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PubMedDate not supplied

Hematological manifestations of congenital cytomegalovirus infection.

Nine infants suffering from congenital cytomegalovirus infection were studied for clinical features with special reference to hematological manifestations. The mean age of appearance of first clinical symptom of disease was 14 days and mean age at presentation was 58 days. Significant pallor, thrombocytopenia and evidence of hemolysis were present in 8 (89%), 4 (44%) and 4 (44%) patients respectively out of three bone marrow examination performed, paucity of erythroid and megakaryocytic cells were seen in two. The other clinical features included hepatomegaly in a (100%) splenomegaly in 6 (66%) each, petachial rash in 5 (55%), Hepatitis in 4 (44%) optic atrophy and corneal opacities in one patient each. Two patients died. Remaining patients showed symptomatic improvement without specific therapy.

Cytomegalovirus Infections↗
PubMed2006

Hemophagocytic syndrome associated with visceral leishmaniasis.

The present paper reports a case of 6-year-old male child, suffering from pallor, fever and hepatosplenomegaly. A clinical diagnosis of enteric fever with a second possibility of malaria was considered. Laboratory findings included bicytopenia, hyperbilirubinemia and raised liver enzymes. Bone marrow examination revealed active hemophagocytosis. On extensive search few amastigote forms of Leishmania donovani were seen. Patient was negative for other viral, bacterial and malaria infections. The final diagnosis of hemophagocytic syndrome associated with visceral leishmaniasis was made. There was response of anti-Leishmanial treatment with improvement in clinical condition.

Animals↗
PubMed2005

Abdominal tuberculosis with autoimmune hemolytic anemia.

An eight-year-old male child presented with progressive distension of abdomen, fever, pallor and jaundice with a history of tubercular contact. Investigations were suggestive of abdominal tuberculosis with autoimmune hemolytic anemia. The child responded well to a course of oral steroids with antitubercular treatment. A literature search did not reveal any previous case report of an association between tuberculosis and autoimmune hemolytic anemia.

Anemia, Hemolytic, Autoimmune↗
PubMed2002

Adrenal cortical and medullar hyperplasia--a retrospective analysis of 6 cases.

The features of the symptoms, laboratory tests and pathological characteristics of adrenal cortical and medullary hyperplasia were studied. In 6 cases of hypercatecholaminenia, plasma norepinephrine (NE), epinephrine (E), catecholamine (CA) and 24-h urinary vanillylmandelic acid (VMA), 17-hydroxycorticosteroid (OHCS) and 17-ketosteroid (KS) were determined. Adrenal glands were examined by CT scan and 131I-MIBG imaging. Pathological examination was performed after operation. The results showed that in 6 cases of hypercatecholaminenia (3 men and 3 women) aged from 34-50 years, the clinical features were just like "pheochromocytoma", for example, episodic headache, perspiration, palpitation, pallor, apprehension, nausea, tremor, anxiety and so on. Plasma levels of CA, NE and E were elevated in all 6 cases. 24-h urinary samples obtained at the onset revealed elevated VMA in 1 case. 24-h urinary cortisol was obviously elevated in all 6 cases. 24-h urinary 17-OHCS, 17-KS was normal. B-type ultrasound, CT, MRI and 131I-MIBG revealed 9 lateral adrenal gland diffuse or nodular enlargement in 6 cases. Pathologic examination showed adrenal cortical and medullary hyperplasia. Clinically, adrenal cortical and medullary hyperplasia resembled "pheochromocytoma". The most significant feature of this disease was both elevated plasma CA and 24-h urinary cortisol obviously. Pathologic examination showed adrenal cortex nodular hyperplasia and medullar diffuse or limit hyperplasia. Whether it is an independent disease or symptoms of the other disease has not final conclusion up till now.

Adrenal Cortex↗
PubMed1986

Pharmacophysiology of atopic dermatitis.

Atopic dermatitis is clearly characterized by altered cutaneous physiologic responses. There is a tendency to acral vasoconstriction. Rubbing causes skin pallor and white dermographism. Vascular instability is demonstrated by responses to cholinergic agents, histamine, and nicotinates. Psychophysiologic studies demonstrate exaggerated vasodilator responses to emotional stress with consequent pruritus and scratching. The itch threshold is low, duration is prolonged, and nighttime scratching movements may be frequent or almost continuous. Regardless of the inciting trigger factors, the scratching causes the damage and the severe dermatitis. Thermal as well as emotional stimuli to sweating cause severe itching in AD, yet the concept of a miliaria-type, poral occlusion mechanism remains unproven. Some studies suggest actually increased sweating along with erythema and pruritus during acute flares of AD. The concept of sweat-borne allergens causing skin reactions during sweating is interesting but has never been proven. Studies of sweat responses to pharmacologic agents have produced conflicting data, and attempts to link these responses to Szentivanyi's beta-adrenergic blockade theory are not convincing. The numerous variables of climate, season, sex, age, and habitus affect sweating greatly. Future studies must carefully control for each of these factors before pharmacologically induced sweat responses can be interpreted clearly. A number of lines of evidence suggest involvement of histamine and other mediators in the evolution of erythema, pruritus, and scratching in AD. Flares of the condition have been reproducibly evoked by only two incitants: experimental emotional stress interviews and specific food challenge in selected sensitive individuals. In the latter, increased plasma histamine has been demonstrated, presumably generated by antigen/IgE stimulated degranulation of mast cells in the gut and/or skin. The demonstrated increased histamine releasability of basophils from atopic individuals may be the result of defective cellular regulatory mechanisms. Recent studies have demonstrated increased cyclic AMP-phosphodiesterase activity in leukocytes from atopic individuals. The resultant decreased intracellular cyclic AMP removes an inhibitory factor, which in turn causes net cellular hyperresponsiveness. This effect has been shown to account, at least in part, for increased histamine release from leukocytes of patients with AD. These and other studies focused upon cell functional regulation are providing better understanding of basic biochemical abnormalities and may lead to improved diagnostic and therapeutic approaches in managing atopic disease.

Biofeedback, Psychology↗
PubMed2001

Combined pre- and post-surgical bupivacaine wound infiltrations decrease opioid requirements after knee ligament reconstruction.

PURPOSE: To test the efficacy of a combination of selective pre- and post-surgical local anesthetic infiltrations of the knee, compared with standard intra-articular injection at the end of surgery alone, to reduce postoperative opioid requirements following arthroscopic cruciate ligament reconstruction (ACLR). METHODS: In a double-blind, randomized, controlled trial, we studied 23 patients (ASA I or II) scheduled for elective ACLR under general anesthesia. The treatment group (n = 12) received infiltrations with bupivacaine 0.25% with epinephrine 1:200,000 presurgically (10 ml into the portals, 10 ml at the medial tibial incision site, 10 ml at the lateral femoral incision site, and 10 ml intra-articularly) and postsurgically (5 ml at the medial tibial incision and 10 ml at the lateral femoral incision). The control group (n = 11) received infiltrations with saline 0.9% in the same manner. All patients received a standard intra-articular local anesthetic instillation of the knee (25 ml of bupivacaine 0.25% with epinephrine 1:200,000) at the completion of surgery. RESULTS: Postoperative opioid requirements were lower in the treatment group (5.8 +/- 2.9 mg morphine equivalent) than in the control group ( 13.7 +/- 5.8 mg; P = 0.008). Treatment patients were ready for discharge approximately 30 min earlier than control patients (P = 0.046). There were no adverse events in the treatment group. In the control group, 2/11 patients vomited and a third experienced transient postoperative diaphoresis, dizziness and pallor. CONCLUSION: We conclude that a combination of selective pre- and post-surgical wound infiltration with bupivacaine 0.25% provides superior analgesia compared with a standard post-surgical intra-articular injection alone.

Adult↗
PubMed2000

Compartment syndrome following intravenous regional anesthesia.

PURPOSE: To present two cases of upper extremity compartment syndrome following intravenous regional anesthesia. CLINICAL FEATURES: Case 1: A 57-yr-old man presented for surgical release of a left-hand Dupuytren's contracture. The procedure was performed under iv regional anesthesia with 360 mg lidocaine and sedation with 150 microg fentanyl and 1.5 mg midazolam. Tourniquet time was 107 min at a pressure of 260 mmHg using three different tourniquet sites. Within minutes of tourniquet release, increased forearm muscle tension, hand anesthesia, pallor, and limited motor function developed. Serum CK and myoglobin levels rose. Myoglobinuria was present. Several fasciotomies and aggressive fluid therapy were performed. Patient made almost full recovery. Case 2: A 73-yr-old woman with controlled hypertension had Dupuytren fasciotomy of her right hand under iv regional anesthesia with 200 mg lidocaine and sedation using 75 microg fentanyl and 1.5 mg midazolam. Tourniquet time was 64 min at a pressure of 250 mmHg using three different tourniquet sites. The patient complained of pain at the iv site during injection of local anesthetic, third tourniquet inflation and after deflation of tourniquet. Thirty minutes after arrival in PACU, her fingers were bluish. She complained of pain and swelling of the forearm. Under general anesthesia, fasciectomy was performed. Myoglobin and CPK levels rose. CPK MB was high but troponin was negative. Three days later she developed pulmonary embolism. She was heparinized and subsequently discharged home. She recovered completely. CONCLUSION: Compartment syndrome may have a rapid and severe onset. Etiology of our cases is still not established. We postulate that increased tissue pressure may be the cause. The anesthesiologists must be aware of compartment syndrome during regional anesthesia.

Aged↗
PubMed2003

Bilateral adrenal non-Hodgkin lymphoma type B.

The adrenal localization of a primary non-Hodgkin lymphoma (NHL) is rare. We report a case of a 66-yr-old woman who had severe asthenia, diffuse skin hitching and abdominal pain. The physical examination revealed poor general conditions, irritability, pallor, dehydration and diffuse skin scratching lesions. The abdomen was painful at left hypochondrium and the Giordano's maneuvre was positive on both sides. A peripheral blood smear showed the presence of big rare lymphocytes with dyshomogeneous chromatin and granulated and hyperbasophil cytoplasm. She underwent abdominal ultrasonography which showed the presence of a hypoechogenic ovoidal mass (major diameter 8.4 cm) within the splenorenal left region. The presence of the suprarenal mass was confirmed by computed tomography (CT) scan which showed an enlarged left adrenal gland (8.1 x 6.2 cm) of solid structure. The right adrenal gland was also enlarged and of round shape (4.5 cm). CT scan-guided fine needle aspiration biopsy was then carried out on the left adrenal mass. It revealed the presence of NHL type B with large cells and modest T-lymphocyte reactive component. The patient was treated with three cycles of cyclophosphamide, adriamycin, vincristine and prednisolone. At the end of the third cycle, there was a transient clinical improvement and the hitching disappeared, but the patient worsened rapidly and she died few weeks later.

Adrenal Gland Neoplasms↗
PubMed2004

Late effects of post-high-dose-rate brachytherapy for oropharyngeal carcinoma: are they severer than post-low-dose-rate?

BACKGROUND: . Late effects by high-dose-rate (HDR) brachytherapy have been believed severer than low-dose-rate (LDR) provided tumor control was constant. Local control of oropharyngeal carcinoma with HDR at Osaka Medical Center was comparable to LDR series from Centre Alexis Vautrin (82%, 79.5%, respectively). To assess the feasibility of HDR brachytherapy, the late effects were compared. PATIENTS AND METHODS: . The data of 29 HDR and 24 LDR patients (median follow-up of 27 and 29.5 months, respectively; p = 0.89) were collected. The HDR schedule was 21 Gy/3.5 fractions/2 days following 46 Gy/23 fractions external beam, while 25 Gy/3 days following 50 Gy/25 fractions external beam was for LDR. Late changes were evaluated using RTOG/EORTC late morbidity scoring scheme. For subclinical late changes, mucosa chapter of Dische score was modified for brachytherapy. Scores were discussed through photos and were agreed on by authors. Late sequelae were estimated, by reviewing charts, concerning frequency, severity, and duration of mucosal damages (erosion and ulcer). RESULTS: . Late changes were of no difference (p = 0.12 for EORTC/RTOG, and p = 0.45, 0.47, 1.00, 0.12, 0.16, 0.95, 0.27, 0.21 for erythema, ulceration, edema, thinning, pallor, telangiectasia, mobility impairment of tongue/faucial pillars, respectively, of the modified Dische score). Late sequelae showed no differences (p = 0.90, 0.12, 0.40 for frequency, severity, duration, respectively, of mucosal damages). CONCLUSION: . The late effects by HDR were not severer than by LDR. HDR oropharyngeal brachytherapy is as safe as LDR.

Adult↗
PubMed1994

[Fulminating E. coli sepsis in Fournier's gangrene].

Fournier's gangrene is a necrotising soft-tissue infection of the scrotum and perineal region caused by gram-negative and gram-positive Enterobacteriaceae. The disease is characterised by its unique appearance, its speed of onset, and its high mortality. CASE REPORT. A 26-year-old male presented to the emergency room complaining of a painful, tremendously swollen scrotum and penis (Fig. 1) that had developed within the past 24 h. Later, slurred speech, pallor, and hypotension were recognised, leading to the patient's admission to the intensive care unit. Suspecting a severe internal haemorrhage, vigorous volume therapy was started using crystalloids and colloids until blood and fresh frozen plasma were available. One hour later, septic shock was presumed and therapy augmented by IV antibiotics, tracheal intubation, and mechanical ventilation. Despite all efforts, the patients condition deteriorated rapidly and he died a few hours later due to multiple organ failure in septic shock. Postmortem, a perforated external hemorrhoidal node was found to be the primary focus of sepsis. Microbiologic cultures revealed Escherichia coli in blood and tissue samples. DISCUSSION. Fournier's gangrene is a rare disease; nevertheless, its clinical picture has to be recognised immediately in order to provide appropriate treatment in time. It occurs predominantly in males after minor trauma, colorectal or urological disease, and perineal or abdominal surgery. Fournier's gangrene usually begins with itching and pain in the scrotal region followed by swelling and dark-blueish discolouration of the scrotum and penis, occasionally including the lower abdominal wall. Fever and chills are usually present. The illness progresses to severe prostation and septic shock with a mortality of 20%-50%. Tissue cultures mostly reveal E. coli, gram-positive enterococci, Pseudomonas, Proteus, and various anaerobes. The treatment should include immediate radical surgical debridement, i.v. administration of broad-spectrum antibiotics, and cardiopulmonary support. CONCLUSION. The dramatic course of Fournier's gangrene requires early recognition, extensive surgical debridement, as well as intensive care treatment in order to prevent irreversible septic shock.

Adult↗
PubMed2002

[The hypothenar hammer syndrome].

The hypothenar hammer syndrome (HHS) is a rare form of secondary Raynaud's phenomenon in workers who frequently use the ulnar side of the palm as a hammer. Clinically the patient with HHS shows neurologic symptoms such as paresthesia, numbness and pain and signs of vascular insufficiency such as coldness, pallor, discoloration and blanching of the affected ulnar sided fingers. The diagnosis is verified by angiography, showing thrombosis or aneurysm of the distal ulnar artery. We report on the treatment and outcome of 5 patients with HHS. All patients were painfree after treatment. But with the ongoing working habit of using the hand as a hammer there is always the risk of a recurrence. Therefore the job environment has to be changed considerably and the most effort should go into prevention and prophylaxis. If the change of the working habit is not possible, the job is clearly at stake.

Accidents, Occupational↗
PubMed2003

Absence of short-term vestibular modulation of muscle sympathetic outflow, assessed by brief galvanic vestibular stimulation in awake human subjects.

There is evidence in experimental animals for a potent vestibulosympathetic reflex, but its existence in humans is controversial. Static head-down neck flexion and off-vertical axis rotation have been shown to increase muscle sympathetic nerve activity (MSNA), but not skin sympathetic nerve activity (SSNA), whereas horizontal linear acceleration decreases MSNA in humans. However, both forms of stimuli also activate other receptors. To examine the effects of a pure vestibular stimulus on MSNA and SSNA, and its potential interaction with the baroreceptors, we used galvanic vestibular stimulation (GVS) in 12 healthy seated subjects. MSNA was recorded in ten subjects via a percutaneous microelectrode in the peroneal nerve; ECG, blood pressure, respiration, skin blood flow and sweating were also recorded. GVS (2 mA, 1 s pulse) was delivered via surface electrodes over the mastoid processes at unexpected times, triggered from the R-wave with a delay of 0, 200, 400 or 600 ms. In addition to causing robust postural illusions, GVS caused cutaneous vasoconstriction and sweat release in all subjects (due to a short-latency increase in SSNA, three subjects), but no significant change in MSNA. The failure of GVS to elicit a change in muscle sympathetic nerve activity, as documented by averaging, suggests that the vestibular system is not engaged in short-term modulation of muscle sympathetic activity. Conversely, phasic vestibular inputs do excite cutaneous sympathetic neurones, consistent with the observation that motion sickness is accompanied by pallor and sweating.

Action Potentials↗
PubMed2000

Imaging and radiological-pathological correlation in histologically proven cases of focal cortical dysplasia and other glial and neuronoglial malformative lesions in adults.

Focal cortical dysplasia (FCD) is a pathological entity first described in 1971. Other more subtle cortical malformations found in patients with epilepsy include microdysgenesis (MD), and glioneuronal hamartias. Although these glial and neuronoglial malformations have distinct histological features, there is terminological confusion in the radiological literature. Few cases have been reported in adults with both imaging and histology. We address these issues, giving a radiological-pathological correlation of histologically proven cortical malformations in adults. We describe clinical, radiological and histological features of 12 cases (five FCD, five MD with glioneuronal hamartias, and two hamartomas), unassociated with other conditions, and discuss them in the light of the literature. FCD is usually seen on MRI as cortical thickening, with or without signal change, which may extend into the adjacent white matter. On histology, abnormal neurons and/or glial cells, blurring of the grey-white matter interface, myelin pallor, demyelination, and gliosis may be found. Glioneuronal hamartias and hamartomas usually appear as complex masses on MRI. FCD and hamartias may be associated, and a combination of imaging findings may be seen on MRI. Atrophy of the ipsilateral hippocampus may be present on MRI in patients with hamartias, and minor cell loss on histology, but not definitive hippocampal sclerosis. Although the imaging findings of cortical malformations are protean, some characteristic MRI features, with histological correlates, may be found. The relevance of most of these observations remains unclear.

Adult↗
PubMedDate not supplied

Association of clinical characteristics of unexplained syncope with the outcome of head-up tilt tests in children.

The aim of this study was to clarify the association of clinical characteristics of unexplained syncope with the outcome of the head-up tilt test (HUT) in children. A total of 47 patients with unexplained syncope were classified into two groups according to their outcomes of HUT: the positive response group and the negative response group. We reviewed their clinical data as well as the results of HUT and analyzed them with logistic regression method. The results showed that the incidence of positive responses to HUT was higher in girls than in boys (8/22 vs 10/7, p < 0.05). Compared with fainted children younger than 12 years of age, 12- to 16-year-old adolescents with unexplained syncope had a high positive outcome of HUT (30 vs 72.9%, p < 0.05). Compared with fainted children with negative response of HUT, children with positive response to HUT often had syncope in special circumstances (e.g., prolonged standing, anxiety and fright, and morning exercise), and they often had prodrome, such as pallor, lightheadedness, and nausea (28/30 vs 8/17, p < 0.05). However, the number and duration of syncopal spells did not relate to the positive responses to HUT. The logistic regression analysis showed that three factors significantly influenced the outcome of HUT: predisposing factors of syncope, prodrome of syncope, and age (p < 0.05; OR = 32.9434, 17.7281, and 2.7842, respectively). Hence, if pubertal girls with unexplained syncope had clear predisposing factors and prodromes, they were likely to have positive responses to HUT, and they were likely to be clinically considered as having vasovagal syncope.

Adolescent↗
PubMed1999

Popliteal entrapment syndrome: non-invasive diagnosis and complete recovery after surgery in an 11-year-old boy.

We present an unusual case of popliteal entrapment syndrome type I in an 11-year-old boy. Presenting symptoms were exercise-related pain and pallor in his left lower leg. The diagnosis of popliteal entrapment syndrome was established by acral plethysmography, colour-coded Doppler sonography and MRI. After myotomy of the gastrocnemius muscle the symptoms resolved completely. Post-operative duplex scan showed normal blood flow, even in plantar flexion of the foot.

Arterial Occlusive Diseases↗
PubMed2004

Sickle beta-thalassemia presenting as orbital compression syndrome.

Orbital compression syndrome is caused by disorders of varying etiologies involving the orbit and presents with ocular and extraocular dysfunction. Ocular involvement of sickle cell disease is uncommon. A 17-year-old male presented with low backache and proptosis of both eyes of 5 days duration without past history of pain crisis or transfusion. Examination revealed pallor, icterus, bilateral proptosis, conjuctival chemosis, and symmetrical restriction of ocular movements with preserved visual acuity. He was drowsy with no other focal deficits. The fundus showed macular edema, venous engorgement, and no hemorrhage. His peripheral smear showed presence of sickle cells. Computed tomography (CT) scan of the orbit revealed orbital subperiosteal hematomas. CT head images showed epidural hematoma in the frontal lobe. High-performance liquid chromatography (HPLC) and mutation studies revealed sickle beta-thalassemia in the patient. He was managed with supportive care, transfusions to keep hemoglobin above 10 g/dl, and hydroxyurea. The patient recovered fully and remained well during follow-up of 12 months. Our case was unique for presenting as orbital compression syndrome without any history of vaso-occlusive crisis.

Adolescent↗
PubMed1999

A case of juvenile myelomonocytic leukemia with ocular infiltration.

We present a case of juvenile myelomonocytic leukemia (JMML) with ocular infiltration. A 1-month-old boy presented with myeloid precursors in peripheral blood and a white blood cell count >10x10(9)/l. His peripheral blood monocyte count was >1x10(9)/l, bone marrow blasts were <20%, and no Ph chromosome was identified. The boy also presented with hepatosplenomegaly, pallor, fever, and skin rash. We diagnosed this case as JMML, although hemoglobin F was within the normal range and no spontaneous colony growth was observed from peripheral blood mononuclear cells. Neither Epstein-Barr (EB) virus nor cytomegalovirus was detected by PCR in bone marrow aspirate or peripheral blood. The patient had several lesions into which JMML cells might have infiltrated, including skin, liver, spleen, oral cavity, right lung, sigmoid colon, and both eyes. To our knowledge, this is the first reported case of JMML with ocular involvement. Since infiltration of JMML cells into both eyes causes blindness, further consideration of the timing of bone marrow transplantation (BMT) in JMML is necessary.

Biopsy↗
PubMed2004

A case of familial Mediterranean fever and polyarteritis nodosa complicated by spontaneous perirenal and subcapsular hepatic hemorrhage requiring multiple arterial embolizations.

The association of familial Mediterranean fever (FMF) and polyarteritis nodosa (PAN) has been well established. These patients have been reported to have an overall better prognosis than other PAN patients. Herein we report a patient with FMF and PAN who died of sepsis following a severe course of recurrent bleeding episodes which required multiple embolization attempts. The 39-year-old Turkish male presented with abdominal pain of 1-month duration. He had been diagnosed with FMF at the age of 24. On admission, he had pallor with general ill appearance. Rebound tenderness was obtained in the right upper abdominal quadrant. He had mild anemia, leukocytosis, thrombocytosis, and hypoalbuminemia. On the 2nd day of his admission, he developed hypotension with a rapid decline in hemoglobin level. Abdominal angiography showed multiple aneurysms in the branches of renal arteries, superior mesenteric artery, and hepatic arterial system including left renal infarct, suggesting PAN. He was put on high-dose steroids and oral cyclophosphamide. Despite medical treatment, he developed intense abdominal pain, hypotension, tachycardia, and a rapid fall in hemoglobin on four occasions. Active bleeding sites were embolized in two different angiography sessions. Although the patient experienced no more recurrent bleeding, he died of multiorgan dysfunction syndrome resulting from sepsis 6 weeks after admission. Polyarteritis nodosa associated with FMF may follow a grave course despite immunosuppressive therapy. Arterial embolization should be considered in the presence of bleeding aneurysms in addition to immunosuppressive therapy.

Adult↗