Testicular infarction secondary to protein S deficiency: a case report

BACKGROUND: Protein S deficiency is an inherited cause of thrombophilia. We present the second reported case in the literature of a man developing testicular infarction secondary to protein S deficiency. CASE PRESENTATION: A 63 year old man presented with sudden onset of pain in his left hemi-scrotum. Despite oral warfarin therapy the plasma INR was only 1.4 at presentation. Doppler ultrasound scan of the scrotum confirmed absent blood flow to the left testis with increased echogenicity. Orchidectomy was performed to remove the necrotic testis. Post-operatively the patient did well and was referred to the Regional Haemophilia and Thrombosis Centre for further management. CONCLUSION: The case highlights a rare but potential complication of protein S deficiency and demonstrates the importance of adequate anticoagulation in these patients

Pseudo-aneurysm of the anterior tibial artery, a rare cause of ankle swelling following a sports injury

BACKGROUND: Ankle pain and swelling following sports injuries are common presenting complaints to the accident and emergency department. Frequently these are diagnosed as musculoskeletal injuries, even when no definitive cause is found. Vascular injuries following trauma are uncommon and are an extremely rare cause of ankle swelling and pain. These injuries may however be limb threatening and are important to diagnose early, in order that appropriate treatment can be delivered. We highlight the steps to diagnosis of these injuries, and methods of managing these injuries. It is important for clinicians to be aware of the potential for this injury in patients with seemingly innocuous trauma from sports injuries, who have significant ankle pain and swelling. CASE PRESENTATION: A young, professional sportsman presented with a swollen, painful ankle after an innocuous hyper-plantar flexion injury whilst playing football, which was initially diagnosed as a ligamentous injury after no bony injury was revealed on X-Ray. He returned 2 days later with a large ulcer at the lateral malleolus and further investigation by duplex ultrasound and transfemoral arteriogram revealed a Pseudo-Aneurysm of the Anterior Tibial Artery. This was initially managed with percutaneous injection of thrombin, and later open surgery to ligate the feeding vessel. The patient recovered fully and was able to return to recreational sport. CONCLUSION: Vascular injuries remain a rare cause of ankle pain and swelling following sports injuries, however it is important to consider these injuries when no definite musculo-skeletal cause is found. Ultrasound duplex and Transfemoral arteriogram are appropriate, sensitive modalities for investigation, and may allow novel treatment to be directed percutaneously. Early diagnosis and intervention are essential for the successful outcome in these patients

Perforated carcinoma of the caecum presenting as necrotising fasciitis of the abdominal wall, the key to early diagnosis and management

BACKGROUND: Necrotising Fasciitis is a life threatening soft tissue infection which requires aggressive, early surgical management. CASE PRESENTATION: We present a rare case of a retroperitoneal perforation of a carcinoma of the caecum presenting as a necrotising fasciitis of the anterior abdominal wall. CONCLUSION: This case highlights the importance of early aggressive debridement to healthy tissue limits, the consideration of a rare underlying cause, and the scope for plastic surgical reconstruction in order that aggressive initial surgery can be adequately performed

Intralipid therapy and adverse reproductive outcome: is there any evidence?

Controversy exists regarding the benefits of intravenous intralipid therapy in patients with a poor reproductive history. It is frequently reported that there is no evidence to support the effectiveness, utility or safety for this treatment. While individual studies may be perceived as weak, a systematic review and meta-analysis were performed to determine if there is any advantage to patients. PubMed, Embase and Scopus searches were performed with the target populations being either recurrent pregnancy loss (RPL), or recurrent implantation failure (RIF) undergoing assisted reproductive technology (ART) and receiving intralipid infusions. These cohorts were compared with either placebo, no intervention or alternative treatments. The most relevant outcome measures were considered to be clinical pregnancy rate (CPR), live birth rate (LBR), implantation rate (IR) and miscarriage rate (MR). Twelve studies encompassing 2676 participants met the criteria for selection and were included and reviewed. Treatment of the target population with intralipid led to an improvement in IR (Odds Ratio (OR): 2.97, 2.05–4.29), pregnancy rate (OR: 1.64, 1.31–2.04), and LBR (OR: 2.36, 1.75–3.17), with a reduction in MR (OR: 0.2, 0.14–0.30). Although intravenous intralipid is not recommended as a routine treatment for recurrent miscarriage or implantation failure, there is enough data to suggest consideration in selected patients where routine testing is unremarkable, standard treatments have failed and immunological risk factors are present. The presence of abnormal uterine natural killer (uNK) cells needs more study as a target marker to determine those who could benefit. Lay summary There is controversy regarding the benefits and efficacy of intravenous intralipid therapy in patients with a poor reproductive history. It is frequently reported that there is no credible evidence to support their use. A situation we frequently face as medical professionals is patients asking us to consider immune therapy (such as intralipid) for reproductive failure where good quality embryos have been used. Intralipid infusions have been reported to improve pregnancy rates with IVF, and reduce the miscarriage risk in selected patient groups, but study results are not universally accepted. We have performed a detailed review and analysis of the literature to determine if there is any benefit to this immune treatment in specific patient groups. Our paper identified and analyzed 12 studies, finding that treatment with intravenous intralipid leads to an improvement in implantation, pregnancy and live birth rates, with a decrease in miscarriage rate. This study shows that there is evidence to suggest consideration of intralipid in certain patients where standard treatments have failed. </jats:sec

Comparing surface immune markers in successful and non-viable ART pregnancies on the day of hCG measurement: a prospective pilot study

Abstract: Blood lymphocyte reference ranges in non-pregnant females are established, but changes in pregnancy are less well understood. The early identification of immunological markers that could suggest an increased risk of early pregnancy loss may allow for timely intervention to improve outcomes. A lymphocytic immunophenotype provides a broad assessment of important immune parameters and potential indicators, which may be of relevance to pregnancy outcome. Comparison of immunophenotype results on the day of a positive hCG after embryo transfer between successful and failed pregnancies allows for this assessment. Baseline non-pregnant lymphocyte percentage and cell/µL profiles were established with a comprehensive panel on 93 age-matched male factor controls. Sixty-five in-vitro fertilisation (IVF) patients had an immunophenotype assessment on the day of a positive hCG, followed by further hCG tests and ultrasound monitoring as required to ultimately evaluate success (live birth) or failure (miscarriage). Thirty-one pregnancies were viable, leading to a live birth, while 34 ended in miscarriage. Total CD56, pNK, NKT, CD4 and CD8 levels were equivalent between all groups. Regardless of the outcome, B lymphocytes increased in pregnancy compared to controls. Of interest, in the later miscarriage cohort, pNK-specific CD69 was reduced (1.6 vs 5.4%, P = 0.02), while CD57+ cells were increased (45.4 vs 38.9%, P = 0.025). Corresponding changes were observed in cell/µL concentrations. Low level CD69 activation and elevated CD56dim and CD57+ NK cells were identified as markers that could potentially identify a pregnancy at risk of miscarriage, with further study needed to explore whether these changes represent cause or effect.Lay summary: Unexplained infertility remains a difficult issue for patients and physicians alike, but despite recent diagnostic strides and innovative methods, there are no clear solutions on the horizon. Pregnancies can still occur in these challenging populations, either spontaneously or by interventions such as IVF. The early identification of various immune markers by blood sampling that may correlate with the subsequent outcome could be beneficial in identifying pregnancies at increased risk of miscarriage and perhaps allowing for timely and effective interventions.</p