Abstract
Introduction
A large proportion of migraine patients remain undiagnosed or misdiagnosed in Italy. In our experience, many migraineurs self-diagnose their condition as “cervical pain attack” or “cervical pain syndrome” (CP), assuming cervical spine pathology as the cause. We aimed to phenotype and classify the headache of patients with self-diagnosed CP, and to describe this sample of patients.
Methods
Consecutive patients aged 18 to 75 years, referred to the Headache Center of the Mondino Institute (Pavia, Italy) for a first visit for headache, completed a questionnaire about CP and were subsequently examined by an experienced clinician.
Results
Out of 207 patients, 132 (64%) believed they suffered from CP. According to ICHD-IIIβ criteria, these patients suffered from migraine or probable migraine in 91% of cases. The great majority of patients who believed that they suffered from CP underwent unnecessary medical exams (including radiation exposure in 40% of cases) and used treatments that were inadequate for their real diagnosis.
Conclusion
The majority of patients with CP suffer from typical migraine. The misdiagnosis produces an economic burden (for patients and the health care system) and leads to impaired quality of life of patients.
Introduction
The majority of migraine patients remain undiagnosed or misdiagnosed in Italy, as in several other countries (1–4). The misdiagnoses reported in the literature include “sinus headache” and tension-type headache (2–4). In our experience, many migraineurs self-diagnose their pain as “cervical pain attacks” or “cervical pain syndrome” (CP), assuming their headache is caused by a cervical spine pathology.
In this study, we aimed at a prospective diagnostic work-up of patients with self-diagnosed CP in a cohort of patients seeking help in a tertiary headache centre. Secondary outcomes were: 1) to evaluate the diagnostic and treatment trajectories of self-diagnosed CP, 2) to identify the reasons why patients believe they suffer from CP, and 3) to compare two subgroups of patients with the same condition (migraine) who did and did not believe they suffered from CP.
Methods
All patients consecutively referred to the Headache Center of the Mondino Institute (Pavia, Italy) for a first visit for headache were enrolled. Inclusion criteria were: age between 18 and 75 years and headache history >1 year. Exclusion criteria were: i) the patient was born outside Italy, ii) a change in the headache characteristics in the last six months, iii) patients with cognitive impairment and/or unable to provide a detailed history.
Enrollment started in November 2013 and was completed in February 2015. The study received approval from the ethics committee, and all patients signed an informed consent form. Patients were asked to complete a questionnaire about the presence of CP self-diagnosis in the waiting room before the visit. The questionnaire included questions about the characteristics of pain attacks (three features), the presence of associated symptoms (from a list of 18), triggers (from a list of 13), aura symptoms, and premonitory symptoms in the 24 hours before the attack (from a list of 12). Patients also answered questions about family history of CP and/or headache, the diagnostic procedures and treatments performed for CP, and the reasons why they believed they suffered from CP.
Patients who did not believe they suffered from CP (NCP) were also asked to answer identical questions.
Afterwards, patients were seen by an experienced headache neurologist. A detailed history was taken and a neurological exam was performed. Paraclinical exams needed to rule out causes of secondary headache (including brain and cervical imaging) were performed when necessary. All patients finally received a diagnosis based on ICHD-IIIβ criteria. The neurologist was blinded to the self-reported diagnosis of the patients. A flow-chart of the study procedures is shown in Figure 1.
Flow-chart of the protocol.
Comparisons between CP and NCP patients with the same condition (i.e. migraine) were performed with the Mann-Whitney test or chi-square test for continuous or categorical variables, respectively. After univariate analysis, stepwise logistic regression analysis with a backward selection, using a p value ≥0.1 for removal from the model, was performed to identify the minimum set of independent predictors of migraine self-diagnosis as CP. All variables with a cutoff p value <0.1 from univariate analyses were included in the model. Statistical analysis was performed with MedCalc Version 13.3.3 (MedCalc Software, Mariakerke, Belgium). Due to the exploratory nature of the study, we report statistical associations at a p value of <0.05.
Results
We enrolled 240 patients. Thirteen were excluded as they did not complete the whole questionnaire. Twenty patients did not meet the inclusion/exclusion criteria (Figure 1). Two hundred and seven patients were included in the data analysis: 75% were female, the average age was 39 ± 13 years. Out of these 207 patients, 132 (64%) believed they were suffering from CP, while 75 (36%) did not. All patients reported head pain during the attacks.
Characteristic of patients reporting suffering from “cervical pain attacks” or “cervical pain syndrome” (CP) and their answers to the questionnaire.
Data are presented as means ± standard deviations (range) for continuous data and as n (%) for categorical data.
median 2, IQR 1–3.
The question was: “Why do you think that your CP attacks are caused by a cervical spine pathology?”; patients could chose more than one option (except for “no specific reason”).
#The question was: “Did any health professional confirm to you that your CP attacks were caused by a cervical spine pathology?”; patients could chose more than one option (except for “nobody”).
Overview of the ICHD-IIIβ diagnoses made by a headache specialist in patients who were originally considering their pain as Cervical Pain (n = 132) (a), and who did not (n = 75) (b). MwoA: Migraine without aura; MwA: Migraine with aura; CM: Chronic migraine; MOH: Medication overuse headache; CTTH Chronic tension-type headache; FETTH: Frequent episodic tension-type headache; HC: Hemicrania continua; HH: Hypnic headache; PEH: Primary exercise headache; CE: Cervicogenic headache.
The comparison between CP and NCP patients was conducted on the group of subjects whose final ICHD-IIIβ diagnosis was migraine, because they represented the largest group (n = 164). We included patients with migraine without aura and/or migraine with aura or chronic migraine. Of these patients, 102 patients reported CP and 62 did not. Comparison of the socio-demographical and/or clinical variables of migraineurs in the two groups (supplementary Table 1) identified 12 characteristics associated with a self-diagnosis of CP: older age (p = 0.038), presence of pericranial tenderness (p = 0.046), longer duration of illness (p = 0.02), longer duration of attacks (p = 0.039), lower incidence of migraine aura (p = 0.008), pain involving the back of the head at the attack onset (p < 0.0001), pain involving the back of the head in whichever phase of the attack (p < 0.0001), presence of imbalance and/or dizziness and/or vertigo with pain (p = 0.007), attacks triggered by weather change (p = 0.008), attacks triggered by bad sleeping (p = 0.03), neck discomfort or stiffness as premonitory symptoms (p = 0.0001), neck stiffness/tenderness at the neurological evaluation (p = 0.046). In the multivariate analysis (Supplementary Table 2), lower educational level (p = 0.004), absence of migraine aura (p = 0.015) and pain involving the back of the head at attack onset (p < 0.0001) emerged as independent predictors of migraine self-diagnosed as CP.
Discussion
To the best of our knowledge, this is the first study that has systematically assessed the headache phenotype of patients with self-diagnosed CP. The results suggest that the phenomenon of self-diagnosing CP is very common in Italy, at least in patients referred to a tertiary headache center.
The majority of these patients suffer from typical migraine, without evidence of causative conditions of the cervical spine. Interestingly, the only patient with cervicogenic headache, associated with a severe osteochondrosis at C2/C3, was in the NCP group. It is noteworthy that the diagnosis of “cervicogenic headache” was made according to the ICHD-3β criteria (5), while it would not have been possible according to the ICHD-II (6), because cervical spondylosis/ostechondrosis was not an acceptable cause.
The reasons why patients believed they suffered from CP are multifaceted. Localization of pain/discomfort in the back of the head/neck appears as the main driving factor. Cultural legacy mixed with a false neuroanatomical attribution of cause and symptom may create strong and diffuse beliefs that go against science.
Another cause of misdiagnosis is pain triggered by weather changes. This could be due to the general belief that weather change may worsen pain in osteoarticular pathologies.
Patients were also convinced that attacks were likely caused by the “cervical spine” when symptoms include nausea/vomiting (35%) or dizziness/vertigo (31%). This is probably a consequence of the fact that patients mistake migraine for CP.
Unfortunately, this lack of awareness seems to also affect physicians, if it is true that 60% of patients reported that the diagnosis of CP was confirmed by a physician. This patient-reported finding needs to be interpreted with caution, as patients may misinterpret what they are told by physicians. Another explanation is that other types of problems in the doctor-patient communication may occur (the terminology used by the doctors, not enough time being dedicated to explanations of the medical condition and its causes, etc.).
It is noteworthy that a sizeable proportion of patients reported that although they did not know why the cervical spine would be the cause of their attacks, they still considered them as “CP attacks”. This might be due to the fact that CP is a passepartout term commonly used in the Italian language without necessarily implying the existence of a serious pathology in the spine. Indeed, many female migraineurs (50% in our sample) call their attacks CP attacks, even if triggered by the menstrual cycle.
The comparison between CP and NCP migraineurs helps to identify other factors that may be involved in this misbelief. A lower educational level was associated with CP belief. This is in keeping with the fact that the lower the educational level, the higher the possibility that patients use the simple axiom that localization of pain = cause. Another reason could be the fact that a lower educational level is associated with a higher age, and such patients are more likely to believe they suffer from cervical problems, although – as already mentioned above – the average age of the patient cohort was 41, with an average onset of CP attacks at 20.2 years. Migraine aura seems to protect migraine patients from mistaking migraine for CP, probably because the frankly neurological nature of the symptoms guides patients and physicians toward a more complex etiology.
Another interesting finding stemming from the present data is the fact that the misdiagnosis drives inappropriate diagnostic investigations or consultations and inadequate treatments. This carries an economic burden (for patients and the health care system), and unnecessary radiation exposure (in 40% of cases).
Interestingly, the self-diagnosis of CP causes a diagnostic delay, as migraineurs who believe they suffer from CP have a longer history of the disorder prior to the diagnosis compared to migraineurs who do not believe they suffer from CP (Supplementary Table 1). Considering that migraine is a treatable condition, one can easily imagine that a lot of suffering is potentially preventable.
It would be interesting to evaluate whether CP-migraine misdiagnosis is also present in other countries, as different sociocultural backgrounds may have an influence on pain perception/interpretation (7). In this setting, we are presently conducting a study in several Neo-Latin countries that share similar languages and cultures (8).
Study limitations
We cannot exclude referral bias. A population-based study would be necessary to define the real prevalence. No patients with cluster headache fell into our population, as the Mondino Institute has a dedicated clinic for those patients.
Clinical implications
The phenomenon of self-diagnosing “cervical pain attacks” (CP) is common (in Italy). The majority of patients who believe they suffer from CP present typical migraine attacks without evidence of pathological conditions of the cervical spine. The self-diagnosis of CP therefore is one of the reasons for the high rates of migraine misdiagnosis in Italy, and drives inappropriate diagnostic investigations or consultations and inadequate treatments. Awareness and education campaigns would be appropriate measures to improve the diagnosis and management of migraine and to avoid unnecessary investigations.
Footnotes
Acknowledgements
This study was carried out in collaboration with UCADH (University Consortium for Adaptive Disorders and Head pain), University of Pavia, Italy. We would like to thank Professor Musicco for his help in the data analysis.
Declaration of conflicting interests
The authors declared the following potential conflicts of interest with respect to the research, authorship, and/or publication of this article: MV, GS, ST, GN, CT have no conflict of interest. TS: the author’s previous and current institutions have received payments for consulting and speaking activities (Actelion, ATI, Biogen Idec, Electrocore, Sanofi Genzyme, Mitsubishi Pharma, Novartis, Teva); grants received (EFIC-Grünenthal, Novartis Pharmaceuticals Switzerland, Swiss MS Society, Swiss National Research Foundation).
Funding
The authors disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by grants from the Italian Ministry of Health to RC 2013–2015.
References
Supplementary Material
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