Recurrent Kimura's disease of head and neck treated with intensity-modulated radiotherapy.

Kimura's disease (KD) is a rare, chronic inflammatory disorder of unknown aetiology, which commonly affects men of the Asian race. Here, we present a case capsule of a 39- year-old man with KD of the left cheek, managed initially by surgery alone. He developed local recurrence after 6 months and was treated with steroids and isotretinoin. Eventually, steroids were discontinued due to toxicity and the lesion progressively increased in size. The patient was successfully treated using intensity-modulated radiotherapy with simultaneous integrated boost as a primary modality with minimal adverse effects. The patient has good local control and cosmetic outcome with no radiation-related toxicity at a follow-up period of 28 months.

Novel Application of 32P Brachytherapy: Treatment of Angiolymphoid Hyperplasia with Eosinophilia in the Right Auricle with 8-Year Follow-Up.

BACKGROUND: Angiolymphoid hyperplasia with eosinophilia (ALHE) is a distinctive benign vascular disease that can be challenging to treat due to inconsistent results for various treatment modalities such as surgical excision, corticosteroids, radiotherapy, laser therapy, and other therapies, so novel approaches are needed to improve treatment outcomes. MATERIALS AND METHODS: ALHE on the right auricle of a 54-year-old Chinese woman underwent brachytherapy using 32P simple drug membranes for five times. The 32P brachytherapy involving simple drug membranes of brachytherapy began by diluting a 32P solution with 0.9% NaCl solution to produce a radioactivity of 69.2-74.7 MBq/mL(1.87-2.02 mCi/mL). The drug membranes were removed between 48 and 72h after application. There were intervals ranging from 65 to 72d between the membrane application periods, and the last treatment was in June 2010. RESULTS: After the 32P brachytherapy, follow-up results over the course of 8 years were promising. The regional symptoms disappeared, the right preauricular swelling decreased, the subcutaneous nodules decreased in size, the exudate disappeared, and the skin appearance improved. CONCLUSIONS: This case indicated that 32P brachytherapy may represent a novel ALHE treatment method that produces a favorable long-term outcome.

Three-dimensional customized bolus for intensity-modulated radiotherapy in a patient with Kimura's disease involving the auricle.

In radiotherapy, a commercial bolus often does not provide a suitable fit over irregular surfaces. To address this issue, we fabricated a customized bolus using 3D printing technology. The aim of our study was to evaluate the application of this 3D-printed bolus in a clinical setting. The patient was a 45-year-old man with recurrent Kimura's disease involving the auricle, receiving radiotherapy in our oncology department. A customized bolus, 5mm in thickness, was fabricated based on reconstruction of computed tomography (CT) images. The bolus was printed on a Dimension 1200 series SST 3D printer. Repeat CT-based simulation indicated an acceptable fit of the 3D-printed bolus to the target region, with a maximum air gap of less than 5mm at the tragus. Most of the surface area of the target region was covered by the 95% isodose line. The plan with the 3D-printed bolus improved target coverage compared to that without a bolus. And the plan with the 3D-printed bolus yielded comparable results to those with the paraffin wax bolus. In conclusion, a customized bolus using a 3D printer was successfully applied to an irregular surface.

Comparison of Local Recurrence Rate of Three Treatment Modalities for Kimura Disease.

Eosinophilic hyperplastic lymphogranuloma, also known as Kimura disease, is a benign and chronic inflammatory condition, predominantly involving the head and neck region. Surgical excision, radiotherapy (RA), surgical resection combined with low-dose postoperative radiotherapy and oral corticosteroids are 4 treatment modalities reported to control this disease effectively. Local recurrence, however, is common and the optimum treatment for Kimura disease is controversial. Thus, the present meta-analysis was performed to identify the treatment modality associated with the lowest local recurrence. Electronic databases (Cochrane Library, Wiley Online Library, PubMed, Chinese National Knowledge Infrastructure, and Wanfang Data) were searched. Data were also obtained from other sources such as related references and communication with the relevant authors. Two reviewers screened the literature according to preselected criteria. All studies involving different treatments for Kimura disease were collected. After data extraction and research quality assessment, the meta-analysis of 22 studies involving 570 patients was conducted using STATA 12.1 software. Meta-analysis revealed that administration of RA or surgical excision alone were inferior in controlling local recurrence compared with surgical resection combined with postoperative RA (risk ratio (RR) = 2.72; 95% confidence interval (CI), 1.47-5.04 and RR = 4.72; 95% CI, 2.53-8.82). Surgical excision alone did not show significant advantage in controlling local recurrence compared with RA alone (RR = 2.13; 95% CI, 0.88-5.17). Surgical resection combined with postoperative RA is superior to either surgery or RA alone in treating Kimura disease. More large scale prospective randomized controlled trials, however, should be conducted to assess the long-term effects and safety issues.

Successful treatment of recurrent Kimura's disease with radiotherapy: a case report.

Kimura's disease is a rare, chronic inflammatory disorder affecting the skin and subcutaneous tissue, predominantly in the head and neck region. It is benign but may be recurrent and difficult to eradicate. A case of recurrent Kimura disease in a 53-year-old man was reported. Radiation therapy was performed for recurrence after surgical excision twice. The prescribed radiation dose was 36 Gy. With a follow-up time of 68 months, the patient was free of the disease.

Kimura's disease: an unusual glandular involvement with blood and tissue eosinophilia.

Kimura's disease is a rare cause of a progressive neck swelling associated with blood and tissue eosinophilia. Though it is a benign disease, however, its unrelating course and unpredictable response to the therapeutic interventions, poses a great challenge to the treating physician, the patients and the caregiver. Here is one such case of Kimura's disease.

Usefulness of thermography techniques for evaluating the disease activity in Kimura's disease.

Kimura's disease (KD) is a rare, chronic inflammatory disorder predominantly affecting the head and neck. A case of KD in a 30-year-old man is reported. Thermography was useful for evaluating the activity of the condition.

Kimura disease: diagnostic imaging findings and surgical treatment.

We have encountered 11 cases of Kimura disease, comprising 10 males and 1 female. The ages at presentation ranged from 16 to 48 years, with a mean of 31.5 years. The sites of the subcutaneous masses were bilateral posterior auricular regions in two cases, frontal region in two cases, temporal region in two cases, head region in one case, parotid region in two cases, parotid and temporal regions in one case, and left earlobe in one case. The interval from onset to surgery ranged from 1 to 10 years, with a mean of 4.7 years. For diagnostic imaging, a combination of magnetic resonance imaging (MRI) and ultrasonography had a high diagnostic value. MRI depicted abnormalities at sites in contact with bone, such as posterior auricular regions, and sites with abundant soft tissue, such as parotid and cheek regions. Diffuse atrophy of subcutaneous fat was observed at the sites of the masses. On ultrasonography, the interior of lymph nodes was homogeneous and hyperechoic, whereas the periphery was hyperechoic, and blood vessels entering lymph nodes were clearly depicted. Surgery was performed in all cases. Postoperative adjuvant radiotherapy was conducted in one patient and radiotherapy and steroid therapy in one other patient. There were two relapses, and both were excised by repeated surgery. Surgical excision of the subcutaneous mass in Kimura disease has the advantages that the treatment period is short and precise histopathologic diagnosis can be obtained.

Radiotherapy in Kimura's disease: a report of eight cases.

Kimura's disease is a rare condition of chronic inflammatory disorder affecting the skin and subcutaneous tissue. It is predominantly in the head and neck region. The lesion is benign but may be persistent/ recurrent and difficult to eradicate. Several forms of treatment have been used, including surgical excision, intralesional and oral corticosteroid, cryotherapy and radiotherapy. The authors report eight cases with histopathology consistent with Kimura s disease who received radiation therapy as a primary treatment or secondary treatment for recurrence after surgical excision in the Division of Therapeutic Radiology and Oncology, Chiang Mai University. The prescribed radiation doses varied from 30-40 Gy. With the mean follow-up time of 21 months, all eight patients were still free from disease at the time of analysis.

Radiotherapy for Kimura's disease: case report and review of the literature.

Kimura's disease is a chronic inflammatory disease of uncertain etiology which involves subcutaneous tissue, presents as a tumor-like lesion and predominantly occurs in the head and neck region. Radiotherapy has been used for treating unresectable or recurrent lesions. We report a 32-year-old male who presented with a mass on the right side of the neck. The mass measured 52 x 40 mm. Peripheral eosinophilia and 5-fold increase in serum IgE level were found. Pathological evaluation following subtotal excision of the mass confirmed Kimura's disease and the patient was given prednisolone p.o. Local recurrence was observed one month later for which the patient received local radiotherapy. No recurrence has been observed up to 30 months in the post-radiotherapy period. Effective radiotherapy given in a favorable schedule and dosage could be a highly effective alternative when other treatment modalities are unsuccessful.

Outcomes of Kimura's disease after radiotherapy or nonradiotherapeutic treatment modalities.

PURPOSE: To evaluate the clinical outcome of Kimura's disease and to identify the optimal treatment regimen for Kimura's disease. METHODS AND MATERIALS: Between 1984 and 2003, 14 patients with Kimura's disease were treated with radiotherapy (RT) and 9 patients were treated with local excision or systemic steroids. The radiation doses ranged from 20 to 45 Gy. Immunohistochemical studies were performed in 13 cases. RESULTS: At RT completion, a marked response in terms of tumor size was noted in most cases. The median follow-up was 65 months. Local control was obtained in 9 (64.3%) of the 14 in the RT group and in 2 (22.2%) of the 9 in the non-RT group. No secondary malignancies were observed in the RT group. CONCLUSION: These results supports the finding that RT is more effective against Kimura's disease. Simple or immunohistochemical features did not influence the treatment outcome.

Angiolymphoid hyperplasia successfully treated with an ultralong pulsed dye laser.

BACKGROUND: Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign vascular proliferation characterized by dermal or subcutaneous nodules or both, primarily in the head and neck. We report the successful treatment of ALHE without scarring using a 595 nm ultralong pulsed dye laser, which applies a unique combination of longer wavelengths, a longer pulse duration, and a higher fluence. OBJECTIVE: To report the use of the 595 nm pulsed dye laser in the treatment of ALHE and to detail the particular aspects of this laser that make it uniquely qualified to treat this entity. METHODS: Case report and review of the literature. RESULTS AND CONCLUSION: This laser may be used as an effective treatment for ALHE and has advantages over alternative treatments and older lasers. Specifically, the longer wavelength (595 nm) penetrates more deeply into dermal tissue, which produces more uniform coagulation across the entire vessel.

Angiolymphoid hyperplasia with eosinophilia of the nail bed and bone: successful treatment with radiation therapy.

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare entity characterized by the presence of one or several papules or nodules in the skin. Histopathology of ALHE shows a marked proliferation of blood vessels with distinctive large endothelial cells and variable inflammatory infiltrates with eosinophils. We report a 32-year-old Caucasian woman with multiple nodules involving the skin, subcutaneous tissue and bone of the distal phalanx of the fingers that were treated successfully with orthovoltage radiation therapy (40 Gy/20 fractions) and without any side-effects after 9 years of follow-up.

Rapid remission of severe pruritus from angiolymphoid hyperplasia with eosinophilia by pulsed dye laser therapy.

A 48-year-old Japanese woman with angiolymphoid hyperplasia with eosinophilia (ALHE) was successfully treated with a flashlamp pulsed dye laser (585 nm, 450 micros pulse duration). The lesion was severely pruritic and had been enlarging slowly for 2 years but was resistant to conventional therapies, including topical, intralesional, and systemic corticosteroid, and cryotherapy. The severe pruritus immediately improved after the first treatment using the pulsed dye laser. The erythema and papules gradually improved without scarring and this was followed by further five treatments over approximately a 4-month interval. No clinical recurrences have been observed 1 year after completion of the treatment. We think that pulsed dye laser therapy is an effective treatment for ALHE in both Japanese as well as Caucasian patients. Pulsed dye laser therapy is also helpful in reducing the pruritus in ALHE patients.

Angiolymphoid hyperplasia with eosinophilia responsive to pulsed dye laser.

Angiolymphoid hyperplasia with eosinophilia is an uncommon, benign, but potentially disfiguring vascular proliferation. Angiolymphoid hyperplasia with eosinophilia commonly affects women in the third decade. Although a benign process, significant morbidity can occur including bleeding, pain, and disfigurement. Several therapies have been attempted including curettage, cryotherapy, retinoids, excision, and various lasers. We report on a case of angiolymphoid hyperplasia with eosinophilia responding to pulsed dye laser therapy (585 nm) (SPTL-1a, Candela Corp, Wayland, Mass).

Radioterapia en la Enfermedad de Kimura: a propósito de un caso / Radiotherapy in Kimura’s disease. report of a case

La Enfermedad de Kimura es un raro trastorno inflamatorio de etiología desconocida que afecta principalmente a varones jóvenes de raza oriental si bien esporádicamente puede afectar a otros grupos de población como en el caso aquí descrito. Se caracteriza por la aparición de nódulos subcutáneos indoloros típicamente en región parotídea que se acompañan de la presencia de adenopatías locorregionales, eosinofilia periférica y elevación de la IgE sérica. La extirpación quirúrgica y el tratamiento con corticoesteroides son de elección en la Enfermedad de Kimura. La radioterapia puede ser una opción y debe considerarse en aquellos pacientes en los cuales la enfermedad reaparece tras la cirugía o el tratamiento prolongado con corticoesteroides. (AU)