Colosplenic fistula diagnosis and management: a case series and review of literature
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ABSTRACT:
SUBMITTER: Hernandez Dominguez O
PROVIDER: S-EPMC11020030 | biostudies-literature | 2024 Feb
REPOSITORIES: biostudies-literature
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Project description:Fetal intestinal volvulus is a rare condition that can lead to hemorrhage, bowel necrosis, and urgent surgical treatment after birth. Thus, prompt diagnosis and treatment are essential to avoiding fetal or neonatal demise. Prenatal ultrasound is a keystone tool in the diagnostic course. However, sonographic findings tend to be non-specific, with limited understanding of the pathophysiology behind their atypical presentation. With a literature review and a case series, we aim to optimize the antenatal diagnosis and management of this rare but life-threatening condition. Six cases from our institution were retrospectively analyzed over 12 years. A literature review was conducted until December 2022. A total of 300 articles matched the keyword "Fetal volvulus", and 52 studies were eligible for the review. Our 6 cases are added to the 107 cases reported in the literature of fetal intestinal volvulus with antenatal ultrasound assessment and without associated gastroschisis or omphalocele. Several prenatal symptoms and ultrasound markers, even if not specific, were more frequently reported. Different experiences of management were described regarding follow-up, the timing of delivery, the mode of delivery, and surgery outcomes. This paper highlights the importance of suspecting and assessing fetal volvulus at routine ultrasound scans, describing the most frequent antenatal presentations and management in order to improve fetal and neonatal outcomes.
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Project description:BackgroundUterine intravenous leiomyomatosis (IVL), a rare type of uterine leiomyoma, is defined by the intravascular proliferation of a histologically benign smooth muscle cell tumor. Pelvic arteriovenous fistula (AVF) is a rare vascular malformation that is most commonly congenital, post-traumatic, or iatrogenic. The link between leiomyomatosis and AVF has received little attention in the medical literature.ResultsWe provide a case series of seven patients, four of whom were from our center, who had IVL complicated by a pelvic AVF. The symptoms of right heart failure were noted as swelling in the abdomen and two legs as well as a significant amount of ascites. Coil embolization of AVFs may be beneficial in minimizing bleeding during IVL surgery. A review of all accessible literature published on IVLs from 2000 to 2020 was conducted, and data were retrieved from 78 papers totaling 262 cases. Complications and recurrence were associated with pelvic mass excision and intravascular remnant tumor, respectively.ConclusionIntravenous leiomyomatosis combined with AVF aggravates congestion symptoms of surrounding organs. It is worth noting the uncommon combination of AVF and IVL, stressing the importance of a thorough assessment and surgical approach in IVL treatment.
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Project description:Dual and H-type tracheoesophageal fistulae can present major diagnostic and management difficulties. A methodological approach with flexible bronchoscopy and a guide wire cannulation technique was used to diagnose, localize, and aid operative surgical management in five children with dual and H-type tracheoesophageal fistulae. All children had successful outcomes.
| S-EPMC7495836 | biostudies-literature
Project description:We report an antenatally diagnosed large distal coronary artery fistula (CAF) arising from an aneurysmal dilation right coronary artery (RCA) and draining in to the right ventricle (RV) just below the septal leaflet of tricuspid valve posteriorly. A postnatal echocardiogram confirmed the diagnosis. On the second day of life, a percutaneous partial closure of the fistula was performed by placing a Flipper coil (Cook Medical, Bloomington, IN) in the RCA just proximal to the drainage site in the cardiac catheterization laboratory. Follow-up echocardiogram on the day following the procedure showed improved forward flow in the descending aorta with decreased RV size. Our case report suggests that antenatal diagnosis of a CAF may aid in early intervention. Partial closure of the fistula in the cardiac catheterization laboratory is safe and effective.
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| S-EPMC10167448 | biostudies-literature
Project description:BackgroundDural arteriovenous fistula (dAVF) rarely presents with a syndrome of reversible parkinsonism and rapidly progressive dementia, which has been described in 19 patients to date. However, its presenting features, pathophysiology, and response to treatment have not been reviewed.MethodsWe report the clinical course and treatment of two novel patients with this syndrome.ResultsDespite severe presentations, both patients recovered fully after fistula obliteration.ConclusionsAtypical parkinsonism with early cognitive changes and other focal neurological signs, along with features such as pulsatile tinnitus or worsening headache, should raise the possibility of dAVF. When treated, this syndrome is nearly always reversible, highlighting the importance of early diagnosis.
| S-EPMC6174482 | biostudies-literature
Project description:IntroductionPancreaticopleural fistula (PPF) is a rare but serious complication of pancreatic disorders. As the clinical presentations of PPF are often deceptive, it can cause a delay in the timely diagnosis and proper treatment. PPF is extremely uncommon in pediatric patients, and diagnostic and management strategies for PPF among pediatric patients are scanty.Patient concernsA 12-year-old girl presented with cough and dyspnea owing to massive right-side pleural effusion confirmed by Chest X-ray. Biochemical examination of pleural effusion revealed a significant elevation of amylase level. Imaging modalities showed dilated pancreatic duct and fistulous tract connecting pancreatic duct and right thorax.DiagnosisChronic pancreatitis with PPF was diagnosed.InterventionsMedical therapy was initially attempted for 2 weeks. Endoscopic therapy with naso-pancreatic drainage tube placement was then performed without any complications after failed medical therapy.OutcomesThe patient has remained healthy and symptom-free during 2 years of follow-up.ConclusionWhen pediatric patients presented with recurrent pleural effusion with unknown etiology, PPF should be taken into consideration. Pleural effusion amylase level is the most important laboratory test and magnetic resonance cholangiopancreatography is recommended to visualize the fistula. Optimal management of PPF should be based on pancreatic duct morphology.
| S-EPMC7306390 | biostudies-literature
Project description:Bullous pemphigoid (BP) is a rare, life-threatening autoimmune blistering disease with pruritus and tension blisters/bullous as the main clinical manifestations. Glucocorticosteroids are the main therapeutic agents for it, but their efficacy is poor in some patients. Tofacitinib, a small molecule agent that inhibits JAK1/3, has shown incredible efficacy in a wide range of autoimmune diseases and maybe a new valuable treatment option for refractory BP. To report a case of refractory BP successfully treated with tofacitinib, then explore the underlying mechanism behind the treatment, and finally review similarities to other cases reported in the literature. Case report and literature review of published cases of successful BP treatment with JAK inhibitors. The case report describes a 73-year-old male with refractory BP that was successfully managed with the combination therapy of tofacitinib and low-dose glucocorticoids for 28 weeks. Immunohistochemistry and RNA sequencing were performed to analyze the underlying mechanism of tofacitinib therapy. A systematic literature search was conducted to identify other cases of treatment with JAK inhibitors. Throughout the 28-week treatment period, the patient experienced clinical, autoantibody and histologic resolution. Immunohistochemical analysis showed tofacitinib significantly decreased the pSTAT3 and pSTAT6 levels in the skin lesions of this patient. RNA sequencing and immunohistochemical testing of lesion samples from other BP patients identified activation of the JAK-STAT signaling pathway. Literature review revealed 17 previously reported cases of BP treated with four kinds of JAK inhibitors successfully, including tofacitinib (10), baricitinib (1), upadacitinib (3) and abrocitinib (3). Our findings support the potential of tofacitinib as a safe and effective treatment option for BP. Larger studies are underway to better understand this efficacy and safety.
2024-11-13 | GSE278926 | GEO
Project description:Breast cancer comprised at least 21.8% of the overall cancer among young adult (YA) women and became the leading cancer in this group in Japan, with 50% adolescent and YAs being diagnosed and 15-44-year-old women showing excellent 5-year survival. Surgical-chemoradiation therapy often results in excellent survivorship with an increased incidence of treatment-induced subfertility. Therefore, adding fertility preservation (FP) to the primary cancer treatment is necessary. Herein, we reported a series of cases of YA women with breast cancer who opted for FP, where their option was tailored accordingly. To date, the selection of oocytes, embryos and ovarian tissue is widely available as an FP treatment. PGT could reduce the risk of BRCA mutation transmission amongst BRCA carriers before pregnancy planning. Otherwise, gonadotropin-releasing hormone analog has no gonadoprotective effect and thus should not be considered as an FP option.
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Project description:BackgroundEncapsulating Peritoneal Sclerosis (EPS) is a rare phenomenon in paediatric patients with kidney failure treated with peritoneal dialysis (PD). This study highlights clinical challenges in the management of EPS, with particular emphasis on peri-operative considerations and surgical technique.MethodsRetrospective analysis of all paediatric patients with EPS treated at the Manchester Centre for Transplantation.ResultsFour patients were included with a median duration of 78 months on PD. All patients had recurrent peritonitis (> 3 episodes), and all had symptoms within three months of a change of dialysis modality from PD to haemodialysis or transplant. In Manchester, care was delivered by a multi-disciplinary team, including surgeons delivering the adult EPS surgical service with a particular focus on nutritional optimisation, sepsis control, and wound management. The surgery involved laparotomy, lavage, and enterolysis of the small bowel + / - stoma formation, depending on intra-abdominal contamination. Two patients had a formal stoma, which were reversed at three and six months, respectively. Two patients underwent primary closure of the abdomen, whereas two patients had re-look procedures at 48 h with secondary closure. One patient had a post-operative wound infection, which was managed medically. One patient's stoma became detached, leading to an intra-abdominal collection requiring re-laparotomy. The median length of stay was 25 days, and patients were discharged once enteral feeding was established. All patients remained free of recurrence with normal gut function and currently two out of four have functioning transplants.ConclusionsThis series demonstrates 100% survival and parenteral feed independence following EPS surgery. Post-operative morbidity was common; however, with individualised experience-based decision-making and relevant additional interventions, patients made full recoveries. Health and development post-surgery continued, allowing the potential for transplantation. A higher resolution version of the Graphical abstract is available as Supplementary information.
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