ReportofCases Jean-PaulMeningaud,MD,PhD,FEBOMS,*PoramatePitak-Arnnop,DDS,Diploma(OMS),†PierreFouret,MD,PhD,‡andJacques-CharlesBertrand,MD§ Kimura’sDiseaseoftheParotidRegion:Reportof2CasesandReviewoftheLiterature

(1)

J Oral Maxillofac Surg 65:134-140, 2007

Kimura’s Disease of the Parotid Region:

Report of 2 Cases and Review of the Literature

Jean-Paul Meningaud, MD, PhD, FEBOMS,*

Poramate Pitak-Arnnop, DDS, Diploma (OMS),†

Pierre Fouret, MD, PhD,‡ and Jacques-Charles Bertrand, MD§

Kimura’s disease (KD) is an uncommon chronic inflammatory disorder of unknown etiology. Clinically, it pre- sents as solitary or multiple subcutaneous nodules, predominantly in the head and neck region, typically in the preauricular region, forehead, and scalp.^1-6Other local- izations such as lacrimo-orbital involvement and upper extremities have been reported.^1,6The soft tissue localization is often associated with regional lymphadenopathy and, occasionally, with enlargement of the major salivary gland.^2-5 Sporadic cases in non-Asian peoples with KD have been previously reported,^4,6,7 but there are no reports of patients from islands in the Southwest Indian Ocean. We report 2 cases of KD with hypereosinophilia in patients who are natives of Madagascar and Mauritius. Our report shows that KD should be considered even in non-Asian patients in the diagnosis of hypereosinophilia-associated disease with head and neck subcutaneous mass.

Report of Cases

CASE 1

A 29-year-old man who was a native of Madagascar with no past medical history gradually developed a large, firm mass over the right preauricular region involving the right

zygomatic and temporal areas over 3 months (Fig 1). The cervical lymph nodes were not palpable; physical examination was otherwise unremarkable. Laboratory results were a marked peripheral hypereosinophilia. Because there was no evidence of allergy or parasitic infestation and because the patient had not taken prior medications, hypereosinophilic syndrome was first considered due to the markedly elevated blood eosinophil count. However, the absence of internal organ involvement made this diagnosis questionable. Com- puted tomography (CT) scan revealed an expansively ill- defined subcutaneous mass occupying the parotidomasseteric region; there was no bone involvement. Nevertheless, it was not determined whether the lesion originated from the parotid gland (Fig 2). Magnetic resonance imaging (MRI) showed that the lesion infiltrating the subcutaneous fatty tissue had inhomogeneous low signal intensity on both T1- and T2-weighted images; the lesion margins were ill- defined without a clear plane of cleavage between the mass and the adjacent parotid gland that was displaced and compressed. No evidence of the invasion of masseter muscle and underlying bone could be seen (Fig 3). Under general anesthesia, surgical removal of the mass with preservation of the facial nerve was performed via rhytidectomy. Patho- logic study of the specimen showed a marked reactive follicular hyperplasia with prominent follicles that were sur- rounded by fibrous tissue. Some follicles contained eosinophilic proteinous material; a few demonstrated folliculolysis. The interfollicular infiltrate was rich in plasma cells and eosinophils that formed scattered eosinophilic microabscesses associated with granulomatous inflammation.

Thin-walled vessels were numerous and often grouped in small foci; their endothelial lining was neither epithelioid nor vacuolated. Reed-Sternberg cells, atypical lymphocytes, or Langerhans cells were not identified. Special stains including periodic acid-Schiff, Giemsa, and Brown and Brenn failed to show any infectious agents (Figs 4-6). Therefore, the definite diagnosis was KD. The patient’s condition has remained satisfactory throughout the 1-year follow-up period.

CASE 2

A 25-year-old Mauritius native male presented with a soft, mobile, painless, and slowly growing, approximately for 18 months, mass over the right parotid region in 1991. With the exception of hypereosinophilia, there was no significant medical history. Lymph nodes from the left spinal, left suprahyoid, left suboccipital, and left axillary groups, as well as from the right inguinal group, were enlarged. CT scan showed a well-circumscribed 8.5⫻ 3.5 cm diameter Received from the Teaching Pitié-Salpêtrière Hospital, Paris,

France.

*Consultant Maxillofacial Surgeon, Department of Maxillofacial Surgery and Stomatology.

†Surgical Fellow, Department of Maxillofacial Surgery and Sto- matology.

‡Professor, Department of Anatomy and Pathologic Cytology.

§Professor and Head, Department of Maxillofacial Surgery and Stomatology.

Address correspondence and reprint requests to Dr Meningaud:

Service de chirurgie maxillo-faciale, CHU Pitié-Salpêtrière, 47 bd de l’hôpital. 75651 Paris Cedex 13, France; e-mail: meningaud@noos.fr

doi:10.1016/j.joms.2005.10.043

(2)

mass that originated from the right parotid gland. The patient benefited from right superficial parotidectomy at an- other hospital. Histologic examination demonstrated lymphoid hyperplasia with vascular proliferation and eosinophilic infiltration; therefore, KD was diagnosed. Post- operative facial paralysis was encountered which was an unsatisfactory surgical outcome. In 1999, the patient was referred to us for consultation because of recurrence of KD as a right preauricular lymphadenopathy confirmed by CT scan (Fig 7). To avoid further injury to the facial nerve, the mass was excised via an intraoral approach. The diagnosis of KD was confirmed pathologically. The postoperative course was uneventful. However, in early 2004 the patient presented with a 4 cm diameter mass in the left retromandibular area. CT scan and MRI revealed hyperplasia of the inferior pole of the left parotid gland consistent with lymphoid hyperplasia (Figs 8, 9). A left superficial parotidectomy via a retromandibular approach was performed. His- tologically, the mass contained the prominent lymphoid hyperplasia compartmentalized by fibrous connective tissue; the interfollicular infiltrate was rich in plasma cells and eosinophils that formed eosinophilic microabscesses. The final diagnosis was KD. At the time of last follow-up there had been no recurrence for 1 year. There was no renal complication.

Discussion

KD is an uncommon chronic inflammatory disorder involving subcutaneous tissue, predominantly in the head and neck region. This disease is most frequently associated with regional lymphadenopathy and/or salivary gland involvement.^3-5,8-10Although KD has long been described as a distinct entity since the original report by Kimura,^2-5 it is often confused with angiolymphoid hyperplasia with eosinophilia (ALHE) in many reports.^11-14A long list of synonyms for KD has added to the confusion. However, KD and ALHE are quite different and should be distinguished using clinical and pathologic features. A brief summary of dis-

tinctive features between these entities is presented inTable 1. KD has a predilection for young to middle- aged Asian men; however, some cases have been reported in non-Asian patients. Although both our patients are natives of islands located in the South- west Indian Ocean, many people from these countries are partially of Asian descent. It is generally assumed that there is a genetic predisposition for KD.

Current theories on precise causation have been the subject of debate. Because of the correlations of KD with immunities, it seems suitable to classify this disease in a category of the reactive immune disorders in etiology, and not with tumors.^8-16 Various leukocytes have been advocated in the disease etio- pathogenesis, including mast cells,^17,18eosinophils,¹⁹ and macrophages.^20,21 Recent studies have also sug- gested that the expression of activated CD4(⫹) T- helper 2 (Th2) cells,^10,22 as well as the overexpres- sion of their cytokines, such as granulocyte-

FIGURE 1. Photograph of the patient case 1 who presented with a 3-month history of progressive swelling of the right preauricular regions involving the right zygomatic and temporal areas.

Meningaud et al. Kimura’s Disease of the Parotid Region. J Oral Maxillofac Surg 2007.

FIGURE 2. A and B, Axial CT scan of case 1 showing an expansive, ill-defined, homogeneously hypoattenuated mass occupying the right parotidomasseteric region and extending posteriorly toward the pos- terior border of mandilar ramus. No bony involvement was evident.

(3)

macrophage colony-stimulating factor, tumor necro- sis factor-␣, interleukin (IL)-4, IL-5, eotaxin, and RANTES,^23,24 may play an important role in the disease development by regulating Ig E production and eosinophilic recruitment. Moreover, a clonal T-cell population may contribute to disease development and recurrence.²⁵ Some antigens responsible for the reactive induction of the immune system have been proposed; such as Candida albicans,¹⁶ Epstein-Barr virus,²⁶and human herpes virus-8.²⁷

Clinically, KD lesions simulate neoplasms in the head and neck region,2-5,10,14,16sometimes in the upper limbs.^28,29The lesions present as deeper masses rather than isolated or grouped red papules, plaques, or mucocutaneous lesions that are commonly seen in ALHE.¹⁵Both diseases can cause proptosis, lid swelling, ocular dysmotility, or a palpable mass when en- countering the orbit and ocular adnexa. Nevertheless, KD has been described in the orbit, eyelids, and lacrimal gland more frequently than ALHE.¹¹ For the

FIGURE 3. MRI of case 1 showing ill-defined inhomogeneous hy- pointense lesions located in the subcutaneous tissue of parotidomasseteric region without a clear plane of cleavage between the mass and the adjacent parotid gland appearing displaced and compressed. No evidence of the invasion of masseter muscle and underlying bone can be seen.

FIGURE 4. Photomicrograph of case 1 showing lymph node-like tissues with markedly reactive follicular hyperplasia and granulomatous inflammation interspersed with normal fatty tissue. (Hematoxylin-eosin stain; magnification⫻25.)

FIGURE 5. Photomicrograph of case1 showing germinal centers with lack of distinctive large epithelioid endothelial cells and vascular proliferation. The interfollicular infiltration was rich in plasma cells and eosinophils forming eosinophilic microabscesses. (Hematoxylin-eosin stain; magnification⫻200.)

FIGURE 6. Photomicrograph of case 1 showing numerous and prominent thin-walled vessels with lack of epithelioid or vacuolated endothelial cells. There was no atypia in the lymphoid cell population.

(Hematoxylin-eosin stain; magnification⫻400.)

(4)

non-Asian with KD, some authors have reported its localization in preauricular subcutaneous tissue and cervical lymph nodes of male Caucasians; most had peripheral hypereosinophilia.⁴Lymph nodes and major salivary glands, especially parotid glands, are commonly affected, resembling malignancies.^2,3 Interest- ingly, no bilateral parotid masses have been previously documented, as has been shown in our second case.

Swelling of the extraocular muscle,³⁰juvenile temporal arteritis,³¹and associated coronary artery disease³² are also grouped in accessory lesions of KD, including sleep apnea in cases of laryngeal involvement.³³

Many patients with KD also develop allergic condi- tions and renal involvement. Asthma, chronic urticaria/

eczema, pruritis, rhinitis, cutaneous eosinophilic vascu-

litis, and alopecia areata are frequently associated.^2,5,14 The coexistences of KD and other cutaneous lesions have also been reported, such as lichen amyloidosus.

It is believed that the inflammatory process of KD results in the basal layer damage necessary for colli- sion occurrence of the later lesions.³⁴ Also, it has a high frequency of an association with nephrotic syndrome; a handful of KD pediatric patients with renal diseases or who require dialysis have been reported.10,22,35,36 This renal involvement has been attributed to the deleterious effects of eosinophil granules and possibly to microemboli from the heart in patients presenting with fibroplastic endocarditis

FIGURE 7. CT scan of case 2 showing a recurrent mass as a right preauricular lymphadenopathy. The patient, subsequently, underwent the excision via an intraoral approach as the second operation.

FIGURE 8. CT scan of case 2 before the third operation shows a lesion in the region of the left parotid.

FIGURE 9. MRI of case 2 before the third operation revealed hyper- plasia of the inferior pole of the left parotid gland consistent with lymphoid hyperplasia.

Table 1. THE COMPARISON BETWEEN KD AND ALHE Kimura’s

Disease ALHE

Gender Predominantly

male

Predominantly female Ethnic group Predominantly

Asian

Asian and non-Asian

Localization Deep,

subcutaneous

Superficial, dermal Clinical presentation Nodule Papule

Lymphadenopathy Frequent Rare

Salivary gland involvement

Frequent Rare

Hypereosinophilia Frequent Rare

Serum Ig E Elevated Normal

Predominant cellular component

Lymphoid infiltrate

Vascular hyperplasia Feature of

endothelial cells

Flat Epithelioid

and/or vacuolized Meningaud et al. Kimura’s Disease of the Parotid Region. J Oral Maxillofac Surg 2007.

(5)

or eosinophilic myocarditis. Most secondary forms are usually caused by an immunoallergic process leading to the deposit of immune complexes in glomeruli.

The effects of polynuclear eosinophils could also be caused by the release of cytokines and other media- tors. Additionally, inflammatory process in KD may be involved in refractory hypertension and anemia in patients with end-stage renal disease requiring dialysis,⁹steroid-resistant nephrotic syndrome,³⁶as well as renal insufficiency.³⁷ Fortunately, both our patients had no history of or were not associated with renal disease. Nevertheless, in our opinion, KD patients should be referred for other physical evaluation, par- ticularly for nephropathy.

Imaging evaluation of soft-tissue lesions has under- gone a dramatic evolution with the advent of CT and MRI imaging, which are very useful for definite pre- operative diagnosis. Ultrasonogram (US) can also be useful. CT scan provides the information of this soft tissue lesion without any bony involvement. On CT scans with contrast enhancement this lesion appears as a homogeneous, slightly hyperattenuated mass.

MRI will differentiate its precise nature from other soft tissues tumors. This lesion tends to be heterogeneous-hypointense, sometimes slightly hyperintense on T1- and T2-weighted imagings. Additionally, it will show heterogeneous enhancement after administration of contrast media. Its discrepancies of enhancement degree, by nature, may be attributable to differ- ing degrees of fibrosis and vascular proliferation.³⁸ The lesions always present a subcutaneous woolly- echotexture mass with well- or ill-defined border on US.³⁹In addition, on gray-scale US, hypoechoic and round features with normal hilar architecture and homogeneous internal echoes are always revealed in nodal lesions. The prominent intranodal vessels with a hilar pattern and low intranodal resistance are always revealed on Doppler US, as well as soft tissue;

parotid lesions also show low-resistance vascularity within.⁴⁰

Concerning the presentation in our first case, the exact plane of the lesions should be established to differentiate them from tumors in other origins. The differential diagnosis in emphasis was made in groups of soft tissue tumors, such as lipoma, rhabdomyoma, neurilemmoma, or schwannoma, and lymphoprolif- erative or granulomatous lesions, including Miku- licz’s disease (sialolymphadenitis) or Castelman’s disease.^16,41 Hyperplasia and benign tumors of subcutaneous vascular proliferations should also be in the differential list, including ALHE, bacillary angiomato- sis, intravascular papillary endothelial hyperplasia, Kaposi’s sarcoma, pseudo-Kaposi’s sarcoma, and various types of hemangiomas. As demonstrated in the second case, when lymph nodes are involved in the submental, submandibular, and upper cervical re-

gions, as well as within the parotid gland, it mimics malignancy. The differential diagnosis with reactive lymphadenopathy, subcutaneous soft tissue tumors with lymph node involvement, Hodgkin’s lymphoma, dermopathic lymphoma, parotid tumor with nodal metastasis, and Mikulicz’s disease or Castelman’s disease should not be excluded.^40,41 Differentiation of Langerhans cell histiocytosis and salivary gland neoplasms from KD is also crucial in the region of the parotid gland.⁴²

Diagnosis is achieved by excisional biopsy, which is also the therapy of choice. Microscopically, as com- pared with ALHE, KD shows a marked lymphoprolif- eration with prominent germinal center and some folliculolysis. Also, KD lacks distinctive large epithelioid endothelial cells. Instead, the angioproliferation involved postcapillary venules presenting with thin- walled vessels^4,15are often accompanied by a characteristic inflammatory infiltration, forming eosinophilic microabscesses. In nodal specimens, florid germinal centers, vascularized germinal centers, increased paracortical post-capillary venules, eosinophilic infiltration, and fibrosis are commonly found. Warthin- Finkeldey polykaryocytes are often positive for lymphoid and macrophagic markers. Fine needle aspiration cytology is characterized by an admix- ture of significant numbers of eosinophils, frag- ments of collagenous tissue, epithelioid endothelial cells, and occasional polykaryocytes (Warthin- Finkeldey-type giant cells) against a background of a polymorphous lymphoid population. Therefore, cytology can be a useful adjunct diagnostic tool in KD.^43,44

Treatment options range from conservative obser- vation for asymptomatic patients to surgical excision, steroid therapy, and radiotherapy for the symptom- atic patient. The lesion is benign but may be persistent and difficult to eradicate. Multiple treatment modalities have been proposed for KD, each with limited success or undesirable side effects and recurrence is common.

Treatments include oral corticosteroids,^7,32,41oral retin- oids,⁴⁵oral pentoxifylline,⁴⁶cyclosporine,^34,47,48surgi- cal excision,³ radiotherapy,^26,41,49 and laser therapy.³³ Overall prognosis is favorable, but multiple relapses are possible. There is no evidence of malig- nant transformation and occasionally spontaneous resolution occurs.⁷

Similar to our opinion, some reports have sug- gested that complete surgical excision with/without initial high-dose corticosteroid whenever feasible is the preferred treatment for a well-circumscribed lesion, despite a high recurrence rate.^3,50 With great emphasis on diffusely infiltrative lesions, complete extirpation is virtually impossible and may result in recurrence.¹⁹When surgery is not possible or in sit- uations of recurrence, conservative treatment with

(6)

either corticosteroids^7,14,32,41 or radiation^26,41 often produces a favorable response. On the other hand, many internists advocated medical management (eg, corticosteroids).^7,14,32,41 Some immunomodulators, such as pranlukast or suplatast tosilate, have been observed to support steroid tapering by the possible immunomodulatory mechanism of IL-4 production of Th2 cells.^51,52Because of their effect on the Th2 cells, cyclosporine,^47,48 interferon-alpha,⁵³ and oral pentoxifylline⁴⁶ may also be introduced in cases of steroid-resistant KD subcutaneous masses or ulcers. If the degree of improvement is unsatisfactory or drug resistance occurs, the more radical excision should be considered later. Nicotine, which is an effective treatment of other neutrophil-associated skin disorders (such as pyoderma gangrenosum and orogenital ulcer- ations caused by Behçet’s disease) may be attributable to prominent eosinophilic infiltration such as KD skin lesions.⁵⁴ Prospective, randomized clinical trials, however, are needed to answer whether these medi- caments are a substitute for, or adjunct to, surgery.

Renal involvement of KD subsides most of the time after administration of corticosteroid.⁹ In steroid-resistant cases, KD masses must be sacrificed.³⁶ Care should be taken in KD cases of renal transplantation because both chronic graft rejection and KD involve the Th2-dominant immune response, which may lead to secondary renal graft failure.¹⁰

KD is an uncommon chronic inflammatory disease presenting as a subcutaneous swelling typically in the head and neck area. It has been described more often in Asians. However, it does occur in non-Asians with a similar clinicopathologic presentation. Our 2 cases suggest that KD is no longer just a disease from Oriental countries, although most cases have been from there. KD is a distinctive entity with elusive etiology. It has characteristic histologic features that are important to recognize, including follicular hyperplasia, eosinophil microabscesses, and angioproliferation. The surgical resection (with a safe margin of healthy tissue) with/without initial high-dose steroid therapy is the treatment of choice with risk of recurrence; regression should be monitored over a pro- longed period of time.

Acknowledgment

The authors acknowledge the support of Dr Laurent Giaoui, attending maxillofacial surgeon of Department of Maxillofacial Sur- gery, Teaching Pitié-Salpêtrière Hospital, for the information on the patient case 2.

References

1. Chang B, Ryan D, Kennedy S, et al: Orbital Kimura’s disease in a white child. Br J Ophthalmol 83:1198, 1999

2. Khoo BP, Chan R: Kimura disease: 2 case reports and a literature review. Cutis 70:57, 2002

3. Arshad AR: Kimura’s disease of parotid gland presenting as solitary parotid swelling. Head Neck 25:754, 2003

4. Chen H, Thompson LD, Aguilera NS, et al: Kimura disease: A clinicopathologic study of 21 cases. Am J Surg Pathol 28:505, 2004

5. Lee MW, Bae JY, Choi JH, et al: Cutaneous eosinophilic vascu- litis in a patient with Kimura’s disease. J Dermatol 31:139, 2004 6. Yoganathan P, Meyer DR, Farber MG: Bilateral lacrimal gland involvement with Kimura disease in an African American male.

Arch Ophthalmol 122:917, 2004

7. Daaleman TP, Woodroof J: Kimura’s disease presenting as subcutaneous facial plaque in an African American. Cutis 66:201, 2000

8. Karavattathayyil SJ, Krause JR: Kimura’s disease: A case report.

Ear Nose Throat J 79:195, 2000

9. Hsu PY, Yang HY, Lin CC, et al: Refractory hypertension and anemia in end-stage renal disease: An unusual manifestation of Kimura’s disease. Ren Fail 25:499, 2003

10. Lu HJ, Tsai JD, Sheu JC, et al: Kimura disease in a patient with renal allograft failure secondary to chronic rejection. Pediatr Nephrol 18:1069, 2003

11. Buggage RR, Spraul CW, Wojno TH, et al: Kimura disease of the orbit and ocular adnexa. Surv Ophthalmol 44:79, 1999 12. Ferri M, Ross C, Harvey JT: Recurrent orbital angiolymphoid

hyperplasia with eosinophilia (Kimura’s disease). Can J Oph- thalmol 34:290, 1999

13. Seregard S: Angiolymphoid hyperplasia with eosinophilia should not be confused with Kimura’s disease. Acta Ophthal- mol Scand 79:91, 2001

14. Ray V, Boisseau-Garsaud AM, Hillion G: Maladie de Kimura à localisation palatine chez un Antillais. Rev Med Interne 24:253, 2003

15. Cawson RA, Binnie WH, Barrett AW, et al (eds): Oral Diseases.

Clinical and Pathological Correlations. Edinburgh, Mosby, 2001, pp 1033-1034

16. Regezi JA, Scuibba JJ: Lymphoid lesions, in Regezi JA, Scuibba JJ (eds). Oral Pathology. Pathologic Correlations (ed 3). Phila- delphia, Saunders, 1999, pp 272-273

17. Wong KT, Shamsol S: Quantitative study of mast cells in Ki- mura’s disease. J Cutan Pathol 26:13, 1999

18. Aoki M, Kawana S: The ultrastructural patterns of mast cell degranulation in Kimura’s disease. Dermatology 199:35, 1999 19. Lee CT, Huang CC, Lam KK, et al: Kimura’s disease in a chronic

hemodialysis patient. Am J Nephrol 21:47, 2001

20. Saita N, Goto E, Yamamoto T, et al: Association of galectin-9 with eosinophil apoptosis. Int Arch Allergy Immunol 128:42, 2002

21. Hosaka N, Minato T, Yoshida S, et al: Kimura’s disease with unusual eosinophilic epithelioid granulomatous reaction: A finding possibly related to eosinophil apoptosis. Hum Pathol 33:561, 2002

22. Rajpoot DK, Pahl M, Clark J: Nephrotic syndrome associated with Kimura disease. Pediatr Nephrol 14:486, 2000

23. Tsukadaira A, Kitano K, Okubo Y, et al: A case of pathophysi- ologic study in Kimura’s disease: Measurement of cytokines and surface analysis of eosinophils. Ann Allergy Asthma Immu- nol 81:423, 1998

24. Kimura Y, Pawankar R, Aoki M, et al: Mast cells and T cells in Kimura’s disease express increased levels of interleukin-4, interleukin-5, eotaxin and RANTES. Clin Exp Allergy 32:1787, 2002

25. Chim CS, Fung A, Shek TW, et al: Analysis of clonality in Kimura’s disease. Am J Surg Pathol 26:1083, 2002

26. Nagore E, Llorca J, Sanchez-Motilla JM, et al: Detection of Epstein-Barr virus DNA in a patient with Kimura’s disease. Int J Dermatol 39:618, 2000

27. Kemeny E, Nagy SZ, Nagy F, et al: Membranous nephropathy accompanied by HHV8-DNA-positive angiolymphoid hyperplasia of the skin with eosinophilia: Lack of HHV8 viral DNA in the kidney biopsy. Clin Nephrol 61:295, 2004

28. Lee JH, Park HJ, Kim YC, et al: Two cases of Kimura’s disease with unusual clinical manifestations. Dermatology 201:162, 2000

(7)

29. Sanders M, Raj R, Miller M, et al: Kimura’s disease: Upper limb involvement in a Pacific Island man. ANZ J Surg 73:465, 2003 30. Kanazawa S, Gong H, Kitaoka T, et al: Eosinophilic granuloma (Kimura’s disease) of the orbit: A case report. Arch Clin Exp Ophthalmol 237:518, 1999

31. Watanabe C, Koga M, Honda Y, et al: Juvenile temporal arteritis is a manifestation of Kimura disease. Am J Dermatopathol 24:43, 2002

32. Horigome H, Sekijima T, Ohtsuka S, et al: Life threatening coronary artery spasm in childhood Kimura’s disease. Heart 84:5, 2000

33. Okami K, Onuki J, Sakai A, et al: Sleep apnea due to Kimura’s disease of the larynx. Report of a case. ORL J Otorhinolaryngol Relat Spec 65:242, 2003

34. Teraki Y, Katsuta M, Shiohara T: Lichen amyloidosus associated with Kimura’s disease: Successful treatment with cyclosporine.

Dermatology 204:133, 2002

35. Hung CC, Liao PL, Chang CT, et al: Steroid-sensitive anemia in a boy on dialysis—An association with Kimura disease. Pediatr Nephrol 15:183, 2000

36. Chartapisak W, Opastirakul S: Steroid-resistant nephrotic syndrome associated with Kimura’s disease. Am J Nephrol 22:381, 2002

37. Romao JE, Saldanha LB, Ianez LE, et al: Recurrence of focal segmental glomerulosclerosis associated with Kimura’s disease after kidney transplantation. Am J Kidney Dis 31:3, 1998 38. Hiwatashi A, Hasuo K, Shiina T, et al: Kimura’s disease with

bilateral auricular masses. AJNR Am J Neuroradiol 20:1976, 1999

39. Ching AS, Tsang WM, Ahuja AT, et al: Extranodal manifestation of Kimura’s disease: Ultrasound features. Eur Radiol 12:600, 2002

40. Ahuja A, Ying M, Mok JS, et al: Gray scale and power Doppler sonography in cases of Kimura disease. AJNR Am J Neuroradiol 22:513, 2001

41. Wade B, Arteaga C, Kraemer P, et al: Le granulome eosinophile des tissus mous ou maladie de Kimura: A propos d’un cas. Med Trop 58:482, 1998

42. Darvishian F, Hirawat S, Teichberg S, et al: Langerhans’ cell histiocytosis in the parotid gland. Ann Clin Lab Sci 32:201, 2002

43. Boccato P, Mannara GM, Rinaldo A, et al: Kimura’s disease of the intraparotid lymph nodes: Fine needle aspiration biopsy findings. ORL J Otorhinolaryngol Relat Spec 61:227, 1999 44. Deshpande AH, Nayak S, Munshi MM, et al: Kimura’s disease.

Diagnosis by aspiration cytology. Acta Cytol 46:357, 2002 45. Boulanger E, Gachot B, Verkarre V, et al: All-trans-retinoic acid

in the treatment of Kimura’s disease. Am J Hematol 71:66, 2002 46. Hongcharu W, Baldassano M, Taylor CR: Kimura’s disease with oral ulcers: Response to pentoxifylline. J Am Acad Dermatol 43:905, 2000

47. Kaneko K, Aoki M, Hattori S, et al: Successful treatment of Kimura’s disease with cyclosporine. J Am Acad Dermatol 41:

893, 1999

48. Nakahara C, Wada T, Kusakari J, et al: Steroid-sensitive nephrotic syndrome associated with Kimura disease. Pediatr Nephrol 14:482, 2000

49. Hareyama M, Oouchi A, Nagakura H, et al: Radiotherapy for Kimura’s disease: The optimum dosage. Int J Radiat Oncol Biol Phys 40:647, 1998

50. Terakado N, Sasaki A, Takebayashi T, et al: A case of Kimura’s disease of the hard palate. Int J Oral Maxillofac Surg 31:222, 2002

51. Tsukagoshi H, Nagashima M, Horie T, et al: Kimura’s disease associated with bronchial asthma presenting eosinophilia and hyperimmunoglobulinemia E which were attenuated by suplatast tosilate (IPD-1151T). Intern Med 37:1064, 1998 52. Ohtsuka Y, Shimizu T, Fujii T, et al: Pranlukast regulates tu-

mour growth by attenuating IL-4 production in Kimura disease.

Eur J Pediatr 163:416, 2004

53. Kanny G, Cogan E, Marie B, et al: Cas de maladie de Kimura traité par interferon et corticothérapie. Rev Med Interne 20:

522, 1999

54. Kanekura T, Usuki K, Kanzaki T: Skin disorders with prominent eosinophilic infiltration treated successfully with nicotine. Re- port of two cases. Dermatology 208:153, 2004

J Oral Maxillofac Surg 65:140-143, 2007

Sialectasis of Stensen’s Duct With an Extraoral Swelling: A Case Report With

Surgical Management

Harold D. Baurmash, DDS*

The definition of sialectasis, according to the Merriam Medical dictionary, is “a dilated salivary duct.” When dealing with Stensen’s duct, such dilations occur as a

consequence of intraductal obstructive objects such as sialoliths or polyps (papillomas),¹ but most commonly with ductal stenosis or narrowing. Ductal stenosis may occur secondary to sialolithotomy, especially if the duct is sutured following stone removal, traumatic ductal injury with resultant fibrosis, or as a consequence of long standing ductal inflammation associated with chronic parotitis.

Dilations will vary in size depending on the severity of the obstruction, the elasticity of the duct, and the degree of gland function. In the case of chronic parotitis, mild to moderate dilations will be encountered, resulting in the so-called “sausage effect” where

*Formerly, Clinical Professor of Oral and Maxillofacial Surgery, Columbia University, School of Dental and Oral Surgery, NY, New York.

Address correspondence and reprint requests to Dr Baurmash:

4666 Hazleton Lane, Lake Worth, FL 33467; e-mail Hali2533@

aol.com

doi:10.1016/j.joms.2005.12.033