Carpal tunnel syndrome caused by remitting seronegative symmetrical synovitis with pitting oedema.

Britta Nijsse, Gerwin Roks - Department of Neurology,

St Elisabeth Hospital, Tilburg, The Netherlands

Correspondence to: Britta Nijsse, b.nijsse@

elisabeth.nl

To cite: Nijsse B, Roks G.

BMJ Case Reports Published online: 28 November 2012

DESCRIPTION

A 73-year-old man presented with paresthesias of

the fingertips and weakness of the abductor pollicis

brevis muscle and the opponens pollicis muscle of

both hands. Carpal tunnel syndrome (CTS) was

suspected and confirmed by electromyography. The

presence of swelling and pitting oedema of both

hands (figures 1 and 2) led to the diagnosis of

remitting seronegative symmetrical synovitis with

pitting oedema (RS3PE).

RS3PE is a rheumatological clinical syndrome,

distinctive of rheumatoid arthritis and polymyalgia

rheumatica. It occurs predominantly in elderly men

and is characterised by rapid-onset symmetrical

synovitis, pitting oedema especially on the dorsum

of the hands, negative rheumatoid factor and a dramatic response to low doses of glucocorticoids (prednisone 10–20 mg/ day).

Few studies report the correlation of RS3PE with

CTS. An incidence of 22–43% is reported.

symptoms completely resolve after treating RS3PE

with steroids. Every neurologist should recognise

CTS, but this does not always relate the swollen

hands to RS3PE. Owing to the presumed paraneoplastic aetiology, it is important to recognise the syndrome. Several reports about RS3PE-associated

malignant conditions were published, mostly

adenocarcinomas or haematological malignancies.

This created the presumption of RS3PE being a

paraneoplastic manifestation. The search for occult

malignancy is particularly crucial in patients with

systemic symptoms and those who are not responsive to steroids.

The symptoms in our patient completely

resolved after steroid treatment and analysis

showed no signs of malignancy.

Learning points:

▸ If Carpal tunnel syndrome (CTS) goes together

with swollen hands and pitting oedema, the

diagnosis of remitting seronegative symmetrical

synovitis with pitting oedema (RS3PE) should

be considered.

▸ Given the presumed paraneoplastic aetiology,

it is important to screen for associated

malignancies.

▸ RS3PE has an excellent response on

glucocorticoids and also the CTS symptoms

resolve.

Competing interests None.

Patient consent Obtained.

REFERENCES

1 Olive A, del Blanco J, Pons M, et al. The clinical spectrum of

remitting seronegative symmetrical synovitis with pitting edema.

The Catalan Group for the Study of RS3PE. J Rheumatology

1997;24:333–6.

2 Bucaloiu ID, Olenginski TP, Harrington TM. Remitting seronegative

symmetrical synovitis with pitting edema syndrome in a rural

tertiary care practice: a retrospective analysis. Mayo Clin Proc.

2007;82:1510–15.

3 Cantini F, Salvarini C, Olivieri I, et al. Remitting seronegative

symmetrical synovitis with pitting oedema (RS3PE) syndrome: a

prospective follow up and magnetic resonance imaging study. Ann

Rheum Dis 1999;58:230–6.

Figure 1 Promine

Casereports

Severe hypertension and pulmonary edema associated with systemic absorption of topical phenylephrine in a child during retinal surgery.

2012

Abdelhalim AA, Mostafa M, Abdulmomen A, Othman EA.

Source

Department of Anesthesia, King Abdul Aziz University Hospital, King Saud University, Riyadh, Saudi Arabia.

Abstract

Topical phenylephrine solutions are widely used in eye procedures to promote pupil dilation without cycloplegia. We report a case of intraoperative severe hypertension and acute pulmonary edema occurring in a child during retinal surgery after possible systemic absorption of topical phenylephrine eyedrops. Our objective is to discuss the proper treatment and preventive strategies for such a complication. A 4-year-old, male patient, 18.4 kg in weight, physical status ASA I was admitted for right retinal detachment surgery. Anesthesia was induced with sevoflurane in oxygen, followed by glycopyrrolate (5.0 μg/kg), propofol 25 mg, fentanyl 50 μg and cisatracurium 0.15 mg/kg given intravenously. Anesthesia was maintained with sevoflurane 2-2.5% in a mixture of nitrous oxide and oxygen (60%:40%). After incision, two drops of 10% aqueous phenylephrine were administered topically by the surgeon to the right eye for further pupil dilation. Few minutes later, the noninvasive blood pressure rose to 220/120 mmHg and the heart rate increased to 140 beats/min. Oxygen saturation (SpO(2)) dropped from 99% (with an inspired oxygen concentration (FiO(2)) of 0.4) to 82%. Auscultation revealed crepitations throughout the chest and a blood-stained frothy fluid was aspirated from the trachea with possible development of acute pulmonary edema. Hydralazine (5 mg) and furosemide (10 mg) were administered intravenously. Seven minutes later, the blood pressure returned to normal and the SpO(2) increased to 92% on FiO(2) of 1.0, with decreased intratracheal secretions. After approximately 20 minutes, the SpO(2) had improved to 99%, with a FiO(2) of 1.0 and the blood pressure was 109/63 mmHg and heart rate was 121 beats/min. The FiO(2) gradually reduced back to 0.4 over 30 min with no further desaturation. The patient was discharged from the post anesthesia care unit 5 h after surgery with adequate spontaneous breathing, SpO(2) 99% on room air, normal blood pressure and pulmonary auscultation. Anesthesiologists and ophthalmologists should be aware of the possible cardiovascular side-effects of topical phenylephrine, and it should be used cautiously with appropriate intraoperative monitoring of hemodynamic variables. Moreover, preventive strategies to minimize systemic absorption of the drug should be taken.

PubMed

Bone edema on MRI is highly associated with low bone mineral density in patients with early inflammatory back pain: results from the DESIR cohort.

Bone oedema on MRI is highly associated with low bone mineral density in patients with early inflammatory back pain: results from the DESIR cohort.

Nov 2012

Briot K, Durnez A, Paternotte S, Miceli-Richard C, Dougados M, Roux C.

Source

1Department of Rheumatology, Cochin Hospital, Paris Descartes University, Paris, France.

Abstract

OBJECTIVES:

 To assess bone mineral density (BMD) at lumbar spine and hip in a large cohort of patients with early inflammatory back pain (IBP) suggestive of axial spondyloarthritis (SpA), and to assess systemic and bone inflammation (according to MRI) as risk factors of low BMD.

PATIENTS AND METHODS:

 332 (52.4% male) patients with IBP suggestive of axial SpA defined by Calin or Berlin criteria were recruited; they had lumbar spine and hip BMD and body composition measurements. Low BMD was defined by Z≤-2 (at least one site). Clinical, biological (erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP)) and imaging (x-rays, spine and sacroiliac joint MRI) parameters were compared in patients with and without low BMD (Z≤-2). Significant parameters in univariate analysis were tested in multivariate models.

RESULTS:

 Patients (mean age 33.8 years) had a short duration of axial symptoms (mean 1.6 years); 71.4% fulfilled the Assessment of Spondyloarthritis International Society criteria for axial SpA and HLA-B27 was present in 62.1%. 43 (13.0%) had low BMD (88% male). Multivariate logistic regression showed that parameters significantly associated with low BMD (any site) were the presence of bone marrow oedema (inflammatory lesions) on MRI (OR 4.63, p=0.001), either ESR or CRP (OR 2.60, p=0.037) and male gender (OR 9.60, p=0.0004).

CONCLUSIONS:

This study conducted in a large cohort of young adults with early IBP suggestive of SpA shows that 13.0% of patients have a low BMD and that the main risk factor associated with low BMD was inflammation on MRI.

PubMed

A Human Disease Model of Drug Toxicity-Induced Pulmonary Edema in a Lung-on-a-Chip Microdevice.

Nov 2012

Huh D, Leslie DC, Matthews BD, Fraser JP, Jurek S, Hamilton GA, Thorneloe KS, McAlexander MA, Ingber DE.

Source

Wyss Institute for Biologically Inspired Engineering at Harvard University, Boston, MA 02115, USA.

Abstract

Preclinical drug development studies currently rely on costly and time-consuming animal testing because existing cell culture models fail to recapitulate complex, organ-level disease processes in humans. We provide the proof of principle for using a biomimetic microdevice that reconstitutes organ-level lung functions to create a human disease model-on-a-chip that mimics pulmonary edema. The microfluidic device, which reconstitutes the alveolar-capillary interface of the human lung, consists of channels lined by closely apposed layers of human pulmonary epithelial and endothelial cells that experience air and fluid flow, as well as cyclic mechanical strain to mimic normal breathing motions. This device was used to reproduce drug toxicity-induced pulmonary edema observed in human cancer patients treated with interleukin-2 (IL-2) at similar doses and over the same time frame. Studies using this on-chip disease model revealed that mechanical forces associated with physiological breathing motions play a crucial role in the development of increased vascular leakage that leads to pulmonary edema, and that circulating immune cells are not required for the development of this disease. These studies also led to identification of potential new therapeutics, including angiopoietin-1 (Ang-1) and a new transient receptor potential vanilloid 4 (TRPV4) ion channel inhibitor (GSK2193874), which might prevent this life-threatening toxicity of IL-2 in the future.

Science Translocational Medicine

An Orally Active TRPV4 Channel Blocker Prevents and Resolves Pulmonary Edema Induced by Heart Failure.

Nov 2012

Thorneloe KS, Cheung M, Bao W, Alsaid H, Lenhard S, Jian MY, Costell M, Maniscalco-Hauk K, Krawiec JA, Olzinski A,Gordon E, Lozinskaya I, Elefante L, Qin P, Matasic DS, James C, Tunstead J, Donovan B, Kallal L, Waszkiewicz A, Vaidya K, Davenport EA, Larkin J, Burgert M, Casillas LN, Marquis RW, Ye G, Eidam HS, Goodman KB, Toomey JR, Roethke TJ,Jucker BM, Schnackenberg CG, Townsley MI, Lepore JJ, Willette RN.

Source

Heart Failure Discovery Performance Unit, Metabolic Pathways and Cardiovascular Therapy Area Unit, GlaxoSmithKline, 709 Swedeland Road, King of Prussia, PA 19406, USA.

Abstract

Pulmonary edema resulting from high pulmonary venous pressure (PVP) is a major cause of morbidity and mortality in heart failure (HF) patients, but current treatment options demonstrate substantial limitations. Recent evidence from rodent lungs suggests that PVP-induced edema is driven by activation of pulmonary capillary endothelial transient receptor potential vanilloid 4 (TRPV4) channels. To examine the therapeutic potential of this mechanism, we evaluated TRPV4 expression in human congestive HF lungs and developed small-molecule TRPV4 channel blockers for testing in animal models of HF. TRPV4 immunolabeling of human lung sections demonstrated expression of TRPV4 in the pulmonary vasculature that was enhanced in sections from HF patients compared to controls. GSK2193874 was identified as a selective, orally active TRPV4 blocker that inhibits Ca(2+) influx through recombinant TRPV4 channels and native endothelial TRPV4 currents. In isolated rodent and canine lungs, TRPV4 blockade prevented the increased vascular permeability and resultant pulmonary edemaassociated with elevated PVP. 

Furthermore, in both acute and chronic HF models, GSK2193874 pretreatment inhibited the formation of pulmonary edema and enhanced arterial oxygenation. Finally, GSK2193874 treatment resolved pulmonary edemaalready established by myocardial infarction in mice. These findings identify a crucial role for TRPV4 in the formation of HF-induced pulmonary edema and suggest that TRPV4 blockade is a potential therapeutic strategy for HF patients.

PubMed-Science Translocational Medicine

Cystoid Macular Edema Global Clinical Trials Review

Cystoid Macular Edema Global Clinical Trials Review, H2, 2012 New Report

Naperville, IL -- (SBWIRE) -- 11/02/2012 -- Cystoid Macular Edema Global Clinical Trials Review, H2, 2012 provides data on the Cystoid Macular Edema clinical trial scenario. This report provides elemental information and data relating to the clinical trials on Cystoid Macular Edema. It includes an overview of the trial numbers and their recruitment status as per the site of trial conduction across the globe. The databook offers a preliminary coverage of disease clinical trials by their phase, trial status, prominence of the sponsors and also provides briefing pertaining to the number of trials for the key drugs for treating Cystoid Macular Edema. This report is built using data and information sourced from proprietary databases, primary and secondary research and in-house analysis by GlobalData's team of industry experts.

Scope

- Data on the number of clinical trials conducted in North America, South and Central America, Europe, Middle-East and Africa and Asia-pacific and top five national contributions in each, along with the clinical trial scenario in BRIC nations

- Clinical trial (complete and in progress) data by phase, trial status, subjects recruited and sponsor type

- Listings of discontinued trials (suspended, withdrawn and terminated)

Reasons to buy

- Understand the dynamics of a particular indication in a condensed manner
- Abridged view of the performance of the trials in terms of their status, recruitment, location, sponsor type and many more
- Obtain discontinued trial listing for trials across the globe
- Espy the commercial landscape of the major Universities / Institutes / Hospitals or Companies

SB Wire

Graft rejection more likely in eyes with corneal edema

Graft rejection more likely in eyes with corneal edema

There was a lower graft rejection rate in eyes with Fuchs’ dystrophy than in eyes with pseudophakic oraphakic corneal edema, a study found.

In addition, female recipients experienced a higher rejection rate than male recipients, while donor age did not play a role in rejection.

 The analysis used data from the prospective Cornea Donor Study to determine the effect of donor and recipient factors on corneal allograft rejection. The study examined 1,090 patients who underwent penetrating keratoplasty, mostly for Fuchs’ dystrophy or pseudophakic corneal edema. Patients were followed postoperatively for up to 5 years.

The 369 eyes with corneal edema were more likely to experience a rejection event than the 676 eyes with Fuchs’ dystrophy  Female patients had a higher likelihood of rejection compared with male patients. The age of the donors, which ranged from 10 years to 75 years, did not correlate with rejection.

The researchers suggested the need for further studies to better understand whether graft rejection is affected by anti-inflammatory postoperative treatment.

Healio

Negative pressure pulmonary edema with laryngeal mask airway use: Recognition, pathophysiology and treatment modalities

Negative pressure pulmonary edema with laryngeal mask airway use: Recognition, pathophysiology and treatment modalities

2012
Rashmi Vandse¹, Deven S Kothari¹, Ravi S Tripathi¹, Luis Lopez¹, Stanislaw P A Stawicki², Thomas J Papadimos^1
1 Department of Anesthesiology, The Ohio State University Medical Center, Columbus, Ohio, USA
² Department of Surgery, The Ohio State University Medical Center, Columbus, Ohio, USA

Correspondence Address:
Thomas J Papadimos
Department of Anesthesiology, Room N431, 410 West Tenth Avenue, Columbus, Ohio 43210
USA

Negative pressure pulmonary edema (NPPE) following the use of the laryngeal mask airway (LMA) is an uncommon and under-reported event. We present a case of a 58-year-old male, who developed NPPE following LMA use. After biting vigorously on his LMA, the patient developed stridor upon emergence, with concurrent appearance of blood-tinged, frothy sputum and pulmonary edema. He subsequently required three days of mechanical ventilation. After discontinuation of mechanical ventilation the patient continued to require additional pulmonary support using continuous positive airway pressure, with a full facemask, to correct the persistent hypoxemia. His roentgenographic findings demonstrated an accelerated improvement with judicious administration of intravenous furosemide.

International Jounral of Critical Illness & Injury Science

Neurogenic pulmonary edema following intracranial coil embolization for subarachnoid hemorrhage -A case report.

Oct 2012

Kim JE, Park JH, Lee SH, Lee Y.

Source

Department of Anesthesiology and Pain Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea.

Abstract

Neurogenic pulmonary edema (NPE) is a well-known complication of acute central neurologic injury, particularly aneurysmal subarachnoid hemorrhage. Both increased intracranial pressure and severe over-activation of the sympathetic nervous system seem to be pathogenetic for the onset of NPE. Although intracranial endovascular therapy is minimally invasive, it may affect brain stem regions and result in sympathetic activation. We now report the case of a 70-year-old woman who suddenly developed pulmonary edema during coil embolization of a ruptured aneurysm. During the intervention, oxygen saturation declined suddenly and a chest radiograph revealed pulmonary edema. The delayed appearance of NPE in this patient implies a risk for sympathetically mediated NPE during endovascular therapy.

PubMed