Current Case Series Research Articles

Transthoracic Real‐Time Three‐Dimensional Echocardiography in the Diagnosis and Description of Noncompaction of Ventricular Myocardium

Numerous modalities have been used to diagnose and characterize noncompaction of ventricular myocardium (NCVM) including magnetic resonance imaging, two-dimensional echocardiography (2DE), contrast-enhanced 2DE, and angiography. The current case series examines the use of real-time three-dimensional transthoracic echocardiography (RT-3DE) in four such cases of NCVM. From December 2003 to March 2004, we performed RT-3DE using a Philips Sonos 7500 echocardiographic scanner equipped with a 2-4 MHz 3D matrix array transthoracic probe, to evaluate four patients with NCVM. The real-time 3D transthoracic probe allows for dataset acquisition in an ultrasound wedge, which can be manipulated instantaneously. In addition, complete 3D volume rendering is acquired, allowing for volumetric imaging. The age range of the patients was 2 months to 42 years. One patient had the codiagnoses of coarctation of the aorta and bicuspid aortic valve. In all four patients, RT-3DE enabled diagnosis and provided detailed characterization of the affected myocardium. Entire trabecular projections and intertrabecular recesses were easily visualized simultaneously, and endocardial borders were clearly demarcated. Wall motion abnormalities of the affected myocardium were clearly visualized. The compacted and noncompacted portions of the myocardium could be differentiated well. Our study provides preliminary data highlighting the utility and feasibility of RT-3DE in the clinical characterization of NCVM. The complex 3D nature of this disorder and the endocardial hypertrabeculation were more readily visualized than with 2DE.

Successful treatment of cervical stenosis with hysteroscopic canalization before embryo transfer in patients undergoing IVF: A case series

The course of the transfer catheter through the cervical canal is one of the most important issues for a successful embryo transfer (ET) during in vitro fertilization (IVF) cycles. Technically difficult ETs due to cervical stenosis are associated with reduced chance of pregnancy after assisted reproductive procedures. In the current case series, we report on three patients with cervical stenosis who underwent IVF-ET cycles. These three patients, in whom ET was classified as “difficult,” failed to conceive with previous ET attempts. An intervention to create a cervical tract was performed with operative hysteroscopy under general anesthesia before transcervical ET. After the hysteroscopic shaving procedure, we observed quite an improvement in access to the endometrial cavity during ET procedure. These patients had significantly easier ET procedures compared with previous attempts and achieved clinical pregnancies. Hysteroscopic revision of the cervical canal results in easier ET and improves pregnancy rates in patients with cervical stenosis and histories of difficult ET.

Diagnosis of conjoined twins before 16 weeks' gestation: the 4‐year experience of one medical center

Descriptions of the rarely occurring condition of conjoined twins are sparse. We report a case series of four conjoined twin pregnancies diagnosed up to 16 weeks of gestation. One was in a quadruplet in vitro fertilization intracytoplasmic sperm injection (IVF-ICSI) pregnancy of cryopreserved embryos and the others were spontaneous conceptions. Three were thoracopagus and had increased nuchal translucency (NT) or big nuchal cysts and one was omphalopagus. All the three affected twin pregnancies were terminated by cervical dilatation and uterine evacuation. In another case, selective fetal reduction, shortly followed by embryo reduction to singleton was performed. Histological analysis of one case revealed dissimilar gender. No further cytogenetic or molecular studies were conducted by parental request. The pathophysiology of increased NT, subcutaneous edema or nuchal cysts in thoracopagus conjoined twins, the possible embryonic events that may lead to dizygotic conjoined twins and the clinical management of conjoined twins in sets of multiple gestation are discussed. The current case series illustrates that there are many problems related to this rare, but intriguing, accident of nature that still need to be overcome.

Synovial Chondromatosis of the Temporomandibular Joint: Report and Analysis of Eleven Cases

Synovial chondromatosis (SC) is a benign monoarticular condition that is uncommon in the temporomandibular joint (TMJ). The purpose of this article is to present 11 additional cases of SC of the TMJ and discuss newer modes of imaging, diagnostic approaches, treatment options, and follow-up data. Medical records of 11 patients with SC treated within our department from 1991 to the present were reviewed. Demographic data, etiology, clinical presentation, diagnostic evaluation, treatment, and follow-up information were collected. Previously reported cases (both TMJ and others) from 1988 to present were identified for comparison and the literature reviewed. There were 8 females and 3 males with an average age of 54 years. Pain and preauricular swelling were the most common presenting clinical complaints. Etiologic factors (parafunction, inflammatory joint disease) were found in 7 of 11 cases. Plain radiographs showed joint calcifications in only 2 of 11 cases. Computed tomography identified calcifications in 3 of 6. Magnetic resonance imaging clearly demonstrated the mass and its extension in 10 of 10 cases. Fine needle aspiration was diagnostic in 4 of 9. All patients were treated with an open arthrotomy. Meniscectomy was required in 7 of 11. Average follow-up was 5.2 years with no recurrences reported. The current case series of SC shows a female predilection with age and presenting complaints similar to those previously reported. A traumatic etiology was not identified, although a weak association is reported in the literature. The superiority of magnetic resonance imaging for both diagnosis and evaluation of extension of disease is shown. In a subset of cases, fine needle aspiration is useful for confirming the clinicoradiographic impression. Complete removal of involved tissue is associated with an excellent prognosis.

Risk factors for emergency peripartum cesarean hysterectomy

Emergency peripartum hysterectomy was performed for management of intractable obstetric hemorrhage that was unresponsive to other treatment. The procedure is often associated with a relatively high morbidity and mortality especially when performed under emergency life-threatening situation. Most current case series reported the incidence of 1—3 per 1000 deliveries. This study was performed as case control study to evaluate data regarding the risk factors of emergency peripartum hysterectomy. The study design was a case-control study of pregnant women who delivered at Ramathibodi Hospital Mahidol University Bangkok Thailand during January 1997 to December 2002. Inclusion criteria was a pregnant woman who underwent emergency peripartum hysterectomy within 24 h after delivery which delivered a single infant or twins and gestational age = 28 weeks. (excerpt)

Treatment of Legionnaires??? Disease

Legionnaires' disease is pneumonia, usually caused by Legionella pneumophila, which can range in severity from mild to quite severe. While it is commonly acquired in the community, it can just as easily be acquired nosocomially from water sources that have not been appropriately decontaminated. While historically initial treatment was always with erythromycin, current case series and treatment recommendations suggest that outpatients receive immediate treatment with one of the following antibacterials: azithromycin, erythromycin, clarithromycin, telithromycin, doxycycline or an extended-spectrum fluoroquinolone. If the symptoms are severe enough to warrant hospitalisation then the patient should receive treatment with parenteral azithromycin or extended-spectrum fluoroquinolones followed by step-down to oral formulations to complete the regimens. While a shorter course of 7-10 days for more severe infections may be possible for intravenous/oral azithromycin, other antibacterials should be administered for a total of 10-21 days and started as soon as possible upon presentation to optimise outcomes.

Factor V inhibitors

Acquired deficiencies of, or inhibitors to, factor V are considered rare events. We report a series of 14 acquired factor V deficiencies, 10 of which were confirmed to have inhibitors to factor V, as identified within Australia in the past 5 years following a multi-laboratory investigation. The initial index case seen by one laboratory was followed within 4 months by a separate similar case. This prompted local contact with colleagues (n = 20) working in other haemostasis referral laboratories to identify the current case series. In total, nearly one-half of all haemostasis referral laboratories contacted had seen a case within the past 5 years. Clinical features and the apparent associated risk of bleeding complications generally varied, as did laboratory findings and the likely causal event. There were three females and 11 males. Age ranged from 44 to 95 years (median, 81 years). The level of inhibitor ranged from undetectable to over 250 Bethesda units. The probable cause leading to development of the inhibitors ranged from exposure to bovine thrombin, exposure to antibiotics, surgery and malignancy. Of additional interest was the apparent association of anti-phospholipid antibodies in many of the cases. For example, in the two similar index cases, with factor V inhibitor titres > 200 Bethesda units, high levels of anti-cardiolipin antibodies (> 70 GPL units) were also detected. Although less clear because of inhibitor interference, many of the cases also showed evident co-associated lupus anticoagulant activity. In conclusion, we report a series of factor V inhibitors recently identified within our geographic region that would represent an annual incidence of around 0.29 cases per million Australians. Although considered a rare finding, there is a high likelihood that most haemostasis referral laboratories will see a case every five or so years.

Radiation dose to otologic structures during head and neck cancer radiation therapy.

Otologic structures are often contained within head and neck cancer radiation treatment ports. The dosimetry to otologic structures has not been routinely analyzed and radiation treatment planning does not currently attempt to specifically avoid the inner ear structures when dosimetry is calculated. Recent studies demonstrate that up to 30% of patients experience sensorineural hearing loss on multimodality therapy with cisplatin and radiation. In the current case series, radiation dosimetry to otologic structures was calculated from computed tomogram treatment plans on patients. Fifteen nasopharyngeal, oral cavity, oropharyngeal, and hypopharyngeal cancer patients were analyzed. Between 8% and 102% of the total dose is delivered to the petrous bone/cochlea, with 4 of 15 patients getting more than 50% of the dose to at least one cochlea The mastoid air cells received between 3% and 75% of the total dose, with higher doses being delivered to patients with bulky high neck metastases or nasopharyngeal tumors. The eustachian tubes received between 20% and 102% of the total dose, with 10 of 15 patients receiving more than 50% of the dose to this anatomic site. We conclude that the cochlea and eustachian tubes receive significant radiation during treatment, particularly in nasopharyngeal cancer patients. Careful design of radiation treatment ports may allow for the reduction of radiation to hearing structures.

Digital Adrenaline Injection Injuries: A Case Series and Review

MW ElMaraghy, AW ElMaraghy, HB Evans. Digital adrenaline injection injuries: A case series and review. Can J Plast Surg 1998;6(4):196-200. Accidental digital adrenaline autoinjection is an uncommon injury, with only a few case reports existing in the literature. A six-case series of digital EpiPen injection injuries, a review of the literature and treatment protocols with their pathophysiological and pharmacological basis are presented. Oral nifedipine, topical 2% nitroglycerine paste, digital block using lidocaine and warm water immersion are ineffective in acutely reversing digital ischemia. Clinical and experimental evidence demonstrating spontaneous resolution of digital vasospasm may support the expectant management of these injuries. If medical intervention is undertaken, safe and rapid reversal of adrenaline-induced digital ischemia can be achieved with local and/or regional infiltration of phentolamine mesylate. Based on a review of the current case series and the literature, the authors’ preferred method of treatment includes simultaneous digital block with a mixture of phentolamine and 2% lidocaine and local injection of phentolamine at the puncture site. Initially, the authors suggest using 5 mg phentolamine mesylate in 2 mL of 2% lidocaine, and injecting 1 mL (2.5 mg) of the solution into the ulnar and 1 mL (2.5 mg) into the radial aspect of the base of the involved digit, followed by injecting the injury site with a 5 mg/mL solution of phentolamine.

Cardiac Papillary Fibroelastoma: A Treatable Cause of Transient Ischemic Attack and Ischemic Stroke Detected by Transesophageal Echocardiography

Transesophageal echocardiography (TEE) is used frequently in patients with cerebrovascular ischemia. On TEE, a typical appearance of a cardiac fibroelastoma is that of a pedunculated, mobile mass attached to a leaflet of a valve. Surgical excision of the lesion may lead to resolution of the symptoms and prevent further cerebrovascular ischemic events; valve replacement is seldom necessary. Herein we describe three patients with cerebral or ocular ischemia in whom histologic study confirmed a cardiac papillary fibroelastoma after initial detection by TEE. Cardiac papillary fibroelastomas should be considered in the differential diagnosis of transient ischemic attack and stroke, even in cases of recurrent events in the same vascular distribution. Although the use of echocardiography in the evaluation of stroke and transient ischemic attack is controversial, TEE must be considered in patients in whom the cause of cerebrovascular ischemia is unclear after noninvasive neurovascular studies or transthoracic echocardiography, even if the patient's cardiac history and the findings on physical examination are normal.