Uncommon Cause Research Articles

Abdominal Cocoon Syndrome in a Liver Transplant Patient: A Case Report and Review of the Literature

Introduction: Cocoon syndrome is an uncommon cause of intestinal obstruction caused by the formation of a fibrous band around small intestines, thought to be caused by chronic peritoneal inflammation and associated with several conditions such as liver cirrhosis and liver transplantation. Case Report: We present a case of a 72-year-old male patient who had liver transplantation three years ago for liver cirrhosis. He had an acute presentation of intestinal obstruction where a computed tomography (CT) scan showed dilated bowel loops and signs of intestinal ischemia, which warranted surgical laparotomy where the diagnosis of cocoon syndrome (sclerosing encapsulating peritonitis) was made and was then treated by adhesiolysis. No immediate or postoperative complications were documented and his life was back to normal. Conclusion: Therefore, an abdominal cocoon is a challenging diagnosis and should be considered in front of intestinal obstruction, especially in those with risk factors such as liver transplantation.

Parada cardiorrespiratória após infarto agudo do miocárdio por vasoespasmo coronariano. Relato de caso e revisão da literatura

Vasospastic angina is an uncommon cause of cardiac arrest and ventricular arrhythmias. However, survivors of these complications are at an increased risk of recurrence, despite normal ventricular function and optimized medical therapy. We describe a case of a 50-year-old former smoker who developed cardiorespiratory arrest secondary to severe coronary vasospasm.

Transverse colon volvulus presenting as bowel obstruction, atelectasis, and displacement of the right lobe of the liver into the left upper abdominal quadrant: a case report

BackgroundTransverse colon volvulus is an uncommon cause of intestinal obstruction. It is a surgical emergency that can lead to bowel infarction, peritonitis, and death.Case presentationWe report a case of transverse colon volvulus in a 35-year-old Congolese immigrant man who had a rare presentation with features of intestinal obstruction associated with right lung collapse and left mediastinal shift.ConclusionThis case is unusual because it presented with respiratory features that mimicked a pneumothorax in addition to features of intestinal obstruction. The use of point-of-care lung ultrasound was helpful in ruling out a pneumothorax, and this could help avoid situations such as unintentional chest drain insertions by other professionals who may encounter a similar case. Because transverse colon volvulus is rare, a high level of suspicion and awareness is required to make an accurate diagnosis.

Clinico-pathological profile of schistosomal appendicitis detected in surgically resected appendices: A retrospective study

BackgroundAppendicular schistosomiasis (AS) is an uncommon cause of acute appendicitis that can only be diagnosed histopathologically. There are few reports of AS and its histopathological profile is unclear in Egypt. Therefore, we aimed to evaluate the prevalence and clinicopathological, profile of AS among patients presented with acute appendicitis for proper management and to reduce the health burden of the disease. MethodsThis is a retrospective study performed at a university hospital comprising all patients with pathological confirmation of appendiceal Schistosomiasis after appendectomies between 2017 and 2021. All data were extracted from patient's files including age, sex, residence, main patient's complaint, Alvarado scores, and laboratory data. The histopathological specimens were reviewed by the histopathologist in this study and special stains were done when indicated. ResultsThe prevalence of AS was 1.07% (20/1870), all of them were adults or old age with a mean age of 39.25, 80% of cases were males. The most frequent clinical presentation in our patients was abdominal pain in the right iliac fossa (96%) with a mean duration of symptoms of 2.5 days. At the time of the operation, two patients had perforations, with no recorded mortality. Only 20% showed gross unusual foci, and lymphoplasmacytic infiltration was seen in 90% of cases. All patients were advised to postoperative praziquantel therapy. ConclusionHistopathological evaluation for schistosomiasis in all surgically removed appendix is recommended in endemic areas for middle and old ages and comprehensive postoperative follow-up with praziquantel therapy is required to ensure disease eradication and prevent further complications.

Solitary rectal ulcer syndrome – An uncommon cause of rectal bleeding in children

Background: Solitary rectal ulcer syndrome (SRUS) is a benign chronic disorder rarely found in children commonly presents with rectal bleeding, mucorrhea, straining during defecation, tenesmus, feeling of incomplete defecation, rectal prolapse and constipation. The pathogenesis is not clearly understood but several factors such as trauma, rectal prolapse, ischemia, behavioral disorders such as excessive straining during defecation and rectal manipulation, sexual abuse and disharmony of the pelvic floor muscles during defection may be involved. SRUS is diagnosed on the basis of clinical symptoms and endoscopic and histological findings. Various treatment protocol for SRUS have been described such as conservative management such as family reassurance, regulation of toilet habits, avoidance of straining, encouragement of a high-fiber diet, topical treatments with salicylate, sulfasalazine, steroids and sucralfate, and surgery. Methods: A comprehensive search is done using PubMed, Scopus, Google scholar and MEDLINE. Results: The result of this review will be published in a journal. Conclusion: This study discusses about the pathogenesis, clinical features, diagnosis and various treatment strategy of solitary rectal ulcer syndrome in children. Bangladesh Medical Res Counc Bull 2023; 49(1): 70-74

An uncommon cause of hypophosphatemic rickets: Answers.

An uncommon cause of hypophosphatemic rickets: Answers.

An uncommon cause of hypophosphatemic rickets: Questions.

An uncommon cause of hypophosphatemic rickets: Questions.

Surgical management of an atypical chronic fracture of the third trochanter of the femur

SummaryFractures of the third trochanter of the femur are an uncommon cause of hindlimb lameness in the horse. The diagnosis is easily made with ultrasonography. Reported prognosis is good after a period of rest and a progressive return to exercise. This case report describes a third trochanter fracture in a 5‐year‐old trotter mare, which is atypical in terms of both its clinical presentation and management. At the dynamic examination, a left hind lameness was only observed when harnessed at the track. Nuclear scintigraphic examination revealed a marked increase in radiopharmaceutical uptake on the third trochanter of the left femur. Radiographic and ultrasonographic findings confirmed an old complete fracture of the third trochanter of the left femur, with a displaced fragment. The mare was referred for surgical treatment in order to remove the fractured third trochanter. At the follow‐up examination, the mare was sound in hand and imaging revealed successful resection of the main fractured fragment. After a period of stall rest, the mare progressively resumed training and then racing 7.5 months after the surgery, performing at its expected level. Surgical treatment may be an option for chronic fractures of the third trochanter.

A Case of Epiploic Appendagitis after COVID-19

Acute epiploic appendagitis is an uncommon cause of abdominal pain resulting from appendageal ischemia caused by torsion or thrombosis of the draining vein. It is frequently misdiagnosed as acute appendicitis or diverticulitis. The coronavirus disease 2019 (COVID-19) pandemic has changed how this rare disease is diagnosed. There was a report of a young men diagnosed with COVID-19 and epiploic appendagitis as a rare cause of abdominal pain. In addition, a 50-year-old men was diagnosed with epiploic appendagitis during the treatment of COVID-19. This paper reports the case of a 53-year-old men who presented with right lower quadrant abdominal pain after COVID-19 and was diagnosed with acute epiploic appendagitis by computed tomography image findings. The thrombotic condition of COVID-19 may contribute to acute appendagitis, but more studies are needed to confirm this hypothesis.

Middle Turbinate Pyocele, an Uncommon Differential for Nasal Mass: A Case report.

Mucoceles are respiratory epithelium-lined cystic lesions found in the paranasal sinuses. Pyocele occurs when a mucocele becomes infected. Mucoceles develop as a result of obstruction of the normal sinus drainage tract with superadded infection. Inflammatory mediators are secreted, which lead to bone resorption and expansion of the mucocele. Concha bullosa is one of the most common anatomical variations in the nasal cavity. It refers to an air-filled cavity within the middle turbinate. Obstruction of the concha bullosa can rarely lead to the formation of a mucocele which may be secondarily infected forming a mucopyocele. The mucocele of the middle turbinate may present as an uncommon cause of nasal obstruction, headache, and other nasal or ocular symptoms, which, if correctly diagnosed, can be completely reversed by endoscopic surgery.

Primary Ovarian Insufficiency and Hormone Replacement Therapy: A Review Article

An uncommon but significant cause of ovarian hormone deficit and infertility in women is Primary Ovarian Insufficiency (POI). In addition to leading to infertility, POI is linked to many health problems, including troublesome menopausal symptoms, lower bone density and an increased risk of fractures, early cardiovascular disease development, a potential early reduction in cognition, and dry eye syndrome. It is essential to provide women with POI with the proper Hormone Replacement Treatment (HRT) to replace premenopausal levels of ovarian sex steroids and reduce the health concerns that come with them. Using HRT formulations that nearly resemble natural ovarian hormone production is advised in this evaluation. HRT should be continued until the typical age of natural menopause, 50 years old. Keywords: Primary Ovarian Insufficiency, Hormone Replacement Treatment, Menopausal Symptoms

Burkholderia cepacia infection associated with sickle cell disease: An uncommon entity.

Burkholderia, a multidrug-resistant Gram-negative bacteria, is an uncommon cause of infection mostly in immunocompromised patients with a clinical profile very similar to tuberculosis. The most common conditions associated with this organism are cystic fibrosis and chronic granulomatous diseases. Bacteremia with it occurs in patients who are chronically ill and associated with significant morbidity and mortality. We are reporting here a case of perisplenic intra-abdominal abscess caused by Burkholderia cepacia in a patient with sickle cell disease (SCD).

An uncommon cause of severe polycystic ovary syndrome (PCOS)

An uncommon cause of severe polycystic ovary syndrome (PCOS)

Postcoital bleeding

Postcoital bleeding (PCB) is defined as any bleeding or spotting unrelated to menstruation that occurs during or after sexual intercourse. It is a relatively common complaint presenting to general practice, and can sometimes prove challenging to diagnose and manage, due to the number of conditions that can cause PCB. It can cause a lot of distress to the patients, as many will associate such bleeding to a malignant cause. At the same time, some patients will take it lightly and present late. There are no current national guidelines in the UK for PCB, which may contribute to inconsistencies in investigation and management. There are many common and uncommon causes of PCB. This article will explore these with the help of a clinical scenario. An aid to diagnosis and management will be discussed.

A235 “WHAT’S SPRUE WITH YOU?”: OLMESARTAN-INDUCED ENTEROPATHY AS AN UNCOMMON MIMICKER OF CELIAC DISEASE

Abstract Background Olmesartan-induced enteropathy is an uncommon mimicker of celiac disease. Symptoms include diarrhea and weight loss and pathology shows villous atrophy, intraepithelial lymphocytes, and subepithelial collagen deposition. In contrast to celiac disease, tissue transglutaminase (TTG) is not elevated and gluten avoidance does not induce clinical or histologic improvement. Both clinical and histologic findings are reversible with cessation of pharmacologic therapy. Purpose To raise clinician awareness of an uncommon but reversible cause of enteropathy. Method A 67-year old man presented with 3 months of progressive diarrhea and 35lb weight loss on a background of hypertension, dyslipidemia, and type 2 diabetes. His medications included olmesartan, amlodipine, metformin, aspirin, and atorvastatin, none of which were new. Infectious stool studies, serum and fecal inflammatory markers, imaging, TTG and IgA were normal. Panendoscopy revealed duodenitis with biopsies consistent with active enteritis, intraepithelial lymphocytosis and villous shortening suggestive of olmesartan-induced enteropathy given negative TTG. Olmesartan was discontinued. Result(s) His symptoms completely resolved over several weeks after discontinuation. Conclusion(s) Olmesartan is an uncommon but reversible mimicker of celiac disease. It was first described in 2012 in a case series of 22 patients, all of whom had negative celiac serology and demonstrated histologic and clinical improvement with cessation of olmesartan. Time from medication initiation to onset of symptoms ranged from 6 months to 7 years, with a mean of 3.1 years. While case reports exist of angiotensin-receptor blockers as causative agents, the most common is olmesartan, implicated in 94% of ARB-induced enteropathy cases in a recent systematic review. The exact mechanism of injury is unknown. The delay in onset of symptoms suggests a cell-mediated immunity phenomenon. An immune-mediated reaction is also supported by the high prevalence of HLA-DQ2/DQ8 haplotypes (71%) among affected individuals vs. 30-40% in the general population. Olmesartan may be implicated more strongly than other ARBs due to its higher affinity for AT1 receptors; theoretically leaving unsaturated AT2 receptors to bind angiotensin. AT2 receptors are known to induce cellular apoptosis, and unopposed cellular death could theoretically lead to villous atrophy. The overall risk of developing enteropathy on olmesartan is rare, but not well quantified. This is likely due to both underrecognition, and variability in study designs leading to inability to perform meta-analysis on currently available data. Data from a French cohort estimates a crude incidence rate of 5.6 per 100,000 person years for patients on olmesartan developing intestinal malabsorption requiring hospitalization. Despite its rarity, prescribers should be cognizant of this side effect which can evolve even after years of medical therapy. Please acknowledge all funding agencies by checking the applicable boxes below None Disclosure of Interest None Declared

Vertebrobasilar dolichoectasia and other arterial abnormalities leading to abducens nerve palsy

IntroductionDolichoectatic vessels can cause cranial nerve dysfunction by either direct compression or ischemia. Abducens nerve palsy due to neurovascular compression by elongated, enlarged, tortuous or dilated arteries is an uncommon but important cause. AimTo highlight neurovascular compression as a cause of abducens nerve palsy and discuss various diagnostic techniques. MethodsManuscripts were identified using the National Institutes of Health PubMed literature search system. Search terms included abducens nerve palsy, neurovascular compression, dolichoectasia and arterial compression. Inclusion criteria required that the articles were written in English. ResultsThe literature search identified 21 case reports where abducens nerve palsy was due to vascular compression. Out of these 18 patients were male and the mean age was 54 years. Eight patients had unilateral right abducens nerve involvement; eleven patients had unilateral left nerve involvement and two patients had bilateral involvement. The arteries causing the compression were basilar, vertebral and anterior inferior cerebellar arteries. A compressed abducens nerve is not usually clearly detected on CT (Computed Tomography) or MRI (Magnetic Resonance Imaging). MRA (Magnetic Resonance Angiography), Heavy T2- WI (weighted imaging), CISS (constructive interference in steady state) and FIESTA (Fast Imaging Employing Steady-state Acquisition) are essential to demonstrate vascular compression of the abducens nerve. The various treatment options included controlling hypertension, glasses with prisms, muscle resection and microvascular decompression.

ROLE OF MULTIDETECTOR CONTRAST ENHANCED CT IN CORRELATION OF BIOCHEMICAL AND RADIOLOGICAL FINDINGS IN ACUTE PANCREATITIS, CASE SERIES

Hyperglyceridemia (HTG) can result from a variety of causes, with gallstones, alcoholic consumption, and HTG being the most common etiologies in order. Type 1 and type 4 hyperlipidemia or type 2 diabetes mellitus (T2DM) are familial forms of HTG, while excessive alcohol consumption, certain drugs, hypothyroidism, and obesity can also lead to HTG. HTG is a relatively uncommon cause of acute pancreatitis (AP), accounting for only 10-15% of cases. In a study of 25 patients admitted with AP, 5 were found to have HTG as the cause. These patients, who were aged 30-45 and included equal numbers of male and female patients, had high triglyceride levels at admission and responded to treatment with intravenous insulin and dextrose. This suggests that HTG should be considered as a potential cause of AP in patients with T2DM and that measuring triglycerides is an important step in the evaluation of all patients with AP

Copper Deficiency Anemia Due to Low-Calorie, Blended Enteral Diet: Case Report.

Copper deficiency is an uncommon cause of hematologic abnormalities in children that is often overlooked or misdiagnosed. When found, specific recommendations on dose, duration, or frequency of copper repletion are lacking in the current literature. This report describes the case of a 4-year-old boy who presented to a university medical center in the midwestern United States with persistent macrocytic anemia found to be caused by copper deficiency secondary to a low-calorie, blended enteral diet. Intravenous copper supplementation was initiated at 40 μg/kg/d for a total of 6 days, followed by enteral supplementation of 2 mg cupric oxide. This repletion regimen ultimately improved the patient's copper deficiency and resolved anemia within 1 month.

Abstract Number ‐ 93: Endovascular thrombectomy for cerebral venous sinus thrombosis using the Penumbra Indigo Aspiration System

Introduction Cerebral venous sinus thrombosis (CVST) is an uncommon cause of stroke that can be difficult to treat. Endovascular thrombectomy techniques have evolved over time, but evidence behind the efficacy and safety of these techniques for CVST is still lacking. The Penumbra Indigo® Aspiration System (Penumbra, Inc., Alameda, California, USA) is designed to remove emboli from the peripheral arteries and veins and can potentially restore blood flow in cases of limb ischemia and venous thrombosis. We report our initial experience in the treatment of CVST using the Penumbra Indigo® Aspiration System. Methods A 35‐year‐old African American male with no major past medical history presented to the hospital with altered mental status. Computed tomography (CT) showed multiple areas of venous infarction. CT vessel imaging revealed extensive dural venous sinus thrombosis involving the superior sagittal sinus (SSS), torcula, left transverse sinus, right paramedian transverse sinus with partial filling defects in the left sigmoid sinus and distal right transverse sinus with a matched defect in the left paramedian anterior frontal lobe corresponding to the area of evolving infarct. Continuous low‐dose heparin infusion was started. Cerebral angiogram revealed evidence of extremely slow perfusion to the brain with no significant drainage through the dural sinuses with complete occlusion of the distal portion of the SSS and bilateral transverse sinuses. Furthermore, angiogram of the right internal jugular (IJ) vein displayed complete occlusion of the bilateral transverse sinuses. Results An Aristotle® 24, 200‐cm Guidewire (Scientia Vascular, Salt Lake City, Utah, USA) was inserted into a 160‐cm‐long Zoom 35™ catheter (Medtronic, Minneapolis, Minnesota, USA), and this system was then advanced within the CAT12 aspiration catheter within a right IJ 12 French Sheath under fluoroscopic guidance across the occlusion to the superior sagittal sinus, Multiple aspiration attempts were then performed. The CAT 12 Aspiration was performed in the SSS and bilateral transverse sinuses. Afterwards, CAT8 and then CAT7 were then advanced into the thrombus more proximally in the SSS and multiple aspiration attempts were performed in the SSS with the aid of the separator device. There was evidence of partial recanalization of the SSS with significant improved flow of bilateral sigmoid sinuses and bilateral transverse sinuses. A decision was made to stop the intervention. There were no complications related to these procedures. Conclusions The Penumbra Indigo® Aspiration System has been used in various arterial and venous systems but this is the first report to demonstrate its technical feasibility in thrombectomy for CVST. Further feasibility studies are necessary to inform its applicability in CVST.

Lumbar Radiculopathy due to Iliacus Abscess: Diagnosis, Treatment, and Review of the Literature

Background: An iliopsoas abscess is a type of deep pelvic infection that presents as an uncommon cause of back pain. Patients generally present with vague and ambiguous symptoms that pose diagnostic difficulty. This case report describes a 19-year-old female who initially presented with low back pain and lower extremity lumbar radiculopathy secondary to complex iliacus deep pelvic infection. This is the second documented case of lumbar radiculopathy as the presenting symptom for iliopsoas abscess. The patient underwent surgical drainage and debridement and was prescribed intravenous culture-tailored antibiotic therapy for 6 weeks.