Indometacina como tratamiento de granuloma eosinófilo multifocal / Indomethacin as a treatment for multifocal Eosinophilic granuloma

Granuloma eosinófilo es la variante más frecuente de histiocitosis de células de Langerhans. La mayoría de las lesiones ocurren en cráneo, costillas, columna vertebral o huesos largos, y pueden ser únicas o múltiples. El tratamiento depende del lugar de la afectación y del número de lesiones. Las opciones terapéuticas incluyen un agente único con prednisona, la combinación de vinblastina y prednisona, curetaje de las lesiones óseas o instilación intralesional de esteroides. Indometacina parece ser efectiva como tratamiento de lesiones de histiocitosis de células de Langerhans del hueso en niños y es bien tolerada. Presentamos el caso de un paciente varón de 4 años de edad con afectación de 2 huesos del cuerpo, cráneo y vértebra, tratado con curetaje de la lesión craneal e indometacina oral durante 19 meses, con completa curación de las lesiones y sin recurrencia 4 meses después de suspenderla. Concluimos que indometacina parece ser efectiva en el tratamiento de lesiones óseas de histiocitosis de células de Langerhans en niños, evitando otras terapias más agresivas. (AU)

Eosinophilic granuloma of the bone is the most common variant of Langerhans cell histiocytosis. Most of the lesions occur in the skull, ribs, spine or long bones and may be single or multiple. Therapy is generally chosen based on the site involved and the number of lesions. Treatment options include single agent with prednisone, the combination of vinblastine and prednisone, curettage of bone lesions or intralesional steroids injection. Indomethacin seems to be effective in treating isolated Langerhans cell histiocytosis of the bone in children and is generally well-tolerated. We present the case of a 4-year-old boy with involvement of 2 bones, skull and vertebra, treated with curettage of the skull and indomethacin for 19 months. There was complete healing of the lesions at the end of the treatment and no evidence of recurrence 4 months post-treatment. We conclude that indomethacin seems to be effective in the treatment of Langerhans cell histiocytosis of the bone in children, avoiding more aggressive therapies. (AU)

Granuloma eosinofílico cervical en el adolescente: reporte de un caso y revisión de la literatura / Cervical eosinophilic granuloma in adolescents: case report and literature review

INTRODUCCIÓN El granuloma eosinofílico (GE) es una patología infrecuente, sobre todo en adultos, que puede afectar la columna cervical. A pesar de la vasta literatura, esta enfermedad afecta principalmente a la población infantil, y no hay un consenso sobre el manejo en adultos. Con el objetivo de aportar conocimiento respecto a esta patología poco frecuente, se presenta un caso clínico de GE cervical en un paciente de 16 años, a quien se trató de manera conservadora, con buenos resultados y retorno completo a sus actividades. CASO CLÍNICO Un hombre de 16 años, seleccionado de rugby, consultó por dolor cervical axial persistente y nocturno de 6 semanas de evolución, sin trauma evidente. Al examen, destacó dolor a la compresión axial sin compromiso neurológico asociado. Los exámenes de tomografía computarizada (TC) y resonancia magnética (RM) revelaron lesión lítica en el cuerpo de C3 de características agresivas, de presentación monostótica en tomografía por emisión de positrones-tomografía computada (TEP-TC) compatible con tumor primario vertebral. Se decidió realizar biopsia percutánea bajo TC, para definir el diagnóstico y manejo adecuado, la cual fue compatible con células de Langerhans. Al no presentar clínica ni imagenología de inestabilidad ósea evidente o compromiso neurológico, se manejó con tratamiento conservador, inmovilización cervical, analgesia oral, y seguimiento estrecho. A los cuatro meses de evolución, se presentó con una TC con cambios reparativos del cuerpo vertebral y sin dolor, y logró retomar sus actividad habituales. CONCLUSIONES El diagnóstico de GE es infrecuente a esta edad, y se debe plantear entre diagnósticos diferenciales de lesiones líticas agresivas primarias vertebrales. Es necesario el uso de imágenes, y la biopsia vertebral es fundamental para confirmar el diagnóstico. Su manejo va a depender de la sintomatología, del compromiso de estructuras vecinas, y de la estabilidad de la vértebra afectada. El manejo conservador con seguimiento clínico e imagenológico es una opción viable.

INTRODUCTION Eosinophilic granuloma (EG) is a rare, tumor-like lesion, infrequently affecting the cervical spine, particularly in adults. Although vastly described in literature, this pathology mainly affects children, and there is still no consensus on its treatment in older patients. With the goal of contributing to increase the knowledge regarding this infrequent pathology, we present a case of a C3 eosinophilic granuloma in a 16-year-old patient, who was treated conservatively, with good results, including complete return to his previous activities. CLINICAL CASE a 16-year-old male, elite rugby player, presented with a history of persistent neck pain, mainly at night, with no previous trauma. Upon physical examination, he reported neck pain with axial compression of the head, without neurological impairment. Both computed tomography (CT) and magnetic resonance imaging (MRI) scan revealed an aggressive lytic lesion in the C3 vertebral body, a with monostotic presentation on positron emission tomography-computed tomography (PET-CT) compatible with a primary spine tumor. A CT-guided percutaneous biopsy was obtained to establish the diagnosis and provide the proper management. The results were compatible with Langerhans cells. As he presented no symptoms or imaging findings of evident bone instability, as well as no neurological impairment, the patient was treated conservatively, with a cervical brace, oral pain medication and close followup. A CT obtained after four months of treatment showed reparative changes of the C3 vertebral body; at this point, the patient reported no neck pain, so he was able to return to his previous activities. CONCLUSIONS Although an EG is rare at this age, it should be considered in the differential diagnosis of primary vertebral aggressive lytic lesions. Imaging and a vertebral biopsy are paramount to confirm the diagnosis. The treatment modality depends on the symptoms, the involvement of adjacent structures, and the stability of the affected vertebra. Conservative management including clinical and imaging followup is a viable option.

Cardiac involvement in the adult primary vasculitides.

INTRODUCTION: Heart involvement in vasculitis is rare, but potentially severe. The ascertainment of cardiac disease in vasculitis is complex and requires an integrated multidisciplinary approach involving the Rheumatologist, Radiologist, Cardiologist, and Heart surgeon. AREAS COVERED: the authors searched PubMed using the keywords 'heart'[Mesh] and vasculitis"[Mesh]. EXPERT OPINION: Virtually any vasculitis can affect the heart, but cardiac involvement is more common in some vasculitides such as Takayasu arteritis, polyarteritis nodosa, and eosinophilic granulomatosis with polyangiitis. Immunosuppressive treatment and when indicated surgery can improve the prognosis.

New perspectives in eosinophilic granulomatosis with polyangiitis (EGPA): report of the first meeting of the European EGPA Study Group.

The European Eosinophilic Granulomatosis with Polyangiitis (EGPA) study group first gathered in Firenze in December 2018. The discussion was centred around the clinical and therapeutic needs in EGPA which still remain unmet. Indeed, EGPA is a puzzling and rare disease which shares clinical features with other anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAVs) and hypereosinophilic syndromes (HESs). Some of the recommendations published in 2015 are based on data derived from EGPA-related diseases, rather than from EGPA itself, and therefore need to be updated. Thus, the aim of the meeting was to stimulate ongoing research, to promote collaborative European studies and to define the main issues on which future studies should be focused. Current fields of research on EGPA include potential serological biomarkers of disease activity and of specific organ involvement, possible links between different genetic variants and clinical phenotypes, and new therapeutic perspectives. Herein, we give an overview of the meeting with the goal to stimulate an international collaboration and new points of discussion.

The outcome of eosinophilic granuloma involving unilateral atlantoaxial joint: A case report and literature review.

RATIONALE: Solitary eosinophilic granuloma (EG), the most benign, common form of Langerhans cell histiocytosis, has a self-limiting process and is associated with a good prognosis. Immobilization is recommended as the first treatment strategy for solitary EG, although the treatment protocols are still controversial. Radiotherapy and surgery are secondary treatment choices. Lesions of the upper cervical spine react differently to treatment because of their specific anatomical and motor features. PATIENT CONCERNS: We discuss the case of a 29-year-old man with axis EG who underwent immobilization, radiotherapy, and finally surgery. DIAGNOSIS: Eosinophilic granuloma (EG). INTERVENTIONS: An initial conservative protocol, including immobilization with a collar and radiotherapy, effectively relieved his neck pain, whereas torticollis secondary to atlantoaxial subluxation was not improved. Therefore, he underwent tumor resection through the anterior approach and spinal reconstruction, fixation, and fusion through the posterior approach. OUTCOMES: The deformity was well corrected and follow-up was satisfactory. LESSONS: Upon review of the literature, we found that EG lesions affecting the axis have more risk factors for instability or deformity, and they have particular anatomical and motor characteristics; thus, they require more consideration and attention in terms of treatment, prognosis, and follow-up.

[A lumbar vertebral eosinophilic granuloma with spinal instability in an adolescent]. / Granulome éosinophile vertébral lombaire isolé avec instabilité rachidienne chez un adolescent.

This is a case report of a rare isolated eosinophilic granuloma of vertebra L3, in a 15-year-old adolescent. Vertebral instability, due to aggravation of the osteolysis, occurred without neurological symptoms despite orthopedic treatment. Surgery was necessary to correct and stabilize the spinal deformation. Postoperative chemotherapy (vinblastine and corticoids) was given as recommended for Langerhans cell histiocytosis (HL2010). No recurrence was observed after 5 years of follow-up.

Management of eosinophilic granuloma in pediatric patients: surgical intervention and surgery combined with postoperative radiotherapy and/or chemotherapy.

BACKGROUND: Eosinophilic granuloma (EG) of spine in pediatric patients presents kinds of clinical manifestation and a difficult management scenario. The choice of treatment, issues of surgical intervention versus conservative treatment, combination therapy or single treatment, all these factors, including neurological deficits, spinal stability, long-term complications, and continued skeletal growth, must be considered. METHODS: From 2008 to the 2015, 31 pediatric patients of spinal EG were retrospectively reviewed. They were 17 males and 14 females, with a mean age 8.89 ± 2.84 years old (range, 3.5-14 years old). All the cases were divided into two groups. Twenty-three cases (Group S) accepted surgical interventions and surgery combined with local low-dose radiotherapy and/or chemotherapy. Eight patients (Group C) accepted radiotherapy and/or chemotherapy. All the patients had imaging studies of the lesion including standard radiography, three-dimensional computed tomography (CT) scan, and magnetic resonance imaging (MRI) prior to and after treatment. Cases in group S underwent surgery-related treatment, which include six patients accepted surgery only, nine patients accepted postoperative low-dose radiotherapy, four patients accepted additional chemotherapy, and four patients accepted both. Cases in group C accepted radiotherapy and/or chemotherapy, which include five patients accepted radiotherapy, one patient accepted chemotherapy, and two patients accepted both radiotherapy and chemotherapy. Clinical symptoms, neurologic status, radiologic manifestations, treatment, outcome, and/or complications were recorded and analyzed. RESULTS: All the 31 patients had pain relief (both in group S and group C) but the patients in group S obtained more prompt pain relief. All the patients in group S obtained local kyphosis correction, reconstruction of stability of spine, and recovery of neurological deficit after treatment. They have no surgery-related complications after treatment and in follow-up. Seventeen patients who received additional postoperative radiotherapy and/or chemotherapy also had good outcomes. There were no severe radiation and chemotherapy complications in procedure. The patients (group C) who accepted non-surgical treatment also have no severe complications, and a new femur lesion was found in one patient in follow-up. CONCLUSION: Surgical intervention, including anterior and/or posterior approach with bone graft or surgery combined with postoperative low-dose radiotherapy and/or chemotherapy is a safe and effective way for treatment of the spinal EG. Compared with radiotherapy and/or chemotherapy, more prompt pain relief can be achieved via surgical intervention or surgery combined radiotherapy and/or chemotherapy.

Current concepts for the diagnosis and management of eosinophilic granuloma of bone.

This review summarizes current concepts in the diagnosis and management of the patients with eosinophilic granuloma. Given the benign biology, the clinical course, and the pediatric group of patients that this condition more commonly affects, a treatment approach that carries a lower risk of complications while ensuring a successful cure is desirable. Variable treatment options have been reported with satisfactory results and a recurrence rate of less than 20 %. In this setting, symptomatic lesions that are accessible in the spine or the extremities may be treated with intralesional methylprednisolone injection after tissue biopsy for histological diagnosis.

MEDICAL MANAGEMENT OF RECURRENT EOSINOPHILIC GRANULOMA IN TWO BLACK RHINOCEROS (DICEROS BICORNIS).

Recurrent eosinophilic granuloma (EG) in two captive eastern black rhinoceros ( Diceros bicornis michaeli) was effectively managed with glucocorticoids and antihistamines. The first case was a female and the second case was a male. The animals were housed at separate institutions and initially presented with hemorrhagic oral lesions. Multifocal lesions occurred in the second case. Multiple biopsies were taken from each animal, all of which were consistent with EG. Each animal was anesthetized multiple times for surgical treatment but experienced frequent recurrence. Due to lack of response to therapy and the risks and adverse events associated with repeated anesthesia, medical treatment was initiated in both cases using a tapering dose of oral dexamethasone. The lesions dramatically improved, but would recur frequently after treatment. Hydroxyzine, an oral antihistamine, greatly reduced the incidence and severity of the lesions. Medical management with glucocorticoids and antihistamines minimized stressful anesthetic events in both cases and contributed to the successful management of this recurrent disease. The exact pathogenesis of EG in black rhinoceros remains unknown but response to antihistamines suggests an allergic etiology.

Spontaneous remission of eosinophilic granuloma of the maxilla after incisional biopsy: a case report.

BACKGROUND: Langerhans cell histiocytosis (LCH), previously known as Histiocytosis X, is an infrequent disease that congregates a wide spectrum of clinical presentations with variable systemic involvement. Unification of these diseases under only one category is based on the almost identical histopathologic features of the lesions, but the etiology and proper approach for each presentation remains controversial. The localized alternative of Langerhans cell histiocytosis (LLCH), known as Eosinophilic Granuloma (EG) of bone, is the predominant clinical presentation of LCH. The maxilla is involved in 1 % of the head and neck region cases, representing an uncommon condition in this area. CASE PRESENTATION: In this clinical case report, it is described a case of a 16-year-old male patient with an asymptomatic osteolytic lesion at first upper left molar apical level, a finding detected on control radiographic images was reported as "Monostotic Eosinophilic granuloma of the maxillary bone", which was later confirmed through an incisional biopsy. A surgical excision was initially planned, but finally it was not performed due to a spontaneous healing of the lesion after the incisional biopsy. CONCLUSIONS: The presented case supports a conservative approach in the management of solitary EG of maxillary and mandibular bone lesions and even supports an expectant attitude in the course of treatment given the possibility of a spontaneous regression after the biopsy, especially in small lesions.

Eosinophilic Granuloma of the Mandible: Is a Conservative Treatment Sufficient for Local Disease Control?

Eosinophilic granuloma is the most benign and common form of the Langerhans cell histiocytosis, a rare proliferative disease that can affect single or multiple organs. In the quite common head and neck manifestation these lesions can be confused in the beginning, with other bone diseases such as odontogenic cysts, periodontal disease, or malignancies. Treatment varies depending on the size, number, localization of the lesions, and patient's general conditions. The authors describe here a patient of a single lesion of eosinophilic granuloma localized in the posterior mandible region treated with a very conservative surgical approach in a patient with poor socio-economic conditions. The authors performed teeth extractions, an excisional biopsy and open curettage and after a follow-up of 24 months without partial mandible resection and reconstruction time the lesion had healed. The clinical situation confirmed that, in carefully selective patients, a conservative approach could be a useful therapeutic opportunity.

Orbital Compartment Syndrome in Eosinophilic Angiocentric Fibrosis.

A 43-year-old man reported right visual loss after biopsy of an intranasal mass, which improved after treatment at another hospital. On first examination at our hospital, his visual acuity was 1.2 OD, and MRI showed a right intraorbital mass involving the ethmoid sinus. His visual acuity decreased to 0.08 OD 12 days after orbital biopsy, with right globe tenting shown by MRI. An emergent lateral canthotomy and cantholysis were performed. The histopathological diagnosis was eosinophilic angiocentric fibrosis, and treatment with intravenous methylprednisolone (125 mg/day) and oral diaminophenyl sulfone (75 mg/day) was started immediately after surgery. One month later, the diaminophenyl sulfone was discontinued and cyclophosphamide (50 mg/day) was started. The methylprednisolone and cyclophosphamide doses were tapered over 9 months and 7 months, respectively. At the 1.5-year follow-up examination, his visual acuity was 1.0 OD. The lesions did not grow in size.

[Surgical treatment combined with oral administration of indomethacin for eosinophilic granuloma of the skull: report of a pediatric case].

A 13-year-old girl presented headache for 5 d upon admission to hospital. An initial CT revealed 3 lesions located in her skull, the sizes of which were 2.5 cm×3.2 cm,1.2 cm×1.0 cm,0.3 cm×0.3 cm, respectively. The largest lesion was resected by surgery and confirmed as eosinophilic granuloma by pathology. After surgery, she took oral indomethacin 25 mg b·i·d for 3 months and tolerated it well. CT scan was performed 3 months and 1 year later, and the results showed that the unresected lesions shrank progressively and the defected bones were regenerated and healed one year later after operation.

Eosinophilic granuloma of the temporal bone in children.

Eosinophilic granuloma (EG) is a bony destructive disease that frequently occurs in children; it is a subtype of Langerhans cell histiocytosis. The aims of this study were to detect the presenting features of temporal bone lesions in children and to evaluate the efficacy of surgery combined with radiotherapy in treatment of the disease. A retrospective study on 12 children with EG of the temporal bone was done. Computed tomography and hearing assessment were performed for all patients. All patients were treated with cortical mastoidectomy followed by postoperative radiotherapy. Follow-up was carried out for at least 2 years. The patients' presenting symptoms were external ear canal mass in 10 patients (83.3%), postauricular swelling in 8 patients (66.7%), and persistent otorrhea in 4 patients (33.3%). Ten patients (83.3%) showed conductive hearing loss, whereas 2 patients (16.7%) showed mixed hearing loss on the affected side. Computed tomography showed osteolytic defects without sclerotic margins filled with soft tissue masses involving the mastoid bone. Histopathologic examination showed eosinophils and Langerhans cells that were immune reactive for CD1 antigen and S-100 protein. Postoperative follow-up showed complete cure of the disease in 10 children (83.3%), with recurrence detected in 2 patients (16.7%) who needed second surgical intervention. We concluded that temporal bone EG in children may present with features that mimic the features of chronic suppurative otitis media. However, computed tomography and histopathologic examination are diagnostic. Cortical mastoidectomy together with postoperative radiotherapy is an achievable treatment in most cases.

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.

Benign tumors of the spine.

Benign tumors in the spine include osteoid osteoma, osteoblastoma, aneurysmal bone cyst, osteochondroma, neurofibroma, giant cell tumor of bone, eosinophilic granuloma, and hemangioma. Although some are incidental findings, some cause local pain, radicular symptoms, neurologic compromise, spinal instability, and deformity. The evaluation of spinal tumors includes a thorough history and physical examination, imaging, sometimes laboratory evaluation, and biopsy when indicated. Appropriate treatment may be observational (eg, eosinophilic granuloma) or ablative (eg, osteoid osteoma, neurofibroma, hemangioma), but generally is surgical, depending on the level of pain, instability, neurologic compromise, and natural history of the lesion. Knowledge of the epidemiology, common presentation, imaging, and treatment of benign bone tumors is essential for successful management of these lesions.

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 eosinofílico da coluna vertebral cervical em criança / Eosinophilic granuloma of the cervical spine in children

Granuloma eosinofílico (GE) refere-se à forma benigna e mais frequente de histiocitose de células de Langerhans. Trata-se de uma doença que acomete principalmente crianças e adolescentes, sendo rara a sua ocorrência em adultos. Constitui a forma localizada de proliferação de histiócitos em crânio e ossos longos. O acometimento vertebral é incomum, sendo a localização cervical a menos descrita. Neste artigo, é descrito um caso de uma criança de 7 anos de idade com GE na coluna vertebral cervical com compressão medular. A criança evoluiu com regressão dos sintomas após tratamento conservador, tendo permanecido assintomática ao longo de seguimento de dois anos.

Eosinophilic granuloma (EG) is a benign and more frequent form of Langerhans cell histiocytosis. Children and adolescent are mainly affected, being rare in adults. There is local proliferation of histiocytes in skull and long bones. Vertebral involvement is uncommon, especially at the cervical local. In this paper, we describe a case of a 7 years old child with EG in the cervical spine with spinal cord compression. The patient presented with regression of symptoms after conservative treatment and remained asymptomatic at the two years follow-up.

Eosinophilic granuloma of the temporal bone in an adult: controversies in the management.

Eosinophilic granuloma is a rare, benign, lesion characterized by uncontrolled proliferation of Langerhan's cells. It is commonly found in a bone and has been described in almost every bone in the body. It usually affects children and is seldom found in adults. A case of 60-year-old adult male with an eosinophilic granuloma of the right temporal bone with infiltration of the temporal and infratemporal fossa and osteolysis of the squama of the temporal bone is presented. Diagnostic procedure and course of treatment are described. Controversies in the management and different approaches in therapy of such lesion are discussed because there is no agreed treatment protocol established.

Histiocitosis de células de Langerhans localizada en hueso malar. Presentación de un caso / Localized langerhans cell histiocytosis of the zygomatic bone. A case report

La histiocitosis de células de Langerhans localizada (HCLL), conocida como granuloma eosinófilo, representa entre el 50 y el 60% de todos los casos de histiocitosis de células de Langerhans. El tratamiento clásico para la HCLL ha sido el curetaje o la resección de las lesiones óseas. Hay publicaciones de casos tratados con inyección intralesional de corticosteroides, combinado con curetaje. Se presenta un caso clínico de un paciente de tres años de edad con diagnóstico de HCLL que compromete en su extensión el hueso malar, tratado con infiltraciones de corticosteroides y posterior curetaje de la lesión. A un año de realizado el tratamiento, el paciente se encuentra asintomático y con una regeneración ósea del hueso malar, evidenciable en la tomografía axial computarizada(AU)

Localized Langerhans cell histiocytosis (LLCH), also known as eosinophilic granuloma, represents 50 to 60% of all cases of Langerhans cell histiocytosis. The standard treatment for LLCH has been lesion curettage or resection. Cases treated with intralesional corticosteroid injections combined with curettage have been described. We report the case of a three-year-old patient diagnosed of LLCH with extensive zygomatic bone involvement, who was treated with corticosteroid infiltrations and subsequent curettage of the lesion. One year after treatment, the patient is asymptomatic with zygomatic reossification evidenced on computed tomography(AU)