A Case Report on Red Ear Syndrome with Tinnitus Successfully Treated with Transcranial Random Noise Stimulation

Abstract

Background: The red ear syndrome represents a rare symptom complex consisting of auricular erythema associated with painful and burning sensations. It has been described in combination with tinnitus rarely. It has been hypothesized to be etiologically related to altered trigeminal afferent input, temporomandibular disorders, and thalamic dysfunction. Objectives: The initial objective of applying transcranial random noise stimulation (tRNS) in a case of red ear syndrome in combination with tinnitus was the alleviation of the phantom sounds. Study Design: This is a case report on the successful treatment of red ear syndrome with tinnitus by means of transcranial random noise stimulation (tRNS) and a short review on the published cases of this condition. Setting: We present the case of a 50-year-old woman reporting a simultaneous onset of constant left-sided tinnitus and feelings of warmth accompanied by an intermittent stabbing and/or oppressive pain stretching from the ipsilateral ear to the head/neck/shoulder region, occasionally accompanied by nausea/vomiting and dizziness. After failure of pharmacological treatment attempts, either because of lacking clinical effects (gabapentin, zolmitriptan, and indomethacin) or because of adverse reactions (pregabaline), the patient was offered an experimental neuromodulatory treatment with bitemporal tRNS primarily targeting the tinnitus complaints of the patient. Methods: tRNS was conducted in 2 – 3 day sessions (stimulation site: bilateral temporal cortex/2.0 mA/10 s on-and-off-ramp/offset 0 mA/20 min/random frequencies 101 – 640Hz / NeuroConn Eldith DC-Stimulator plus). Results: In 3 consecutive pain attacks repeated sessions of tRNS resulted in substantial alleviation of pain intensity and a prolongation of the interval between attacks. This was an expected finding as the proposed tRNS treatment was initially offered to the patient aiming at an alleviation of the tinnitus complaints (which remained unaffected by tRNS). Limitations: The reported data derive from compassionate use treatment in one single patient. Application of a sham condition would have been desirable, but is not possible in the context of compassionate use treatment. Nevertheless, we would consider it rather unlikely that the reported effects are purely unspecific as the patient did exclusively report symptom alleviation of pain-related parameters without affecting the tinnitus. Conclusions: This case report demonstrates the feasibility and therapeutic potential of applying neuromodulatory treatment approaches in red ear syndrome, a rare form of trigemino-autonomal headache. Therefore, it deserves detailed observation in clinical routine applications as well as controlled trials further investigating its neurobiological effects. Key words: Red ear syndrome, pain, trigemino-autonomal headache, chronic tinnitus, transcranial electrical stimulation, random noise stimulation

Tinnitus Patients with Comorbid Headaches: The Influence of Headache Type and Laterality on Tinnitus Characteristics

“…Our findings are in line with earlier studies, which demonstrated that tinnitus severity is higher in patients with comorbid headache ( 24 ) and correlates with headache frequency ( 25 ). A potential interaction between tinnitus and migraine ( 26 – 30 ) or other trigeminoautonomal headache syndromes ( 31 , 32 ) has been described in many studies.…”

Brain stimulation methods for pain treatment

“…Not surprisingly, the direction of electrode polarization is critical in terms of the after-effects. Through stimulation of the motor cortex, tDCS can be used for: chronic pain (Deer et al 2014;Souto et al 2014;Hodaj et al 2016), pain after endoscopic retrograde cholangiopancreatography (Borckardt et al 2011), trigeminal pain including refractory orofacial pain (Antal et al 2010;Hansen et al 2011;DosSantos et al 2012;Hagenacker et al 2014;Fricova et al 2016;Kreuzer et al 2017), fibromyalgia (Valle et al 2010;Cummiford et al 2016;Castillo-Saavedra et al 2018), major depression (Vigod et al 2014;Tortella et al 2015;Brunoni et al 2016;Liu et al 2017), bipolar disorder (Bersani et al 2015;Pereira-Junior et al 2015;Tortella et al 2015), schizophrenia (Agarwal et al 2013;Palm et al 2016;Gögler et al 2017), Alzheimer's disease (Nardone et al 2012;Bystad et al 2016;Narita and Yokoi 2017), modulation of associative learning (Branscheidt et al 2017), Parkinson's disease (Benninger et al 2010;Hendy et al 2016;Schabrun et al 2016), pain after stroke (Schjetnan et al 2013;Chhatbar et al 2016;Russo et al 2017) and tinnitus (Garin et al 2011;…”

“…This problem does not occur when tACS is used in the "ripple" frequency range (Moliadze et al 2010). Ripples Kringelbach et al 2007;Pereira et al 2007Pereira et al , 2013Holslag 2018;Johnson et al 2008 Strong Holtzheimer and McDonald 2014;McNamara et al 2001;George and Belmaker 2007;Prikryl et al 2013;Fregni et al 2005;Ferreri et al 2003;Li et al 2013;Politi et al 2008;Shen et al 2016Shen et al , 2017 Non-invasive Low spatial targeting compared to DBS, higher cost compared to other non-invasive methods rTMS pain Rokyta and Fricová 2012;Fricová et al 2013;Kohútová et al 2017;Robertson et al 2010;Thomas et al 2007, Shupak et al 2006Thomas et al 2001 Non-invasive, low cost Less pronounced effect compared to rTMS tDCS chronic pain, trigeminal pain including refractory orofacial pain, fibromyalgia, major depression, bipolar disorder, schizophrenia, Alzheimer's disease associative learning, Parkinson's disease, pain after stroke, tinnitus Souto et al 2014;Hodaj et al 2016;Borckardt et al 2011;Antal et al 2010;Hansen et al 2011;DosSantos et al 2012;Hagenacker et al 2014;Kreuzer et al 2017;Fricova et al 2016;Castillo-Saavedra et al 2018;Valle et al 2010;Tortella et al 2015;…”

Daily high-frequency transcranial random noise stimulation of bilateral temporal cortex in chronic tinnitus – a pilot study

“…hf- tRNS has also been found to be able to enhance mathematical ability speed 11 and perceptual learning 5 . In a case report the successful treatment of a patient suffering from “Red Ear Syndrome” by high-frequency tRNS was registered 12 . The “Red Ear Syndrome” contains a symptom complex of auricular erythema, attack-like local pain and tinnitus perception 13 .…”