Idiopathic red ear syndrome: A rare case report

Red ear syndrome (RES) is a very rare disorder that is characterized by a unilateral or bilateral attack of paroxysmal burning sensation and reddening of the external ear. The duration of symptoms ranges from a few seconds to hours. It can occur spontaneously or be triggered by rubbing of the ear, heat or cold stimulation, brushing of hair, and neck movement. Diagnosis and treatment of this condition are challenging. The pathophysiology of RES is still unclear and hypotheses involving peripheral or central nervous system mechanisms have been proposed. RES is regarded as refractory to medical treatments, although some migraine preventative treatments have shown moderate benefit mainly in patients with migraine‐related attacks. We report a case with Idiopathic RES who presented with paroxysmal redness of the bilateral pinnae partially benefitted by medical treatment.     

Red ear syndrome and migraine: report of eight cases

We describe eight idiopathic cases of red ear syndrome in seven children and one adult. All were migraineurs with a history of paroxysmally painful and red ear, unilateral or alternating, in isolation or associated with migraine attacks. The reported duration of these episodes varied from 30 minutes to 1 hour. Neurologic examination, brain MRI and CT scans, and x-rays of the cervical spine were normal. The close temporal relationship between the "red ear episodes" and migraine attacks suggests an association between the two conditions.     

Circadian and seasonal variation of migraine attacks in children

OBJECT: To investigate the rhythmicity of migraine episodes without aura in a pediatric population. METHODS: Time of occurrence of 2517 migraine attacks in 115 children was recorded, by means of a diary, both by hourly and monthly intervals. RESULTS: A significant circadian variation, characterized by a peak in the afternoon (P < .001) and one in the early morning (P= .002) was found. A seasonal peak was also observed between November and January, while a nadir was observed in July. CONCLUSIONS: The clustering of attacks in the morning and midday and in autumn-winter, with a minimum frequency in July, suggests that school activities may represent an important cause of migraine.     

Circadian rhythm of core body temperature in subjects with chronic fatigue syndrome

The pathophysiological basis for chronic fatigue syndrome (CFS) remains poorly understood. Certain symptoms of CFS, namely fatigue, neurocognitive symptoms and sleep disturbance, are similar to those of acute jet lag and shift work syndromes thus raising the possibility that CFS might be a condition associated with disturbances in endogenous circadian rhythms. In this study, we tested this hypothesis by examining the circadian rhythm of core body temperature (CBT) in CFS and control subjects. Continuous recordings of CBT were obtained every 5 min over 48 h in a group of 10 subjects who met the Center for Disease Control (CDC) definition of CFS and 10 normal control subjects. Subjects in the two groups were age, sex and weight‐matched and were known to have normal basal metabolic rates and thyroid function. CBT recordings were performed under ambulatory conditions in a clinical research centre with the use of an ingestible radio frequency transmitter pill and a belt‐worn receiver‐logger. CBT time series were analysed by a cosinor analysis and by a harmonic‐regression‐plus‐correlated‐noise model to estimate the mean, amplitude and phase angle of the rhythm. The goodness of fit of each model was also compared using the Akaike Information Criterion (AIC) and σ². Average parameters for each group were compared by Student’s t‐test. By cosinor analysis, the only significant difference between CFS and control groups was in the phase angle of the third harmonic (P=0·02). The optimal harmonic‐regression‐plus‐correlated‐noise models selected were ARMA(1,1): control 7, CFS 6; ARMA(2,0): control 1, CFS 4; and ARMA(2,1): control 2 subjects. The optimal fit ARMA model contained two harmonics in eight of 10 control subjects but was more variable in the CFS subjects (1 harmonic: 5 subjects; 2 harmonics: 1 subject; 3 harmonics: 4 subjects). The goodness of fit measures for the optimal ARMA model were also better in the control than the CFS group, but the differences were not statistically significant. We conclude that, measured under ambulatory conditions, the circadian rhythm of CBT in CFS is nearly indistinguishable from that of normal control subjects although there was a tendency for greater variability in the rhythm. Hence, it is unlikely that the symptoms of CFS are because of disturbance in the circadian rhythm of CBT.     

Fatal stroke in migraine: a case report with autopsy findings

A 50-year-old woman with migraine was admitted to hospital shortly after having abruptly developed hemiparesis. CT scan revealed infarction in the territory of the right middle cerebral artery. Death ensued after three days due to cerebral edema with herniation. Autopsy revealed no pathologic findings in the heart or in the extra- or intracranial arteries. It is suggested that the fatal stroke may have resulted from arterial spasm caused by ergotamine overdosage and possibly complicated by thrombosis.     

Familial hemiplegic migraine with prolonged aura and multimodality imaging: a case report

We report a case of familial hemiplegic migraine with prolonged aura where multimodality imaging showed hemispheric cytotoxic edema along with evidence of hypometabolism in the affected hemisphere while there was no evidence of hypoperfusion of the affected hemisphere demonstrating that neuronal depression is a more plausible explanation in its pathogenesis.     

Immunoadsorption therapy for Klinefelter syndrome with antiphospholipid syndrome in a patient: A case report

BACKGROUNDKlinefelter syndrome (KS) is a genetic disease of male sex chromosome malformations that affects sperm production and reduces testosterone production. It has been reported that there is currently more than 10 cases of KS combined with antiphospholipid syndrome.CASE SUMMARYHere, we describe a 31-year-old male patient with chromosome 47, XXY type, who suffered deep vein thrombosis of the lower limbs accompanied by abnormal antiphospholipid antibody, lupus anticoagulant and factor VIII. After treatment with immunoadsorption therapy, glucocorticoids, cyclophosphamide, intravenous immunoglobulin and anticoagulant therapy, the patient showed dramatic symptomatic improvement. During the follow-up, the patient did not develop any new thrombotic events.CONCLUSIONImmunoadsorption combined with glucocorticoid and cyclophosphamide shock comprehensive treatment has achieved significant results for patients with KS combined with antiphospholipid syndrome.     

New onset migraine with aura following head injury: a case report

A patient is described who developed recurrent headaches following a traumatic injury in the vicinity of the right greater occipital nerve (GON) that fulfill IHS criteria for migraine with aura. Both aura and headache can be triggered by pressure over the GON and relieved by local anesthetic blockade.     

Alpha rhythm in classical migraine (migraine with aura): abnormalities in the headache-free interval

The alpha rhythm of 18 patients with classical migraine (migraine with aura) was studied by EEG spectrum analysis for evidence of neural abnormalities during the asymptomatic period. The temporal relationship of the findings to attacks was studied by serial records in 11 cases. Increased frequency dispersion and frequency asymmetries of the alpha rhythm were found. The records were, however, mostly normal when separated from attacks by at least 10 asymptomatic days. The abnormalities increased significantly before the onset of prodromal symptoms and clearly outlasted the headache phase. The results give evidence of a fluctuating asymmetric neural disorder in classical migraine.     

Dental management of a patient with Moebius syndrome: A case report

BACKGROUNDMoebius syndrome (MBS) is a nonprogressive and rare congenital neuromuscular disorder involving the facial nerve and abductor nerve; it mainly manifests as facial paralysis and eye strabismus paralytic symptoms. Tissues in the oral cavity are also compromised, characterized by microstomia, micrognathia, tongue malformation, cleft lip, high arched palate or cleft palate, bifid uvula, and dental malocclusion. Therefore, dentistry plays a fundamental and crucial role in caring for these individuals. However, there is limited available data on MBS treatment, particularly regarding dental management.CASE SUMMARYThis case report presents dental treatment of a 21-year-old man with MBS and discusses crucial interactions among oral complications of MBS. In this case, the patient was clinically characterized by congenital neuromuscular disorder, occlusal disorders, and tooth and gum problems. It is noteworthy that the patient presented early eruption of deciduous teeth 2 mo after birth, which has not been reported in other MBS cases and suggests a potentially new clinical manifestation of this syndrome. It is important to note that MBS cannot be cured, and oral manifestations of this syndrome can be managed by a multidisciplinary health care team that helps the patient maintain oral hygiene and dental health. After a series of oral treatments, no obvious poor oral hygiene, swollen gums, or abnormal imaging results were observed after 2 years of follow-up.CONCLUSIONThis case addressed the oral clinical manifestations of MBS and difficulties experienced during dental management, and suggested early tooth eruption as a potentially new clinical manifestation of this syndrome. Knowledge of the loop-mediated association among oral complications of this syndrome is essential to perfecting treatments.     

Agraphia as the only symptom of migraine aura: a case report

A 37-year-old female migrain patient is described, who suffered from episodes with pure lexical agraphia as the only symptom of migraine aura. The MRI showed multiple subcortical white matter lesions. Central inflammatory and demyelinating disorders were excluded. This case suggests that, during a migraine aura, even very circumscribed brain areas involved in complex neuropsychological processing can be affected.     

Primary and secondary red ear syndrome: implications for treatment

Approximately 57 patients with red ear syndrome have been reported in the literature since it was initially described in 1994. The clinical phenotype therefore continues to be defined and no consistent treatment response has been described. We report three new cases of red ear syndrome and suggest that the disorder may exist as a primary trigeminal or cervical autonomic cephalalgia, coexist with other rare trigeminal autonomic cephalalgias, or may be secondary. The pathogenesis in these different circumstances may be distinct, the triggers specific and, when secondary, the disorder may be more resistant to treatment.     

Adult-onset ophthalmoplegic migraine with recurrent sixth nerve palsy: a case report

We describe a patient with ophthalmoplegic migraine and left sixth nerve palsy, in whom disease's onset occurred at middle age.     

Sporadic hemiplegic migraine: report of a case with clinical and radiological features

A case of visual hallucination, headache and left hemiparesis is reported. The patient had a history of recurrent attacks of similar semiology for the previous 15 years. MRI brain revealed a cortical hyperintensity on T2W, FLAIR and diffusion weighted imaging (DWI) in the right cerebral hemisphere with a normal ADC (apparent diffusion coefficient) map and MR angiogram. Detailed workup for MELAS was negative. A diagnosis of sporadic hemiplegic migraine was made and he was managed conservatively. He made a gradual complete recovery over 2 weeks. He was discharged on flunarizine for prophylaxis and has remained asymptomatic over the ensuing 4 months. This interesting condition is reviewed and discussed herein.     

Posterior reversible encephalopathy syndrome in a patient with metastatic breast cancer: A case report

BACKGROUNDPosterior reversible encephalopathy syndrome (PRES) is a neurotoxic encephalopathic state with clinical symptoms such as headache, altered consciousness, visual disturbances, and seizures. Vasogenic edema occurs predominantly in the posterior occipital and parietal lobes of the brain. PRES is caused by various diseases, and its mechanism remains unclear. However, it can be easily diagnosed based on characteristic lesions on magnetic resonance imaging.CASE SUMMARYA 51-year-old woman with unremarkable past medical history presented with progressively worsening back pain since 2 mo. Physical examinations revealed paralumbar muscle tenderness, a large lesion on the right breast and several mass-like lesions on both breasts. The blood pressure (BP) was elevated (150/90 mmHg), and did not respond to antihypertensive medication. On the seventh day of hospitalization, she exhibited a confused mental status and generalized tonic-clonic seizures. On magnetic resonance imaging, bilateral cortical and subcortical edema of the occipital lobes, suggestive of PRES, was observed. The serum calcium was 15.8 mg/dL. After two days of treatment with nicardipine, elcatonin, and zolendronic acid, her BP was 130/91 mmHg and serum calcium was 10.1 mg/dL. The patient regained consciousness and her mental status improved. Fluorodeoxyglucose-positron emission tomography revealed right breast cancer with extensive metastases. CONCLUSIONAlthough rare, hypercalcemia can lead to PRES by causing uncontrolled hypertension. Prompt diagnosis can help prevent severe mental disturbances and even death.     

Monitored anesthesia care for craniotomy in a patient with Eisenmenger syndrome: A case report

BACKGROUNDEisenmenger syndrome (ES) is an uncorrected congenital cardiac defect with a left-to-right shunt, leading to pulmonary arterial hypertension. Patients with ES are susceptible to hemodynamic alterations during noncardiac surgery with general anesthesia, which increases perioperative morbidity and mortality. Monitored anesthesia care (MAC) is often used during minor procedures in patients with cardiac disease. However, few reports on MAC in patients with ES exist.CASE SUMMARYA 49-year-old man was admitted for a severe headache lasting 30 d. He had been diagnosed with a large perimembranous ventricular septal defect (VSD) with bidirectional shunt flow and pulmonary arterial hypertension 10 years ago. A round mass in the right frontal lobe was revealed by Magnetic resonance imaging. Stereotactic aspiration using a neuronavigation system was performed under MAC. The patient was stayed in the hospital for 5 d, and discharged without complications.CONCLUSIONMAC may be effective for craniotomy in patients with ES.