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Kimura's disease soa
1. 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
2. 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
3. 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
16. 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
17. 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
18. 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.
19. 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
20. BPNST vs Kimura’s Lymph Node
Source: http://www.columbianeurosurgery.org/conditions/neurofibroma/
21. 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.
3 distinct lumps within left arm
1st Lesion (1.5 x 1 x 2 cm ): Lying over the lower medial aspect of arm in close relation to the medial cutaneous nerve of the forearm
2nd Lesion (3.5 x 0.7 x 1.5 cm ): Adjacent to 1st lump following the course of the median nerve
3rd Lesion (1.3 x 1.3 x 2.6cm): Within flexor compartment of forearm in close relation to the median nerve and/or anterior interosseous nerve
All the lesions have identical signal characteristics with high T2 and intermediate T1 signal. They enhance homogeneously post contrast.
Compatible with multiple peripheral nerve sheath tumours
3 distinct lumps within left arm
1st Lesion (1.5 x 1 x 2 cm ): Lying over the lower medial aspect of arm in close relation to the medial cutaneous nerve of the forearm
2nd Lesion (3.5 x 0.7 x 1.5 cm ): Adjacent to 1st lump following the course of the median nerve
3rd Lesion (1.3 x 1.3 x 2.6cm): Within flexor compartment of forearm in close relation to the median nerve and/or anterior interosseous nerve
All the lesions have identical signal characteristics with high T2 and intermediate T1 signal. They enhance homogeneously post contrast.
Compatible with multiple peripheral nerve sheath tumours
3 distinct lumps within left arm
1st Lesion (1.5 x 1 x 2 cm ): Lying over the lower medial aspect of arm in close relation to the medial cutaneous nerve of the forearm
2nd Lesion (3.5 x 0.7 x 1.5 cm ): Adjacent to 1st lump following the course of the median nerve
3rd Lesion (1.3 x 1.3 x 2.6cm): Within flexor compartment of forearm in close relation to the median nerve and/or anterior interosseous nerve
All the lesions have identical signal characteristics with high T2 and intermediate T1 signal. They enhance homogeneously post contrast.
Compatible with multiple peripheral nerve sheath tumours
3 distinct lumps within left arm
1st Lesion (1.5 x 1 x 2 cm ): Lying over the lower medial aspect of arm in close relation to the medial cutaneous nerve of the forearm
2nd Lesion (3.5 x 0.7 x 1.5 cm ): Adjacent to 1st lump following the course of the median nerve
3rd Lesion (1.3 x 1.3 x 2.6cm): Within flexor compartment of forearm in close relation to the median nerve and/or anterior interosseous nerve
All the lesions have identical signal characteristics with high T2 and intermediate T1 signal. They enhance homogeneously post contrast.
Compatible with multiple peripheral nerve sheath tumours
Intra op findings:
2 x 2cm nerve sheath tumour adjacent to median nerve just proximal to cubital fossa
1 x 2cm tumour arising from lateral cutaneous nerve of arm just proximal to cubital fossa all tumours arising from nerve sheaths.
1 x 1cm tumour arsing just posterior to Anterior Interosseous Nerve (AIN) at AIN branching junction from median nerve in flexor compartment of forearm
All tumours surrounding but not involving main nerves
Intra op findings:
2 x 2cm nerve sheath tumour adjacent to median nerve just proximal to cubital fossa
1 x 2cm tumour arising from lateral cutaneous nerve of arm just proximal to cubital fossa all tumours arising from nerve sheaths.
1 x 1cm tumour arsing just posterior to Anterior Interosseous Nerve (AIN) at AIN branching junction from median nerve in flexor compartment of forearm
All tumours surrounding but not involving main nerves
Intra op findings:
2 x 2cm nerve sheath tumour adjacent to median nerve just proximal to cubital fossa
1 x 2cm tumour arising from lateral cutaneous nerve of arm just proximal to cubital fossa all tumours arising from nerve sheaths.
1 x 1cm tumour arsing just posterior to Anterior Interosseous Nerve (AIN) at AIN branching junction from median nerve in flexor compartment of forearm
All tumours surrounding but not involving main nerves
All the three lumps are lymph nodes
Preserved nodal architecture with reactive follicular hyperplasia
Prominent eosinophilic infiltrate in the interfollicular and sinusoidal areas, with infiltration into perinodal tissue
Eosinophilic infiltration of germinal centres resulting in folliculolysis and necrosis and Eosinophilic microabscesses present
No evidence of malignancy.
All the three lumps are lymph nodes
Preserved nodal architecture with reactive follicular hyperplasia
Prominent eosinophilic infiltrate in the interfollicular and sinusoidal areas, with infiltration into perinodal tissue
Eosinophilic infiltration of germinal centres resulting in folliculolysis and necrosis and Eosinophilic microabscesses present
No evidence of malignancy.
All the three lumps are lymph nodes
Preserved nodal architecture with reactive follicular hyperplasia
Prominent eosinophilic infiltrate in the interfollicular and sinusoidal areas, with infiltration into perinodal tissue
Eosinophilic infiltration of germinal centres resulting in folliculolysis and necrosis and Eosinophilic microabscesses present
No evidence of malignancy.
All the three lumps are lymph nodes
Preserved nodal architecture with reactive follicular hyperplasia
Prominent eosinophilic infiltrate in the interfollicular and sinusoidal areas, with infiltration into perinodal tissue
Eosinophilic infiltration of germinal centres resulting in folliculolysis and necrosis and Eosinophilic microabscesses present
No evidence of malignancy.
All the three lumps are lymph nodes
Preserved nodal architecture with reactive follicular hyperplasia
Prominent eosinophilic infiltrate in the interfollicular and sinusoidal areas, with infiltration into perinodal tissue
Eosinophilic infiltration of germinal centres resulting in folliculolysis and necrosis and Eosinophilic microabscesses present
No evidence of malignancy.
Unknown etiology
Chronic inflammatory process
? Aberrant immune reaction to an unknown antigenic stimulus
? Genetic
Mast cells play important role by regulating IgE synthesis and orchestrating eosinophilic infiltration