A case of recurrent cavernous sinus dural arteriovenous fistula arising after superselective shunt occlusion and detected by venous arterial spin labeling

Background: Superselective shunt occlusion (SSSO) for cavernous sinus dural arteriovenous fistula (CSDAVF) avoids the risk of cranial nerve palsy, unlike entire sinus packing, but requires paying attention to recurrence. Distinguishing between true and paradoxical worsening of postoperative ophthalmic symptoms using a less-invasive modality is often difficult. Here, we report a case of true worsening of neuro-ophthalmic symptom by recurrent CSDAVF detected by venous-arterial spin labeling (ASL) on magnetic resonance imaging. Case Description: A 55-year-old woman with neither contributory medical history nor previous head trauma presented with neuro-ophthalmic symptoms and pulsatile tinnitus. Digital subtraction angiography (DSA) revealed CSDAVF with multiple shunted pouches. She underwent successful transvenous SSSO, but neuroophthalmic symptom worsened after SSSO and venous-ASL revealed increased signal intensity in the right superior orbital vein (SOV). DSA confirmed recurrent CSDAVF and additional transvenous embolization was performed. Neuro-ophthalmic symptoms and venous-ASL hyperintensity on SOV improved postoperatively. Conclusion: Venous-ASL is noninvasive and seems useful for detecting true worsening of neuro-ophthalmic symptoms of recurrent CSDAVF.

Human Monoclonal Antibodies against NS1 Protein Protect against Lethal West Nile Virus Infection

Therapeutic antibodies against flaviviruses often promote neutralization by targeting the envelope protein of the virion. However, this approach is hindered by a possible concern for antibody-dependent enhancement of infection and paradoxical worsening of disease.

Left hand extensor tenosynovitis due to <i>Histoplasma capsulatum</i> complicated by immune reconstitution inflammatory syndrome

We describe a case of left hand extensor tenosynovitis due to histoplasmosis in a patient with dermatomyositis on chronic immunosuppression. Treatment involved surgical debridement and antifungal therapy. The patient experienced paradoxical worsening of tenosynovial inflammation during de-augmentation of immunosuppression felt to be immune reconstitution inflammatory syndrome.

ANCA associated glomerulonephritis in tuberculosis: a paradoxical reaction

We present a case of antineutrophil cytoplasmic antibodies (ANCA)-associated rapidly progressive glomerulonephritis in the context of treatment of pulmonary tuberculosis (TB). A 42-year-old woman was treated for drug-susceptible pulmonary TB and represented with paradoxical worsening of symptoms and radiological features. She was HIV negative. A severe acute kidney injury with features of glomerulonephritis was evident on admission. Perinuclear ANCA and antimyeloperoxidase antibodies were present in serum and renal biopsy was consistent with ANCA-associated vasculitis. The patient was successfully treated with both antituberculous therapy and immunosuppression (corticosteroids and mycophenolate mofetil) with subsequent clinical improvement and amelioration of renal function. We propose this is the first case that describes the association between paradoxical reactions during TB treatment and ANCA-associated glomerulonephritis.

843 Paradoxical Waking Hypoxemia that Improves with Sleep

Introduction We present a case of paradoxically worsened hypoxia during wake phase of polysomnography while undergoing a CPAP titration study. Nighttime hypoxemia is a common feature in obstructive sleep apnea, due to obstructive events that manifest while sleeping. Excluding OSA, there remains an extensive differential for disease processes that cause hypoxemia while asleep; however, none of these processes can explain waking hypoxemia that improves upon sleeping. Report of case(s) A 70 year old male with severe OSA diagnosed by home sleep test (REI 46.5, nadir O2=76%) underwent polysomnography with PAP titration and demonstrated several hours of interrupted sleep without hypoxia and minimal obstructive events on CPAP 9–13 cmH2O. During the study, while awake at CPAP of 14 cmH2O, he developed hypoxia to mid-high 80s and supplemental oxygen bleed in was added starting at 3L and increased to 5L during a prolonged period of wakefulness. On CPAP 15 cmmH2O with 5L bleed-in, the patient fell asleep and oxygen saturation again increased to low 90s. He underwent an extensive workup for other cardiopulmonary causes of hypoxemia, with pulmonary function testing showing moderate obstructive ventilatory defect and mild DLCO impairment. An echocardiogram with saline contrast bubble study was relatively unremarkable, without evidence of right to left shunting. He underwent a chest CTA which was negative for pulmonary embolism, though it did reveal an enlarged pulmonary artery consistent with pulmonary hypertension. His chronic hypoxemia was treated with 2L supplemental oxygen during the day and bleed-in with CPAP at night. Conclusion Though nocturnal hypoxemia is common with OSA, polysomnography with paradoxical hypoxemia during wake phase has not been reported. Notably, the patient was without prolonged hypoxia during his sleep phase while on CPAP treatment with minimal apneic/hypopneic events. Pulmonary hypertension can also present as nocturnal hypoxemia, but it should worsen with sleep, rather than improve. There are case reports of right to left shunting worsened by PAP, though his hypoxemia persisted despite PAP. His paradoxical worsening hypoxemia with wakefulness is still unexplained. Support (if any):

Disseminated coccidioidomycosis in a patient who is immunocompromised in the setting of immune reconstitution inflammatory syndrome

Coccidioidomycosis is a systemic fungal infection first described in 1892. More than 95% of annual cases occur in Arizona and California. It is an opportunistic infection (OI) transmitted via inhalation of airborne spores (arthroconidia) and rarely via percutaneous inoculation into a tissue or solid organ transplantation in patients who are immunocompromised and with HIV. With the advent of antiretroviral therapy (ART), the incidence of OIs has markedly reduced; however, OIs continue to occur, particularly in patients who present late for medical care or delay ART initiation. In rare cases, immunodeficient individuals may experience a paradoxical worsening or unmasking of OI symptoms, known as the immune reconstitution inflammatory syndrome (IRIS). We present a case of a 31-year-old man with disseminated coccidioidomycosis affecting the spleen, lymph nodes, lungs, bone marrow, and adrenals who developed IRIS after the initiation of ART.

Paradoxical reaction to increased doses of intrathecal baclofen in a patient with Leigh syndrome

In clinical practice, intrathecal baclofen (ITB) therapy is used to control spasticity. After initial placement of the ITB pump, clinicians incrementally increase the dose until effectiveness in alleviating spasms and spasticity is optimized. However, this case describes a 4-year-old male with Leigh syndrome who developed a paradoxical worsening of spasticity and pain with incremental increase of his ITB pump. In this rare genetic disease with a poor prognosis, an ITB pump was trialed and implanted and titrated upwards with initial improvement. However, his spasticity and pain then began to worsen with each dosage increase. Subsequently, his symptoms improved significantly when the dose was weaned. This is the first case that describes this paradoxical reaction in a pediatric population and discusses recommendations about how clinicians should safely titrate the pump for patient care.

Progressive multifocal leucoencephalopathy as a manifestation of immune reconstitution inflammatory syndrome in a patient with HIV infection: a case report

Immune reconstitution inflammatory syndrome (IRIS) is defined as paradoxical worsening of a known condition or the appearance of a new condition after initiating antiretroviral therapy (ART) in HIV-infected patients. IRIS results from restored immunity to specific infectious or non-infectious antigens. Immune reconstitution following initiation of ART may lead to activation of an inflammatory response to detectable or latent JC virus (JCV) infection, an etiological agent of progressive multifocal leucoencephalopathy (PML). We present an interesting case of IRIS manifesting as PML in a newlydiagnosed HIV-infected patient started on ART.