Abstract: Kimura Disease Is A Rare Form of Chronic Inflammatory Disorder Involving

Kimura Disease
A Case Report
Agus Priyo Wibowo , Ni Ketut Sungowati2, Juanita2, Imeldy Prihatni Purnama2
1
1
Resident Department of Anatomical Pathology, Faculty of Medicine, Hasanuddin University , Dr.Wahidin Sudirohusodo Hospital
2
Lecturer Department of Anatomical Pathology, Faculty of Medicine, Hasanuddin University , Dr.Wahidin Sudirohusodo Hospital
Makassar – Indonesia
Abstract : Kimura disease is a rare form of chronic inflammatory disorder involving

subcutaneous tissue, predominantly in the head and neck region and frequently associated
with regional lymphadenopathy. The etiology of Kimura disease is still unknown but may be
due to impairment with immune system. Kimura's disease should be suspected when the
clinical finding of painless unilateral cervical adenopathy and hypereosinophilia presented.
And an definite diagnose arise by histopathology examination, with hematoxylin-eosin
staining showed follicular hyperplasia with perifollicular fibrosis, dense eosinophil infiltrate,
and proliferation of capillary vessel lined by normal, flat, spindle-shaped endothelial cells.
We reported A 37 years old man with history of soft tissue tumor on facialis dextra region,
lymphadenopathy colli dextra, and exopthalmic bulbus oculi dextra over the last 10 year’s.
Laboratory assessment on hematologic review showed that eosinofil 45.800/ul.
Keywords : Kimura disease, case report
Introduction
Kimura’s disease is a rare chronic benign disorder young men of Asian race but is rare ,
usually affecting in western countries.1,2 Kimura’s disease is usually seen in young adults,
with most patients being 20-40 years of age. Men are affected more commonly than women,
with a 3:1 ratio.3 The major physical manifestation of the disorder is slowly enlarging
subcutaneous masses, often in the head and neck area and usually in association with
peripheral blood and tissue eosinophilia, incombination with markedly increased serum IgE
concentrations.1 The etiology of Kimura Disease is still unknown but may be due to
impairment or interference with immune regulation, atopic reaction to a persistent antigenic
stimulus by arthropod bites, virus, and neoplasm.3 Peripheral eosinophilia and the presence of
eosinophils in the inflammatory infiltrate suggest that Kimura Desease might be a kind of
hypersensitivity reaction.
Case report
A 37 years old man presented to oncologic surgery department with history of soft tissue
tumor on facialis dextra region, lymphadenopathy colli dextra, and exopthalmic bulbus oculi
dextra over the last 10 year’s and diagnose by Soft tissue tumor facial dextra with differential
diagnose tumor of fascial dextra suspek hemangioma. Laboratory assessment on hematologic
review showed that eosinofil 45.800/ul, the other value is on normal range. On radiologic
expertise of AP lateral skull x-ray note with soft tissue density regio mandibulae dextra and
on MSCT scan evaluation note with soft tissue tumor regio facialis dextra, lymphadenopathy
regio colli dextra, exopthalmic bulbus oculi dextra and retension cyst sinus maxillaris sinistra.
Tissue was accepted from oncologic surgery department and evaluated on Patologi Anatomi
laboratory departemen, we accepted about four divers tissue that some coated with skin, size
of tissue about 10x7x4cm, 7x5x2cm, 7x3x2cm, and 4x2x1cm, soft to firm in consistency.
Histopathology examination, stain with hematoxylin-eosin showed on dermis and subcutis,
hyperplasia follicles of varying sizes with prominent germinal centre and perifolikular
fibrosis [Fig1]. Eosinophilic micro abscesses seen in focal areas, folliculolysis by eosinophils
was also seen[Fig 2, Fig 3]. Many blood vessels with flat endothelial cells were seen and
lined by normal, flat, spindle-shaped endothelial cells [Fig 4].
Figure 1: Hyperplasia of lymphoid follicles with prominent germinal centre, eosinophil
infiltration seen between follicles and perifolikular fibrosis. H and E stain (4x objective)
Figure 2: Eosinophil infiltration, follicles with prominent germinal centre. H and E

stain(10x objective)
Figure 3: Folikel with abundant eosinofil. H and E stain(40x objective)
Figure 4 : Proliferation of capillary vessel lined by normal, flat, spindle-shaped endothelial

cells with many eosinofile. H and E stain(40x objective)
Discussion
Kimura disease is a chronic inflammatory disorder involving subcutaneous tissue and lymph
nodes predominantly in the head and neck region frequently involving periauricular and
submandibular region.4 More common among Asians, predominant in young men aged
between 20-40years. The onset is insidious, manifests as a painless enlarging nodular mass
located deep in the subcutaneous tissues and in almost all cases it involves regional
lymphnodes. The lesions are single in 60% of cases, occasionally an isolated enlarged
lymphnode may be the only presentation. Some cases can present with generalized
lymphadenopathy. Peripheral blood eosinophilia and elevated serum Ig E levels are the
constant features.5 Definitive diagnosis can be obtained by histological examination of the
excised lesion.6 Histopathology shows markedly hyperplastic follicles with reactive germinal
centre. Diffuse eosinophilia, eosinophilic microabscess and infiltration of germinal centre
sometimes resulting in folliculolysis. Vascular hyperplasia with normal, flat endothelial.
Immunohistochemistry shows deposits of IgE in the germinal centre.7 Ultrasound, MSCT and
Magnetic Resonance Imaging (MRI) might be diagnostic and can help staging the extent and
progression of the disease as well as the lymph node involvement.
Differential diagnosis includes mainly angiolymphoid hyperplasia with eosinophilia(ALHE)
the vascular proliferation is most significant in ALHE, forming aggregates or lobules
comprised of plump endothelial cells . Others are Castleman disease, drug reactions, parasitic
infestations, dermatopathic lymphadenopathy.8
Conclusion
Kimura Disease is diagnosed based on histopathological findings of the excised lesion.
Hyperplastic follicles whit abundant eosinophil and angiolymphoid proliferation with normal
endotel are characteristic findings. Above case was diagnosed as Kimura Disease on
histopathology which showed the characteristic features.
References
1. Kimura T, Yoshimura S, Ishikaura E. Unusual granulation combined with hyperplastic
change of lymphatic tissue. Trans Soc Pathol Jpn 1948;37:179-80.
2. Sorbello M, Laudini A, Morello G, Sidoti MT, Maugeri JG, Giaquinta A, et al.
Anaesthesiological implications of Kimura’s disease: a case report. J Med Case Reports
2009;25(3):7316.
3. Tseng CF, Lin HC, Huang SC, Su CY. Kimura’s disease presenting as bilateral parotid
masses. Eur Arch Otorhinolaryngol 2005;262(1):8–10.
4. S Massimiliano, L Alessandro, M Gianluigi et al. Anaesthesiological implications of
Kimura Disease. A case report. J Med Case Reports. 2009; 3: 7316.
5. Loachim LH, Medeiros JH. Lymphadenopathies. Lymphadenopathies associated with
clinical syndromes. Kimura lymphadenopathy. Loachim’s lymph node pathology. 4th edition.
Lippincott Williams and Wilkins. Philadelphia: 190-192.
6. Tseng CF, Lin HC, Huang SC, Su CY. Kimura’s Disease presenting as bilateral parotid
masses. Eur Arch Otorhinolaryngol. 2005 Jan; 262(1): 8-10.
7. Kou TT, Shih LY, Chan HL. Kimura’s Disease. Involvement of regional lymph node and
distinction from angiolymphoid hyperplasia and eosinophilia. Am J Surg Pathol. 1998 Nov;
12(11): 843-854.
8. Kristina Buder, Sabrina Ruppert, Axel Trautmann, Eva-Bettina Bröcker, Matthias
Goebeler, Andreas Kerstan. Angiolymphoid hyperplasia with eosinophilia and Kimura’s
disease – a clinical and histopathological comparison. Deutsche Dermatologische
Gesellschaft (DDG). 2014

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Kimura Disease

Abstract : Kimura disease is a rare form of chronic inflammatory disorder involving

Figure 2: Eosinophil infiltration, follicles with prominent germinal centre. H and E

Figure 4 : Proliferation of capillary vessel lined by normal, flat, spindle-shaped endothelial

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