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LETTER TO EDITOR
Year : 2013  |  Volume : 61  |  Issue : 6  |  Page : 660-662

Intraneural capillary hemangioma: A rare cause of proximal median neuropathy


1 Department of Neurology, Nizam's Institute of Medical Sciences, Hyderabad, Andhra Pradesh, India
2 Department of Pathology, Nizam's Institute of Medical Sciences, Hyderabad, Andhra Pradesh, India
3 Department of Radiology, Vijaya Diagnostic Center, Hyderabad, Andhra Pradesh, India

Date of Submission10-Sep-2013
Date of Decision26-Oct-2013
Date of Acceptance18-Dec-2013
Date of Web Publication20-Jan-2014

Correspondence Address:
Yerasu Muralidhar Reddy
Department of Neurology, Nizam's Institute of Medical Sciences, Hyderabad, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.125280

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How to cite this article:
Reddy YM, Meena AK, Uppin MS, Vadapalli R, Challa S. Intraneural capillary hemangioma: A rare cause of proximal median neuropathy. Neurol India 2013;61:660-2

How to cite this URL:
Reddy YM, Meena AK, Uppin MS, Vadapalli R, Challa S. Intraneural capillary hemangioma: A rare cause of proximal median neuropathy. Neurol India [serial online] 2013 [cited 2019 Sep 15];61:660-2. Available from: http://www.neurologyindia.com/text.asp?2013/61/6/660/125280


Sir,

Proximal median neuropathy is distinctly uncommon compared with median entrapment at the carpal tunnel. Causes include external compression from casting, trauma, venipuncture, anatomic variations and compressive mass lesions including tumor or hematoma. [1] Intraneural tumors are far more rare causes of median neuropathy. Intraneural hemangioma of the median nerve is a rare condition and only ten cases have been described in the literature. [2],[3],[4],[5],[6],[7],[8],[9] Hence the report of this unusual case.

An 18-year-old medical student presented with complaints of inability to make fist with right index finger and thumb and paresthesias of right lateral three fingers of 31/2 months duration. There was no history of trauma, venipuncture, or casting. Examination revealed pointing index sign of right hand. There was weakness of flexor digitorum superficialis (FDS), flexor carpi radialis (FCR), pronator teres (PT), flexor digitorum profundus (FDP), flexor pollicis longus (FPL), abductor pollicis brevis (APB), and opponens pollicis (OP). There were no sensory deficits. Nerve conduction studies revealed decreased compound muscle action potential (CMAP) with normal distal latency, conduction velocity, and F wave latency of right median nerve. Right median sensory nerve action potential from the second digit was absent. Electromyographic examination of APB, FCR, FDS, and PT showed no spontaneous activity, normal motor unit action potentials but reduced recruitment. High-resolution ultrasonography (HRUS) revealed well-defined hypo-echoic lesion of 15 × 6 mm-size with loss of fascicular architecture in median nerve just proximal to elbow with increased colour flow. Magnetic resonance neurography (3T) of the nerve showed fusiform thickening with enlargement with loss of fascicular architecture measuring 8.5 mm × 1.86 cm with minimal soft tissue edema at the level of distal arm. At the level of cubital fossa the nerve measured 5.6 × 5.5 mm anteroposteriorly. The nerve was hyperintense on T1 weighted, T2 weighted and short-time inversion recovery sequences. It showed diffusion restriction on diffusion-weighted imaging. Post-contrast fat saturation images showed focal enhancing intra and perineural mass with cuffing, enhancing vessels [Figure 1], [Figure 2], [Figure 3], [Figure 4]. There were no flow voids. Exploration was performed using pneumatic tourniquet. It revealed enlarged median nerve from distal arm till the level of FDS. There was reddish yellow soft tissue mass around the nerve in the distal arm and cubital fossa with intraneural extension and adhesion to the surrounding tissues. The nerve was released by cutting nerve sheath longitudinally; perineural adhesions were broken and interfascicular dissection was done using operative microscope. Marginal resection was done. Histopathology showed capillary sized proliferating vascular channels lined by plump endothelial cells containing intraluminal red blood cells with scant intervening fibroblasts consistent with capillary hemangioma. Patient started improving clinically one month after exploration. He is able to hold pen and write sentences.
Figure 1: Clinical photograph showing right hand pointing index sign suggesting proximal median neuropathy

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Figure 2: (a) High-resolution ultrasonography of the lesion at the cubital fossa with Color flow on Doppler (b) DWIBS sequence showing median nerve-thickening just proximal to the elbow joint with focal fusiform intraneural mass (c) Post - contrast fat sat PROSET images showing focal enhancing intra and perineural mass with cuffing, enhancing vessels

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Figure 3: Intraoperative photograph showing swollen median nerve with blood vessels over it in the distal arm

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Figure 4: (a) The lesion seen in perineurium. (b) The lesion is comprised proliferating small capillary channels lined by endothelium (c) S-100 immunostain positive in the nerve fibers indicating intraneural lesion

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The causes of proximal median neuropathy are exhaustive. [1] Intraneural tumors are very rare causes of median neuropathy. Capillary hemangioma is a hamartoma, an abnormal, localized proliferation of vascular endothelial cells. Benign intraneural hemangioma originating from peripheral nerves is rare. In the literature, such tumors were reported in median nerve, ulnar nerve, [2],[3],[4],[5],[6],[7],[8],[9] digital nerves, [10],[11] and posterior tibial nerve. [12] Most patients present with a painful, soft mass along the path of a nerve with signs and symptoms of nerve compression and entrapment. The diagnosis is usually made on per operative and histopathological examination. Till date 10 cases of intraneural hemangioma of median nerve were reported. Most of them presented with carpal tunnel syndrome. [2],[3],[4],[5],[6],[7],[8] Raynauds phenomenon as presenting symptom was also reported. [6] This is first case of intraneural capillary hemangioma causing proximal median neuropathy.

HRUS not only helps in diagnosis but also aids in assessing the size, location, and relation to the nerve and surrounding structures. HRUS shows varied echogenicity with multiple cystic spaces. [13] Doppler ultrasound may reveal no detectable or only weak Color Doppler signals. Kim et al. [14] diagnosed intraneural hemangiomas in ulnar nerve at multiple levels by multilevel ulnar neuropathy by ultrasonographic evaluation. This patient showed hypoechoic fusiform enlargement of the nerve with loss of fascicular architecture from distal arm till the elbow with Color flow on Doppler.

Magnetic resonance imaging is more superior to ultrasound in delineating the tumor and its relationship to surrounding structures. [15] Hemangiomas appear hyperintense on T1, T2- weighted and fat-suppressed sequences. Flow voids, feeding and draining vessels may be seen. These lesions usually enhance on gadolinium administration. Kerimoglu et al. [10] diagnosed digital nerve hemangioma using MRI which showed draining and feeding vessels suggesting hemangioma. Neurography in this patient showed similar features as mentioned in addition to diffusion restriction. However, there were no flow voids, feeding, or draining vessels.

Total surgical excision along with nerve graft offers cure. However, this is not feasible many times and should be resorted to as last option. Partial resection can relieve symptoms partly. Tumor may recur with partial resection. In our case we did microsurgical interfascicular dissection with marginal resection.

 
 » References Top

1.Gross PT, Jones HR Jr. Proximal median neuropathies: Electromyographic and clinical correlation. Muscle Nerve 1992;15:390-5.  Back to cited text no. 1
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2.Coessens B, De Mey A, Lacotte B, Vandenbroeck D. Carpal tunnel syndrome due to an haemangioma of the median nerve in a 12-year-old child. Ann Chir Main Memb Super 1991;10:255-7.  Back to cited text no. 2
[PUBMED]    
3.Kojima T, Ide Y, Marumo E, Ishikawa E, Yamashita H. Haemangioma of median nerve causing carpal tunnel syndrome. Hand 1976;8:62-5.  Back to cited text no. 3
    
4.Patel CB, Tsai TM, Kleinert HE. Hemangioma of the median nerve: A report of two cases. J Hand Surg Am 1986;11:76-9.  Back to cited text no. 4
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5.Peled I, Iosipovich Z, Rousso M, Wexler MR. Hemangioma of the median nerve. J Hand Surg Am 1980;5:363-5.  Back to cited text no. 5
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6.Prosser AJ, Burke FD. Haemangioma of the median nerve associated with Raynaud′s phenomenon. J Hand Surg Br 1987;12:227-8.  Back to cited text no. 6
[PUBMED]    
7.Oztekin HH, Karaarslan AA. Carpal tunnel syndrome due to a cavernous hemangioma of the median nerve. Acta Orthop Traumatol Turc 2003;37:170-2.  Back to cited text no. 7
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8.Doðramaci Y, Kalaci A, Sevinç TT, Yanat AN. Intraneural hemangioma of the median nerve: A case report. J Brachial Plex Peripher Nerve Inj 2008;22:3-5.  Back to cited text no. 8
    
9.Kline SC, Moore JR. Intraneural hemangioma: A case report of acute cubital tunnel syndrome. J Hand Surg Am 1992;17:305-7.  Back to cited text no. 9
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10.Kerimoglu U, Uzumcugil A, Yilmaz G, Ayvaz M, Leblebicioglu G, Altinok G. Intraneural hemangioma of digital nerve diagnosed with MR imaging. Skeletal Radiol 2007;36:157-60.  Back to cited text no. 10
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11.Kon M, Vuursteen PJ. An intraneural hemangioma of a digital nerve: Case report. J Hand Surg Am 1981;6:357-8.  Back to cited text no. 11
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12.Mestdagh H, Lecomte-Houcke M, Reyford H. Intraneural haemangioma of the posterior tibial nerve. J Bone Joint Surg Br 1990;72:323-4.  Back to cited text no. 12
[PUBMED]    
13.Keng CY, Lan HH, Chen CC, Gueng MK, Su YG, Lee SK. Soft tissue hemangiomas: High-resolution grayscale and color doppler ultrasonographic features in 43 patients. J Med Ultrasound 2008;16:223-30.  Back to cited text no. 13
    
14.Kim DH, Kang JW, Park JW. Multilevel ulnar neuropathy caused by multiple intraneural hemangiomas. Muscle Nerve 2010;41:562-6.  Back to cited text no. 14
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15.Hems TE, Burge PD, Wilson DJ. The role of magnetic resonance imaging in the management of peripheral nerve tumours. J Hand Surg 1997;22:57-60.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]

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