Kimura’s disease a case report


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Kimura’s disease a case report

  1. 1. ISSN 0975-8437 INTERNATIONAL JOURNAL OF DENTAL CLINICS 2011:3(1):115-116 CASE REPORTKimura’s Disease: A case reportShilpa.C.Patel, Vishal.R. DaveAbstract Kimura’s disease is a rare inflammatory disorder of unknown cause, primarily seen in youngAsian males. The disease is characterized by a triad of painless subcutaneous masses in head and neckregion, blood and tissue eosinophilia and moderately elevated serum immunoglobulin E levels. Here, wepresent a case report of this rare entity of unusual age presentation with classical histopathogical features.Key Words: Kimura’s Disease; Eosinophilia; Parotid Gland; Lymphadenopathy Received on: 22/09/2010 Accepted on: 06/12/2010Introduction Kimura’s disease (KD) is a rare chronic number. The histopathological feature ofangiolymphoid proliferative soft tissue disorder excisional biopsy was matched with that ofof unknown origin.(1) It occurs most often in incisional one. The patient was supplementedyoung and middle aged asian male. It is most with oral Dexamethasone for total duration of 6common in head and neck region, with a weeks (0.5 mg twice daily for 1 week and thenpredilection for preauricular area. Typical tapered over 5 weeks). The patient wasclinical presentations are painless subcutaneous apparently normal without any local recurrencemasses, regional lymph node enlargement, blood & systemic involvement within 6 month ofand tissue hyper eosinophilia, and markedly follow-up period.elevated serum IgE levels.(1) According to the Discussionprevious medical literatures, Kimura’s disease Kimura’s disease first described in1937has a high recurrence rate so early & definitive in Chinese literature by H.T.Kimm and C.Szetodiagnosis of the disease is vital for effective and they termed it as “eosinophilic hyperplastictreatment plan.(1) Here we report a case of lymphogranuloma”.(2) The disease becameKimura’s disease with clinico-pathological widely known as Kimura’s disease after Kimuradifferential diagnosis. and colleagues reported two cases of unusualCase Report granulation combined with hyperplastic changes A 65 year old man reported to of lymphoid tissue.(1) The etiopathogenesis ofoutpatient department with chief complaint of KD remains unknown and it is consideredpainless swelling on right side of face in parotid nowadays as an allergic disease and it seems toregion since 4 months but it was painful on be a systemic immunological disorder.mastication. Here we summarized details of Eosinophilia and increased serum IgE levelspresent case in the following Table-1. With a make KD be considered a CD4 (+) T helper 2provisional diagnosis of salivary gland (Th2) allergic reactions. Th2 cells would produceneoplasm, an incisional biopsy was performed. interleukins (IL) IL-4, IL-5 and IL-13, which, inThe section stained with Haematoxylin & Eosin turn, would act in B cells favoring the productionrevealed unencapsulated ill-defined lesion with of antigen-specific IgE. Th2 cell proliferationlymphoid follicle formation, fibrosis and chronic and the overexpression of cytokines would playinflammatory cell infiltration with predominant an essential role in the development of theeosinophils (Figure 1a, 1b, 1c, 1d). Inter disease.(3)follicular areas show increased number of It occurs predominantly as a unilateralvenules with endothelial cell proliferation. In manifestation in the head and neck and it issome areas lymphoid follicle were infiltrated by frequently associated with regionaleosinophils with resultant eosinophilic micro lymphadenopathy with or without involvementabscesses. With a diagnosis of Kimura’s disease, of salivary glands.(4) In the present case, ethe patient was undergone superficial lobe presence unilateral parotid gland swelling withparotidectomy with removal of multiple regional regional lymphadenopathy led to the clinicallymph nodes with vicinity. diagnostic dilemma of other pathologic entities The excisional biopsy specimen affecting salivary glands & lymph nodes. Thereceived was firm nodular soft tissue measuring differential diagnosis of Kimuras diseaseapproximately 4.2X2X1.5 cm3 and two in frequently in previous literatures are©INTERNATIONAL JOURNAL OF DENTAL CLINICS VOLUME 3 ISSUE 1 JANUARY-MARCH 2011 115
  2. 2. ISSN 0975-8437 INTERNATIONAL JOURNAL OF DENTAL CLINICS 2011:3(1):115-116Lymphomas, salivary gland neoplasms, benign (AIL). Constant classical features of KD includelymphoepithelial lesions (BLL / Mikulicz’s numerous lymphoid follicles, mixeddisease), Angiolymphoid hyperplasia with inflammatory infiltrate composed mainly ofeosinophilia (ALHE/ Epitheloid hemangioma) eosinophils and increased amount of postand angioimmunoblastic lymphadenopathy capillary venules.(5)Age/Sex location Physical findings lymphadenopathy Systemic involvement Laboratory investigation65/Male Parotid (right) 3.5X3.5 cm firm, Regional No renal involvement Eosinophilia (36%) Elevated nontender & normal lymphadenopathy erythrocyte sedimentation rate overlying skin (ESR) Raised IgE levelsTable-1 clinical & investigation details of present caseFigure 1a. Lymphocytic infiltration with follicle formation and fibrosis .(4x), 1b. Well defined lymphoid follicle withprominent germinal centre, (10x) 1c. Chronic Inflammatory cell infiltration with predominant eosinophils withmicroabscesses.(40x), 1d. Interfollicular areas showing mixed inflammation with prominent eosinophils andproliferation of venules.(40x), 1e. Interfollicular areas showing increased number of venules with endothelial cellproliferation.(40x) Till date there is no definite treatment Head, Department of Oral & Maxillofacial Pathology,for KD is well established.(2) However the MGM Dental College & Hospital, Navi Mumbai, 2.treatment plan should aim at preservation of vital Dr. Vishal.R.Dave, M.S, Assistant Professor,structures associated with the lesions & cosmetic Department of ENT, GCS Medical College, Ahmedabad, Gujarat, India.rehabilitation, while preventing recurrences. ReferencesTreatment options range from observation & 1. Chung YG, Jee WH, Kang YK, Jung CK, Parkfollow-up of mild & symptomatic cases to GS, Lee A, Bahk WJ, Cho HM, Park JW.conservative surgical approach, medication & Kimura’s disease involving a long bone. Skeletalradiotherapy in symptomatic & recurrent cases. Radiology 2010; 39(5):495-500.If the lesion is primary, localized or present in 2. Pitak-Arnnop P, Bellefqih S, Chaine A,young age then surgical approach is more Dhanuthai K, Bertrand JC, Bertolus C. Head andpreferable. According to previous literature, only neck lesions of Kimuras disease: Exclusion ofsurgical approach had high incidence of human herpesvirus-8 and Epstein-Barr virus by in situ hybridisation and polymerase chain reaction.recurrence.(1) If the lesion is recurrent with An immunohistochemical study. Journal ofsystemic involvement, application of medication Cranio-Maxillofacial Surgery 2010; 38 (4):266-like corticosteroid and immunosuppressive 70.agents have been shown to decrease size of the 3. Briggs PL. Kimura disease is not angiolymphoidlesion.(2) Irradiation should be considered in hyperplasia with eosinophilia: clinical andpatients resistant to the steroid or to prevent the pathological correlation with literature reviewpatient from deleterious effect of long term use and definition of diagnostic criteria. Anaisof steroid.(5) Brasileiros de Dermatologia 2006;81(2):167-73. 4. Kung ITM, Gibson J, Bannatyne P. KimurasConclusion Disease: A Clinico-Pathological Study Of 21 In conclusion this paper draws attention Cases And Its Distinction From Angiolymphoidon such a rare chronic inflammatory disease Hyperplasia With Eosinophils.which mimics neoplastic conditions. This disease Pathology1984;16(1):39-44.should be considered in differential diagnosis of 5. Jani A, Coulson M. Kimuras disease. A typicalpatients presented with head & neck mass and case of a rare disorder. Western Journal oflymphadenopathy and investigated accordingly Medicine 1997; 166 (2) this disease has good prognosis. Knowledge Address for Correspondenceof signature features of Kimura’s disease put the Dr. Shilpa C Patel, M.D.S, 801, Osho Kabir, Plot No 42, Sector 10,physicians in a better position to evaluate its New Panvel, Maharashtra,India- 410206.clinical outcome and optimal treatment regimen. Email: dr_shilpa0769@yahoo.comAffiliations of Authors: 1. Dr. Shilpa.C.Patel,M.D.S., Oral & Maxillofacial Pathology, Professor & Source of Support: Nil, Conflict of Interest: None Declared©INTERNATIONAL JOURNAL OF DENTAL CLINICS VOLUME 3 ISSUE 1 JANUARY-MARCH 2011 116