Traumatic ulcerative granuloma with stromal eosinophilia - clinical case report, literature review, and differential diagnosis.

BACKGROUND: Traumatic ulcerative granuloma with stromal eosinophilia (TUGSE) is a rare self-limiting condition of the oral mucosa. The lesion manifests as an isolated ulcer that can be either asymptomatic or associated with mild to severe pain, and in most cases, it affects the tongue. TUGSE lesions may mimic malignancy such as squamous cell carcinoma, CD30 positive lymphoproliferative disorder, or infectious diseases such as primary syphilis, tuberculosis, or Epstein-Barr virus mucocutaneous ulcer. Histologically dominating cells are lymphocytes, histiocytes, and eosinophils. CASE PRESENTATION: We describe a TUGSE case of a patient with a solitary ulcer on the lower left retromolar buccal plane. Upon presentation, the patient reported a swelling on the buccal mucosa of the left lower jaw since 1 year with rapid growth over the last days and mild pain while chewing. The diameter of the intraoral lesion on the lower left retromolar buccal plane was approximately 4 × 3 cm; the lesion presented as indurated base with a central superficial ulceration of 2 × 1 cm, indicative for a malignant process. Histologically, the ulceration showed an expanding, infiltrative, and vaguely granulomatous morphology, involving the superficial mucosa and the fatty tissue, and extended between the deep striated muscle fibers. The lesion was rich in lymphocytes, histiocytes, and eosionophils intermingled with activated T-blasts without phenotypic abnormalities. TUGSE was then diagnosed based on the phenotype (especially the lacking expression of CD30, the retained T-cell phenotype, and the absence of Epstein-Barr virus), the clinical presentation, and the morphology. Twenty-six months after diagnosis, no recurrence of the ulceration was seen. CONCLUSIONS: As TUGSE may mimic malignancy or infectious diseases, biopsy is mandatory and should be combined with thorough clinical examination. A screening for infectious diseases (mainly syphilis, Epstein-Barr virus, and HIV infections) must be performed routinely. In most cases, the lesions resolve spontaneously, obviating the need of further actions other than clinical follow-up. The pathogenesis of TUGSE lesions is still under debate, although local traumatic events and a locotypic immune response have been suggested to be major contributing factors.

Rare case of eosinophilic granulomatosis with polyangiitis in two patients with α-1-antitrypsin deficiency (PiSZ).

We present two cases of eosinophilic granulomatosis with polyangiitis occurring with α-1-antitrypsin deficiency, both PiSZ phenotype. The simultaneous occurrence of these two conditions has seldom been described in the literature, despite evidence of an association between α-1-antitrypsin deficiency and other forms of vasculitis. Both patients had pulmonary involvement and reported intermittent exacerbations of vasculitic symptoms. Both patients were managed on low-dose oral steroids and azathioprine remaining well with occasional exacerbations. It is important to consider whether there is an association between eosinophilic granulomatosis with polyangiitis and α-1-antitrypsin deficiency, as this may lead to more severe pulmonary symptoms during exacerbations. If a genetic association between the two conditions is found, clinicians should be aware of the possible need to screen for α-1-antitrypsin deficiency in appropriate patients.

Traumatic ulcerative granuloma with stromal eosinophilia around mini dental implants without the protection of a denture base.

This is a report of a case of an unusual oral lesion after the placement of mini implants for the retention of a mandibular overdenture. A patient received four 2-mm-wide dental implants in the anterior mandible and had her mandibular denture relined with a soft material. After 3 months, she was not wearing her mandibular denture, and two nodular ulcerated lesions were observed near the mini implants. The lesions ceased following excision and regular denture wearing. Clinical and microscopic examination led to the diagnosis of traumatic ulcerative granuloma with stromal eosinophilia (TUGSE). TUGSE is rare lesion with a benign course that may occur following injury of the oral mucosa by mini implants under certain circumstances.

An unusual affliction of the tongue.

A dramatic presentation of a large ulcer on the dorsal tongue of a recently hospitalized patient is presented. The lesion was found to be nosocomial in origin, and consistent with traumatic ulcerative granuloma with stromal eosinophilia (TUGSE). A review of the current pathogenic mechanisms, differential diagnosis and management of TUGSE is included.

Eosinophilic granulomatous gastrointestinal and hepatic abscesses attributable to basidiobolomycosis and fasciolias: a simultaneous emergence in Iraqi Kurdistan.

BACKGROUND: Deep eosinophilic granulomatous abscesses, as distinguished from eosinophilic subcutaneous abscesses, are rare. Most reports are from the Far-East and India where the most commonly attributed cause is Toxocara. Sulaimaniyah in Northeastern Iraq has experienced an outbreak of eosinophilic granulomatous liver and gastrointestinal (GI) abscesses beginning in 2009. The purpose of this study was to determine the etiology and guide treatment. METHODS: The study was an ongoing investigation of patients having a histopathologic diagnosis of eosinophilic granulomatous abdominal abscesses in Sulaimaniyah hospitals from May 2009 to August 2012. Tissues were examined for organisms, and Enzyme Linked Immunoabsorbent Assays (ELISA) were performed for serum antibodies to Fasciola hepatica, Toxocara, and Echinococcus granulosus. RESULTS: Fourteen patients had granulomatous inflammation surrounding a central necrotizing eosinophilic exudate identified in surgical pathology specimens from abdominal surgeries. Two children and four adults had abscesses that formed GI masses. These patients included a 39 year old male with oropharyngeal and transverse colon disease, and a 48 year old male with liver and GI abscesses. All sites demonstrated a Zygomycete fungus surrounded by eosinophilic Splendori-Hoeppli material consistent with basidiobolomycosis. Five of the six patients with fungal disease were treated by surgery and 4 to 7 months of itraconozol. One child died of intestinal perforation while receiving IV amphotericin B; two adults required additional surgery for recurrent GI obstruction. Eight patients had isolated liver abscesses with no organisms identified by histopathology: ELISA results for F. hepatica were positive for five, borderline for one, and negative for two patients. These eight patients were treated for fascioliasis by surgical resection of localized abscesses and albendazol. One patient serologically positive for F. Hepatica was found to have a common duct fluke two years after initial diagnosis. Serological testing for Toxocara and Echinococcus granulosus was negative in all 14 patients. CONCLUSIONS: Basidiobolomycosis and F. hepatica are implicated as the cause of abdominal eosinophilic granulomatous abscesses in 12 of 14 patients identified over a period of 40 months in northern Iraq. Treatment was complicated by chronic biliary tract disease in fascioliasis and perforation and recurrent intestinal obstruction with basidiobolomycosis.

Feline eosinophilic granuloma complex(ities): some clinical clarification.

PRACTICAL RELEVANCE: The feline eosinophilic granuloma complex (EGC) comprises a group of clinically well recognised but poorly understood dermatoses that are common in cats. In many cases, lesions are severe and can be accompanied by varying degrees of (and sometimes considerable) pruritus and/or pain. In addition, lesions can be chronic and recurrent. It is, therefore, important to achieve a prompt and accurate diagnosis in order to provide optimal, often life-long, treatment for affected cats. PATIENT GROUP: There is no age predisposition or well documented breed predilection for the development of EGC lesions in cats. Some studies have reported a possible female predisposition, but this has not been consistently documented. CLINICAL CHALLENGES: The clinical diagnosis of EGC lesions is usually straightforward, but investigation of the potential underlying aetiology can pose a challenge for the clinician. Information on the indication for various diagnostic tests and their interpretation is lacking, and the tendency for these cases to be managed with chronic medical intervention prior to achieving a definitive diagnosis can further complicate the interpretation of any diagnostic investigation. In addition, successful therapeutic management of these cases can be challenging. Some cats suffer only a single episode of disease that resolves with treatment, while others have recurrent lesions and some of these can be refractory to treatment. The individual variation in both the clinical nature of the disease and the response to therapy could be related to disease severity, but could also be explained by differences in the underlying aetiopathogenesis. EVIDENCE BASE: This article reviews the published literature to discuss the complex aetiology of the EGC and present an overview of the different clinical presentations and diagnosis. A further and particular aim has been to provide some evidence-based recommendations for the management of this unusual group of dermatoses.

Granuloma eosinófilo craneal solitario: caso clínico / Solitary eosinophilic granuloma of the skull: a clinic case

La histiocitosis de las células de Langerhan (HCL), es una enfermedad Granulomatosa de etiología no bien precisada todavía, la cual puede tener diferentes manifestaciones y localizaciones,1 aunque algunos la consideran una proliferación neoplásica de las células de Langerhan que aparecen en el escenario de sitios nodulares o no.2 La Histiocytosis X incluye tres componentes: El Granuloma Eosinófilo, la enfermedad de Hand-Schüller-Christian y el síndrome de Letterer-Siwe.5 El Granuloma Eosinófilo es un tumor óseo benigno muy raro, que se presenta en más del 90 por ciento de los casos en niños menores de 10 años, 6 teniendo además cierta predilección por el sexo masculino. Esta lesión es usualmente encontrada en los huesos largos y planos. Se trata de una paciente femenina de 50 años de edad con antecedentes de procesos alérgicos y asma bronquial grado II y fumadora inveterada, que hacía 8 meses, previo a su ingreso, había comenzado a presentar cefalea holocraneal, fue notando irregularidades en su cuero cabelludo y fue hospitalizada porque al realizarle una radiografía simple de cráneo se evidenció una imagen radiotransparente de bordes mal definidos, lo que fue constatado por tomografía simple de cráneo, la paciente fue operada, realizándole una craniectomía y excéresis de un tejido Granulomatoso sobre la duramadre con colocación de injerto de cemento quirúrgico en el área de la craniectomía. Se le diagnosticó un Granuloma Eosinófilo. Posterior a su recuperación fue tratada con oncología donde se le aplicó radioterapia. La evolución de la paciente fue satisfactoria, ahora se encuentra asintomática.

The Langerhans cells hystiocitosis is a Granulomatous ill, it has unknown etiology today. This disease can be many places and clinical manifestations, although some authors consider it a neoplasic proliferation of the Langerhans cells which can be on nodular region scenery or not. X Hystiocitosis include three diseases: Eosinophilic Granuloma, Hand-Schuller- Chritian disease and Letterer-Siwe syndrome. Eosinophilic Granuloma is a very uncommon benign bone tumor, which may be present in more than 90 percent in childhood under 10 years old. They have predominance on male patients. Usually it is found on long and flat bones. The case report is about 50 years old, female patient, with allergic history and bronchial asthma II degree, she come to the hospital because she don’t filled well since 8 months ago and she complained mild headache, furthermore she noticed irregularity over her skull, on vertex region, at her admission she was screening with cranial X ray, where was found a radio lucid images over the painful area, moreover it had irregular edge whit more than 10 cm long. After that she was screening on CT-scan, where was found a very large osteolytic lesion without intracranial grow. She was approached with craniectomy and graft of surgical cement. She was diagnosed Eosinophilic Granuloma on pathologic department. After her discharge she was treated by oncology, here she received holocraneal radiotherapy. Her outcome has been satisfactory because she is asymptomatic and her CT-scan showed not evidences of recidivism.

Traumatic ulcerative granuloma with stromal eosinophilia of the retromolar region.

Traumatic ulcerative granuloma with stromal eosinophilia (TUGSE) is a rare chronic benign lesion of the oral mucosa. Clinically, it may mimic squamous cell carcinoma as well as other malignant lesions. Most cases of TUGSE are reactive. A case of TUGSE of the retromolar region is reported. An asymptomatic ulceration with indurated borders, limited to the gingiva, was localized in the right retromolar region. An incisional biopsy was taken. Six weeks after the biopsy, the residual ulceration spontaneously disappeared. After 6 months of follow-up, the lesion had not recurred.Traumatic ulcerative granuloma with stromal eosinophilia can be interpreted as a subset of lymphoproliferative disorders.A malignant lymphoid proliferation should be suspected when atypical histologic findings and monoclonality are observed. Given the benign nature of TUGSE, overtreatment should be prevented in patients affected by this disease.

Eosinophilic oesophagitis: an enigmatic, emerging disease.

Eosinophilic oesophagitis is a disease that has emerged in recent years. It is often associated with dysphagia and oesophageal food impaction in adults. The disease is characterised by infiltration of eosinophilic granulocytes into the oesophageal mucosa. This infiltrate may be responsible for the subtle peristaltic abnormalities that can be found in these patients. Endoscopic findings are usually absent or nonspecific, although a discrete circular ring pattern of the mucosa may be noticed. Occasionally, overt endoscopic abnormalities (such as exudative changes and shearing of the mucosa) can be found. The presence of at least 15 intraepithelial eosinophilic granulocytes per high-power field in random biopsies from the whole length of the oesophagus is considered to be diagnostic. Gastro-oesophageal reflux needs to be excluded as it may lead to eosinophilic infiltration as well. Adequate diagnosis is relevant for treatment and the prevention of unnecessary further investigations. The disease responds well to the ingestion of fluticasone propionate and its long-term prognosis is generally good. But when fluticasone is discontinued recurrent symptoms are common, and some cases are severe, needing treatment with systemic corticosteroids.

Eosinophilic granuloma arising from the pelvis in children: A report of three cases.

Eosinophilic granuloma (EG) is a benign tumor-like condition which is characterized by a clonal proliferation of Langerhans-type histiocytes and defined as a local form of Langerhans cell histiocytosis (LCH). The radiographic appearances of EG are quite different depending on the phase of the disease and the site of involvement. A status of EG in the bone is divided into acute and chronic phases. Radiologically acute phase of EG is difficult to differentiate from a malignant bone tumor such as Ewing's sarcoma or acute osteomyelitis. Chronic phase of EG may mimic a chronic osteomyelitis or a benign bone tumor. We report 3 children's cases of EG in the pelvis which showed quite different radiological features and clinical courses. A 6-year-old boy (Case 1) had an osteolytic lesion with slightly defined margins in the right acetabulum. A 4-year-old boy (Case 2) had a radiologically similar-looking lesion in the left acetabulum. These lesions resembled radiologically chronic osteomyelitis (Brodie's abscess) or a benign bone tumor and healed spontaneously after biopsy. A 2-year-old boy (Case 3) had an osteolytic lesion with ill-defined margin in the ilium. It was difficult to differentiate from a malignant tumor such as Ewing's sarcoma, or acute osteomyelitis. The lesion became enlarged after needle biopsy. In spite of an additional curettage, the osteolytic lesion remained in the left pelvis in 1 year. Treatment for EG is controversial. Curettage of the affected site and bone grafting is usually accomplished. However, some EG heal spontaneously. It is of great importance to understand the natural course of EG and this knowledge will give us the opportunity to avoid unnecessary treatment. EG with poor osteolytic margins may progress further after biopsy. EG with well-defined margins may heal spontaneously after biopsy only.

Oral eosinophilic granulomas in tigers (Panthera tigris)--a collection of 16 cases.

Oral eosinophilic granulomas were diagnosed in 16 tigers (Panthera tigris). All lesions were located on the hard or soft palate and typically consisted of flat or slightly raised circular ulcers. Histologic features of these lesions were essentially identical to those seen in oral eosinophilic granulomas of domestic cats and dogs. No clinical signs were noted in eight cases, though various degrees of inappetence, excessive salivation, and dysphagia were noted in the other eight tigers. Six cases were not treated. Treatment for the remaining 10 cases centered on corticosteroids and additional treatments included surgical removal, cryotherapy, antibiotics, and chlorpheniramine. Treatment with corticosteroids did appear to be effective in some cases, though lesions would worsen after cessation of therapy and no cases were cured. In addition, three cases developed complications possibly related to this corticosteroid therapy. The etiology of these lesions remains unknown, though an underlying allergic condition is likely.

Gastric eosinophilic granuloma in China: case series.

AIM: Eosinophilic granulomas are benign localized lesions in the stomach that are distinct from eosinophilic gastritis. The aim of this study was to identify the potential etiological factors and clinical features of gastric eosinophilic granulomas (GEG). METHODS: Clinical manifestations, histopathological features, diagnosis and treatments of 48 hospitalized cases of GEG were analyzed retrospectively. A modified Giemsa staining was employed to detect Helicobacter pylori in 23 of these 48 patients. RESULTS: There was a significant gender difference among patients with GEG with male patients significantly more affected than female patients. The final diagnosis was made after operating the patients (the misdiagnosis level was rather high before resection). Lymphoid follicles were found in 68.6% of the tissues surrounding the lesion. H. pylori infection was detected in 69.6% of patients. In 11 patients eosinophilia in the peripheral blood was observed. CONCLUSION: The results suggest that H. pylori infection, estrogen status and local allergic reactions may be associated with the development of GEG.

Granuloma eosinofílico primário do pulmão: registros clássicos / Primary eosinophilic granuloma of the lung: classical records

Foram analisados, retrospectivamente, cinco casos de granuloma eosinofílico primário do pulmão (GEPP) identificados durante admissão no Hospital Universitário Lauro Wanderley da Universidade Federal da Paraíba (HULW/UFPB), no interregno de 1982 a 1998, admitidos com história diagnóstica de fibrose intersticial difusa (FID). Foram revisados os seguintes aspectos: formas de apresentação clínica e epidemiológica, presunção diagnóstica na admissão, doenças concomitantes e evolução terapêutica. A presunção clínica do diagnóstico foi confirmada mediante a citometria com níveis elevados de CD1 no lavado broncoalveolar (LBA). A biópsia pulmonar aberta foi indicada pelos sintomas persistentes em acompanhamentos ambulatoriais sem melhora clínica. O relatório anatomopatológicodemonstrou histiocitose de células de Langhans. A terapia à base de corticosteróides não apresentou boa eficácia, uma vez que, observando a evolução clínica dos casos apresentados, ocorreram dois óbitos e os demais não tiveram uma melhora significativa da enfermidade de base, porém existiu um prejuízo nessa avaliação, em virtude do não-abandono do vício tabágico. Enfatiza-se a fisiopatogenia e a importância dessa enfermidade, apesar de rara e pouco conhecida, como causa de FID, ilustrando um dos casos que se reveloubastante exuberante no diagnóstico de GEPP.

We studied retrospectively five cases of eosinophylic granuloma of the lung identified during admission at HULW/UFPB from 1982 to 1998. They were admitted with diagnostical history of difuse intersticial fibrosis. The following aspects were reviewed: clinical and epidemilogical presentation, diagnostic presumption at admission, concomitant diseases and evolution. The diagnostic was confirmed with bronchoalveolar lavage (BAL) and lung biopsy. The anatomicopathologic exams report histiocytosis of Langhans'cells. The clinical course was variable. There are no accepted successful treatment, although three patients responded poorly to corticosteroids and twodied, in spite of all effort in the treatment with corticosteroids too.

[Eosinophilic ulcer of the oral mucosa: 11 cases]. / Ulcère éosinophilique de la muqueuse buccale: 11 cas.

BACKGROUND: The eosinophilic ulcer is a lesion of the oral mucosa, that has been infrequently described in the literature. This is a benign and self-limiting lesion of unknown origin. CASE REPORTS: In this article, eleven new cases of eosinophilic ulcer of the oral mucosa are presented. The clinical, histologic and evolutive features are reviewed. DISCUSSION: Recurrent trauma is clearly involved in the pathogenesis of this entity.