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PubMed1976

Acquired angioedema with lymphoproliferative disorder: association of C1 inhibitor deficiency with cellular abnormality.

A patient with a lymphoproliferative disorder, angioedema, and an acquired deficiency of the inhibitor of the activated first component of complement was studied. The patient's complement profile revealed depletion of the first component of complement, which has not been seen in angioedema of the hereditary type. There was no evidence for C1-depleting activity in the patient's plasma. The majority of the patient's peripheral blood mononuclear cells resembled B cells in their memebrane receptor properties and in that they carried easily detectable immunoglobulin, predominantly IgM. However, these cells were unusual in that they phagocytosed both latex particles and C3-coated erythrocytes. Morphological study of the cells infiltrating the patient's lung revealed immature, atypical, and plasmacytoid lymphocytes and immunoblasts. Both the patient's peripheral blood mononuclear cells and a suspension of cells from the pulmonary infiltrate were capable of depleting the first component of complement and its inhibitor from homologous plasma. Normal ABO-compatible cells did not possess this property. The data suggested that the patient's abnormal lymphoid cells may have interacted with the complement system to produce a biochemical defect and a clinical syndrome closely resembling angioedema of the hereditary type.

Aged↗

PubMed1983

The ICU chest film: cardiac versus pulmonary disease.

We have discussed several diseases that diffusely affect the pulmonary parenchyma. The diagnostic problem is to separate cardiac pulmonary edema from noncardiac pulmonary edema, diffuse interstitial fibrosis, and lymphangitic spread of carcinoma. Frequently, this may not be possible by radiographic means alone, and additional historic and physiologic information must be obtained. It is also important to know that cardiac pulmonary edema may present in a focal or regional distribution in patients with chronic obstructive pulmonary disease. Several additional radiographic tests may be used to evaluate abnormal pulmonary parenchymal densities seen on the portable chest radiograph, when the differential diagnosis includes increased extravascular water, pneumonia, and pulmonary fibrosis. The easiest of these tests to perform is the gravitational shift test.

Heart Diseases↗

PubMed1995

Association of p53 mutations with metastatic prostate cancer.

In prostate cancer, mutation of the p53 tumor suppressor gene has been associated with locally advanced disease and hormone-resistant disease that is predominantly localized to bone. However, little is known regarding the status of the p53 gene in metastatic prostate cancer that has not been treated with hormonal manipulation. We evaluated formalin-fixed, paraffin-embedded malignant tissues from 86 patients with various stages of prostate cancer, including pathologically confined, locally advanced, and metastatic disease, to detect abnormal p53 nuclear protein accumulation using immunohistochemistry. No abnormal p53 immunostaining was detected in 18 patients with prostate cancer confined to the gland. Two tumors from 21 patients with locally advanced disease (extracapsular extension and/or seminal vesicle invasion) had abnormal nuclear p53 accumulation, and a mutation in exon 7 of the p53 gene was detected in tumor DNA from one patient using single-strand conformation polymorphism-direct sequencing analysis. Of the remaining 47 patients studied in whom tissues from the prostate gland and a metastatic site (44 lymph node, 2 bone, and 1 lung) were available, only 3 had received hormonal therapy prior to obtaining metastatic tissue. In four patients both primary and metastatic tumors demonstrated accumulation of p53 protein, whereas seven additional patients exhibited p53 accumulation only at the metastatic site. In three patients the metastatic tumors harbored missense single-base substitutions in exon 5, as detected using single-strand conformation polymorphism-direct sequencing. These results indicate that p53 abnormalities are associated with lymph node metastases derived from prostate cancer patients that had not undergone hormonal therapy.

Adenocarcinoma↗

PubMed1993

Power of the MAC (morphology-antibody-chromosomes) method in distinguishing reactive and clonal cells: report of a patient with acute lymphatic leukemia, eosinophilia, and t(5;14).

We present a patient with acute lymphatic leukemia, eosinophilia, and a 5;14-translocation, a rare but well-documented condition. In order to clarify whether granulocytes were involved in the disease, we applied the MAC (Morphology-Antibody-Chromosomes) technique to samples of the bone marrow and, during a central nervous system relapse, to those of the cerebrospinal fluid. The karyotype of the blast cells was 47,XY, + X,t(5;14)(q31;q32),i(7)(q10). Interphase cytogenetic study by in situ hybridization with an X-specific alphoid probe revealed the abnormality in CD10, CD19, and TdT (terminal deoxynucleotidyl transferase) positive lymphoid cells, whereas CD13 positive, Sudan black B positive, eosinophilic, and basophilic granulocytes as well as monocytes and small lymphocytes did not have the abnormality. Our results show that the eosinophilic and basophilic granulocytes in this subtype of acute leukemia do not belong to the malignant clone but are reactive. This study also confirmed the usefulness of the MAC technique in distinguishing neoplastic and reactive cells in malignancy.

Adult↗

PubMed2000

Familial amyloidotic polyneuropathy (ATTR Ser50Ile): the first autopsy case report.

We report an autopsy case of a pedigree of familial amyloidotic polyneuropathy (FAP) with a mutation of isoleucine-50 transthyretin (ATTR Ser50Ile). A 47-year-old man started developing severe diarrhea and weight loss at age 41 years, followed by urinary incontinence, autonomic-nervous-system abnormalities and serious heart failure; the diagnosis of FAP (ATTR Ser50Ile) was made on the basis of genetic, histochemical and immunohistochemical analysis. Six years after the initial symptoms, he died of septic shock. Autopsy revealed suppurative peritonitis, perforation of the sigmoid colon and marked systemic amyloid deposition. The total amount of amyloid deposited in the heart was greatly increased and was much lower in the thyroid gland and kidneys compared with amyloid deposits in ordinary FAP (ATTR Val30Met). Amyloid deposition in peripheral vessel walls was prominent, particularly in lymphatics and veins. His elder sister, 54 years old, started to develop orthostatic hypotension at age 49 years, followed by dysesthesia, diarrhea and severe congestive heart failure. Endomyocardial biopsy revealed severe TTR-amyloid deposition; ultrastructural examination demonstrated that amyloid fibrils were deposited disproportionately and extended radially around microvessels.

Aged↗

PubMed2005

Chronic and delayed-onset mustard gas keratitis: report of 48 patients and review of literature.

PURPOSE: To report the clinical features of 93 eyes of 48 patients with chronic and delayed-onset mustard gas keratitis. Clinicopathologic correlation in 5 eyes and a review of related literature are presented. DESIGN: Retrospective, noncomparative case series. PARTICIPANTS: Forty-eight Iranian survivors of Iraqi chemical warfare with chronic or delayed-onset mustard gas keratitis. METHODS: We reviewed the symptoms, clinical findings, course, and treatment of our patients and reviewed the literature. In 5 patients, histopathologic features of corneal and conjunctival specimens were evaluated. MAIN OUTCOME MEASURES: Ocular findings, clinical course, treatment measures, and histopathologic studies. RESULTS: Of 48 patients, 31 (64.6%) had chronic symptomatology, whereas 17 (35.4%) experienced delayed-onset lesions. Visual acuity at referral ranged from hand motions to 20/20. Ocular surface changes included chronic blepharitis and decreased tear meniscus in all patients, limbal ischemia (81.3%), and conjunctival vascular abnormalities (50%). Corneal signs in order of frequency were: scar or opacity (87.5%), neovascularization (70.8%), thinning (58.3%), lipoid deposits (52.1%), amyloid deposits (43.8%), and epithelial defects and irregularity (31.3%). Many patients received conservative treatment; others underwent allograft stem cell transplantation (20 eyes of 17 patients), penetrating keratoplasty (12 eyes of 12 patients), and lamellar keratoplasty (4 eyes of 3 patients). Conjunctival specimens were evaluated by light microscopy. Decreased goblet cell density, attenuated or thickened epithelium, scarring in the substantia propria associated with plasmacytic and lymphocytic infiltration, and dilated lymphatic vessels were noted. Excised corneal buttons disclosed absence of epithelium and Bowman's layer, fibrovascular pannus, stromal scarring, and vascularization. CONCLUSIONS: Mustard gas causes chronic and delayed destructive lesions in the ocular surface and cornea, leading to progressive visual deterioration and ocular irritation. The pathophysiologic features of these changes are not clearly identified. Excised conjunctival and corneal specimens revealed a mixed inflammatory response without any specific features. Based on the clinical appearance of the lesions and the histopathologic findings, an immune-mediated component seems possible. This article contains additional online-only material available at.

Adult↗

PubMed1981

Adenocarcinoma of the uterine tube: potential for lymph node metastases.

A clinicopathologic study of 15 cases of primary adenocarcinoma of the uterine tube occurring over a 12-year period is presented. The mean age of the patients was 54.8 years. Predominant symptoms were abnormal bleeding, abnormal vaginal discharge, and pelvic pain. Abnormal vaginal cytology was noted in two instances (13%). A pelvis mass was detected in two thirds of patients. The neoplasm was bilateral in three instances (20%). Lymph nodes were involved in eight cases (53%), and metastatic spread to the para-aortic nodes was present in five (33%). The most common primary treatment was total abdominal hysterectomy and bilateral salpingo-oophorectomy. Various combinations of adjuvant radiation therapy and chemotherapy were also employed. Six patients (40%) are alive without evidence of recurrent cancer. It is concluded that lymphatic spread, especially to the para-aortic nodes, is a major pattern of disseminaton for adenocarcinoma of the uterine tube. The presence of tumor in capillary-like spaces bears a strong relationship to lymph node metastases, and the para-aortic nodes are a frequent site of involvement even when the disease is apparently limited to the tube.

Adenocarcinoma↗

PubMed1999

Erdheim-Chester disease: clinical, radiologic, and histopathologic findings in five patients with interstitial lung disease.

Erdheim-Chester disease is a clinicopathologic entity defined by a characteristic pattern of symmetric osteosclerosis caused by an infiltrate of mononuclear cells that include prominent numbers of foamy histiocytes. About half of patients have extraskeletal manifestations, including involvement of the hypothalamus/posterior pituitary, orbit, retroperitoneum, skin, lung, and heart. Pulmonary involvement is an uncommon but important manifestation of Erdheim-Chester disease because it causes significant morbidity and mortality. A review of the Mayo Clinic files produced four patients with confirmed Erdheim-Chester disease in whom lung biopsy had been performed. One additional patient was included from the University of Pittsburgh. Four patients were women. The mean age was 53.6 years (range 25-70 years). All patients had bilateral and symmetric sclerotic bone lesions characteristic of Erdheim-Chester disease, although in three the skeletal abnormalities were discovered only after lung biopsy. Four patients had dyspnea, and one also had a dry cough. One patient died 17 months after diagnosis. Chest radiographs showed diffuse interstitial infiltrates in all patients, with an upper zone predominance in three. Thoracic computed tomography (CT) scans showed thickening of the visceral pleura and interlobular septa with patchy associated fine reticular and centrilobular opacities and ground glass attenuation. Lung biopsy specimens showed an infiltrate of foamy histiocytes, lymphocytes, and scattered Touton giant cells with associated fibrosis in a striking lymphatic distribution. The infiltrate involved visceral pleura, interlobular septa, and bronchovascular bundles. Immunohistochemical stains were positive for CD68 in all cases and S-100 protein in four cases. Stains for CD1a were consistently negative. Ultrastructural studies in one case showed no Birbeck granules. Although in bone the histologic features of Erdheim-Chester disease may overlap with Langerhans' cell histiocytosis, its expression in the lung is distinct. Lung involvement in Erdheim-Chester disease has emerged as a unique radiographic and histologic entity.

Adult↗

PubMed1995

[A case of primary pulmonary non-Hodgkin's lymphoma showing a fine granular pattern around the main tumor on thin-slice CT].

A 61-year-old woman was admitted for evaluation of an abnormal shadow found incidentally on a chest radiograph. A 35 x 20 mm nodule with an ill-defined margin was found at the left hilum on conventional CT. Thin-slice (2 mm) CT was done, and a fine granular zone was found around the lesion. Left upper lobectomy was done and the lesion proved to be a primary pulmonary non-Hodgkin's lymphoma (small cell type, B-cell). The granular pattern seen on CT was found to correspond to intraseptal micronodules scattered along the lymphatic vessels, a finding which is considered to be pathognomonic of pulmonary lymphomas by many pathologists. We know of no previous description of how these histologic characteristics can appear on high-resolution thin-slice CT. These findings will be helpful for the CT diagnosis of pulmonary lymphoma.

Diagnosis, Differential↗

PubMed1977

Human immunodeficiency disease: impairment of cellular interactions leading to abnormal mediator production in mixed lymphocyte culture reaction.

Leukocyte migration inhibitory factor (LMIF) production in mixed lymphocyte culture (MLC) reactions is the result of cellular interactions based on two separate phenomena: the capacity of lymphocytes to stimulate in MLC, and the capacity of lymphocytes to respond in MLC. Puromycin-treated lymphocytes are capable of stimulating allogeneic cells for LMIF production, but are unable to respond with synthesis of LMIF (one-way MLC-LMIF test). We have studied the stimulating and responding capacity of lymphocytes from patients with different immunodeficiency syndromes in a one-way MLC-LMIF assay. Lymphocytes from patients known to have qualitative and quantitative defects of T cell or B cell functions (Hodgkin's disease, mycosis fungoides, thymoma, chronic lymphatic leukemia) were found to respond poorly as measured by mediator production although their stimulating fuction was frequently retained. Patients with advanced solid tumors often had both MLC-stimulating and responding functions depressed. There was no apparent correlation between mitogen response and MLC-induced LMIF responses or between MLC proliferative response (as measured by thymidine incorporation) and mediator production. Studying of stimulatory and responding capacity of lymphocytes in the MLC-LMIF assay provides a new tool for assessing immunocompetence and allows for in vitro evaluation of cellular interactions that may play an important role in vivo.

Candidiasis, Cutaneous↗

PubMed1995

Surgical predictors of para-aortic metastases in early-stage cervical carcinoma.

Radical pelvic surgery for cervical carcinoma is contraindicated in the presence of para-aortic node metastases. However, the incidence of para-aortic nodal involvement is very low in early-stage disease. Therefore, it may not be necessary to subject all patients to para-aortic lymphadenectomy prior to radical hysterectomy. Medical records for 408 patients with early-stage cervical carcinoma treated at the Pennsylvania State University-M.S. Hershey Medical Center were reviewed to ascertain if clinical factors can be utilized intraoperatively to accurately predict those patients at minimal risk for para-aortic lymph node metastases. The presence of clinically suspicious (abnormally enlarged or firm) pelvic or para-aortic lymph nodes or extracervical spread of tumor at the time of exploration were significant predictors of para-aortic metastases (P < 0.001). The majority of patients (85%) had none of these risk factors, and no patient had para-aortic metastases in the absence of these predictors. Suspicious pelvic or para-aortic lymph nodes were present in the minority of patients (15%) and identified all patients with para-aortic metastases. Therefore, para-aortic lymphadenectomy may be safely omitted at the time of exploration for radical hysterectomy in the absence of enlarged or abnormally firm pelvic or para-aortic lymph nodes. In the presence of either of these factors or extracervical spread of disease a para-aortic lymphadenectomy is necessary to rule out metastases.

Adenocarcinoma↗

PubMed1997

Localization of ectopic parathyroid glands using technetium-99m sestamibi imaging: comparison with magnetic resonance and computed tomographic imaging.

The aim of the study was to compare the accuracy of technetium-99m sestamibi imaging for localization of ectopic parathyroid glands in patients with hyperparathyroidism with that of magnetic resonance (MR) and computed tomographic (CT) imaging. Eleven patients with primary (n=3) or secondary (n=8) hyperparathyroidism were studied with 99mTc sestamibi parathyroid imaging CT and MR imaging. Images of the neck were acquired at 10 min and 2-3 after tracer injection. The three patients with primary hyperparathyroidism and five patients with secondary hyperparathyroidism underwent parathyroidectomy. The ectopic glands were confirmed by histopathological examination of the resected specimens. In respect of 20 parathyroid glands in the eight patients explored surgically, the sensitivity and specificity of sestamibi imaging were 70% (14/20) and 88%, respectively, those of CT, 40% (8/20) and 88%, and those of MR imaging, 60% (12/20) and 88%. Of these patients, three had parathyroid adenomas while five had hyperplasia (17 glands). Sestamibi imaging localized eight ectopic parathyroid glands, which were surgically confirmed (six were located in the thymus and two in the mediastinum). In one patient explored surgically, the ectopic gland was located outside the field of the MR coil. Although the remaining three cases of secondary hyperparathyroidism were not confirmed surgically, these patients demonstrated sestamibi uptake in five parathyroid glands, including three ectopic glands. MR images demonstrated abnormal parathyroid glands in the same regions as sestamibi imaging. Our data indicate that 99mTc-sestamibi imaging should be used initially to localize the ectopic parathyroid glands in patients with hyperparathyroidism for anatomical guidance prior to MR or CT imaging.

Choristoma↗

PubMed2001

Trisomy 8 as sole karyotypic aberration in an ovarian metastasizing Sertoli-Leydig cell tumor.

Sertoli-Leydig cell tumors (SLCTs) represent a rare group of sex-cord stromal tumors of the ovary of unknown pathogenesis. We report a SLCT of intermediate differentiation with peritoneal recurrence and lymph node metastasis 12 months after removal, including cytogenetic analysis by comparative genomic hybridization and fluorescence in situ hybridization, which showed trisomy 8 as sole unbalanced karyotypic aberration. Our results provide evidence that a simple numeric chromosomal abnormality in SLCT may be associated with a malignant phenotype and suggest that the molecular pathogenesis of SLCT may be different from ovarian granulosa-stromal cell tumors.

Adolescent↗

PubMed1991

Nasopharyngeal carcinoma with bone marrow metastasis.

Five of 23 patients with recurrent nasopharyngeal carcinoma (NPC) were diagnosed to have bone marrow metastasis. They all had advanced local-regional disease, and were treated with neoadjuvant chemotherapy and definitive radiotherapy after the initial diagnosis. Bone marrow metastasis developed 4-24 months later. The clinical features were anemia (5 of 5), leukopenia (3 of 5), thrombocytopenia (4 of 5), sepsis (3 of 5), tenderness of the sternum (3 of 5), and fever (4 of 5). Patients frequently had elevation of serum lactic dehydrogenase (LDH), alkaline phosphatase (ALK-P), and IgG and IgA antibody titers to Epstein-Barr viral capsid antigen when bone marrow involvement was diagnosed. However, clinical manifestations and laboratory tests were not specific. It is important that three patients had normal bone scans. All five patients had a rapid downhill course; four patients died within 23 days, and the fifth 3 months after the diagnosis of bone marrow metastasis. We concluded that bone marrow was a common metastatic site in NPC patients. Bone marrow metastasis adversely affected patients' survival and required a high index of suspicion for diagnosis. We suggested that bone marrow biopsy should be considered as a routine staging procedure in NPC patients and indicated especially when patients presented with abnormal blood counts, sepsis, bone pain, or tenderness of the sternum. It may be positive in the face of a normal bone scan.

Adult↗

PubMed1992

Ga-67 uptake by metastatic carcinoma of the penis.

Six patients with metastatic carcinoma of the penis underwent Ga-67 citrate scanning. Intense radioactive uptake was noted in the metastatic inguinal lymph nodes that was not influenced by the presence or absence of infection. There was a correlation between clinical improvement and decrease in abnormal groin uptake. The results reported here suggest the possibility of using Ga-67 citrate scanning in the staging, management, and follow-up of this relatively rare condition, although further experience is needed to establish its precise role.

Adult↗

PubMed1997

Is there an anatomic explanation for chest pain in patients with pulmonary sarcoidosis?

BACKGROUND: Chest pain (CP), its cause unknown, is a common and often prominent symptom of sarcoidosis. METHODS: We determined the frequency and character of CP in patients with pulmonary sarcoidosis and examined its relationship with (1) length of time since diagnosis, (2) roentgenograhic stage, and (3) radiographic abnormalities on spiral chest computed tomography (CT). RESULTS: Twenty-two patients were studied: 14 of 22 patients (64%) had CP, with 4 of 14 (29%) identifying pain as their primary symptom. Eleven of 14 (79%) had pleuritic CP; 12 of 22 (54.5%) described CP as substernal; and 5 of 22 (22.7%) described CP between the scapula. There was not a significant correlation between CP and the presence or degree of lymphadenopathy. There was no significant correlation between CP and the presence or location of pleural disease. Abnormalities of other thoracic structures also had no significant correlation with the presence of CP. CONCLUSIONS: We conclude that there is no "anatomic reason" for CP in patients with pulmonary sarcoidosis that is evident on chest CT.

Adult↗

PubMed1975

Lymphangitic pulmonary metastases secondary to breast cancer with normal chest x-rays and abnormal perfusion lung scans.

Three patients with advanced breast cancer are described who developed lymphangitic pulmonary metastases with normal chest x-rays and abnormal perfusion lung scans. The abnormal lung scans can probably be attributed to embolization of the pulmonary vasculature, first described in association with lymphangitic pulmonary metastases in 1903. The importance of recognizing this condition in patients with breast cancer is shown by their frequently good responses to combination chemotherapy.

Adult↗

PubMed2001

Lymph node staging by positron emission tomography in patients with carcinoma of the cervix.

PURPOSE: The aim of this study was to compare the results of computed tomography (CT) and positron emission tomography (PET) with [18F]-fluoro-2-deoxy-D-glucose (FDG) for lymph node staging in patients with carcinoma of the cervix and to evaluate the relationship of the imaging findings to prognosis. PATIENTS AND METHODS: We retrospectively compared the results of CT lymph node staging and whole-body FDG-PET in 101 consecutive patients with carcinoma of the cervix. Patients were treated with standard irradiation and chemotherapy (as clinically indicated) and observed at 3-month intervals for a median of 15.4 months (range, 2.5 to 30 months). Progression-free survival was evaluated by the Kaplan-Meier method. RESULTS: CT demonstrated abnormally enlarged pelvic lymph nodes in 20 (20%) and para-aortic lymph nodes in seven (7%) of the 101 patients. PET demonstrated abnormal FDG uptake in pelvic lymph nodes in 67 (67%), in para-aortic lymph nodes in 21 (21%), and in supraclavicular lymph node in eight (8%). The 2-year progression-free survival, based solely on para-aortic lymph node status, was 64% in CT-negative and PET-negative patients, 18% in CT-negative and PET-positive patients, and 14% in CT-positive and PET-positive patients (P <.0001). A multivariate analysis demonstrated that the most significant prognostic factor for progression-free survival was the presence of positive para-aortic lymph nodes as detected by PET imaging (P =.025). CONCLUSION: This study demonstrates that FDG-PET detects abnormal lymph node regions more often than does CT and that the findings on PET are a better predictor of survival than those of CT in patients with carcinoma of the cervix.

Actuarial Analysis↗