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PubMed1993

Congenital histidine-rich glycoprotein deficiency.

The proband, a 43-year-old woman, suffered from right transverse sinus thrombosis during oral contraceptive treatment. A month after stopping the drug, her plasma activities of antithrombin III, protein C, protein S, heparin cofactor II, plasminogen and plasminogen activator inhibitor were normal, but her plasma histidine-rich glycoprotein (HRG) level was only 21% of the normal level of 109.5 +/- 51.5% (mean +/- 2 SD). The HRG concentrations in her plasma determined on four different occasions over 6 months were similar. She showed no clinical signs of liver insufficiency or sepsis. Low levels of plasma HRG (20% to 35% of normal) were also found in her aunt, uncle and two daughters. These results suggest that congenital HRG deficiency is inheritary in this family.

Adult↗
PubMed2001

De novo production of IgG antinuclear antibodies in a neonate.

We describe the first case in which de novo production of multiple IgG antinuclear antibodies (ANA) occurred in a female neonate of an ANA negative mother. The infant presented at 4 weeks of age with hemorrhagic panencephalitis, diffuse intraparenchymal hemorrhages, and straight sinus thrombosis. She had been vaccinated against hepatitis B at birth. No other cause was found and maternal prenatal care was unremarkable. The infant's screening ANA test by ELISA was positive at 6 weeks with specificity for ssDNA, Sm, and nRNP/Sm. At 8 weeks antibodies to dsDNA and centromere were detected as well. By 8 months, she still had slightly elevated anti-dsDNA, Sm, and nRNP/Sm antibodies. The ANA test by immunofluorescence was abnormal at 8 weeks through 13 weeks with centromere and then homogeneous pattern. Based on similarities with other reported cases, we speculate that hepatitis B vaccination may have been involved in the development of antinuclear antibodies.

Adult↗
PubMed1983

Intracranial complications of acute and chronic infectious ear disease: a problem still with us.

Among 334,884 admissions to the North Carolina Baptist Hospital from 1963 through 1982, 100 patients had central nervous system (CNS) complications of middle ear disease. The complications occurred predominantly in young patients, 85 of the 100 being less than 20 years of age. Meningitis occurred in 76 patients; the acute form was more prevalent (63 cases). The less common nonmeningitic complications included brain abscess (n = 6), effusion (n = 5), lateral sinus thrombosis (n = 5), otitic hydrocephalus (n = 5), and empyema (n = 3). Overall mortality was 10%. One patient with brain abscess died; 9 of the 76 patients with meningitis died (12%), with 4 of those deaths occurring among the 13 patients with chronic meningitis (31%). Because these complications have declined markedly since the advent of antibiotics, many contemporary otolaryngologists have been unexposed to these complications. However, as this series shows, they do still occur, their natural history remains the same, and the resulting mortality is still alarmingly high. A plea is made for otolaryngologists to maintain an awareness of these complications and to work with pediatricians and neurosurgeons for the best team care of patients with CNS complications of middle ear disease.

Adolescent↗
PubMed2001

Classification of venous ischaemia with MRI.

PURPOSE: Venous ischaemia is diagnosed by angiography and estimated with SPECT and PET. But venous ischaemia presents different features due to aetiology, type of onset, time course and collateral circulation. The purpose of this study was to analyse and to classify VI with MRI. METHODS: An analysis of 12 cases of dural arteriovenous fistula (DAVF) with venous ischaemia, 4 cases of sinus thrombosis, and a case of cortical venous thrombosis was performed. Venous ischaemia is classified with MRI as Type 1: no abnormality, Type 2: T2WI showed high signal intensity area and Gd-MRI showed no enhancement, Type 3: T2WI showed high signal intensity area and Gd-MRI showed enhancement, Type 4: venous infarction or haemorrhage. RESULTS: Type 1 was 8 cases. Type 2 was 3 cases and indicated cytotoxic oedema. Type 3 was 2 cases and indicated vasogenic oedema because of the destruction of blood brain barrier. Type 4 was 4 cases. CONCLUSIONS: The classification may be a useful indicator of severity of venous ischaemia and treatment.

Adult↗
PubMed1983

Masked mastoiditis.

With the advent of broad-spectrum antibiotics, the clinical course of middle ear disease has been altered. One result has been the occasional suppression of the presenting signs and symptoms of mastoiditis secondary to acute middle ear disease, causing the clinician to have a false sense of security following apparent resolution of the middle ear infection. The course may be so insidious that the first awareness of the mastoiditis may be following presentation of an intracranial complication such as meningitis, lateral sinus thrombosis, or brain abscess. The authors report 9 patients seen in the past 5 years with masked mastoiditis, ranging in age from 4 months to 43 years. The presenting symptoms were all vague and non-classical; however, intracranial complications of meningitis, facial paralysis, brain abscess, and papilledema were present on admission in 7 patients, and another 2 patients had unsuspected epidural abscess upon surgical exploration. The mastoid radiographs were uniformly positive, while the CT scan was positive in 3/9 patients. All patients recovered following mastoidectomy and intracranial therapy except one patient who expired after brain herniation. The clinician is urged to maintain a high suspicion of masked mastoiditis in the high-risk patient, including newborn, diabetic, elderly, immunosuppressed or debilitated patients.

Adolescent↗
PubMed1986

Clinical implications of orbital cellulitis.

Periorbital and orbital cellulitis are clearly two distinct disorders with different etiologies. Periorbital cellulitis is well-documented as a more common infectious process limited to the eyelids in the preseptal region. In contrast, orbital cellulitis represents a more severe, but less common, infection of the orbit posterior to the septum with or without subperiosteal abscess, orbital abscess, or cavernous sinus thrombosis. A retrospective review of the clinical and laboratory data of 137 cases of orbital and periorbital cellulitis was performed. Periorbital cellulitis was documented in 98 cases (71%), in contrast to orbital cellulitis which was noted in 39 (28%) patients. All patients with orbital cellulitis received intravenous antibiotics, however, surgical therapy was necessary for progression of orbital cellulitis in 49% of patients. The majority of patients underwent incision and drainage of an abscess in the orbit. Complications as a result of orbital cellulitis occurred in nine (23%) of the patients. A distinction is made between the clinical entity of periorbital cellulitis and orbital cellulitis. Separating these clinical entities on the basis of physical examination and radiographic studies is important because the medical and surgical management of these two disease processes is different.

Abscess↗
PubMedDate not supplied

Malignant external otitis: further considerations.

Malignant external otitis is an infection which begins in the external auditory canal. It is uniformly caused by the Gram negative Pseudomonas aeruginosa organism and mainly affects elderly diabetics. It spreads to the soft tissues beneath the temporal bone and, if not properly treated leads to facial nerve palsy, mastoiditis, sepsis, osteomyelitis of the base of the skull, sigmoid sinus thrombosis, multiple cranial nerve palsies and death. Experience with 72 patients in varying stages of the disease is summarized. Stressed are the diagnostic criteria of nonresponsiveness to the usual methods of treatment, continued suppuration, and the continuing reformation of granulation tissue in the floor of the external auditory canal. Medical treatment is recommended with hospitalization and intravenous carbenicillin and gentamicin. Minor surgical debridement is helpful. All patients should be treated medically for as long as improvement continues, reserving surgical intervention only in the event a plateau is reached or symptoms and signs become worse under treatment. With or without a major surgical procedure, it is imperative to continue treatment for at least seven days after apparent cure in order to avoid recurrent disease possibly at a site distant from the canal.

Adult↗
PubMedDate not supplied

Hypercoagulability in otologic patients.

INTRODUCTION: Three patients with otologic disorders developed complications related to hypercoagulability. This report was prepared to increase the awareness among otolaryngologists of the diagnosis and management of hypercoagulable states, and to encourage expert consultation when indicated. MATERIALS AND METHODS: One patient with dural venous sinus thrombosis complicating an otitis media, one patient with a large glomus jugulare tumor, and one patient with a sudden sensorineural hearing loss were treated by the otolaryngology service. Suspicious thromboembolic events were evaluated by the hematology service, and appropriate anticoagulative therapy was recommended. RESULTS: One patient with an inherited hypercoagulopathy and two patients with acquired hypercoagulopathies were treated for otologic problems. Two of the patients were stabilized and discharged on life-long anticoagulation therapy. The third patient, in spite of intensive medical and surgical support, eventually succumbed to complications to which an acquired hypercoagulable state made a significant contribution. Expert consultants made the hematologic diagnoses and treatment recommendations. CONCLUSION: Otolaryngologists should be aware that hypercoagulable states may now be more accurately diagnosed and characterized, and that thorough investigation of thromboembolic events may affect treatment decisions.

Adult↗
PubMed1991

Prospective observations of 100 high-risk neonates by high-field (1.5 Tesla) magnetic resonance imaging of the central nervous system: I. Intraventricular and extracerebral lesions.

The results of observations of the first 100 neonates at the University of Texas Health Science Center (Houston) who received magnetic resonance imaging of the central nervous system by means of a high-field image (1.5 T) are reported. All were assessed prospectively to be at risk neurodevelopmental delay. This first report specifically addresses the appearance of primarily hemorrhagic intracranial lesions, including intraventricular hemorrhage (n = 28), and extracerebral lesions, which include 3 cases of venous sinus thrombosis (n = 20). The signal intensities of hemorrhage underwent a characteristic evolution with time with only minor variations in the study group. Magnetic resonance imaging detected direct evidence of hemorrhage for up to 2 months, but hemosiderin was detected as a late indicator of hemorrhage for up to 9 months. Magnetic resonance imaging was equal in benefit to head ultrasonography and computed tomography for the diagnosis of intraventricular hemorrhage, but magnetic resonance imaging was also able to approximate the time of onset of hemorrhage. Magnetic resonance imaging was superior for the evaluation of extracerebral hemorrhage; ultrasonography failed to detect any of these lesions and computed tomography detected only 3 of 7. Short-term neurological abnormality was assessed, but the ability of magnetic resonance imaging to predict long-term neurodevelopmental delay is unknown and is the subject of an ongoing project.

Cerebral Hemorrhage↗
PubMed1997

Potentially prothrombotic abnormalities of coagulation in benign intracranial hypertension.

OBJECTIVE: Benign intracranial hypertension (BIH) may be caused by intracranial venous sinus thrombosis. Cerebral angiograms may, however, be normal in patients with BIH that are associated with conditions with an increased risk of venous thrombosis. This raises the possibility that unrecognised non-occlusive venous thrombus might impede CSF drainage. This study therefore examined the strength of the association between risk factors for thrombosis and BIH. METHODS: The incidence of prothrombotic abnormalities among a mixed prospectively and retrospectively investigated cohort of 38 patients with BIH, was compared with healthy obese subjects, and patients with other neurological diseases. Prothrombotic abnormalities investigated included anticardiolipin antibodies, lupus anticoagulant, antithrombin III, proteins C and S, plasma fibrinogen, kaolin cephalin clotting time, prothrombin time, and full blood counts. RESULTS: Evidence for the presence of an antiphospholipid antibody was found in 32% of cases. Cases of familial deficiency of antithrombin III, thrombocytosis, and polycythaemia were also noted. Additionally, an increased concentration of plasma fibrinogen was found in 26%. A coagulation abnormality was more often detectable in those subjects with normal or low body mass index and in those tested within six months of onset. CONCLUSION: There is a thrombotic pathogenesis in some cases of BIH.

Adolescent↗
PubMed2004

Acute mastoiditis in infancy: the Soroka experience: 1990-2000.

UNLABELLED: Acute mastoiditis (AM) is an uncommon but serious complication of acute otitis media (AOM). In the pre-antibiotic era, AM was seen more frequently than it is today, but it was rare in infants. However, in the last two decades an increase in the incidence of AM in infancy has been reported in the literature. During the years 1990-2002, we treated 113 patients with 128 episodes of AM; of them, 24 were infants (median age 6 months; 18 males) who suffered from 26 episodes of AM. Twenty developed AM as a complication of their first episode of AOM. One of the four infants with a prior history of AOM suffered from common variable immunodeficiency. A significant increase in the incidence of AM in infants was recorded during the study period (P = 0.01). The most common presenting clinical signs were post-auricular swelling and fever >38 degrees C (77% and 77%, respectively, of all patients). Seventeen episodes of AM were not treated with prior antibiotics. Tympanocentesis was performed in all episodes of AM. Middle ear fluid culture was positive in 17 (65%) of the 26 AM episodes. The most common pathogens cultured were Streptococcus pneumoniae (10 infants, 58% of all pathogens, 3/10 intermediately susceptible to penicillin) followed by Streptococcus pyogenes (4, 23%), non-typable H. influenzae (2, 12%) and S. aureus (1, 6%). Temporal bone CT showed bone destruction in 14 patients; 3 infants had subperiosteal abscesses and 3 lateral sinus thrombosis. Ten infants underwent mastoid surgery due to non-resolution of symptoms and signs with antibiotic therapy. Eight underwent cortical mastoidectomy with two patients undergoing ventilation tube introduction only. The remainder of the infants healed with conservative treatment. CONCLUSIONS: (1) A significant increase in the incidence of AM in infants was recorded over the last decade, though a specific reason for this trend remains uncertain; (2) Most of the cases of AM followed the infant's initial AOM episode, and most of the infants had not received prior antibiotic therapy; (3) The clinical signs and symptoms of AM were more severe in infants than in older patients; (4) While S. pneumonia was the most common pathogen isolated in middle ear fluid cultures, the involvement of S. pyogenes in AM was higher than that reported in AOM.

Abscess↗
PubMed1966

Human trichinosis: report of four cases, with emphasis on central nervous system involvement, and a survey of 500 consecutive autopsies at the Ottawa Civic Hospital.

In 1963, acute trichinosis was recognized in four patients at the Ottawa Civic Hospital. One presented with cavernous sinus thrombosis and sixth nerve palsy. A second had a severe systemic infection with myocarditis; the signs of myocarditis appeared in the third week. Electrocardiographic abnormalities included T-wave flattening, prolongation of PR interval and QRS complex, and non-specific changes in 25%. Central nervous system involvement occurred in the second week with general symptoms (headache, delirium and psychotic behaviour), followed in the third week by focal signs (nerve palsy, convulsion, pareses and coma).The incidence of trichinosis in the U.S.A. fell from 15.9% before 1948 to 4.5% in the period 1948-1963. The Canadian incidence in the period from 1940 to 1943 was 5.6%. In a survey in Ottawa using the Baermann digestion and compression methods, four positive cases were found out of 500 diaphragms examined. These figures indicate the success of the public health regulations aimed at controlling trichinosis. A gastrocnemius muscle biopsy is still an invaluable diagnostic tool, especially in critically ill patients with negative skin tests and no eosinophilia.

Adrenocorticotropic Hormone↗
PubMed1984

Minimal change nephrotic syndrome in children: deaths during the first 5 to 15 years' observation. Report of the International Study of Kidney Disease in Children.

Of 521 children with a previously untreated nephrotic syndrome, as defined by proteinuria greater than or equal to 40 mg/h/m2 and serum albumin less than or equal to 2.5 g/dL, entering the International Study of Kidney Disease in Children between January 1967 and April 1976, 389 showed minimal changes on renal biopsy. Of these, seven boys and three girls died, all before July 1972. Infection was the cause of death in six patients. One child died of dural sinus thrombosis, one died as a result of cardiorespiratory failure following salt-poor albumin infusion, and another died from chronic renal failure due to focal and segmental glomerulosclerosis not apparent on initial biopsy. The mode of death in the remaining child was uncertain. Analysis according to histopathologic subgroups of minimal change disease showed no statistically significant differences in the incidence of deaths, although mesangial abnormalities and tubular atrophy were associated with higher mortality than nil disease or focal glomerular obsolescence. Nine of the ten children who died had either failed to respond to initial prednisone therapy (initial nonresponders, n = 5), or responded but relapsed during the initial 8 weeks of treatment (early relapser, n = 4), even though only one quarter of the total sample were nonresponders or early relapsers (P less than .0005). Nearly one fifth of all initial nonresponders with minimal change nephrotic syndrome died. Thus the pattern of response to initial steroid therapy in patients with minimal change nephrotic syndrome may have prognostic significance.

Adolescent↗
PubMed2004

Maternal brain death: medical, ethical and legal issues.

CASE PRESENTATION: We present the case of a pregnant woman who experienced a cerebral venous sinus thrombosis resulting in brain death at 13 weeks gestation. We discuss the management of the mother and foetus following this tragic event. We also discuss the complex medical, legal and ethical issues that arose following maternal brain death. The central question is whether continuing maternal organ supportive measures in an attempt to prolong gestation to attain foetal viability is appropriate, or whether it constitutes futile care. DISCUSSION: Successful maintenance of maternal brain somatic function to facilitate foetal maturation in utero has been reported. While the gestational age of the foetus is central to resolving this issue, there is no clear upper physiological limit to the prolongation of somatic function following brain death. Furthermore, medical experience regarding prolonged somatic support is limited. Finally, the legal rights conferred on the foetus may vary significantly depending on the jurisdiction in which the maternal brain death occurs and may have important implications. CONCLUSIONS: A consensus building approach, involving the family, is essential to resolving these potentially conflicting issues.

Adult↗
PubMed2001

[A point mutation at Arg169 (CGG-->TGG) in hereditary protein C deficiency].

We investigated a 56-year-old Japanese man with protein C deficiency, who was referred to our hospital because of venous sinus thrombosis and pulmonary thromboembolism. Protein C (PC) activity and the corresponding antigen level in plasma were 66% and 106% of the normal values, respectively. Both the activity and antigen levels of protein C were reduced by approximately 50% in plasma from the patient's mother. All exons and their flanking intron regions were amplified by PCR from genomic DNA. Sequencing analysis of the PCR fragments revealed that the patient was heterozygous for a C to T substitution at nucleotide position 6218, resulting in a single amino acid substitution of arginine (CGG) by tryptophan (TGG) at codon 169 of the heavy chain. We analyzed the patient, his mother, and normal controls by a Sac II digestion study of exon 7 and found that the patient and his mother had the same C to T point mutation at base 6218. This mutation could have been responsible for the defective activation of the molecule and the resulting thrombotic disorder. The patient is now being treated with warfarin, and so far no further clinical thrombotic episode has occurred.

DNA Mutational Analysis↗
PubMed2002

Sudden onset headache: a prospective study of features, incidence and causes.

Sudden onset headache is a common condition that sometimes indicates a life-threatening subarachnoid haemorrhage (SAH) but is mostly harmless. We have performed a prospective study of 137 consecutive patients with this kind of headache (thunderclap headache=TCH). The examination included a CT scan, CSF examination and follow-up of patients with no SAH during the period between 2 days and 12 months after the headache attack. The incidence was 43 per 100 000 inhabitants >18 years of age per year; 11.3% of the patients with TCH had SAH. Findings in other patients indicated cerebral infarction (five), intracerebral haematoma (three), aseptic meningitis (four), cerebral oedema (one) and sinus thrombosis (one). Thus no specific finding indicating the underlying cause of the TCH attack was found in the majority of the patients. A slightly increased prevalence of migraine was found in the non-SAH patients (28%). The attacks occurred in 11 cases (8%) during sexual activity and two of these had an SAH. Nausea, neck stiffness, occipital location and impaired consciousness were significantly more frequent with SAH but did not occur in all cases. Location in the temporal region and pressing headache quality were the only features that were more common in non-SAH patients. Recurrent attacks of TCH occurred in 24% of the non-SAH patients. No SAH occurred later in this group, nor in any of the other patients. It was concluded that attacks caused by a SAH cannot be distinguished from non-SAH attacks on clinical grounds. It is important that patients with their first TCH attack are investigated with CT and CSF examination to exclude SAH, meningitis or cerebral infarction. The results from this and previous studies indicate that it is not necessary to perform angiography in patients with a TCH attack, provided that no symptoms or signs indicate a possible brain lesion and a CT scan and CSF examination have not indicated SAH.

Acute Disease↗
PubMed2005

Delayed otogenic hydrocephalus after acute otitis media in pediatric patients: the changing presentation of a serious otologic complication.

CONCLUSIONS: The clinical presentation of otogenic dural sinus thrombosis (DST) as a complication of acute otitis media (AOM) can be masked by antibiotic treatment. Morning episodes of vomiting and/or headache, visual impairment and a history of AOM seem to be indicative of otogenic hydrocephalus. We therefore advocate that the MRI scans of patients with similar symptoms should be carefully studied to facilitate the early diagnosis of a potentially life-threatening complication. OBJECTIVE: To describe the frequency, pathognomonic signs, clinical course and outcome of otogenic hydrocephalus and DST as complications of AOM in pediatric patients. MATERIAL AND METHODS: We undertook a retrospective chart review of all pediatric patients (age 1-14 years) treated for otitis media and its complications at an academic medical center between 1999 and 2003. The main outcome measures were otologic and ophthalmologic findings and CT and MRI scans at the beginning of treatment and 3 months later. RESULTS: We report on five cases with otogenic DST following AOM. All but one of them presented initially with diplopia caused by otogenic hydrocephalus. In four cases the otologic complaints had already disappeared by the time of MRI confirmation of the diagnosis. Only one child was referred with severe otologic symptoms. Management included systemic antibiotics, short-term heparin anticoagulation and surgical decompression. In our cases, even after intensive i.v. antibiotic treatment, only surgery led to a significant improvement in the clinical condition.

Acute Disease↗
PubMed2003

Clinical picture of patients with cerebral venous thrombosis and patterns of dural sinus involvement.

BACKGROUND/OBJECTIVES: Cerebral venous thrombosis may cause focal brain lesions or an isolated intracranial hypertension without focal signs. We investigated whether these different clinical pictures correspond to different patterns of dural sinus thrombosis. METHODS: Forty-eight patients with cerebral venous thrombosis were classified in 2 groups based on the clinical and neuroradiological picture at admission: one group had clinical signs of intracranial hypertension, without focal neurological signs; the other group had focal neurological signs. The extension of thrombosis in dural sinuses was compared between the groups. RESULTS: The amount of thrombosed sinuses was significantly different in the two groups, the involvement of the dural sinuses being greater in patients with isolated intracranial hypertension. CONCLUSIONS: The clinical picture of cerebral venous thrombosis is related to the pattern of dural sinus involvement. The presence of isolated intracranial hypertension is more frequent in patients with a more extended thrombosis of the dural sinuses.

Angiography, Digital Subtraction↗