Rejected

Three patients with eosinophilic meningitis (EoM) were investigated in two hospitals in Porto Alegre, Southern Brazil. These patients had a common exposure after the ingestion of raw mollusks in a religious ritual. Two of them had an uncommon presentation with intense lower distal extremities pain and small fiber neuropathy as defined by an electroneuromyography (ENMG) study. All three patients were positive for Angiostrongylus cantonensis serology and recovered after antihelminthic and anti-inflammatory treatment. Increased awareness of A. cantonensis infection is important to avoid new infections and to improved recognition and handling of cerebral angiostrongyliasis.

Hepatitis is a rare complication of herpes simplex virus (HSV) which can lead to acute liver failure, liver transplant, or death. This complication is more commonly seen in neonates, immunocompromised, or pregnant patients. Early recognition of disease facilitates prompt treatment with antiretrovirals and prevent its progression. To our knowledge, only 30 cases have been reported. Our patient presented with headaches and elevation of transaminases followed by vesicular rash. Culture tested positive for HSV1 and HSV2 and the patient was successfully treated with Acyclovir.

Atraumatic limb pain and limp is a common pediatric presentation in the emergency department in the United States. In a majority of cases, these presentations are benign. However, in cases where pediatric patients are repeatedly presenting for atraumatic limb pain, further investigation is required. We present such the case of a 14-year-old female with acute worsening of progressive atraumatic knee pain who was found to have a Brodie's abscess, a subacute pyogenic form of osteomyelitis. This is a particularly challenging diagnosis, as it often presents with no associated symptoms such as fever or weight loss. The consequences of missing this diagnosis include permanent disability and potential amputation, but excellent outcomes can be expected for those who undergo timely surgical debridement and irrigation. We discuss the etiology, common presentations, and treatment of this rare but potentially limb-threatening disease in the hope that clinicians will consider this diagnosis in cases of persistent or progressive atraumatic limb pain.

Langerhans cell histiocytosis (LCH) is a rare systemic disorder characterized by an infiltration of CD1a+/langerin+ histiocytes, commonly involving bone, skin, and lymph nodes in children. Hepatic involvement is rarely observed in multisystem LCH. We describe an exceptional case of hepatic LCH in an adult preceding the diagnosis of multisystem LCH, mimicking anti-mitochondrial antibody (AMA)-negative primary biliary cholangitis (PBC). A 65-year-old man presented with intermittent pruritus, weakness, dyspnea, fever, and chills that have been progressive for four years. Physical examination was unremarkable. Laboratory work revealed cholestatic biochemistry profile. Liver biopsy showed portal non-necrotizing granuloma encasing a damaged duct (florid duct lesion), and multifocal lobular Kupffer cell clusters, suggestive of PBC. Tests for autoimmune diseases including AMA were negative. Endoscopic retrograde cholangiopancreatography (ERCP) was negative for biliary obstruction. One month after the liver biopsy, he developed flaky, red, and burning rash on the right scalp, forehead, and epigastric skin. A skin biopsy at an outside institution revealed LCH. Subsequent re-examination of the liver biopsy showed that the histiocytes within the florid duct lesion were positive for CD1a and S-100. Concurrently, a small focus of LCH was noted in his gastric biopsy performed for gastritis symptoms. Hepatic LCH may mimic AMA-negative PBC histologically and clinically and may present as a harbinger of multisystem LCH. While rendering the diagnosis would be challenging without prior history of LCH and with focal involvement, awareness of such presentation and communication with clinical colleagues may be helpful.

Antiphospholipid syndrome (APS) is a systemic autoimmune disorder with marked thrombotic and inflammatory features driven by the presence of antiphospholipid antibodies (APLA). Here, we report a case of APS with a rare, atypical manifestation and discuss a differential diagnosis. A 53-year-old male without significant past medical history presented with new onset of episodic tongue stiffness and dysarthria which lasted for about a minute over a period of three months. This was associated with intermittent right retro-orbital sharp pain radiating to the parietal area. He also reported swelling and stiffness of the third and fourth right proximal interphalangeal (PIP) joints lasting throughout the day. A physical exam revealed tongue fasciculations. As the MRI showed patchy white matter hyperintensities neurology initially suspected multiple sclerosis. However, cerebrospinal fluid (CSF) analysis including neuromyelitis optica (NMO) antibodies and oligoclonal antibodies was negative. Rheumatological work up was remarkable for positive antinuclear antibodies (ANA); anticardiolipin antibodies and lupus anticoagulant were positive 12 weeks apart. This, alongside with stable white matter changes on imaging was suspicious for an extra-criteria manifestation of antiphospholipid antibody syndrome. The most commonly described neurological manifestations of APS are headache, transient ischemic attack (TIA), and stroke. Tongue stiffness as an initial symptom is quite unusual and, to the best of our knowledge has not been reported in medical literature. In patients with isolated neurological findings of unclear etiology, an autoimmune disease such as APS should be considered, and appropriate diagnostic work up should not be postponed. Unfortunately, positive laboratory markers can have a wide differential diagnostic panel. In addition, APS may mimic many diseases both in clinical presentation and MRI findings thus making the correct diagnosis challenging. However, studies show that, unlike multiple sclerosis (MS), white matter changes in APS remain static during the course of the disease. Identification of atypical presentations of APS is critical as prompt and correct medical management can improve patients' quality of life and clinical outcomes.

Currently, the management strategies aimed at the resolution of migraine are pharmacological. Most of these therapies are known to alter the serotonin balance of the brain. Furthermore, therapies blocking the calcitonin gene-related peptide (CGRP) have also proven to be quite effective in their treatments. However, apart from being expensive, these therapies do not influence premonitory and aura symptoms. This suggests an incomplete approach and an inadequate understanding of the migraine pathophysiology. Recent metabolic studies have indicated that migraine should be considered as an adaptive response to the mismatch between the cerebral energy reserves and expenditure. Therefore, understanding the underlying metabolism helps derive possible novel therapeutic modalities for migraines. In this review, we highlight the underlying metabolic abnormalities found in migraine patients. This will form the basis of our evidence-based discussion on metabolic therapeutic options for migraines.

Idiopathic hypereosinophilic syndrome (IHES) is a rare myeloproliferative disease characterised by multisystem dysfunction and persistent, extreme eosinophilia of unknown cause. Here we present a 42-year-old patient complaining of moderate to severe chest pain and shortness of breath, and typical ischaemic electrocardiography changes were recorded. He was initially suspected of having acute coronary syndrome, however the coronary angiogram excluded coronary abnormalities. Bone marrow biopsy, left ventriculography, transthoracic echocardiography and cardiac magnetic resonance examinations confirmed the diagnosis of IHES and IHES-mediated cardiac involvement. The patient's illness was alleviated during the first hospitalisation, whereas it had rapidly worsened one month after discharge. In addition, simultaneous pulmonary and skin-infiltrating lesions occurred during the second hospitalisation. The patient's condition improved markedly with combined glucocorticoid, hydroxyurea and warfarin therapy, as well as treatment for heart failure. In this report the diagnostic modalities and treatment strategies for IHES are discussed and reviewed.

The programmed intermittent epidural bolus technique has shown superiority to continuous epidural infusion techniques, with or without patient-controlled epidural analgesia for pain relief, reduced motor block and patient satisfaction. Many institutions still use patient-controlled epidural analgesia without a background infusion, and a comparative study between programmed intermittent epidural bolus and patient-controlled epidural analgesia without a background infusion has not yet been performed. We performed a randomised, two-centre, double-blind, controlled trial of these two techniques. The primary outcome was the incidence of breakthrough pain requiring a top-up dose by an anaesthetist. Secondary outcomes included: motor block; pain scores; patient satisfaction; local anaesthetic consumption; and obstetric and neonatal outcomes. We recruited 130 nulliparous women who received initial spinal analgesia, and then epidural analgesia was initiated and maintained with either programmed intermittent epidural bolus or patient-controlled epidural analgesia using ropivacaine 0.12% with sufentanil 0.75 µg·ml . The programmed intermittent epidural bolus group had a programmed bolus of 10 ml every hour, with on-demand patient-controlled epidural analgesia boluses of 5 ml with a 20 min lockout, and the patient-controlled epidural analgesia group had a 5 ml bolus with a 12 min lockout interval; the potential maximum volume per hour was the same in both groups. The patients in the programmed intermittent epidural bolus group had less frequent breakthrough pain compared with the patient-controlled epidural analgesia group, 7 (10.9%) vs. 38 (62.3%; p < 0.0001), respectively. There was a significant difference in motor block (modified Bromage score ≤ 4) frequency between groups, programmed intermittent epidural bolus group 1 (1.6%) vs. patient-controlled epidural analgesia group 8 (13.1%); p = 0.015. The programmed intermittent epidural bolus group had greater local anaesthetic consumption with fewer patient-controlled epidural analgesia boluses. Patient satisfaction scores and obstetric or neonatal outcomes were not different between groups. In conclusion, we found that a programmed intermittent epidural bolus technique using 10 ml programmed boluses and 5 ml patient-controlled epidural analgesia boluses was superior to a patient-controlled epidural analgesia technique using 5 ml boluses and no background infusion.

Barré's 1926 report "Sur un syndrome sympathique cervicale postérieure et sa cause fréquente: l'arthrite cervicale" is arguably the first description of what we now call cervicogenic headache. Barré's contribution to the subject and significant insights, which have stood the test of time, are insufficiently recognised. This article is an English translation of Barré's French original.

Treating neuropathic pain is a critical clinical issue. Although numerous therapies have been proposed, effective treatments have not been established. Therefore, safe and feasible treatment methods are urgently needed. In this study, we investigated the therapeutic effects of autologous intrathecal administration of bone-marrow-derived mononuclear cells (MNCs) on neuropathic pain. We generated a mouse model of neuropathic pain by transecting the spinal nerve and evaluated neuropathic pain by measuring the mechanical threshold in the following 14 days. Mice in the MNC injection group had a higher mechanical threshold than those in the buffer group. We assessed the effect of MNC treatment on the dorsal root ganglia and spinal cord by immunohistochemistry, mRNA expression, and cytokine assay. The migration and accumulation of microglia were significantly suppressed in the MNC group, and the mRNA expression of inflammatory cytokines such as interleukin (IL)-6, IL-1β, and tumor necrosis factor alpha (TNF-α) was markedly downregulated. Furthermore, MNC administration tended to suppress various cytokines in the cerebrospinal fluid of the model mice. In conclusion, our results suggest that intrathecal injection of MNCs relieves neuropathic pain and might be a promising cell therapy for the treatment of this condition.