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PubMed1987

Polyarteritis nodosa and familial Mediterranean fever.

A 22-year-old familial Mediterranean fever (FMF) patient was hospitalized for continuous fever, myalgia, hypertension, vertigo and a petechial rash. Laboratory findings revealed hyperglobulinaemia, thrombocytosis and a leukaemoid reaction. While on steroid therapy the patient sustained a haemorrhage into a renal aneurysm which responded to gel foam embolization. After 12 months of follow-up his condition remained stable under treatment with cyclophosphamide, prednisone and antihypertensive medications. This case provides the fourth example of polyarteritis nodosa associated with FMF.

Abdomen↗
PubMed1993

Systemic-onset juvenile chronic arthritis and bone marrow hypoplasia.

A two- and-a-half-year-old girls with systemic-onset JCA developed pancytopaenia 21 days after her illness began. Bone marrow examination revealed hypoplasia, with no evidence of erythrophagocytosis. Two weeks later peripheral blood specimens showed evidence of bone marrow regeneration. No definite cause for the hypoplasia was found subsequently. Persistent haematological abnormalities after resolution of the hypoplasia included anaemia of chronic inflammation, leucocytosis and thrombocytosis. Unexplained bone marrow hypoplasia has not previously been described in systemic-onset JCA.

Arthritis, Juvenile↗
PubMed1996

Correlates of disablement in polyarticular juvenile chronic arthritis--a cross-sectional study.

To assess the impact of disease on the functional outcome of patients with polyarticular juvenile chronic arthritis (JCA), the relationship between impairments and functional limitations was studied. Therefore, variables from the impairment domain were correlated with variables of the functional limitation domain and outcome variables were analysed for differences as a result of inflammatory disease, rheumatoid factor (RF), disease duration and age at onset. Twenty-three patients with polyarticular JCA were subjected to auxologic evaluation, a laboratory check, radiographic evaluation, joint count on tenderness and swelling, joint mobility/deformity examination, functional assessment of skills, health assessment and psychosocial evaluation. Inflammatory disease parameters, like CRP, ESR, thrombocytosis and leucocytosis, were increased in 6/23 patients. The parameters of the impairment domain, like joint tenderness and swelling, showed mild outcome, while parameters of the functional limitation domain showed more severe outcome. Generally, perceived competence was found to be normal. A clinically relevant number of patients (10/13) showed low scores on the activity factor of the Child Behaviour Check List (CBCL). A significant relationship was found between inflammatory disease variables and functional limitation outcome. RF seropositivity was not a good outcome predictor. Disease duration and age of onset showed no significant difference in the outcome of the domains. Significant correlation was found between the parental report of the Childhood Health Assessment Questionnaire (CHAQ) and all impairment parameters. Joint swelling showed a significant relationship with CHAQ and Juvenile Arthritis Functional Assessment Report (JAFAR). Disability outcome did not correlate with functional limitation. In general, children with polyarticular JCA function rather well when using a multidomain evaluation approach. Compensatory and adaptational mechanisms might contribute to the poor correlation between impairment and functional limitation parameters. Laboratory evaluation of inflammatory disease, a joint count of swollen joints and parent's report of the child's health status related best in our study.

Adolescent↗
PubMed1999

Haematological abnormalities in children with tuberculosis.

Over a 16 month period 307 children with suspected tuberculosis (TB) and an available full blood count (FBC) seen at Tygerberg Hospital in South Africa were evaluated and categorized as confirmed (A), probable (B), and no TB (C) according to WHO criteria. There was no difference in the mean age of the 168 group A (33.6 months), 83 group B (34.4 months), and the 56 group C (31.6 months) children. A lower mean haemoglobin (Hb 10.2 vs. 10.8 g/dl) was the only significantly different haematological parameter in children with TB compared with the comparison group (Group C). There were no differences in median total white cell count, neutrophils, lymphocytes, monocytes, platelets, or the proportion of children in each group with anaemia, microcytosis, neutrophilia, neutropenia, lymphocytosis, lymphopenia, monocytosis, thrombocytosis or thrombocytopenia. The most common haematological abnormalities in children with TB were the presence of anaemia, neutrophilia, and monocytosis but these changes were found with equal frequency in control patients. Although haematological abnormalities are fairly common in children with TB, in a developing country these abnormalities also occur frequently in children with other non-tuberculosis respiratory infections. An FBC has no diagnostic predictive value when investigating a child for TB.

Child, Preschool↗
PubMed2005

Kawasaki disease presenting with hemorrhagic pleural effusion.

Hemorrhagic pleural effusion associated with Kawasaki disease KD is very rare and has not been reported in the medical literature. We describe a 5-year-old male with incomplete (atypical) KD who presented with fever and severe respiratory distress due to bilateral pleural effusions. Other features included unilateral cervical lymphadenopathy, swelling of the hands and feet followed by periungual desquamation, elevated erythrocyte sedimentation rate, thrombocytosis and sterile pyuria. Pleural fluid analysis revealed a hemorrhagic exudative effusion. Therapy with high-dose i.v. immunoglobulin resulted in dramatic clinical improvement and resolution of pleural effusion. An echocardiogram obtained at presentation and at 6 weeks of illness was normal. Pediatricians should be aware that hemorrhagic pleural effusion can be a presenting manifestation of KD.

Child, Preschool↗
PubMed1995

A phase II trial of zeniplatin in metastatic melanoma.

A third-generation platinum analogue, zeniplatin, was administered at a dose of 145 mg/m2 intravenously over 60-90 minutes every 21 days as the initial chemotherapy to 21 patients with metastatic melanoma. Prehydration and mannitol diuresis was introduced after the first 7 patients. There were 17 males and 4 females. The median age was 52 (range: 29-81). ECOG performance status was 0 in 10 patients, 1 in 8 patients and 2 in 3 patients. Major disease sites were lymph nodes, skin, lung, liver, and bone. Patients received a median of 2 cycles (range: 1-7). Two patients achieved partial responses. One with nodal disease progressed after 166 days and the other with buccal mucosal disease after 142 days. A third patient showed partial regression of nodal disease but developed cerebral metastases. Gastrointestinal toxicity included WHO grade 3 vomiting in 8 patients and nausea in 2. Antiemetics were used, but ondansetron was not available. WHO grade 3 hematologic toxicities included neutropenia in 8 patients and anemia and thrombocytopenia in 1 patient. Thrombocytosis was seen in 35% of courses. Dosage reduction was required in 15% of courses and escalation in 5% of courses. Three patients developed phlebitis related to the infusion. One patient developed a reversible rise in serum creatinine, but, unlike other studies, no severe nephrotoxicity was reported. Zeniplatin demonstrated only modest activity in melanoma with significant gastrointestinal and hematologic toxicity.

Adult↗
PubMed1988

Hypertension complicating essential thrombocythemia.

Essential thrombocythemia (ET) is a myeloproliferative disorder characterized by marked thrombocytosis with associated hemorrhagic, thrombotic and embolic complications caused by platelet dysfunction. In this report we describe two cases of ET and moderate to severe hypertension associated with renal artery stenosis and renal microvascular lesions. In both cases treatment of the hematologic disorder resulted in reduction of blood pressure and improvement of clinical symptoms.

Adult↗
PubMed1992

Clinical aspects of chronic myeloproliferative diseases.

The nonleukemic chronic myeloproliferative disorders, polycythemia vera, essential thrombocytosis, and myeloid metaplasia with myelofibrosis, are clonal disorders with similar but distinct clinical and laboratory findings. This review will discuss the diagnostic criteria for each disease, the variable clinical picture, and the therapeutic modalities, actual and theoretical.

Chronic Disease↗
PubMed1993

Case report: cerebral venous thrombosis as a manifestation of acute ulcerative colitis.

Ulcerative colitis is associated with a number of extraintestinal complications, including the infrequent occurrence of thromboembolic disease. Cerebral venous thrombosis is extremely rare, with only 15 cases reported in the literature. The outcome in this group of patients is poor; the result in more than 80% of the cases is permanent neurologic sequelae or death. The precise mechanisms involved in thrombogenesis remain unclear. A hypercoagulable state may occur in ulcerative colitis because of well-documented associations of thrombocytosis, elevated factors V and VIII and fibrinogen, and decreased antithrombin III. Treatment regimens for cerebral venous thrombosis remain controversial and include anticoagulation with heparin, surgical thrombectomy, and systemic and local infusion of fibrinolytic regimens. A conservative approach with antiedemic agents, anticonvulsants, antiplatelet therapy, and acetazolamide also may be beneficial and offers a substantially reduced hemorrhagic potential. A case of primary superior sagittal sinus thrombosis associated with active ulcerative colitis treated by the latter method is reported. The patient's neurologic recovery was complete, with recanalization of the thrombosed venous sinus being demonstrated by magnetic resonance imaging studies.

Adult↗
PubMed2005

Diagnostic evaluation of patients with a high suspicion of malignancy: comorbidities and clinical predictors of cancer.

BACKGROUND: The diagnosis of cancer is based on the demonstration of malignant cells obtained via biopsy or needle aspiration. For some patients, diagnostic options may be limited either because of tumor location, underlying comorbid conditions, or lack of access to care. METHODS: 275 of 282 consecutive patients presenting to the University of Texas M.D. Anderson Cancer Center with a suspicion of cancer between April 1, 2000 and January 23, 2003 were evaluated retrospectively. We analyzed differences in means of diagnosis, complication rates, clinical characteristics, and comorbid medical conditions between patients with and without a cancer diagnosis. Logistic regression analysis was used to determine the independent predictors of a diagnosis of cancer. RESULTS: 179 (65%) patients had a cancer diagnosis. Endoscopic ultrasonography with fine needle aspiration (EUS/FNA) and image-guided percutaneous biopsy (IGPB) were the most commonly used diagnostic techniques. Complications occurred in 6% of all cases. Independent predictors of a cancer diagnosis included age of 50 years or older, jaundice, weight loss, percentage of monocytes greater than 7, and platelet count greater than 440x10/L; the ROC statistic was 0.796 (CI, 0.738-0.854; P<0.001). Controlling for age, there was no difference in comorbidity between patients with and without a cancer diagnosis. CONCLUSIONS: EUS/FNA and IGPB play an important role in the diagnosis of certain types of malignancy and are associated with a low risk for complications. Advanced age, prior history of malignancy, weight loss, abnormally high percentage of monocytes, and thrombocytosis may be predictive of a cancer diagnosis in patients with suspected malignancy. Comorbid medical conditions are common among patients and occur at rates similar to the general population. Further study is necessary to determine organ-specific predictors of malignancy and to better understand the relationship between cancer and coexisting medical conditions.

Aged↗
PubMed2006

Pseudohyperkalemia in patients with increased cellular components of blood.

OBJECTIVE: We performed a study to investigate the difference between serum and plasma potassium concentration in patients with increase in one or more of the cellular components of blood. DESIGN AND METHODS: This study was performed in two phases. During the first phase, we performed a cross-sectional comparison of the difference between serum and plasma potassium concentration (Dk) in 341 patients with the various clinical conditions where pseudohyperkalemia has been described, as well as with secondary or spurious erythrocytosis and in 30 normal controls. A cut-off value of Dk discriminating polycythemia vera from other erythrocytoses was estimated. In the second phase we studied the significance of this cut-off value as predictor of polycythemia vera in 90 naive patients who were referred with an elevated hematocrit. RESULTS: Dk was significantly increased in the groups with platelet, erythrocyte or with a mixed type disorder compared to the controls (P < 0.01). Among these groups, Dk was significantly increased in the groups with thrombocytosis and mixed type disorder, compared to the group with erythrocytosis (both P < 0.01). A cut-off value of Dk discriminating polycythemia vera from other erythrocytoses was estimated (0.70 mmol/L). Dk (> or = 0.70 mmol/L), platelet and white blood cell count were identified as significant independent predictors of polycythemia vera. CONCLUSIONS: The Dk is increased in patients with erythrocytoses, thrombocytoses or both. This phenomenon is more profound in patients with a mixed type disorder, such as polycythemia vera patients, compared to those with erythrocytoses alone.

Blood Cell Count↗
PubMed1993

Primary angiosarcoma of the spleen. A clinicopathologic study of 40 cases.

Forty primary splenic angiosarcomas occurring in 21 men and 19 women, 19-84 years old (median 59 years) are reported. Patients presented with splenomegaly (35 of 38, 92%), abdominal pain (33 of 40, 83%), and systemic symptoms such as fatigue (2 of 40, 5%), fever (4 of 40, 10%), and/or weight loss (16 of 40, 40%). Five (13%) experienced splenic rupture associated with hemoperitoneum. Abnormal laboratory findings included cytopenia (31 of 34, 91%), leukocytosis (8 of 21, 38%), and thrombocytosis (1/39, 3%). Most spleens weighed 500-1,000 g (mean, 1,180 g). The cut splenic surfaces showed multiple hemorrhagic nodules that were frequently associated with infarction, although some had a diffuse pattern of involvement. Microscopically, there were a variety of histologic patterns displayed by the vasoformative component. A honeycomb or sponge-like pattern was common in some, whereas others simulated a cavernous hemangioma or normal splenic sinuses (pseudosinusoidal pattern). Papillary endothelial tufts and solid proliferations of spindled to round to epithelioid cells were also seen. Factor VIII-related antigen was detected in 19 of 23 cases, BMA-120 in 18 of 23, UEA-1 receptor in 18 of 23, and vimentin in 23 of 23 as well as CD68 antigen in 1 of 23 cases. S-100 protein and cytokeratin were not found in any of the 23 cases studied. Metastases in 22 of 32 patients (69%) were to the liver (13 patients), bone or bone marrow (7 patients), lymph nodes (1 patient), and brain (1 patient). Three patients had concomitant malignancies and one had a prior history of a mixed B-cell lymphoma 5 years previously that had been treated with chemotherapy. Follow-up in 38 patients revealed that 30 (79%) are dead at a median interval of 6 months (range 0-48 months) and 8 are alive 5-21 months after diagnosis. These findings indicate that splenic angiosarcoma is an aggressive neoplasm with a high metastatic rate and an abysmal prognosis. Recognition of the wide range of histologic patterns is of diagnostic value but no apparent prognostic significance.

Adult↗
PubMed1995

Extrapulmonary inflammatory myofibroblastic tumor (inflammatory pseudotumor). A clinicopathologic and immunohistochemical study of 84 cases.

Inflammatory myofibroblastic tumor (IMT) or inflammatory pseudotumor is a spindle cell proliferation of disputed nosology, with a distinctive fibroinflammatory and even pseudosarcomatous appearance. Although the lung is the best known and most common site, inflammatory myofibroblastic tumor occurs in diverse extrapulmonary locations. We report our experience with 84 cases occurring in the soft tissues and viscera of 48 female patients and 36 male patients between the ages of 3 months and 46 years (mean, 9.7 years; median, 9 years). A mass, fever, weight loss, pain, and site-specific symptoms were the presenting complaints. Laboratory abnormalities included anemia, thrombocytosis, polyclonal hypergammaglobulinemia, and elevated erythrocyte sedimentation rate. Sites of involvement included abdomen, retroperitoneum, or pelvis (61 cases); head and neck, including upper respiratory tract (12 cases); trunk (8 cases); and extremities (3 cases). The lesions ranged in size from 1 to 17 cm (mean, 6.4; median, 6.0). Excision was performed in 69 cases. Eight had biopsy only. Five patients received chemotherapy or radiation in addition to undergoing biopsy or resection as initial treatment. Sixteen patients had multinodular masses involving one region. Clinical follow-up in 53 cases revealed that 44 patients were alive with no evidence of disease, four were alive with IMT, and five were dead. Thirteen patients had one or more recurrences at intervals of 1-24 months (mean, 6 months; median, 10 months). No distant metastases were documented. The five patients who died had complications either due to the location of the lesion (heart, peritoneum, retroperitoneum, or mesentery) or related to treatment (lymphoproliferative disorder following hepatic transplantation; sepsis following wound infection). The abdominal masses were the largest. All tumors were firm and white with infiltrative borders and focal myxoid change. Three basic histologic patterns were recognized: (a) myxoid, vascular, and inflammatory areas resembling nodular fasciitis; (b) compact spindle cells with intermingled inflammatory cells (lymphocytes, plasma cells, and eosinophils) resembling fibrous histiocytoma; and (c) dense plate-like collagen resembling a desmoid or scar. Immunohistochemistry demonstrated positivity for vimentin, muscle-specific actin, smooth muscle actin, and cytokeratin consistent with myofibroblasts. Based on this series, inflammatory myofibroblastic tumor is a benign, nonmetastasizing proliferation of myofibroblasts with a potential for recurrence and persistent local growth, similar in some respects to the fibromatoses.

Adolescent↗
PubMed1991

Bilateral lower limb salvage with free flaps in a patient with sickle cell ulcers.

A patient with long-standing bilateral circumferential lower extremity sickle cell ulcerations refractory to conservative management was successfully treated with bilateral free latissimus muscle transfers. This report confirms the value of free tissue transfer in the treatment of these difficult skin ulcerations. Exchange transfusions that brought the SS hemoglobin below 30% were crucial to the prevention of sickling in the microcirculation of the flap during its obligate period of ischemia. Furthermore, they protected the flap during a period of ischemia that exceeded 4 hours following a postoperative arterial thrombosis. In the presence of severe thrombocytosis associated with sickle cell disease, prophylactic treatment with aspirin may be of significant value.

Adult↗
PubMed1975

Myeloproliferative disorders.

Forty-three operative procedures were performed on a population of 250 patients with myeloproliferative disorders, including polycythemia vera, myeloid metaplasia (MM) and chronic myelogenous leukemia (CML). The overall operative mortality was approximately 7% and the incidence of excessive bleeding which could be related to coagulopathy was 5%. Twenty-one patients with MM or CML underwent splenectomy for palliation of symptoms related to the enlarged spleen or hematologic problems. Eighty-four percent of the latter group were improved. Adverse hematologic effects which could be attributed to splenectomy in these patients were confined to two patients who developed marked thrombocytosis. Among the 23 patients with MM, 9 had portal hypertension. Three underwent portacaval shunt and one a splenorenal shunt for bleeding varices. One of the patients died of hepatic necrosis. Estimated hepatic blood flow determinations (EHBF) in 4 patients with portal hypertension demonstrated a marked absolute increase and an increase in the ratio of EHBF/Cardiac Index. Absence of any evidence of intrahepatic or extrahepatic obstruction in these patients and the demonstration that splenectomy relieved portal hypertension defined at surgery in 4 patients, suggests that augmented adhepatic flow contributes to portal hypertension in some cases. The review leads to the conclusions that: 1) Operative procedures in prepared patients with myeloproliferative disorders are not associated with prohibitive mortality and morbidity rates. 2) Splenectomy is indicated for patients with increasing transfusion requirements and symptomatic splenomegaly or hypersplenism and should be performed early in the course of disease. 3) When associated portal hypertension and bleeding varices are present, hemodynamic studies should be carried out to define if splenectomy alone, or a portal systemic decompressive procedure is indicated.

Adult↗
PubMed1977

Antithrombin III deficiency causing postsplenectomy mesenteric venous thrombosis coincident with thrombocytopenia.

The most commonly recognized cause of mesenteric venous thrombosis following splenectomy is hypercoagulation secondary to reactive thrombocytosis. A case is reviewed in which hypercoagulation followed splenectomy for idiopathic thrombocytopenic purpura (ITP) in spite of persistent thrombocytopenia. Episodic mesenteric venous occlusion occurred due to antithrombin III deficiency. This hypercoagulable state may be the cause of primary acute mesenteric venous occlusive disease. Symptoms and signs suggesting thrombosis in the portal circulation demand immediate coagulation studies since even in the thrombocytopenic patient thrombotic proglems can occur. Surgical intervention is the treatment of choice for segmental small bowel ischemia; warfarin therapy is indicated when there is evidence of antithrombin III deficiency.

Adult↗
PubMed1994

Autologous splenic transplantation for splenic trauma.

OBJECTIVE: The authors reviewed the experimental evidence, surgical technique, complications, and results of clinical trials evaluating the role of autologous splenic transplantation for splenic trauma. SUMMARY BACKGROUND DATA: Splenorrhaphy and nonoperative management of splenic injuries have now become routine aspects in the management of splenic trauma. Unfortunately, not all splenic injuries are readily amenable to conventional spleen-conserving approaches. Heterotopic splenic autotransplantation has been advocated for patients with severe grade IV and V injuries that would otherwise mandate splenectomy. For this subset of patients, splenic salvage by autotransplantation would theoretically preserve the critical role the spleen plays in the host's defense against infection. METHODS: The relevant literature relating to experimental or clinical aspects of splenic autotransplantation was identified and reviewed. Data are presented on the experimental evaluation of autogenous splenic transplantation, methods and complications of autotransplantation, choice of anatomic site and autograft size, and results of clinical trials in humans. RESULTS: The most commonly used technique of autotransplantation in humans involves implanting tissue homogenates or sections of splenic parenchyma into pouches created in the gastrocolic omentum. Most authors have observed evidence of splenic function with normalization of postsplenectomy thrombocytosis, immunoglobulin M levels, and peripheral blood smears. Some degree of immune function of transplanted grafts has been demonstrated with in vivo assays, but the full extent of immunoprotection provided by human splenic autotransplants is currently unknown. CONCLUSIONS: Multiple human and animal studies have established that splenic autotransplantation is a relatively safe and easily performed procedure that results in the return of some hematologic and immunologic parameters to baseline levels. Some aspects of reticuloendothelial function are also preserved. Whether this translates into a real reduction in the morbidity or mortality rates from overwhelming bacterial infection is unknown and requires further investigation.

Animals↗
PubMed1992

The fibrinolytic system and proteins C and S in treated polycythaemia rubra vera.

This study was designed to assess whether factors other than high haemoglobin, thrombocytosis and abnormal platelet function predispose to thrombosis in polycythaemia rubra vera (PRV). Components of the fibrinolytic system and concentrations of the naturally occurring anticoagulants were measured in patients and controls in the resting state; the fibrinolytic capacity was reassessed after venous occlusion. The results were related to presence or absence of a history of thromboembolism. Under resting conditions, patients with PRV had reduced plasminogen activator inhibitor antigen levels and higher fibrin plate lysis area and tissue plasminogen activator activity. Protein C, protein S and factor V levels were reduced. Those patients with a history of thromboembolism had decreased tissue plasminogen activator activity after venous occlusion compared to those who had not experienced a thrombosis. We conclude that reduced fibrinolytic capacity may predispose to thrombosis in PRV. Despite treatment to normalize haemoglobin levels, the patients have persistent activation of their fibrinolytic systems. This, and reduced levels of proteins C and S, may be secondary to a chronic, clinically occult, disseminated intravascular coagulation.

Adult↗