IJCR (2020), Volume 4

  • Successful non-operative management of extensive pneumomedia- stinum and pneumoperitoneum

    Background: A 70 year old male presented with acute left flank pain and was found to have extensive free air in both the mediastinal and peritoneal cavities. He was managed successfully without surgery. Case: A 70 year old male presented with left flank pain that began an hour prior to arrival. He had a history of obstructive sleep apnea that had been treated with continuous positive airway pressure therapy (CPAP) at home for many years. His vital signs, physical exam, and laboratory testing were all unremarkable. However, imaging findings were impressive for massive pneumoperitoneum and pneumomediastinum. Given his clinical stability, operative exploration was deferred, and he was admitted for observation and bowel rest. He was discharged less than 48 hours after admission without incident. His history of home CPAP therapy was the only attributable cause for the ominous findings seen on his imaging. Recognizing that this was a case of benign free air saved this patient from the morbidity and potential for death that comes with an unnecessary operative exploration. Conclusion: Continuous positive airway pressure therapy can result in benign free air within the mediastinal and peritoneal cavities. Evaluating the complete clinical scenario will allow for recognition of similar cases in which these grim findings are not indicative of surgical disease, thus avoiding the iatrogenesis of unnecessary surgery.

  • Shewanella, not just found in the marine environment. A rare case of Shewanella algae bacteremia

    A 63 year old Caucasian female with ampullary pancreatic cancer s/p pancreaticoduodenectomy in 2016 was admitted to our cancer center with complaints of fever (temperature 103° F) and abdominal pain. She was found to have multiple liver abscesses. Her blood culture grew out Shewanella algae. We herein report on this rare human bacterial infection.

  • A suspected insulinoma with the unexpected histopathological finding of a nesidioblastosis – A case report and literature review

    Introduction: Nesidioblastosis is a hyperplasia of the beta cells that causes persistent hyperinsulinemic hypoglycemia, unfortunately, it is difficult to diagnose through imaging studies. Case presentation: A 68-year-old woman with palpitations, diaphoresis, dizziness and alterations in the level of consciousness that improve with food intake. Laboratories: glucose 35.0 mg/dl, insulin 12.5 mUI/ml, proinsulin 14.1 pmol/L and peptide C 2.55 ng/ml. Octreoscan reveals an abnormal epigastric uptake area and the tomography shows a focal hypervascular lesion in pancreatic body of 12×11 mm. A distal pancreatectomy was performed without palpating the tumor in transoperative period, and a diffuse nesiodioblastosis was reported in the histopathological study. The patient persists with hypoglycemia and an additional pancreatic resection was performed, resecting 90% of the organ. Although an insulinoma was not located in the piece, an area surrounded by connective tissue was observed where islet hyperplasia was more accentuated. Discussion: The pathophysiology of nesidioblastosis is unknown, its main differential diagnosis is insulinoma and this should be suspected when the tumor can not be identified. Most agree that a pancreatectomy that involves 60-80% of the total organ can control glucose levels with low risk of diabetes and pancreatic insufficiency. Conclusion: The nesidioblastosis should be suspected when a hyperinsulinemic hypoglycemia is difficult to control and when a tumor can not be identified. The extension of the pancreatectomy should be individualized and if an insulinoma is not localized and a nesidioblastosis is suspected, an intraoperative histopathological examination with frozen section evaluation for the margins could determine the extent of pancreatectomy.

  • Rare case of ectopic pleomorphic adenoma on the plane of the cheek

    Pleomorphic adenoma is a benign tumor of the salivary gland with the highest incidence rate among all salivary gland tumors in the general population. Even though it’s common, ectopic presentation of the same is rare. When it happens, it presents in the head and neck region. We discuss in this paper a rare case of ectopic pleomorphic adenoma on the plane of the cheek.

  • Phosphate Enema Causing Life-Threatening Rectal Perforation

    We report a near fatal rectal perforation due to a phosphate enema in an elderly male. The presentation in septic shock within 4 hours of the enema is quite rare. Early recognition and prompt management are essential for a good outcome. A defunctioning colostomy is standard for these cases but we recommend a distal rectal washout since intraluminal faeces in a loaded rectum could be a cause of ongoing sepsis. Although enemas are commonly used for constipation in the elderly, suppositories and oral preparations should be used preferentially where appropriate.

  • Methemoglobinemia – A review and recommendation for management – Acute methemoglobinemia with hemolytic anemia following suicidal use of aniline based substance

    Methemoglobinemia is a rare disease classification related with congenital or acquired (usually iatrogenic) hemoglobin oxygenation disorder. Despite the fact that number of potentially methemoglobin forming agents is very long, methemoglobinemia is still a case-report of patients admitting to the Emergency Departments. The patient was brought after consuming resin hardener for suicidal purposes from the Center of lower reference to the hospital Emergency Department with suspected burns of the gastrointestinal tract. The patient presented cyanosis, dyspnoea and brown-colored urine on admission.

  • Surgical difficulties with a giant carcinoid in the anterior mediastinum

    Atypical carcinoid in the anterior mediastinum is a rare neoplasm. In our case the huge mediastinal carcinoid was treated with surgery using a median sternotomy and clamshell incision because of the extension of the tumor. We describe our difficulties under the surgery and take a look at the literature.

  • A rare case of Hepatic Sinusoidal obstruction syndrome due to a drug use

    Here, we report a case of hepatic sinusoidal obstruction syndrome(HSOS) of a 30 years old female patient with Ulcerative colitis(UC) who was being treated by mesalazine. The patient was admitted to the emergency unit by harsh and cramping abdominal pain, she had increased AST-ALT-Bilirubine and GGT levels in the blood laboratory analysis. In the Portal venous Doppler US; Splenomegaly and coarse granular appearance in liver parenchymal echogenecity were observed. In the abdominal CT, Splenomegaly was confirmed. In the Dynamic Abdominal MRI, pathognomonic liver finding which was the patchy contrast-enhancing reticular appearance, was visualized and the exact diagnosis was handled by true-cut biopsy. The aid of Dynamic Abdominal MRI to the early diagnosis of HSOS was also demonstrated in this case.

  • 15q11.2 Deletion Syndrome: Expanding the Phenotype

    Background: Although multiple reports exist in the literature of patients with 15q11.2 deletion syndrome, the variability of the phenotype has made clinical delineation difficult. Neuro-developmental scores have not been previously reported. We present clinical findings in a group of 16 patients referred to our center for evaluation and management of neurodevelopmental difficulties. Methods: All patients were seen in our center between 2005 and 2016. They were seen by a clinical geneticist and their diagnosis of 15q11.2 deletion syndrome was confirmed by a microarray analysis. Bayley Scales of Infant Development was done and provided a mental developmental index and a motor developmental index. The data collected was then compared to previous reports in the literature of patients with 15q11.2 deletion syndrome. Results: The reviewed group consisted of 10 males and 6 female patients between the ages of 3 and 15 years. The most common clinical findings included developmental delay (94%), hypotonia (88%), ADHD (75%), anxiety (50%), feeding difficulties (44%), autism (31%), dolichocephaly (25%), 2-3 toe syndactyly (19%), seizure activity (19%), and congenital heart disease (13%). Additional findings included prominence of the metopic suture, epicanthal folds, micrognathia, ankle torsion, incontinence, and sleeping difficulties. Our developmental evaluation by the Bayley Scales of Infant Development indicated an average Mental Developmental Index of 75 (NL = >85) and Motor Developmental Index of 75 (NL = >85) in the patients less than 3 years old. Close to 43% of patients had an occipitofrontal circumference (OFC) greater than or equal to the 97th. Conclusion: The data provided here intends to expand the phenotype of the 15q11.2 deletion syndrome. Neurodevelopmental scores have not been previously reported in 15q11.2 del syndrome which were found to be in the mild developmental delay range.

  • A severe case of toxic epidermal necrolysis which started with agep-like features: Does a new clinical type of drug eruption?

    Acute generalized exanthematous pustulosis (AGEP) and toxic epidermal necrolysis (TEN) are adverse cutaneous reactions. Despite the fact that these 2 cutaneous reactions differ in presentation, prognosis, pathologic features, and treatment, overlap can exist between them, creating a diagnostic challenge. It has rarely been reported that AGEP overlaps or mimics TEN, while TEN which starting by multıple pustules like AGEP has not been reported. The present study reports a rare case of a 14-year-old male patient with the clinical manifestations of multiple purulent bullae caused by amoxycillin ingestion. On 3. day dermatological examination revealed flaccid bullae and epidermal splitting with positive Nikolsky’s sign on right pectoral area, erosions on oral and anogenital mucosa. Histopathologic examination showed a massive inflammatory infiltration and epidermal necrosis indicated Steven Johnson syndrome. Later on added systemic symptoms with haemodynamic, respiratory imbalance and the patient was exitus. The case is discussed to reveal whether a new clinical pattern of drug eruption has been identified. We describe a patient with TEN who presented initially with clinical features of acute generalized exanthematous pustulosis (AGEP). To the best of our knowledge, one report drug eruption with purulent bulla and epidermal necrolysis has previously been documented, and therefore the present study is the second case report of its kind. Because the initial presentation of such adverse drug reactions may vary, diagnosis is difficult and dermatologists must careful the possibility of mimics among adverse cutaneous reactions.