Multiple Sites of Kimura’s Disease Occuring Along Multiple Peripheral Nerves in the Same Patient:

A Case Report

Kumaran Rasappan, Vaikunthan Rajaratnam, Chin Teck Yew, Lang Tee Ung

Introduction

There can be multiple differential diagnosis for asymptomatic swellings in the limbs

Radiological imaging guide identification of the lump

However, radiological diagnosis may not always be accurate

We report a case of an asymptomatic swelling in a patient who’s histological diagnosis differed from radiological diagnosis

History 25 year old Chinese male

Noticed swelling over medial aspect of left elbow incidentally

4 months duration

Noted it getting slightly bigger over the last month

Asymptomatic

Examination

2 x lumps over anterior medial aspect of elbow and distal arm

Non tender, firm, deep

Slightly mobile transversely but not mobile longitudinally

Tinnel’s negative

Completely asympthomatic

Source: http://img05.deviantart.net/3a9f/i/2011/148/e/8/hanging_right_arm_by

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• (1.5 x 1.0 x 2 cm) Lump

• Close relation to the medial cutaneous nerve of forearm

Axial Saggital

Coronal

Source: http://img05.deviantart.net/3a9f/i/2011/148/e/8/hanging_right_arm_by

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• (3.5 x 0.7 x 1.5 cm) Lump

• Following the course of the median nerve

Axial Saggital

Coronal

Source: http://img05.deviantart.net/3a9f/i/2011/148/e/8/hanging_right_arm_by

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• (1.3 x 1.3 x 2.6cm) Lump

• Close relation to the median nerve and/or anterior interosseous nerve

Axial Coronal

Source: http://img05.deviantart.net/3a9f/i/2011/148/e/8/hanging_right_arm_by

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Compatible with multiple peripheral nerve sheath tumours

Source: http://img05.deviantart.net/3a9f/i/2011/148/e/8/hanging_right_arm_by

_treeclimber_stock-d3hfvaj.png

Source: http://img05.deviantart.net/3a9f/i/2011/148/e/8/hanging_right_arm_by

_treeclimber_stock-d3hfvaj.png

Source: http://img05.deviantart.net/3a9f/i/2011/148/e/8/hanging_right_arm_by

_treeclimber_stock-d3hfvaj.png

Histological Images

Preserved nodal architecture with reactive follicular

hyperplasia

Intermediate power

Histological Images

Germinal centre hyperplasia

Intermediate power

Histological Images

Interfollicular eosinophilia

Intermediate power

Histological Images

Eosinophilic mirco abscess

with folliculolysis and necrosis

Intermediate power

Histological Images

Intermediate power

The findings are representative of Kimura's Disease

Kimura’s disease / Eosinophilic Lymphogranuloma

First described in 1937 in the Chinese literature by HT Kimm and C Szeto

Definitive histological description published in 1948 by Kimura et al

Endemic in Asia (China and Japan) but rare

Only about 200 cases reported since 1948

M/F sex ratio 7 : 3.5

Peak age incidence is during 3rd decade

DDx: Angiolymphoid hyperplasia with eosinophilia

Clinical presentation of Kimura’s Disease Usual location: Head and Neck

Accompanied by satellite adenopathies with hypertrophy of salivary glands

Nodules can be pruritic or painful but the overlying skin is normal

Extra membranous glomerulonephritis is found in half of pts

Cutaneous eosinophilic vasculitis has been described

Epitrochlear, axillary, inguinal and popliteal lesion have been described

Eyes, ears, sperm ducts and nerves very rarely affected

Our Case

EMBASE, PubMed, CINAHL and Google Scholar were used

Only 4 cases involving the peripheral nerves were found 2 case reports describe involvement of the superficial radial nerve 1 case report describes involvement of the median nerve 1 case report describes involvement of the ulnar nerve

No case reports of Anterior Interosseous Nerve or medial cutaneous nerve of forearm

No case reports of multiple peripheral nerve involvement in the same patient

First such finding of 3 locations of Kimura’s Disease in 3 different peripheral nerves in the same patient.

Take Home Message MRI for multiple unknown lumps in limb

Take MRI report only as a guide

Benign Peripheral Nerve Sheath Tumour (BPNST) vs Kimura’s Disease

BPNST vs Kimura’s Lymph Node

Source: http://www.columbianeurosurgery.org/conditions/neurofibroma/

References

Song KS, Woo DE. Kimura’s disease involving the peripheral nerve of the arm in a child: a case report. Journal of Pediatric Orthopaedics B. 2015 Jan 1;24(1):63-6.

Vermaak PV, Lyons CB, Harley OJ. Kimura’s disease affecting the superficial branch of the radial nerve. Journal of Hand Surgery (European Volume). 2014 Feb 1;39(2):208-9.

Lee YS, Ang HK, Ooi LL, Wong CY. Kimura's disease involving the median nerve: a case report. Annals of the Academy of Medicine, Singapore. 1995 May;24(3):462-4.

Martorell M, Pérez-Vallés A, García-García JA, Calabuig C, Aguilella L. Angiolymphoid hyperplasia with eosinophilia involving the cubital nerve. Acta neuropathologica. 2004 Apr 1;107(4):372-6.

Larroche C, Bletry O. Kimura's disease. Orphanet encyclopedia, February. 2005 Feb.

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