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J Cutan Pathol 2018 Oct 25. Epub 2018 Oct 25.

Dartmouth Geisel School of Medicine, Hanover, New Hampshire.

A relationship between Kimura disease (KD) and angiolymphoid hyperplasia with eosinophilia (ALHE) has been debated. Given substantial clinical and histological overlap, these entities were once considered to represent a disease spectrum; however, they are now widely considered to be nosologically distinct. A diagnosis of either condition is further complicated by resemblance to various malignancies, which must be carefully excluded. Read More

Actas Dermosifiliogr 2018 Oct 6. Epub 2018 Oct 6.

Sección de Dermatopatología, Departamento de Anatomía Patológica, Hospital Clínico Universidad de Chile, Universidad de Chile, Santiago, Chile.

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign vascular proliferation characterized by solitary or multiple angiomatous lesions. It is most common in young or middle-aged women, and the lesions typically affect the head and neck, showing a particular predilection for the periauricular region. The differential diagnosis in patients with ALHE is broad and includes both benign and malignant conditions. Read More

Ann Pathol 2018 Dec 16;38(6):386-390. Epub 2018 Aug 16.

Service de pathologie, centre hospitalier Montbéliard, 12, rue Docteur Flamand, 25200 Montbéliard, France.

We report the case of a Caucasian 73-year-old woman, without medical history, who is operated of two lesions of the forehead and the scalp measuring 0.4 and 1cm. There is no adenopathy. Read More

Dermatol Surg 2018 08;44(8):1147-1149

Department of Medicine, University of British Columbia, Vancouver, British Columbia, Canada Division of Dermatology, Department of Medicine, University of Ottawa, Ottawa, Ontario, Canada Division of Plastic Surgery, Department of Surgery, University of Ottawa, Ottawa, Ontario, Canada Division of Dermatology, Department of Medicine, University of Ottawa, Ottawa, Ontario, Canada.

J Surg Case Rep 2018 Jul 2;2018(7):rjy145. Epub 2018 Jul 2.

ENT Department, AL-Farwaniya Hospital, Kuwait.

Introduction: Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare entity that usually occurs as 0.5-3 cm, pink-to-brown nodules on the skin surface and subcutaneous tissues of the head and neck (Al-Muharraqi MA, Faqi MK, Uddin F, Ladak K, Darwish A. Angiolymphoid hyperplasia with eosinophilia (epithelioid hemangioma) of the face: an unusual presentation. Read More

Cancer Biother Radiopharm 2018 Sep 29;33(7):282-284. Epub 2018 Jun 29.

Department of Radiation Oncology and Nuclear Medicine, The Third Affiliated Hospital of Guangzhou Medical University , Guangzhou, P.R. China .

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 P simple drug membranes for five times. The P brachytherapy involving simple drug membranes of brachytherapy began by diluting a P solution with 0. Read More

Ophthalmic Plast Reconstr Surg 2018 Jul/Aug;34(4):e121-e123

HanGil Eye Hospital, Incheon, South Korea.

Kimura disease has been known as a chronic inflammatory disease of unknown etiology, since the first report in 1937 and characterized by an increased eosinophil fraction in peripheral blood, increased serum immunoglobulin E (IgE) levels, and lymphoid granuloma with eosinophilic infiltration on histopathological examination. The authors present a 30-year-old man who visited their clinic with a 3-month history of protrusion and conjunctival injection of the right eye. Surgical mass resection was performed, and initial histopathology revealed Kimura disease. Read More

Dermatol Surg 2018 May;44(5):714-720

All authors are affiliated with the Department of Dermatology and Venereology, All India Institute of Medical Sciences, New Delhi, India.

Background: Although insulated intralesional radiofrequency ablation (IL-RFA) is being increasingly used in other specialties, not much information on its safety and efficacy in dermatology is available.

Objective: To describe our experience with insulated IL-RFA for various dermatological conditions.

Methods: This is a retrospective review of the patients who underwent IL-RFA in the past 3 years. Read More

BMJ Case Rep 2018 Apr 20;2018. Epub 2018 Apr 20.

All India Institute of Medical Sciences, Department of Dermatology and Venereology, New Delhi, India.

A 21-year-old woman presented with multiple erythematous to skin-coloured dome-shaped firm papules and plaques over the right ear concha and external auditory canal for 1 year. It was associated with occasional itching and bleeding. Her main concern was cosmetic disfigurement. Read More

Chin Med J (Engl) 2018 Apr;131(8):1007-1008

Department of Dermatology, Peking University People's Hospital, Beijing 100044, China.

Pol J Radiol 2017 20;82:607-611. Epub 2017 Oct 20.

Department of Radiodiagnosis, Byramjee Jeejeebhoy Government Medical College and Sassoon General Hospitals, Poona, Maharashtra, India.

Background: Kimura disease is an uncommon, benign, chronic, idiopathic disease that is seen mainly in the Asian population, particularly in females. It mainly affects the salivary glands and in particular parotid and cervical lymph nodes. So far, the diagnosis of Kimura disease has been based on histopathology. Read More

Indian J Dermatol Venereol Leprol 2018 May-Jun;84(3):325-327

Department of Dermatology, Venereology and Leprosy, Gandhi Medical College, Hyderabad, India.

J Med Case Rep 2018 Apr 2;12(1):89. Epub 2018 Apr 2.

Department of Pathology, Tishreen University Hospital, Tishreen University, Lattakia, Syria.

Background: Angiolymphoid hyperplasia with eosinophilia is a benign neoplasm that includes blood vessel proliferation and a dense eosinophilic inflammatory infiltrate. Mostly, it affects middle-aged adults manifesting as flesh/plum-colored pruritic nodules and papules, most commonly affecting the ear and the periauricular area.

Case Presentation: In this case, we report a 13-year-old Caucasian girl with bilateral, huge, protruding, and yellowish nostril masses which were peculiar in location and of gross appearance. Read More

J Dermatol 2018 Oct 31;45(10):e284-e285. Epub 2018 Mar 31.

Department of Dermatology, Kyoto University Graduate School of Medicine, Kyoto, Japan.

Skinmed 2018 1;16(1):71-72. Epub 2018 Feb 1.

Department of Dermatology and Cutaneous Biology, Sidney Kimmel Medical College at Thomas Jefferson University, Philadelphia, PA.

An otherwise healthy man in his 50s presented complaining of pruritic lesions on the left side of his scalp. The lesions had slowly been growing in size over the preceding 30 years. They would occasionally bleed, and this is what ultimately prompted him to seek medical advice. Read More

Urol J 2018 Sep 26;15(5):295-296. Epub 2018 Sep 26.

Chengdu University of Traditional Chinese Medicine, Sichuan Province, China.

Herein we report a case of Kimura's disease with unusual manifestations. A 46-year- old Chinese man presented with mass in scrotum which gradually increased in size for approximately 7 years. A computerized tomographic scan of abdomen revealed a soft-tissue density shadow in scrotum and enlargement of lymph nodes in groin and retroperitoneum. Read More

J Cutan Pathol 2018 Jun 6;45(6):395-402. Epub 2018 Apr 6.

Dermatopathologie Friedrichshafen, Friedrichshafen, Germany.

Background: Accurate distinction of epithelioid hemangioma (EH) from its malignant mimics is paramount but remains challenging due to its wide morphological spectrum and lack of objective molecular markers. FOSB oncogenic activation was recently identified as a key event in endothelial proliferation. We sought to investigate the FOSB staining pattern in EH with angiolymphoid hyperplasia with eosinophilia (EH-AHLE) morphology and to evaluate its value in differential diagnosis of epithelioid vascular tumors. Read More

Dermatol Online J 2017 Oct 15;23(10). Epub 2017 Oct 15.

Department of Dermatology, Shaanxi University of Traditional Chinese Medicine Affiliated NO.1 Hospital.

Kimura disease is a rare, benign, chronic, immune-mediated inflammatory disorder. We report a 46-year-old man who presented with a cutaneous nodule behind his left ear. Surgical removal of the growth confirmed the histological diagnosis of KD. Read More

Dermatol Pract Concept 2018 Jan 31;8(1):28-32. Epub 2018 Jan 31.

Department of Dermatology Acibadem University School of Medicine, Istanbul, Turkey.

Background: Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare vascular proliferative disorder mainly located in the periauricular region. The etiopathogenesis of ALHE is unknown, and it is still controversial as to whether the entity represents a benign vascular neoplasm or an inflammatory process.

Aim: Recently, the intracytoplasmic staining pattern of Wilms tumor 1 (WT1) on immunohistochemistry has highlighted true vascular neoplasms, such as microvenular hemangioma, tufted angioma, and spindle cell hemangioma, which has made it helpful to distinguish ALHE from vascular malformations, as there is a negative staining pattern in the other entities. Read More

Arch Bronconeumol 2018 Jun 3;54(6):340-342. Epub 2018 Feb 3.

Department of Pulmonology, Centro Hospitalar de Trás-os-Montes e Alto Douro, Vila Real, Portugal.

Medicine (Baltimore) 2017 Dec;96(48):e8990

Department of Otolaryngology-Head and Neck Surgery.

Rationale: Kimura disease is a rare benign, chronic inflammatory disorder that typically presents with slowly enlarging, nontender, subcutaneous swellings in the head and neck region. The occurrence of Kimura disease in the oral cavity is extremely rare.

Patient Concerns: A 16-year-old boy presented with a complaint of a right painless buccal mass of 3 years' duration. Read More

J Clin Exp Hematop 2017 ;57(3):147-152

Department of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Hospital and Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences.

A 42-year-old man with eosinophilia and high serum immunoglobulin E (IgE) developed a lacrimal gland mass on the left side. Excisional biopsy revealed hyperplasia of lymphoid follicles, and infiltration with lymphocytes and eosinophils around lacrimal gland acini, leading to the pathological diagnosis of Kimura disease. IgE-positive cells were mainly found along follicular dendritic cells, and a small number of IgG4-positive cells was present. Read More

Am J Surg Pathol 2019 Jan;43(1):26-34

Dermatopathology Department, Dermatopathologie Friedrichshafen, Friedrichshafen, Germany.

There is a wide clinicopathologic spectrum of vascular proliferations characterized by the presence of epithelioid endothelial cells, comprising epithelioid hemangioma (EH)-pseudomyogenic (epithelioid sarcoma-like) hemangioendothelioma (PM-HAE), epithelioid hemangioendothelioma, and epithelioid angiosarcoma. Immunohistochemical FOS-B expression as well as FOS-B rearrangement (fluorescent in situ hybridization [FISH]) have recently been described as diagnostically relevant underpinnings of EH (restricted to osseous lesions) and PM-HAE. The aim of this study was to clinicopathologically characterize and to elucidate FOS-B expression in patients with eruptive lesions of the cellular variant of cutaneous EH. Read More

Indian J Otolaryngol Head Neck Surg 2017 Dec 5;69(4):571-574. Epub 2017 Jan 5.

Department of Otolaryngology and HNS (ENT), AIIMS, Virbhadra Marg, Rishikesh, Uttarakhand 240201 India.

Kimura's disease is a rare disorder of uncertain aetiology seen in Asian population, and presents as painless lymphadenopathy or subcutaneous masses in head-neck region. Peripheral eosinophilia, elevated levels of serum IgE, lymphoid proliferation and eosinophilic infiltration are its characteristic pathological features. We report a case of 40 year old male patient who presented with swelling behind right ear after successful ear surgery. Read More

An Bras Dermatol 2017 May-Jun;92(3):392-394

Department of Dermatology at Hospital Federal da Lagoa - Rio de Janeiro (RJ), Brazil.

Angiolymphoid hyperplasia with eosinophilia is a rare and benign vascular tumor whose etiology remains uncertain. It clinically presents itself by angiomatous papules or nodules located on the head and neck. Many controversies in the literature are found in relation to angiolymphoid hyperplasia with eosinophilia and Kimura's disease - its main differential diagnosis - due to their clinical and histopathological similarities. Read More

Mol Clin Oncol 2017 Nov 19;7(5):874-876. Epub 2017 Sep 19.

Department of Dermatology, The First Affiliated Hospital of Sun Yat-sen University, Guangzhou, Guangdong 510080, P.R. China.

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare chronic inflammatory disorder of unknown etiology that most commonly presents as painless lymphadenopathy or subcutaneous masses in the head and neck region. The pathogenesis of the disease is not yet clear. The differential diagnosis is determined using characteristic histological features, such as significant vascular proliferation, lymphocytes and eosinophil inflammation in the dermis. Read More

Eur J Dermatol 2018 Feb;28(1):85-86

Division of Dermatology, Department of Medicine of Sensory and Motor Organs, Tottori University Faculty of Medicine, Yonago, Japan.

Int J Dermatol 2017 Dec 23;56(12):1373-1378. Epub 2017 Oct 23.

Department of Dermatology, Hedi Chaker Hospital, Sfax, Tunisia.

Background: Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign vascular proliferation characterized by dermal or subcutaneous red or brown papules or nodules, most commonly on the head and neck.

Objective: The aim of this study was to review the epidemiological and clinical characteristics of ALHE, focusing particularly on the histological and therapeutic features.

Methods: We performed a retrospective study of all cases of ALHE diagnosed in our dermatology and pathology departments between 2004 and 2015. Read More

Arch Plast Surg 2017 Sep 15;44(5):439-443. Epub 2017 Sep 15.

Department of Pathology, Chung-Ang University Hospital, Chung-Ang University College of Medicine, Seoul, Korea.

Kimura disease is a rare idiopathic chronic inflammatory disorder. It typically presents in the head and neck area, whereas bilateral involvement is unusual. Its diagnosis requires it to be differentiated from other inflammatory diseases and from head and neck tumors. Read More

JAAD Case Rep 2017 Sep 8;3(5):416-419. Epub 2017 Sep 8.

Department of Dermatology, Weill Cornell Medicine, New York, New York.

Clin Nephrol 2017 Oct;88(10):221-225

Kimura disease (KD) is a rare inflammatory soft tissue disorder of unknown origin most frequent in Asians, the prevalence of which is growing in Western countries. Painless papules and/or nodules with a predilection for the head and the neck region, lymphadenopathies, parotid gland involvement, eosinophilia, and raised IgE levels are parts of its presentation. Renal involvement with various forms of glomerulonephritis, including membranous nephropathy (MN), can occur and is generally associated with a proteinuria that encompasses nephrotic syndrome. Read More

J Cutan Aesthet Surg 2017 Apr-Jun;10(2):116-118

Department of Dermatology, Topiwala National Medical College and BYL Nair Hospital, Mumbai, Maharashtra, India.

Int J Surg Case Rep 2017 10;39:84-87. Epub 2017 Aug 10.

Division of Plastic Surgery, King Saud University, Riyadh, Saudi Arabia.

Introduction: Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign reactive inflammatory lesion. The usual presentation is a single or multiple skin nodules of the head and neck. Involvement of the hand is very rare and there have been no comprehensive reviews on ALHE of the upper limb. Read More

Iran J Otorhinolaryngol 2017 Jul;29(93):221-224

Department of Otorhinolaryngology, Maulana Azad Medical College, New Delhi, India.

Introduction: Kimura's disease (KD) is an allergic inflammatory disorder of unknown etiology endemic in Orientals. Kimura's disease was first mentioned by Kimm and Szeto in China in 1937. Kimura's disease is commonly encountered in Asia and is mostly reported in Japan, China, Singapore and Honkong. Read More

J Clin Diagn Res 2017 Jun 1;11(6):ME01-ME04. Epub 2017 Jun 1.

Resident, Department of Medicine, Krishna Institute of Medical Sciences University, Karad, Maharashtra, India.

Kimura's Disease (KD) is a rare chronic inflammatory disorder presenting as multiple painless solitary subcutaneous nodules, predominantly in the head and neck region and frequently associated with regional lymphadenopathy and/or salivary gland involvement. Because of painless nature and indolent course, there is usually a delay in the patient's presentation. KD may radiologically mimic other chronic inflammatory conditions like tuberculosis, vascular malformations and neoplasms. Read More

Indian Dermatol Online J 2017 Jul-Aug;8(4):267-270

Department of Pathology, PGIMER, Dr. Ram Manohar Lohia Hospital, New Delhi, India.

Angiolymphoid hyperplasia with eosinophilia is a rare vascular disease characterized by multiple erythematous to violaceous papules, commonly present on the head and neck region. We report a case of a 23-year-old female who presented with multiple, erythematous asymptomatic papules on the preauricular region and pinna, which on biopsy was suggestive of angiolymphoid hyperplasia. The lesions were treated with a novel combination of radiofrequency ablation and topical timolol. Read More

J Comp Pathol 2017 Jul 3;157(1):57-60. Epub 2017 Jul 3.

Department of Veterinary Pathology, School of Veterinary Medicine, Japan.

A 5-year-old male miniature dachshund was presented with a dermal nodule on the left forelimb that increased to 5 mm in diameter over a 2-month period. Grossly, the nodule was firm, and both the external and cut surfaces were homogeneously pale pink in colour. Microscopically, the nodule was comprised of mainly plump endothelial cells and inflammatory cells; among the latter, lymphocytes were predominant, with few scattered plasma cells, mast cells and macrophages. Read More

Am J Otolaryngol 2017 Sep - Oct;38(5):626-629. Epub 2017 Jul 12.

Department of otolaryngology, Hubei Material and Child Health Hospital, Wuhan, China.

Purpose: The purpose of this study was to investigate the clinical features of eosinophilic hyperplastic lymphogranuloma (EHLG) in the head and neck.

Materials And Methods: Collecting the patients who diagnose with EHLG by pathological examination. The EHLG patients with the masses involved regions, such as involved inguinal region, chest wall, abdominal wall, anterior superior iliac spine or clavicle, instead of head and neck were excluding. Read More

Dermatol Surg 2017 07;43(7):981-983

Department of Dermatology, Penn State Hershey Medical Center, Hershey, Pennsylvania.

Dermatol Online J 2017 Jun 15;23(6). Epub 2017 Jun 15.

Weill Cornell Medical College, Department of Dermatology, New York, New York.

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a distinctive lymphocyte rich vasoformative proliferation comprising epithelioid-appearing endothelial cells with partially canalized capillary vessels in a lymphohistiocytic and eosinophil rich environment. ALHE presents clinically as single or multiple pink-brown dome-shaped papules or nodules, most commonly on the ear and peri-auricular area, followed by the face, and scalp. ALHE involving the subungual unit is rare with only 5 previously reported cases and all involved the underlying bone. Read More

BMC Surg 2017 May 26;17(1):63. Epub 2017 May 26.

Department of Surgical Oncology, Osaka City University Graduate School of Medicine, 1-4-3 Asahi-machi, Abeno-ku, Osaka, Japan.

Background: Kimura's disease (KD; eosinophilic granuloma of soft tissue) is an inflammatory granulomatous disorder of unknown cause with eosinophilic infiltration that occurs mainly in soft tissue. KD occurs mainly in the head and neck, but development in the axillary region is very rare.

Case Presentation: A 74-year-old Japanese woman was evaluated for a mass that she noted in the left axillary region. Read More

J Clin Diagn Res 2017 Mar 1;11(3):ZD21-ZD23. Epub 2017 Mar 1.

Head, Department of Oral and Maxillofacial Surgery, Vall d'Hebrón Hospital. Barcelona, Spain.

Angiolymphoid Hyperplasia with Eosinophilia (ALHE) is an atypical vascular tumour occurring primarily in the head and neck area, which must be distinguished from Kimura's disease. The lesions can appear as single or multiple grouped intradermal papules or subcutaneous nodules. We report a rare case of ALHE in a 57-year-old female with a large lesion of three nodules involving the right occipital artery which had a long term evolution and we treated it by surgical excision. Read More

J Craniofac Surg 2017 May;28(3):e207-e208

Oral and Maxillofacial Surgery, Armed Forces Medical College, Pune, India.

Kimura disease (KD) is a rare chronic inflammatory disorder of unknown etiology representing as solitary or multiple subcutaneous nodules predominantly in the head and neck region. Common sites of involvement are preauricular region, forehead and scalp. The soft tissue localization is often associated with regional lymphadenopathy and enlargement of the major salivary gland. Read More

Actas Dermosifiliogr 2018 Jan - Feb;109(1):73. Epub 2017 Apr 20.

Unidad de Gestión Clínica de Dermatología Médico-Quirúrgica y Venereología, Complejo Hospitalario Universitario de Granada, Granada, España.

Korean J Thorac Cardiovasc Surg 2017 Apr 5;50(2):114-118. Epub 2017 Apr 5.

Department of Pathology, Inje University Haeundae Paik Hospital, Inje University College of Medicine.

Kimura disease (KD) is an immune-mediated chronic inflammatory disease of unknown etiology. KD has many complications associated with hypereosinophilia, including various forms of allergic reactions and eosinophilic lung disease. Additionally, hypereosinophilia is associated with hypercoagulability, which may lead to thromboembolic events. Read More

Eur Ann Otorhinolaryngol Head Neck Dis 2017 Sep 18;134(4):287-289. Epub 2017 Mar 18.

Clinique Clarós, C/ Los Vergós, 31, CP 08017 Barcelona, Spain.

Head Neck Pathol 2017 Mar 28;11(1):78-87. Epub 2017 Feb 28.

Department of Pathology, Southern California Kaiser Permanente Group, Woodland Hills Medical Center, 5601 De Soto Avenue, Woodland Hills, CA, 91365, USA.

The 2017 fourth edition of the World Health Organization Classification of Tumours, specifically as it relates to the ear (Chap. 9), has several changes. Importantly, the number of entities has been significantly reduced by omitting tumors or lesions if they do not occur exclusively or predominantly at this site or if they are discussed in detail elsewhere in the book. Read More

Arch Pediatr 2017 Apr 21;24(4):367-370. Epub 2017 Feb 21.

Service de pédiatrie, CHU de Reims, 47, avenue Cognacq-Jay, 51100 Reims, France.

A subcutaneous mass of the skull in children can have many different causes (infectious, tumoral, and inflammatory). We report on the case of a 5-year-old patient with a subcutaneous mass of the skull evolving over several months. The first pathological analysis concluded in Kimura disease. Read More

G Ital Dermatol Venereol 2017 Apr;152(2):185-186

Division of Dermatology, Department of Clinical and Experimental Medicine, University of Pisa, Pisa, Italy.

J Cutan Aesthet Surg 2016 Oct-Dec;9(4):281-283

Department of DVL, Katuri Medical College and Hospital, Guntur, Andhra Pradesh, India.