Cases reported "Granuloma, Lethal Midline"

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1/34. Lethal midline granuloma starting as granuloma laryngis.

    We report a case of lethal midline granuloma in a 34-year-old male patient. The patient was referred to our hospital because of long-lasting hoarseness. He was treated for granuloma laryngis. After two years nasal obstruction developed followed by ulceration of the hard palate and destruction of part of the nose and the upper lip. A biopsy demonstrated polymorphic infiltrate consisting of small lymphocytes, plasma cells, macrophages, atypical lymphoid cells and eosinophils. radiotherapy was very effective and led to long-term remission. The different etiological aspects of this syndrome are discussed.
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keywords = nasal, nose
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2/34. Lethal midline granuloma: report of three cases.

    Lethal midline granuloma (LMG) is a rare clinical entity characterized by progressive relentless ulcerations and necrosis of midfacial structures. It occurs more frequently in Oriental than in Western populations with no demonstrable etiology. Treatment and outcome for cases differ, but their pathological distinction may not always be possible from routine biopsy specimens. The histological features often seen include widespread coagulative necrosis, heavy inflammatory infiltrates, and atypical pleomorphic cells. However, the paucity of these atypical cells in biopsy specimens and the degree of necrosis can make the diagnosis of a neoplastic lesion very difficult. Because of the progress in pathology methodology including immunohistochemistry, most cases have been proven to be malignant lymphomas of T-cell lineage. We present 3 patients for whom an initial clinical diagnosis of LMG was made. From their several oral biopsies and nasal specimens, difficulties were encountered in differentiating "midline granuloma" from other possible diseases using histomorphological criteria alone. After extensive evaluations, malignant T-cell lymphoma was the specific disease entity identified in only one case by cell membrane immunostaining technique. A literature review was carried out, and recent concepts of the etiology and pathogenesis of this disease are presented.
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ranking = 0.52495413841957
keywords = nasal
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3/34. Midfacial granuloma syndrome or an inflammatory non-specific disease? A case report.

    We report a case of idiopathic midline destructive disease in a 57-year-old man. The patient had a non-specific histological pattern in biopsies obtained from the nose and upper lip, characterized by a granulomatous reaction with progressive destruction of the tissues. The patient's general medical history was non-contributory. Clinical and laboratory data did not support any feasible etiology for this destructive process. The patient was treated with prednisone until the discovery of type II diabetes mellitus (never diagnosed before) and was then in turn treated only with oral antidiabetic therapy. Follow-up controls revealed progressive reduction of the symptoms and of the nasal and lip lesions and total remission of symptoms up to 2 years after the onset of the disease. We discuss the diagnostic and subsequent therapeutic problems in the management of the midline necrotizing lesions.
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ranking = 1.4750458615804
keywords = nasal, nose
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4/34. Differential diagnosis of midline facial granulomas.

    The list of differential diagnoses in the case of midline facial granulomatous lesions is long. Intensive investigation into the true origin of disease must be done before definitive treatment can be begun. Extensive and repeated biopsies are necessary. The diagnosis may narrow down to the nonspecific midline lethal granuloma. Evidence in the literature coupled with our experience strongly indicates that this disease does not exist as a pathologic entity but merely as a clinical syndrome which upon further evaluation will reveal either Wegener's granulomatosis or a reticulum cell neoplasm. Treatment of the syndrome consists of corticosteroid drugs, irradiation, and chemotherapy until a definitive diagnosis is made. Midline granulomas have been and will continue to be a challenging diagnostic and therapeutic problem for the otolaryngologist.
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ranking = 0.47504586158043
keywords = nose
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5/34. T/NK cell non-Hodgkin's lymphoma of the sinonasal tract.

    Non-Hodgkin's lymphoma of the sinonasal tract is now recognized as an important cause of destructive midfacial lesions formally designated as idiopathic inflammatory processes, and commonly treated with local radiotherapy in a bid to halt the destructive process. However, left untreated, the natural history of this disease remains largely unknown. We report a case which demonstrates the slow and apparently indolent natural course that these lymphomas, if left untreated, may display, before finally evolving into overwhelming and fatal disease. We also take the opportunity to present a brief synopsis of the evolution of our understanding of this condition and to review the modern literature on it.
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ranking = 2.6247706920979
keywords = nasal
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6/34. Sinonasal (angiocentric) T/NK cell lymphoma: report of a case with a -12-year history free of recurrent/residual disease and a sudden deterioration.

    This report describes a case involving a 78-year-old Caucasian male, whose medical history was significant for sinonasal (angiocentric) T/NK lymphoma, who was treated by surgery and radiation in 1988. After the treatment, the patient was apparently free of residual/recurrent disease for a period of 12 years. There was periodical clinical follow up including repeated biopsies. After this period, the patient suffered from sudden deterioration of the health status with multiorgan involvement by the disease and he died. The diagnosis was confirmed by an autopsy. To our knowledge, this is the fifth reported case with extended survival (more than 12 years) free of recurrent/residual disease after the initial treatment, in which the patients ultimately died because of the disease. These findings suggest the importance of prolonged clinical follow-up in patients with this diagnosis.
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ranking = 2.6247706920979
keywords = nasal
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7/34. Lethal midline granuloma (peripheral T-cell lymphoma) after lymphomatoid papulosis.

    A Japanese woman with an 8-year history of lymphomatoid papulosis (LP) had lethal midline granuloma (LMG) develop at the age of 51 years. There were histologic similarities between LP and LMG seen in this patient. Surface phenotypic studies on nasal and cutaneous lesions demonstrated a population of T-cells expressing CD2, CD4, CD25, CD30, and histocompatibility antigen-DR (HLA-DR). Genotypic analyses of nasal and skin biopsy specimens disclosed a clonal rearrangement of the beta T-cell receptor gene with the same rearrangement pattern. These data indicate that this patient had LMG characterized by clonal peripheral T-cell lymphoma, which probably resulted from progression of the LP.
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ranking = 1.0499082768391
keywords = nasal
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8/34. Midline malignant B-cell lymphoma with leukemic transformation.

    This article presents a case of lethal midline granuloma on the palate of a 44-year-old woman, which had been identified histologically as B-cell lymphoma with leukemic transformation in the terminal stages. At the first visit, physical and laboratory examinations showed no remarkable findings except for a necrotizing ulcer of the palate, and the biopsy specimens only showed massive inflammatory cell infiltration and necrosis of the granulation tissue. There was a short-term resolution after treatment with cyclophosphamide and prednisolone, but the disease reactivated and the necrotic ulcerative lesion progressively advanced into the nasal cavity. Specimens from the third biopsy exhibited histologic features that were consistent with malignant lymphoma of the diffuse, mixed B-cell type. Chemotherapy with the regimen of cyclophosphamide, doxorubicin, vincristine, and prednisolone was adopted but was interrupted after a short time because of bone marrow suppression. Subsequently, large numbers of enlarged abnormal lymphocytes with a few vacuoles in the cytoplasm appeared in the circulating blood, indicating leukemic transformation of the midline lymphoma. The patient died on the seventh day after the initiation of chemotherapy. In the presentation of this case, the authors mention clinically important matters regarding midline lethal lymphoma and briefly discuss the pathophysiology and pathogenesis.
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ranking = 0.52495413841957
keywords = nasal
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9/34. Midline lethal granuloma--a clinical enigma.

    Midline Lethal granuloma is characterized by progressive destruction of nose, paranasal sinuses and palate. Till date, the diagnosis of this mutilating process remains as enigma due to the non specific histological and systemic findings. However, over the years the clinicians have been able to divide the "Lethal midline granuoloma syndrome" into clinical entities: Idiopathic midline destructive disease, Wegener's granulomatosis, polymorphic retiaculosis and Non-Hodgkins lymphoma. This article attempts to distinguish between these disease entities in the light of 2 case reports of Idiopathic midline destructive disease.
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keywords = nasal, nose
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10/34. Centrofacial malignant granulomas. Clinicopathologic study of 40 cases and review of the literature.

    An important problem in the treatment of centrofacial ulcerations is to establish a precise diagnosis, since similar clinical and microscopic findings can result from many different causes (as in the centrofacial malignant granuloma syndrome [CFMG]). A comprehensive surgical biopsy protocol (known as SNFMI/GMCF), involving microbiology, parasitology, immunology and pathology laboratories, allowed us to evaluate and to treat 40 cases of CFMG, who form the basis of this report. In 13 of them, specific diagnoses were found and curative treatments could be given. In the remaining 27, the optical microscopy pattern met the criteria for CFMG without identifiable origin or the presence of so-called lethal midline granulomas; however, a more precise evaluation with the help of immunofluorescence studies led to the recognition of malignant lymphoma (ulcerative lymphoma of the midface [ULM]). Most of these lymphomas belonged to the T cell lineage; the others were of B lymphoid origin, or, more rarely, of histiocytic origin. patients with ULM received radiotherapy and chemotherapy with a response rate of 70.3%; however, the toxicity was significant, with frequent occurrence of chemotherapy-induced neutropenia followed by severe infectious facial cellulitis. Six patients were enrolled in a preliminary open trial of treatment with recombinant alpha-2b interferon with little success. Three patients were treated with radiation therapy only, and survived. Thus, CFMG is a syndrome with specific causes and treatments, requiring multiple extensive biopsies to make the correct diagnosis. The recognition of ULM as the cause of the previously called "lethal midline granulomas" leads logically to the use of chemotherapy with growth factors in order to ameliorate its bad prognosis.
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ranking = 0.47504586158043
keywords = nose
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