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Düşünen Adam The Journal of Psychiatry and Neurological Sciences 2013;26:211-214 Case Report / Olgu Sunumu DOI: 10.5350/DAJPN2013260212 Isolated Axillary Betul Tekin Guveli1, Fikret Aysal2, Azize Esra Gursoy3, Involvement: A Case Report Mehmet Kolukisa3, Suna Askin4, Ahmet Hakyemez5, Arif Celebi3

1Neurologist, Bakirkoy Training and Research Hospital for Psychiatry Neurology and Neurosurgery, 1st Department of Neurology, Istanbul - Turkey 2Neurologist, Bakirkoy Training and Research Hospital for Psychiatry Neurology and Neurosurgery, 2nd Department of Neurology, Istanbul - Turkey 3Neurologist, Bezmialem Vakif University, Faculty of Medicine, Department of Neurology, Istanbul - Turkey 4Neurologist, Okmeydani Training and Research Hospital, Department of Neurology, Istanbul - Turkey 5Neurologist, Istanbul Training and Research Hospital, Department of Neurology, Istanbul - Turkey

ABSTRACT Isolated involvement: a case report Isolated axillary neuropathy is a rare condition. Trauma to the shoulder, especially dislocation of the shoulder is the most common cause. The other causes of axillary neuropathy are injection to the shoulder, carrying heavy backpacks and acute idiopathic neropathy. Differential diagnosis should be made especially between cervical 5-6 radiculopathy and upper truncus brachial plexopathy. Case: A 32-year-old man admitted with progressive atrophy and weakness of the right shoulder which developed after deep pain. In his neurological examination, abduction weakness and atrophy of the right shoulder was determined. Magnetic resonance imaging findings of cervical spine and right shoulder were normal. Electrophysiologic examination revealed reduced compound muscle action potential amplitude of the right axillary nerve, recorded from , compared to the left side. On needle EMG, subacute neurogenic signs in the right deltoid and teres minor muscles which are innervated by the axillary nerve were detected. Etiological evaluation revealed no cause. It has been suggested that isolated axillary neuropathy may be associated with Personage-Turner syndrome. Antiinflammatory medications and physical therapy provided partial improvement. The aim of our presentation was to discuss the differential diagnosis, treatment options and etiologic causes of axillary neuropathy. Key words: Axillary nerve, acute idiopathic brachial plexus neuropathy, EMG

ÖZET İzole aksiller sinir tutulumu: Olgu sunumu Address reprint requests to / Yazışma adresi: Neurologist Betul Tekin Guveli, Bakirkoy İzole aksiller sinir tutulumu nadir görülen bir durumdur. En sık karşılaşılan neden omuz travması, özellikle omuz Training and Research Hospital for Psychiatry çıkığıdır. Omuza yapılan enjeksiyon, sırt çantası kullanımı veya akut idiyopatik brakiyal pleksus nöropatisi diğer Neurology and Neurosurgery, 1st Department etiyolojik nedenler olabilir. C5/C6 radikülopati, brakiyal pleksus üst trunkus tutulumu ile ayırıcı tanısı yapılmalıdır. of Neurology, Istanbul - Turkey Olgu: Sağ omuzda şiddetli ağrı sonrası güçsüzlük ve incelme gelişen, nörolojik muayenede sağ omuz Phone / Telefon: +90-212-543-6565 abdüksiyon zaafı ve atrofisi olan 32 yaşındaki erkek hastanın servikal spinal ve omuz MR incelemeleri normaldi. E-mail address / Elektronik posta adresi: Elektrofizyolojik incelemede, sağ aksiller sinir bileşik kas aksiyon potansiyeli, deltoid kasından kayıtlama ile sola [email protected] göre düşüktü. İğne elektromiyografisinde aksiller sinir inervasyonlu deltoid ve teres minör kaslarında subakut Date of receipt / Geliş tarihi: dönem nörojenik tutulum bulguları saptandı. Etiyolojik araştırmada neden bulunamadı. İzole aksiller sinir February 27, 2012 / 27 Şubat 2012 tutulumunun Personage-Turner Sendromuna bağlı olabileceği düşünüldü. Antienflamatuar ilaçlar ve fizik Date of acceptance / Kabul tarihi: tedavi ile hasta kısmen düzeldi. Vaka; ayırıcı tanı, tedavi ve etiyolojik nedenleri tartışmak amacı ile sunuldu. April 24, 2012 / 24 Nisan 2012 Anahtar kelimeler: Aksiller sinir, akut idiyopatik brakiyal pleksus nöropatisi, EMG

INTRODUCTION passes laterally and posteriorly, and located under the . Then it travels through the quadrangular he axillary nerve is comprised of fibers from the space formed by neck of the , long head of the TC5-C6 cervical nerve roots. It leaves upper truncus , teres minor and teres major muscles. It divides and of brachial plexus. Axillary nerve first into anterior and posterior branches in the deltoid extends from the lateral side of the , then muscle. While its motor fibers innervate the deltoid and

Düşünen Adam The Journal of Psychiatry and Neurological Sciences, Volume 26, Number 2, June 2013 211 Isolated axillary nerve involvement: a case report

Left Right

Figure 1: Axillary nerve motor conduction study. (Stimulation: Erb. Record: Deltoid muscle)

Table 1: Other motor nerve conduction studies Table 2: Sensory nerve conduction studies

Latance(ms) Amp(mV) C.V.(m/s) Peak latance (ms) Amp (μV) C.V.(m/s)

Right Right median nerve Recording: APB Recording: Wrist Stimulation Stimulation Wrist 3.8 8.8 1. palm 3.4 26.6 Antecubital fossa 8.6 8.4 56.3 3. palm 3.6 29.6 Right 2. palm 3.6 30.6 50.0 Recording: ADM Right ulnar nerve Stimulation Recording:Wrist Wrist 3.1 11.1 Stimulation Below elbow 8.2 10.3 51.0 5. palm 3.4 15.3 48.5 Above elbow 10.6 9.6 50.7 (Amp: Amplitude, ms: milisecond, μV: mikrovolt, C.V.: conduction velocity, m/s: meter/second) (ADM: Abductor digiti minimi muscle, APB: Abductor policis brevis muscle, Amp: Amplitude, mV: milivolt, ms: milisecond, CV: conduction velocity, m/s: meter/second) clinic due to severe right shoulder pain. He could not teres minor muscles, its sensory fibers carry sensory recall a specific precipitating traumatic event. The information from the shoulder joint, as well as the skin cervical spinal and the shoulder MR findings were covering the inferior region of the deltoid muscle (1). normal and he has been treated with with anti- Isolated axillary nerve involvement generally appears inflammatory drugs. It was determined that the pain secondary to the trauma and reveals itself through complaints lessened, however there was weakness and limitation of the shoulder movements (2). slimming on the shoulder. Neurological examination revealed muscle strength (Medical Research Council CASE scale) as 3 in shoulder abduction, atrophy of deltoid muscle and hypoesthesia in the area of axillar nerve It has come to the knowledge that a 32 year old male sensory distribution. patient has referred to a physical therapy rehabilitation At the sixth month of the disease onset, nerve

212 Düşünen Adam The Journal of Psychiatry and Neurological Sciences, Volume 26, Number 2, June 2013 Tekin-Guveli B, Aysal F, Gursoy AE, Kolukisa M, Askin S, Hakyemez A, Celebi A

Figure 3: Denervation potentials in right deltoid muscle Figure 2: Reinnervation potentials in the right deltoid muscle conduction study (NCS) performed by surface the C5-C6 cervical nerve roots except teres minor and stimulation along the posterior border of the deltoideus muscle confirmed the diagnosis of the sternocleidomastoid muscle and recording from the isolated axillary nerve involvement. deltoid muscle, revealed that right axillary nevre We evaluated ANA, ds DNA, anticardiolipin compound muscle action potential amplitude was antibodies, brucella, syphilis and tumour markers lower compared to the left side (Figure 1). NCSs of the including CEA, AFP, PSA, CA-125 for differential other motor and sensorial showed normal results diagnosis of vasculitis, because it is well known that (Table 1,2). vasculitic neuropathy could manifest with an isolated Needle electromyography (EMG) studies revealed nerve involvement at disease onset and later with reduced recruitment of polyphasic motor unit potentials, mononeuritis multiplex. with prolonged duration and normal and increased Isolated axillary nerve involvement is a rare amplitude in the right deltoid and teres minor muscles condition. Injury to the axillary nerve is most commonly (Figure 2), with resting fibrillation potentials and reported following trauma of the shoulder. Visser et al. positive sharp waves in deltoid muscle (Figure 3). EMG (3) reported axillary nerve involvement in anterior findings regarding the right teres major, biceps, shoulder dislocations as common as 42%. Various brachioradial, suprasinatus, infraspinatus, rhomboideus causes including deep in the major, serratus anterior, triceps, flexor carpi radialis, deltoid muscle, carrying heavy backpacks and long extensor digitorum communis, first dorsal interosseus duration of general anesthesia at a position have also were all within the normal limits. been described (1). Our patient could not recall a specific precipitating traumatic event. We excluded primary DISCUSSION shoulder disorders including injuries and calcified tendinitis and cervical spinal disorders The reason of the diagnostic delay of our presenting including spondylosis and cervical discopathy with case might be that the isolated axillary nerve involvement normal findings on MR examination. We established is a rarely encountered condition. Because of the low the diagnosis isolated axillary neuropathy as a amplitude of right axillary nerve CMAP, an axonal manifestation of idiopathic acute brachial plexus damage was taken into consideration. Normal findings neuropathy. of median nerve sensory conduction study from the Acute brachial plexus neuropathy was described for first finger and the lateral cutaneous nerve of the forearm the first time in the 1940s and there are synonyms such conduction study suggested that lesion might be as “Parsonage-Turner syndrome, neuralgic amyotrophy, localized to anterior root or anterior horn. Normal and acute brachial neuralgia”. It is usually sporadic but needle EMG findings of the muscles innervated from recently SEPT9 gene mutations have been identified in

Düşünen Adam The Journal of Psychiatry and Neurological Sciences, Volume 26, Number 2, June 2013 213 Isolated axillary nerve involvement: a case report hereditary form, which is inherited as an autosomal Acute brachial plexus neuropathy should be dominant trait and has an episodic course (1). It may be diagnosed, after the exclusion of all other causes which seen in all age groups and more common in males (3:2). may cause shoulder pain and weakness. Its recurrence Its incidence reported annually 2-3/100 000 (1). Its is rare and prognosis is good (4,7). Treatment of acute etiology is unknown, but viral infections, vaccinations brachial plexus neuropathy is usually anti-inflammatory and immunologic causes may be responsible. It is drugs and physiotherapy. Tsairis at al. (8) reported, that characterized by severe shoulder pain at night which is 99 patients with neuralgic amyotrophy who improved followed by weakness and atrophy of at least one after 2 years 80% and 3 years 90% of them. shoulder muscle in a few weeks. “Lasegue sign of the In conclusion, idiopathic axillary nerve palsy is an arm” is called increasing of pain with compression or infrequent neuromuscular disorder. The diagnostic movement of the affected shoulder. Paresthesia, evaluation of idiopathic axillary nerve palsy should be hypoesthesia, and rarely allodynia may occur at the based on history and physical findings and confirmed beginning of attacks. Autonomic disorders may be by electromyography. In the differential diagnosis of accompanied by vegetative or trophic skin changes, the isolated axillary nerve palsy the diagnosis of acute edema, temperature regulation disorders, increased brachial plexus neuropathy should be kept in mind sweating (4,5). The affected most especially in patients with painful onset and commonly. Suprascapular and axillary nerve development of weakness and atrophy in weeks. The involvement is usually together with multiple nerve preferred treatment is conservative, with analgesia and involvement. Isolated axillary nerve involvement physiotherapy as the primary treatment methods with reported in 10% cases (2,6). a relatively good outcome.

REFERENCES

1. Stewart JD. Focal Peripheral Neuropathies. Canada: JBJ 5. Misomare GW, Lehman DE. Parsonage-Turner Syndrome (acute Publishing, 2010, 173-177. brachial neuritis). J Bone Joint Surg Am 1996; 78:1405-1408.

2. Bonnard C, Anastakis DJ, van Melle G, Narakas AO. Isolated and 6. Goslin KL, Krivickas LS. Proximal neuropathies of the upper combined lesions of the axillary nerve a review of 146 cases. J extremity. Neurol Clin 1999; 17:525-545. Bone Joint Surg Br 1999; 81:212-217. 7. van Alfen N, van Engelen BG. The clinical spectrum of neuralgic 3. Visser CP, Coene LN, Brand R, Tavy DL. The incidence of nerve amyotrophy in 246 cases. Brain 2006; 129:438-450. injury in anterior dislocation of the shoulder and its influence on functional recovery. J Bone Joint Surg Br 1999; 81:679-685. 8. Tsairis P, Dyck PJ, Mulder DW. Natural history of brachial plexus neuropathy. Report on 99 patients. Arch Neurol 1972; 4. Favero KJ, Hawkins RH, Jones MW. Neuralgic amyotrophy. J 27:109-117. Bone Joint Surg Br 1987; 69:195-198.

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