about the characteristics of their headache and associated features.