Background

Several different headaches came together in this patient’s case. They are described briefly below.

Postoperative headaches after craniotomy

According to the International Headache Society (IHS), immediate post-craniotomy headache occurs in 80% of cases, but recedes within seven days in most patients [1]. If the postoperative headache persists for more than three months, it is regarded as “chronic post-craniotomy headache” [1].

Treatment of refractory headache

If no relief can be obtained despite therapy, one is dealing with headaches refractory to treatment [2]. According to the American Headache Society, a multidisciplinary approach that includes psychological support is an essential element in the treatment of such refractory headaches [2].

Current approaches in the treatment of chronic migraine

Current approaches to the treatment of chronic migraine consist of prophylaxis and acute therapy [3,4]. The use of topiramate and botulinum toxin has proven efficacy in prophylaxis [3-5]. Analgesics and non-steroidal anti-inflammatory drugs (NSAIDs) are recommended for acute treatment [3,4].

Psychological approaches to treatment and co-analgesics

Headache patients have an increased risk of developing depressive and affective disorders [6]. The incidence is considerably higher with very frequent or daily headaches, so that the use of antidepressants (mostly tricyclics) as co-analgesics is almost standard practice for such headaches [3].

Interdisciplinary therapy for headache

Interdisciplinary therapy of chronic headaches is the international scientific and ethical standard [3]. An interdisciplinary team should involve at least two medical specialties and at least one psychological specialty [7].

Neural therapy

There are numerous indications for the injection of procaine (a sympatholytic neural therapy agent) into painful areas of the body (“interference fields”) and into or around nerve fascicles of the autonomic nervous system, including headaches and unexplained pain in scar tissue [8,9]. Neural therapists attribute considerable significance to stellate blocks, based on the anatomy of the stellate ganglion [10].

In this case report, we discuss the recommendations that should be made regarding treatment of atypical postoperative headache and advice to patients.

Case presentation

This case concerns a young female patient who had a sports-related accident in 2009 at the age of fourteen. The following day she developed a sudden sensorimotor paresis in both legs and her right arm. In view of her symptoms, cranial magnetic resonance imaging (MRI) was performed; this showed by accident an arteriovenous malformation (AVM) in the right temporal lobe. The patient wanted to proceed immediately with surgical removal of the AVM, but her attending doctors assessed the risk of an unsuccessful operation as too high and preferred to wait with the intervention. When a later MRI showed that the AVM had grown further, the patient was operated in 2010 at the age of 15. The operation was successful from a medical perspective, but since then the young woman has suffered from intolerable headaches.

The postoperative symptoms led to a series of evaluations by various specialists. The patient saw a total of 10 different doctors and therapists and was also treated regularly by her family doctor. At these visits she frequently heard statements such as “You are simulating” or “That will pass”. She often had the feeling that the doctors did not take her seriously. After two years, the suffering of the patient had still not been relieved despite various therapeutic measures. Several attempts at therapy, both with and without the use of medication, were fruitless. The attending doctors told her that she had to learn to live with her pain. Her despair continued to increase.

Constraints due to the headaches and psychosocial effects

Feelings of hopelessness and sadness dominated the patient’s life during these two years. Due to the pain she would often lie in bed for days on end. Her ability to work was assessed as 40% and a disability pension was considered. The activities that young men and women usually engage in, such as sports, visiting clubs, dancing or drinking, had become impossible for the patient or exacerbated the pain. She could not tolerate crowds of people. She was insulted with terms such as “cripple” due to her surgical scar, which extended over a considerable part of the right side of her head. She withdrew from social life. Many of her friends were unable to cope with her problems and turned away from her. The patient fell into a deep depression, which resulted in two attempts at suicide.

Localisation and character of the pain

The headaches involved stabbing pains in the area of the surgical scar on the right frontotemporal part of the patient’s head. The intensity varied and sometimes reached a value of 10 out of 10 on the visual analogue scale (VAS). The headaches continued for the whole day. The patient slept badly and had headaches if she woke up at night. Additional characteristics were loss of appetite, nausea and hypersensitivity to light and noise.

Diagnoses

Initially it was said that the patient suffered from recurrent or refractory headaches resulting from the craniotomy. A subsequent neurological examination resulted in diagnosis of symptomatic, daily migraine following surgical removal of the AVM.

Final attempt

The patient had almost given up hope of an effective treatment when her mother arranged an appointment with a pain specialist. The patient immediately felt that he took her seriously and that he would do his best to help with her problems. From that point on the patient started to regain her courage.

Therapy

The first method that resulted in effective treatment of the patient’s headache was infiltration of the local anaesthetic procaine, based on the pain specialist’s intuition. Procaine was applied in various ways for this patient. The initial emphasis was on the local use of procaine, which was infiltrated into the scar tissue. Subsequently the specialist injected the medication around the right stellate ganglion, in order to determine whether the patient had “sympathetically maintained pain” (SMP). The patient responded well to the stellate blocks. A therapeutic plan was developed that envisaged application of procaine in various ways at regular intervals. These measures succeeded in temporary reduction of the intensity of the patient’s headache to 1 out of 10 on the VAS.

The treatment with procaine was supported by transcutaneous electrical nerve stimulation (TENS) of the stellate ganglion. The possibility of implanting a neurostimulator was rejected after careful balancing of the benefits and risks.

The patient was prescribed topiramate for migraine prophylaxis by a consulting neurologist. This medication did result in significant pain relief, but it soon had to be discontinued due to marked side effects. The alternative use of antidepressants (duloxetine and escitalopram) played a significant role in the treatment of the patient, as it enabled her to keep her mood and thoughts under control.

The pain specialist would have liked to treat the patient as part of an interdisciplinary team, but she rejected this proposal. A multidisciplinary therapy concept was then developed.

Discussion

This case report demonstrates the effects of surgical removal of an AVM on the life of a young woman. Her social life was severely limited by the postoperative pain, she withdrew and lost many of her friends, she was only partially able to work and suffered from a deep depression that involved several attempts to take her own life.

In fact the patient’s suffering was not alleviated for years despite various therapeutic measures, and her despair continued to increase. A vicious circle developed. Neither she nor her family were aware of the risk factors for depression until then.

In principle it is unwise to discontinue the search for the cause of pain and for an effective treatment prematurely. This case involved a very young pain patient. Statements made by the medical staff such as “You have to learn to live with your pain” can have devastating effects on such a young person in puberty. This case report emphasises the importance of taking the patient’s suffering seriously and persisting in the search for a practical solution. An interdisciplinary therapeutic approach should be sought as soon as possible [3]. If the patient rejects such an approach, a multidisciplinary therapy concept should be considered [11,12].

Psychological support should be included in this concept, especially if the individual’s despair is as great as in our patient [7]. Psychological counselling was also valuable to her for another reason: because of her suffering, the patient received appropriate attention and care from her family and various medical practitioners. She might have missed both elements in this period of her life. It is therefore difficult in hindsight to assess the extent to which her pains were unconsciously reinforced.

The doctor-patient relationship played a crucial role in this case. The patient appreciated the fact that the pain specialist dedicated so much time to her during the consultation, and that there was also room for topics other than the therapeutic procedure. According to the patient, the doctor-patient relationship contributed significantly to the success of the therapy. In order to develop such a constructive relationship it is almost essential to take a holistic approach to the patient [13,14]. To a certain extent, patient care should involve more than just academic medical knowledge – and of course it must be completely professional [13]. This aspect is especially important if the patient is experiencing a psychological crisis at the onset of therapy. This crisis should be overcome jointly, in the context of the medical staff and the environment of those involved. The patient should be listened to and taken seriously – two fundamental aspects of an effective doctor-patient relationship [13].

The medicinal migraine therapy referred to above and recommended in the current specialist literature did not result in the pain relief that was hoped for. The migraine-prophylactic agent topiramate as well as the analgesics and NSAIDs that were used for acute therapy were not effective enough or were discontinued due to their side effects. It should be noted that aspirin and paracetamol were not employed as part of the therapeutic approach, but were already in use before the actual diagnosis of migraine [4,15].

The use of procaine as part of the neural therapy approach was the decisive step towards success in this case. Application of TENS also had a positive effect on the therapy. This also applies to the consistent use of antidepressants as co-analgesics in the patient’s treatment plan.

Conclusions

In summary it can be said that the case should have and could have proceeded more effectively from the start if the specialists who were consulted had paid more attention to the doctor-patient relationship, and if they had given the patient the feeling that she was taken seriously. An effective form of pain therapy that went beyond the standard medication should have been sought sooner.

The following recommendations can be made:

The suffering of a patient in pain must be taken seriously. Every effort should be made to find a feasible solution. This is especially important in the case of a very young patient. Various specialists should collaborate in an interdisciplinary therapeutic approach. A psychological specialty should be included. The doctor-patient relationship is crucial for the success of therapy and should ensure that the patients are listened to, taken seriously and approached holistically. This relationship should transcend academic medical expertise to some extent. If the patient is experiencing a severe psychological crisis, this should be overcome jointly. In this regard, the medical personnel are just as important as the patient’s environment. Application of the neural therapeutic agent procaine can provide significant pain relief for refractory postoperative pain from scar tissue. TENS is a passive physical method that (together with other measures) can alleviate pain by neural modulation [16]. The use of antidepressants can have a beneficial effect on the efficacy of treatment [3].

Consent

Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

List of abbreviations

NSAIDs: Non-steroidal anti-inflammatory drugs; MRI: Magnetic resonance imaging; AVM: Arteriovenous malformation; VAS: Visual analogue scale; TENS: Transcutaneous electrical nerve stimulation

Competing interests

The authors declare that they have no competing interests.

Authors’ contributions

LG interviewed the patient, studied patient’s records, performed literature review, and drafted the manuscript. HS designed the study, performed the physical examinations of the patient, carried out the interventions, and edited the manuscript. All authors read and approved the final manuscript.

Acknowledgements

We thank language editor David Tracey for providing translation and medical writing services.