Anesth Pain Med 2012; 7: 16～21 ￭Case Report￭

Auriculotemporal and greater auricular nerve blocks have roles in patients with Ramsay Hunt syndrome with involvement -A report of two cases-

Department of Anesthesiology and Pain Medicine, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea

Hyun Seung Jin, Woo-Seok Sim, and Hee-Jin Roe

Ramsay Hunt syndrome (RHS) refers to herpes zoster infection of CN VIII or cervical spinal nerves are also involved in the the of the . Cases complicated by disease process, in which symptoms and the clinical course are multicranial nerve involvement in the process of reactivation of the heavier and the prognosis worse, emphasizing the greater need virus, which are known to show virulent clinical course and worse prognosis, are not common in literature as in practice, and there for appropriate and earlier treatment. Although pain physicians has been only one reported case of suspected co-involvement of may actually encounter cases with multicranial nerve invol- the trigeminal nerve in Korean literature. Therefore, in cases of vement in RHS once in a while in practice, there has been RHS with severe rash over the face and neck, it is pertinent to give consideration to such multiple involvement in their early presen- only one such case report in Korean literature, where the tation. Facial nerve palsy and herpes related pain are the two authors finally concluded RHS was mistaken for a trigeminal worrisome complication, which could be alleviated by early treatment herpes zoster, rather than co-involement [1]. In search for with neural blockade in addition to oral medication. Especially, foreign literature related to Koreans, one article reported of a nerve blocks are known to decrease the extent of nerve infla- mmation or damage, thereby facilitating recovery and probably Korean patient with concurrent involvement of trigeminal and preventing postherpetic neuralgia. We report two rare cases of facial nerve, and another report by Korean authors evaluated Ramsay Hunt syndrome with trigeminal nerve involvement, where the clinical characteristics and prognosis of over 300 patients early implementation of blockade of somatic peripheral nerve with RHS and multicranial nerve involvement [2], but none of branches, in addition to the conventional treatment, promoted early recovery. (Anesth Pain Med 2012; 7: 16∼21) the articles discussed about the optimal treatment in such cases, especially related to somatic peripheral nerve blocks. Key Words: Herpes zoster, Polyneuropathy, Ramsay Hunt The goals of all treatment modalities of acute herpes zoster syndrome, Somatic peripheral nerve branch block. infection including RHS, are to reduce edema and infla- mmation of the involved nerves and associated pain, thus Ramsay Hunt syndrome (RHS) is herpes zoster infection of reducing central sensitization and thereby preventing posther- the geniculate ganglion, characterized by vesicular skin lesions petic neuralgia (PHN) or nerve palsy. Especially, the usefulness in the auricle of , external ear, palatal and tongue mucosa of neural blockade with local anesthetics with or without and associated pain, usually accompanied by facial nerve palsy. steroids, has been proven in numerous studies [3,4]. Cranial nerve (CN) VIII, and rarely other than The authors are presenting, with review of literature, two rare cases of RHS with co-involvement of the trigeminal

Received: October 20, 2011. nerve, where early suspicion and diagnosis of multicranial Revised: October 24, 2011. nerve involvement, and early institution of blockade of specific Accepted: November 18, 2011. somatic peripheral nerve branches in the involved dermatome Corresponding author: Woo-Seok Sim, M.D., Ph.D., Department of Anesthesiology and Pain Medicine, Samsung Medical Center, combined with conventional treatments, were associated with Sungkyunkwan University School of Medicine, 50, Irwon-dong, good prognosis. Gangnam-gu, Seoul 135-710, Korea. Tel: 82-2-3410-0356, Fax: 82-2-3410-0361, E-mail: [email protected]

16 Hyun Seung Jin, et al：Somatic peripheral nerve block in Ramsay Hunt syndrome 17 󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏

examination revealed no specific abnormalities, and was CASE REPORTS prescribed with oral valacyclovir 1 g daily for 1 week under the impression of recurrent herpes zoster virus infection of the Case 1 trigeminal nerve. The skin lesion seemed to improve after the A 64-year-old man presented with a 3-day history of initiation of such treatment, but the pain did not subside, and right-sided headache and earache accompanied by vesicular starting from the 5th day of oral medication, the patient newly skin lesions. The pain was stinging in nature, and started from developed difficulty in mouth opening and lip movement on the right ear and surrounding temporal area that spread through the right side and blinking of the right eye (Fig. 1). He also the inner ear toward the mandible, and multiple papules and complained of tingling and numbness on the right side of the vesicles developed in the face along the gingiva and tongue, and visited the department, dermatome. The patient immediately visited otolaryngology and where Bell’s palsy due to herpes zoster virus, RHS was dermatology outpatient clinic, but the otolaryngological physical diagnosed. He was admitted to the neurology department, and the laboratory examination on admission showed slightly increase in neutrophil count and ESR, and right facial nerve lesion was observed in both nerve conduction and corneal reflex test. The aggravation of pain and development of new clinical symptoms such as facial nerve palsy seemed to be a sign of progression of the inflammation even after the previous 7-day oral valacyclovir treatment. This raised the possibility of zoster meningitis, encephalitis or multiple cranial nerve involvement including the trigeminal nerve, and a 7-day intravenous treatment with 650 mg of acyclovir every 8 hr was initiated. The cranial nerve magnetic resonance imaging (MRI) showed multiple enhancement around the fundus of internal auditory canal, labyrinth segment of the facial nerve, geniculate fossa, which were all compatible with his clinical RHS with right facial nerve involvement (Fig. 2). Steroid therapy was commenced with oral prednisolone 60 mg daily for 3 days and maintained on 50 mg daily for another 3 days. Fig. 1. This picture shows the skin lesion and facial on admission in the first patient. On hospital day 5, the movement of the right eyelid began to

Fig. 2. This picture shows the two sections of cranial nerve MRI of the first patient, showing enhancement around the fundus of right internal auditory canal, the labyrinthine segment of facial nerve, and geniculate fossa. Arrows indicate the lesion. 18 Anesth Pain Med Vol. 7, No. 1, 2012 󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏 show improvement, whereas the persisting severe pain his admission. However, he had a history of hearing loss necessitated supplementation with carbamazepine 200 mg/d and immediately after admission, and the otolaryngologic exami- gabapentin 300 mg/d. After his discharge on hospital day 8, nation revealed a mild unilateral sensorineural hearing loss, prednisolone was tapered off, while oral carbamazepine and which improved quickly and returned to normal within 5 days gabapentin was maintained. However, the patient came to our of admission. Although the possibility of RHS could not be outpatient clinic due to unrelenting pain. At his first visit, totally ruled out, no further radiologic examination was taken, facial muscle movement and skin sensation had both improved and the treatment was focused on reducing the skin lesions to grade 4/5 by Medical Research Council (MRC) scale, and accompanying pain with antivirals and analgesics, since whereas his pain intensity was describe as 70−80/100 mm by apparent facial nerve palsy was absent throughout the course visual analogue scale (VAS). The most painful area was and the cochlear symptoms resolved. He was discharged after around the right side cheek and chin, which included intravenous injection treatment for 7 days with improvement of electric-shock like pain spreading from the auricle forward acute skin lesions, and was maintained thereafter on ibuprofen, Ⓡ accompanied by dull ache with pruritis of the same area, acetaminophen and codein (Mypol cap , Sungwon Adcock stabbing pain of the tongue, dull ache of the teeth and Pharm., Korea) every 8 h. He visited our clinic due to gingiva. Spontaneous pain and hyperalgesia was observed in all remaining pain, and at his first visit, his pain score on areas. He was taking carbamazepine 600 mg/d, amitriptylin 10 aggravation reached VAS 80−90/100 mm, and its relief of mg/day, pregabalin 150 mg/d, without a significant about 50% after the medication, was only transient. There was improvement in his pain. A stellate ganglion block (SGB) was no observable facial nerve palsy, while skin pigmentation given using 8 ml of 1% lidocaine, which resulted in a relief without vesicles were diffusely scattered around the ear auricle of pain to VAS 50/100 mm at 1-week follow up. Pain and right mandibular nerve dermatome, and crusted skin lesion persisting around the area ahead of the auricle, and below the remained in the external auditory canal. The most severe pain lower lip, raised suspicion of the involvement of the trigeminal was the earache, and all the above mentioned skin areas were nerve, and the cranial nerve MRI findings were reassessed, to accompanied by spontaneous pain and hyperalgesia. Concu- reveal an ill-defined signal change along the spinal nucleus of rrence of the trigeminal nerve lesion with RHS was suspected, trigeminal nerve suggesting a change following viral infection. and somatic and sympathetic nerve blocks were given During the following month, he was given 4 times of SGBs, alternatively, including a right SGB, a right greater auricular twice each of auriculotemporal nerve block (ATNB) and nerve block (GANB) using a mixture of 20 mg of triam- block (MNB), to report a decreased pain score of cinolone in 5 ml of 0.375% ropivacaine, and a right mental VAS 30/100 mm. The somatic blocks and sympathetic blocks nerve block with 10 mg of triamcinolone in 2 ml of 0.375% were given alternatively. Thereafter, 3 more SGBs, and another ropivacaine, and oral gabapentin was commenced. Brain MRI each of ATNB and MNB was given alternatively, in the was carried out to discern the possibility of progressive cranial following 3 month, which was associated with a stepwise neuropathies, which detected an enhancement of the origin of decrease of pain, and the oral medication was tapered off trigeminal nerve and an indefinite signal change in the accordingly. Thereafter, he is on observation at a 3-month geniculate ganglion, suggestive of polyneuropathy of both interval, with his pain nearly subsided and only complaining of trigeminal and facial nerves (Fig. 3). For the following month, a decreased sensory of grade 4/5 in his right lower lip. 4 times of SGB, twice each of ATNB, GANB and MNB were given. The somatic and sympathetic nerve blocks were given Case 2 alternatively during the first half of the follow up period, and An 89-year-old male was hospitalized in a dermatology simultaneously thereafter. In addition, gabapentin was increased department, due to a 4 days onset of vesicular eruptions to 1,200 mg/d, with supplementary tramadol 150 mg/d. His followed by throbbing pain in the right ear, root of nose, pain score around the ear has decreased to VAS 20−30/100 mandible and tongue with a pain scale of VAS 80−90/100 mm with almost no hyperalgesia, and has no complaint other mm. He was diagnosed as herpes zoster infection of trigeminal than the intermittent mild tingling sense in right side of the nerve, and treated for 7 days. He did not complain of tongue. He is under regular follow up for stepwise tapering off apparent facial nerve symptoms except for a slightly heavy of the oral medications. tongue and lips on the right side during the first few days of Hyun Seung Jin, et al：Somatic peripheral nerve block in Ramsay Hunt syndrome 19 󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏

Fig. 3. This picture shows the four sections of brain MRI of the second patient, showing the clearest image of enhancement around the origin of the trigeminal nerve, and geniculate fossa of the facial nerve. Arrows indicate the lesion.

associated pain, and facial nerve paralysis. The 8th nerve DISCUSSION features such as tinnitus, hearing loss, nausea, vomiting are regularly present. We would like to make two points; First, all pain Although Ramsay Hunt, who first reported RHS, had already physicians should be familiar with the atypical presentation of mentioned the co-involvement [6], concurrent involvement of RHS, which might mimic , or show dual one or more cranial or cervical nerves other than the 8th involvement of both facial and trigeminal nerve. Second, in nerve in RHS is rare [1,2,7]. They include various cranial such cases, severe presentation should be expected, and early nerves V, IX, X, XI, and XII, and even the spinal nerves C2 implementation of the nerve blockade of the specific peripheral and C3, leading to a complex form of neurologic disturbance somatic branches in the involved dermatome, together with with symptoms such as trigeminal neuralgia, hoarseness, and SGBs, should be considered to minimize the complications. swallowing difficulty or rash typical to herpes zoster Herpes zoster oticus occurs when the latent varicella-zoster cephalicus. virus is reactivated along the distribution of the sensory nerves The suggested explanation of such multiple involvements innervating the ear, which usually includes the geniculate refers to their embryologic and anatomical correlation. The ganglion. When associated with facial nerve palsy, the is in close proximity to the geniculate infection is called RHS. The annual incidence of RHS has ganglion within the bony facial canal, and the gasserian, been reported as 5 per 10,000 [5]. The syndrome presents as a geniculate and other cranial and 2nd−3rd cervical ganglia triad of vesicular lesion around the ear, palate and tongue, comprise a chain in which inflammation of a single ganglion 20 Anesth Pain Med Vol. 7, No. 1, 2012 󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏 could extend to nearby ganglia [6]. In addition, contiguous sympathetic stimulation, and deafferentation causing synaptic cranial neuropathies may be partly due to the selective reorganization in the central nervous system. Therefore, vulnerability of blood vessels to varicella-zoster virus and the management of the acute pain, which represents the common blood supply [6]. inflammation and damage of the nervous system, is critical in In our cases, the first patient presented with a trigeminal improving the outcome. In fact, acute pain, along with rash in addition to the typical clinical symptoms of RHS, in patient’s age, severity of rash and many other factors, were which the diagnosis of polyneuropathy was confirmed by shown to be a reliable risk factor or predictor of PHN [11]. radiologic studies, whereas the second patient, concluding Neural blockades minimize this inflammatory process [12,13], clinically and radiologically, seemed to have had a mild RHS reduce the abnormal activation of the sympathetic nervous without facial nerve palsy upon trigeminal herpes zoster with a system, and steroids used in the blockade are known to further possible involvement of the vestibulocochlear nerve. reduce inflammation and control C-fiber conduction through Cases with multiple nerve involvement are known to show membrane stabilization. As a matter of fact, articles reporting more severe symptoms and relate to worse prognosis compared that the early implementation of nerve blocks in AHZ lead to to simple cases with only 7th, or 7th and 8th nerve involve- complete recovery of pain and prevention of PHN after AHZ, ment, which calls upon the need for early accurate diagnosis have been published [12,13]. In addition, according to the and more aggressive treatments. However, since the ear has articles which reviewed the effect of neuraxial and sympathetic complex innervations as to be commented later, and the facial blockade on herpes zoster related pain [3,4], although yet nerve symptoms might be a late manifestations [1], or even controversial, it has been a general opinion that, neural absent [8] in RHS, such multicranial involvement may be a blockades, both epidural analgesia and sympathetic blocks, are diagnostic challenge to a naive physician in the early stages of effective in the relief of acute pain in AHZ, and prevention of the disease. PHN in patients with AHZ. Unfortunately, there were no Another main concern of such cases of complicated RHS, is articles that studied the role of peripheral somatic nerve how to minimize the two dreaded complications, namely facial blockade separately. nerve palsy and herpes zoster related pain. The mechanism of The majority of the literature on the subject had facial nerve paralysis in RHS, is known to be the infla- methodologic shortcomings reducing the reliability and clinical mmation and demyelination of the facial nerve from the virus, applicability of the results, and the effect of nerve block in which cause edema and compression of the nerve within the the treatment of acute pain in AHZ, although apparent, is facial canal. Spontaneous and complete recovery from facial known to be transient, lasting for no longer than 1 month, nerve paralysis occurs only in the minority of cases. Therefore, questioning its role in the development of PHN [14]. However, correct diagnosis and subsequent proper treatment, including a as of now, it would be wise to consider its early combination therapy of steroids and antivirals, and neural implementation, since a neural block performed by a skilled blockade, are essential in improving the outcome. Consideration medical personnel is safe without serious complication, and is of early treatment of all Ramsay Hunt syndrome patients with generally accepted in evidence-based practice. A concern may a course of acyclovir (800 mg, five times daily for 7−10 be raised regarding the risk of spread of infection or days) and oral prednisone (60 mg daily for 3−5 days) was secondary bacterial infection induced by injecting into the skin recommended, which have been shown to be both safe and lesion, but the possibility has been reported to be very low effective [6,7]. The earlier initiation of treatment resulted in [13]. better outcome [9,10]. In addition, sympathetic nerve blocks One more important issue is to become familiar with the are known to reduce edema within the facial canal by complicated somatic innervations of the skin around the ear. improving the blood flow [4], and repetitive SGBs could External ear and the anterior part of the auricle is supplied promote recovery of the facial nerve paralysis [1]. mainly by the auriculotemporal nerve, derived from the third Another serious complication, is pain, both acute pain due to branch of trigeminal nerve, the mandibular nerve, with minor acute herpes zoster (AHZ), and postherpetic neuralgia (PHN). contribution from cranial nerves VII, IX, X. On the other This herpes related pain, is mediated by the inflammation and hand, the posterior part of the auricle, cervical area below the damage of the nerves by the virus that induces peripheral and ear and the lower part of the mandible opposing the central sensitization, abnormal activation of C-fibers by mandibular nerve dermatome, are innervated by the greater Hyun Seung Jin, et al：Somatic peripheral nerve block in Ramsay Hunt syndrome 21 󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏󰠏 auricular nerve (GAN) which originates from the cervical plexus and is composed of branches of C2−3. Therefore, the REFERENCES ATN and GAN, are the main branches innervating the ear. Especially, the GAN is divided into two branches that each 1. Park JM, Yu SJ, Park AR, Lee SM. Treatment of Ramsay Hunt communicates with the facial nerve. This complicated dual syndrome that is mistaken for trigeminal herpes zoster. 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