Cases reported "Lymphatic Diseases"

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1/436. Primary amyloidosis presenting as massive cervical lymphadenopathy with severe dyspnea: a case report and review of the literature.

    amyloidosis is an end point of several diseases in which the unifying characteristic is the association and extracellular deposition of the insoluble protein fibrils within various tissues and organs. We have discussed a variety of clinical findings of primary amyloidosis with emphasis on its otolaryngologic manifestations. This case represents the second report in the literature of a neck mass resulting from a plasma cell dyscrasia and the first report of AL disease producing such extensive cervical lymphadenopathy as to cause significant respiratory compromise.
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2/436. Rapid progression of squamous cell carcinoma of the cervix after hyperbaric oxygenation.

    The role of hyperbaric oxygenation in the treatment of radiation-induced sequelae and chronic ulcer is well established. On the contrary, a possible cancer-causing or growth-enhancing effect by hyperbaric oxygenation was highly controversial. Herein, we present a 55-year-old Chinese woman with recurrent squamous cell carcinoma of the cervix on her left inguinal area. She received concurrent chemoradiation therapy followed by radical inguinal lymphadenectomy due to persistent tumor mass. The patient was complicated with severe radiation fibrosis and unhealed wounds, so she was treated with hyperbaric oxygenation (HBO). However, the patient died of complications of the disease after completing HBO therapy I month later and autopsy of the patient showed carcinomatosis of the abdominal cavity and lower abdominal wall. Because previous studies have been inconclusive regarding the effect of HBO on tumor cells, we reviewed the possible relation between the HBO and tumor cells.
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3/436. Destructive bone disease in early syphilis.

    Although destructive bone disease is a well-known complication of tertiary syphilis, osteitis or osteomyelitis are not commonly recognized as complications of early (primary or secondary) syphillis. A patient with secondary syphilis characterized by generalized lymphadenopathy, perianal condyloma lata, and positive rapid plasma reagin (RPR) and fluorescent treponemal antibody-absorption (FTA-ABS) tests also complained of headache, right should pain, and right anterior chest pain and swelling. Roentgenograms showed mottled osteolytic lesions consistent with previously described luetic bone disease. biopsy confirmed the diagnosis of syphilitic osteomyelitis, and treatment with penicillin resulted in prompt resolution of symptoms.
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4/436. Rosai-Dorfman disease (sinus histiocytosis with massive lymphadenopathy) of the pancreas: first case report.

    Rosai-Dorfman Disease is a histiocytic proliferative disorder which primarily affects lymph nodes. Extranodal involvement occurs in about one half of the patients and the head and neck area represents the region most commonly involved. We present the case of a 48 year-old female who was found with a pancreatic mass during evaluation for abdominal pain. She underwent a distal pancreatectomy and splenectomy. Her pathology showed sinus histiocytosis with massive lymphadenopathy (SHML) involving the pancreas and lymph nodes and focally the spleen. The histiocytes characteristically contained one or more viable lymphocytes in the cytoplasm. The lymphocytes had penetrated the cytoplasm in a process known as "emperipolesis", where the lymphocytes continued to have free movement in the histiocyte. The histiocytic cells were positive with S-100 protein and CD68. Rosai-Dorfman Disease (SHML) can affect the peripancreatic lymph nodes with possible secondary pancreatic involvement and present as a pancreatic mass.
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5/436. Potential association between calcified thoracic lymphadenopathy due to previous histoplasma capsulatum infection and pulmonary mycobacterium avium complex disease.

    BACKGROUND: Among patients with pulmonary disease due to mycobacterium avium complex (MAC) seen recently at our center, a substantial number have had extensive calcified mediastinal, hilar, and peribronchial lymphadenopathy, a finding historically inconsistent with pulmonary MAC disease. METHOD: We retrospectively studied the frequency of calcified lymphadenopathy in the chest and prevalence of known risk factors for MAC infection in 79 patients with pulmonary MAC disease who were referred to our hospital over a 1-year period. RESULTS: Calcified intrathoracic adenopathy was present in 25 of the 79 patients (32%). Residential histories revealed that 20 of the 25 patients (80%) with such calcified chest adenopathy reported living for substantial periods in the regions indigenous for histoplasma capsulatum. In contrast, the residences of patients without calcified chest adenopathy were more evenly distributed throughout the country. Nineteen of these 25 patients (76%) with calcified chest adenopathy had no known predisposing risk factor for the infection; in contrast, the proportion of patients with no calcified adenopathy who also had no identifiable classic risk factor tended to be lower (32/54, 59%). CONCLUSION: In this retrospective study, we observed that (1) a large number of patients with pulmonary MAC disease had no identifiable risk factor, (2) calcified chest adenopathy was present in one third of the patients, (3) the residential history of those with calcified adenopathy mirrored the endemic region of histoplasmosis, and, (4) conversely, those patients with pulmonary MAC who lived outside the histoplasmosis belt had no such adenopathy. Thus, we hypothesize that previous fungal infection may predispose the lungs of certain patients to subsequent invasion by MAC, presumably by airway distortion and/or parenchymal damage.
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6/436. Systemic lupus erythematosus and Castleman's disease.

    Lymphadenopathy is a common finding in systemic lupus erythematosus (SLE), yet lymphoid malignancy is rare. Typically, adenopathy associated with SLE responds to glucocorticoid therapy. We evaluated a patient with a diagnosis of SLE who had progressive lymphadenopathy despite receiving aggressive corticosteroid therapy for SLE associated thrombocytopenia. Histopathology initially revealed an aggressive plasmacytosis characteristic of Castleman's disease (CD). CD, or angiofollicular hyperplasia, is a rare lymphoproliferative neoplasm that has features overlapping many autoimmune diseases. This disorder should be considered in autoimmune diseases with unremitting or progressive adenopathy.
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7/436. Reversible monoclonal lymphadenopathy in autoimmune lymphoproliferative syndrome with functional FAS (CD95/APO-1) deficiency.

    The FAS (CD95/APO-1) receptor and its ligand play an important role in the initiation of apoptosis under many physiologic conditions. Loss of function mutations of the FAS gene have been described in lpr mice and in humans with autoimmune phenomena, recurrent lymphadenopathies, and hepatosplenomegaly. This syndrome is now called autoimmune lymphoproliferative syndrome type I (ALPS I). Recently, patients with similar clinical symptoms due to a functional FAS deficiency without FAS gene mutations have been distinguished. This disease has been termed autoimmune lymphoproliferative syndrome type II (ALPS II) or autoimmune lymphoproliferative disease (ALD). This report is the first description of the lymph node pathology and immunohistochemistry in a patient with ALPS II. After recurrent bacterial infections, a 4-year-old child developed cervical giant lymphadenopathy suggesting lymphoma. Lymph node histology resembled the findings in Epstein Barr virus-associated posttransplant atypical lymphoproliferations. Confluent sheets of immunoblasts, however, showed a monoclonal expression of IgG/lambda and a monoclonal rearrangement of the JH chain. The same clone was also present in the peripheral blood. Although high-grade lymphoma could not be excluded, the patient's parents insisted on the patient's leaving the hospital with only antibiotic treatment. Surprisingly, the giant lymphadenopathy completely resolved within 7 weeks, and the clone was no longer detectable in the peripheral blood. Twelve months later the patient was still free from lymphoma and was doing well. Retrospectively, transient monoclonal B-cell populations could be identified in an archival frozen blood sample taken when the patient was 3 years old. Increased FAS-independent spontaneous apoptosis was a feature of the patient's lymphocytes and could be the molecular basis for self-elimination of B-cell clones. We conclude that the diagnosis of a FAS-FAS-L deficiency should be considered in children with an otherwise unexplained atypical lymphoproliferation and that a diagnosis of lymphoma in patients with functional FAS deficiency should be made with considerable reservation.
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8/436. bartonella henselae associated with Parinaud's oculoglandular syndrome.

    bartonella henselae was recovered from the conjunctival scraping of a 38-year-old woman who presented with a 2-week history of tender preauricular lymphadenopathy and a 1-day history of a red left eye. Dry adherent colonies were observed on agar plates at 21 days of incubation, and the isolate was identified through conventional and molecular tests. polymerase chain reaction (PCR) amplification of a specific region of the 16S rRNA gene and confirmation by a separate PCR reaction with hybridization of the product with a B. henselae-specific probe confirmed the isolate as B. henselae. This is the first reported isolation of the causative agent of cat scratch disease from ocular tissue in a patient with Parinaud's oculoglandular syndrome.
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9/436. Primary sjogren's syndrome with severe central nervous system disease.

    OBJECTIVE: central nervous system (CNS) involvement in primary sjogren's syndrome (pSS) is controversial with regard to frequency, significance, and etiology. methods: We describe a young woman with pSS and severe CNS disease and review the literature on the pathophysiology, clinical significance, symptoms, diagnostic examinations, and treatment of CNS disease with concomitant pSS (CNS-SS). RESULTS: Our patient with pSS had a 5-month history of benign lymphadenopathy and myositis, after which she developed severe CNS disease, vasculitic lesions on her hands, and a neurogenic bladder attributable to spinal cord involvement. The diagnosis was based on the clinical picture and the results of a brain magnetic resonance imaging (MRI) scan, electroencephalography (EEG), and cerebrospinal fluid (CSF) analysis. The disease did not respond to corticosteroids, but the administration of cyclophosphamide resulted in recovery. In the literature, the incidence of CNS-SS varies widely, from rare to incidence rates of 20% to 25%. The clinical picture is diverse, ranging from mild cognitive symptoms to fatal cerebrovascular accidents. The pathophysiology of CNS-SS is unclear, specific diagnostic methods are not available, and diagnosis is based on the clinical picture and a combination of examinations. MRI is the most sensitive test and cerebral angiography the most specific. CSF reflects involvement of the leptomeninges, and EEG is nonspecific. There are no controlled studies of the treatment of CNS-SS. Regimens for vasculitis are commonly used. CONCLUSIONS: CNS-SS is uncommonly recognized and difficult to diagnose. Increasingly accurate and available diagnostic examinations will yield more information about the association of CNS disease with pSS.
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10/436. association of reticular dysgenesis (thymic alymphoplasia and congenital aleukocytosis) with bilateral sensorineural deafness.

    Reticular dysgenesis is a rare congenital disorder characterized by severe combined immunodeficiency and profound neutropenia, curable to date, only by bone marrow transplantation. This report describes the association of bilateral sensorineural deafness with this disease.
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