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Clinical Case Reports Jun 2024Acute chest pain can be the first manifestation of multiple endocrine neoplasia type 1(MEN1)-associated thymic neuroendocrine neoplasms (NEN). Comprehensive treatment...
KEY CLINICAL MESSAGE
Acute chest pain can be the first manifestation of multiple endocrine neoplasia type 1(MEN1)-associated thymic neuroendocrine neoplasms (NEN). Comprehensive treatment may be an effective strategy for MEN1-associated NEN.
ABSTRACT
Multiple endocrine neoplasia type 1(MEN1)-associated thymic neuroendocrine neoplasms (NEN) is caused by the mutation of tumor suppressor gene. Patients with MEN1-associated NEN initially presenting with acute chest pain are very rare. In the manuscript, we reported a case of a 45-year-old man who developed MEN1-associated NEN with acute chest pain as initial symptom. Thoracoscopic thymotomy was performed and thymic NEN was successfully removed. Genetic test showed a germline mutation of gene in this patient. Immunohistochemical staining exhibited Syn(+), CgA(+), INSM1(+), CD56(+) and Ki67-positive cells (2%) in MEN1-associated NEN. Further evaluation unveiled MEN1-associated benign tumors including digestive NEN and pituitary gland adenoma. The 99mTc-HYNIC-TOC scintigraphy showed that focally increased radioactivity in the mid-upper abdomen. This patient was administered with 50Gy/25F of radiation dose to treat the postoperative lesions. Subsequently, sandostatin LAR (30 mg per week) was used as systemic therapy. He had no recurrence or metastasis for 6-month follow-up. Thus, acute chest pain can be the first manifestation of MEN1-associated NEN, and comprehensive treatment including surgery, radiation and systemic treatment may be an effective strategy for MEN1-associated NEN.
PubMed: 38883224
DOI: 10.1002/ccr3.9031 -
Frontiers in Medicine 2024Renal arteriovenous malformations (rAVMs) are congenital abnormal pathways between renal arteries and veins that are rare in the general population. It is often...
BACKGROUND
Renal arteriovenous malformations (rAVMs) are congenital abnormal pathways between renal arteries and veins that are rare in the general population. It is often misdiagnosed as malignant renal tumors with abundant blood supply, and the definitive diagnosis primarily relies on angiography. Multimodality imaging, including contrast-enhanced computed tomography (CT), magnetic resonance imaging (MRI), and positron emission tomography (PET)/CT plays an important role in the differential diagnosis of renal space-occupying lesions.
CASE PRESENTATION
A 56-year-old man presented with abdominal distension, loss of appetite, and back pain without obvious cause 2 years ago, without nausea vomiting, or frequent urination. Gastroscopy and colonoscopy showed multiple polyps in the duodenum and colon. Abdomen contrast-enhanced CT revealed a mass of 1.6 × 1.4 cm in the left kidney, which was considered to be a malignant tumor. PET/CT was performed for further diagnosis; the F-fluorodesoxyglucose (F-FDG) PET/CT scan showed mild uptake in the left renal mass, while no uptake of F- prostate-specific membrane antigen (PSMA) was observed. Following a multidisciplinary discussion, the possibility of renal AVMs was considered and subsequently confirmed by renal angiography as the diagnosis. Then, selective segmental renal artery embolization was performed for treatment.
CONCLUSION
Renal AVMs are extremely rare in clinical practice. Due to limited research on the application of F-FDG and F-PSMA PET/CT to renal AVMs, its role remains largely unexplored. With the increasing popularity of PET/CT imaging, comprehensive imaging of the disease has become indispensable. We report the first case of PSMA PET/CT imaging in renal AVMs, and when PSMA expression is absent in a renal mass, the possibility of renal AVMs should be considered.
PubMed: 38882665
DOI: 10.3389/fmed.2024.1420473 -
The Pan African Medical Journal 2024Acute chylous peritonitis is an uncommon medical condition that can occur suddenly, resulting in the buildup of chylous fluid in the peritoneal cavity. It is considered... (Review)
Review
Acute chylous peritonitis is an uncommon medical condition that can occur suddenly, resulting in the buildup of chylous fluid in the peritoneal cavity. It is considered idiopathic because the exact cause is often unknown. The symptoms of acute chylous idiopathic peritonitis can mimic other abdominal emergencies, making it challenging to diagnose and manage, requiring a multidisciplinary approach. We present a case report of acute idiopathic chylous peritonitis miming acute abdomen, how was successfully treated with surgery, and provide a comprehensive review of the available literature on this topic. Chylous peritonitis is a rare condition whose clinical presentation mimics an acute abdomen. It is necessary to undertake careful exploration. An emergent laparotomy is indicated to treat the peritonitis and search for and treat the underlying cause.
Topics: Humans; Abdomen, Acute; Chylous Ascites; Acute Disease; Diagnosis, Differential; Laparotomy; Male; Female
PubMed: 38881770
DOI: 10.11604/pamj.2024.47.131.42794 -
International Journal of Surgery Case... Jun 2024Disseminated Peritoneal Leiomyomatosis (DPL) is a rare benign proliferation of solid peritoneal lesions along the abdominopelvic cavity comprised of smooth muscle and...
INTRODUCTION AND IMPORTANCE
Disseminated Peritoneal Leiomyomatosis (DPL) is a rare benign proliferation of solid peritoneal lesions along the abdominopelvic cavity comprised of smooth muscle and connective tissue. Though hormonal and iatrogenic causes have been theorized, the exact etiology remains unknown. Most patients with DPL are frequently premenopausal with a history of myomectomy or prior hysterectomy. These patients can present asymptomatically or with abnormal uterine bleeding and abdominal discomfort. DPL is a rare entity with less than 150 cases reported in the literature, showcasing the need of awareness of this poorly understood neoplasm. Imaging, if performed, is helpful as positron emission tomography (PET) can differentiate DPL from malignant peritoneal disease. Treatment involves medical and surgical options based on patient's clinical presentation, with medical treatment with gonadotropin-releasing hormone agonist being first line.
CASE PRESENTATION
We report a case of a previously healthy female presenting for desired laparoscopic tubal ligation with incidental countless peritoneal nodules suspicious for carcinomatosis found during the operative event but proven leiomyomas after histologic examination.
CLINICAL DISCUSSION
Differentiating DPL from mimickers such as leiomyosarcoma, endometriosis, and carcinomatosis remains a challenge as macroscopic appearances are similar ultimately requiring histology evaluation.
CONCLUSION
Awareness of the entity is crucial to avoid misdiagnosis and unnecessary anxiety associated with a presumptive diagnosis of malignancy for a largely benign entity.
PubMed: 38878730
DOI: 10.1016/j.ijscr.2024.109908 -
Journal of Medical Case Reports Jun 2024Glycated hemoglobin is a well-known marker for evaluating long-term glycemic control. However, the accuracy of glycated hemoglobin measurement can be affected by the...
BACKGROUND
Glycated hemoglobin is a well-known marker for evaluating long-term glycemic control. However, the accuracy of glycated hemoglobin measurement can be affected by the presence of hemoglobin variants, which makes the determination and interpretation of glycated hemoglobin values in terms of glycemic control not only difficult but also misleading. Here we present the first ever case of a patient with type 2 diabetes with hemoglobin E from Nepal, diagnosed incidentally because of spurious glycated hemoglobin levels.
CASE PRESENTATION
A 45-year-old Hindu Mongolian female with a history of type 2 diabetes for around 9 years but not very compliant with follow-ups was referred to our facility for plasma fasting and postprandial blood glucose levels and glycated hemoglobin. Fasting and postprandial blood sugars were found to be high. A consistent very low glycated hemoglobin by two different high-performance liquid chromatography (HPLC) methods compelled us to call the patient for a detailed clinical history and for the records of investigations done in the past. The patient has been a known case of type 2 diabetes for around 9 years and presented irregularly for follow-up visits. Around 4 years ago, she presented to a healthcare facility with fatigue, severe headaches, pain in the abdomen, discomfort, and dizziness for a couple of months, where she was shown to have high blood glucose. She was referred to a tertiary-level hospital in Kathmandu, where she was prescribed metformin 500 mg once daily (OD). Due to her abnormal hemoglobin A1c reports, she was then sent to the National Public Health Laboratory for repeat investigations. Her blood and urine investigations were sent. Complete blood count findings revealed high red blood cell and white blood cell counts, a low mean corpuscular volume, and a high red cell distribution width-coefficient of variation. Other parameters, including serum electrolytes, renal function tests, liver function tests, and urine routine examinations, were within normal limits. A peripheral blood smear revealed microcytic hypochromic red cells with some target cells. Hemoglobin electrophoresis showed a very high percentage of hemoglobin E, a very low percentage of hemoglobin A2, and normal proportions of hemoglobin A and hemoglobin F. A diagnosis of homozygous hemoglobin E was made, and family screening was advised.
CONCLUSIONS
Clinicians should be aware of the limitations of glycated hemoglobin estimation by ion exchange high-performance liquid chromatography in patients with hemoglobin E and other hemoglobin variants. If the clinical impression and glycated hemoglobin test results do not match, glycated hemoglobin values should be determined with a second method based on a different principle, and glycemic status should be confirmed through alternative investigations, preferably those that are not influenced by the presence of hemoglobin variants (for example, boronate affinity chromatography, fructosamine test, glycated albumin test, the oral glucose tolerance test, continuous glucose monitoring, etc.). Consistent or even doubtful results should also raise the suspicion of a hemoglobin variant, which should be confirmed through further evaluation and investigations.
Topics: Humans; Female; Diabetes Mellitus, Type 2; Middle Aged; Glycated Hemoglobin; Incidental Findings; Hemoglobins, Abnormal; Blood Glucose; Hypoglycemic Agents
PubMed: 38877536
DOI: 10.1186/s13256-024-04518-y -
BMC Surgery Jun 2024The Russia-Ukraine war is associated with critical and severe thoracoabdominal injuries. A more specific approach to treating patients with thoracoabdominal injury...
The Russia-Ukraine war is associated with critical and severe thoracoabdominal injuries. A more specific approach to treating patients with thoracoabdominal injury should also include minimally invasive technologies. It remains unclear about the utility of using video-assisted thoracoscopic surgery (VATS) and laparoscopy in patients with thoracoabdominal injury. The aim of this study was to investigate and evaluate the utility of video-assisted thoracoscopic surgery, laparoscopy as well as magnetic tool applications for the management of severe thoracoabdominal injury in combat patients injured in the ongoing war in Ukraine and treated in the Role 2 deployed hospital. Patients and methods 36 male combat patients thoracoabdominal injury were identified for the study during the first 100 days from February, 24 2022. These individuals were diagnosed with thoracoabdominal GSW in the Role 2 hospital (i.e. deployed military hospital) of the Armed Forces of Ukraine. Video-assisted thoracoscopy surgery (VATS) and laparoscopy with application of surgical magnetic tools were applied with regards to the damage control resuscitation and damage control surgery. Results In 10 (28%) patients, VATS was applied to remove the metal foreign body fragments. Both thoracotomy and laparotomy were performed in 20 (56%) hemodynamically unstable patients. Of these 20 patients, the suturing of the liver was performed in 8 (22%) patients, whereas peri-hepatic gauze packing in 12 (33%) patients. Massive injury to the liver and PI 2.0-3.0 were diagnosed in 2 (6%) patients. Lethal outcome was in 1 (2.8%) patient. Conclusions Thoracoabdominal gunshot injuries might be managed at Role 2 hospitals by using video-assisted thoracoscopy (VATS) and laparoscopy accompanied by surgical magnetic tools. Damage control surgery and damage control resuscitation must be applied for patients in critical and severe conditions.
Topics: Humans; Wounds, Gunshot; Ukraine; Male; Adult; Thoracic Injuries; Thoracic Surgery, Video-Assisted; Laparoscopy; Abdominal Injuries; Hospitals, Military; Young Adult; Treatment Outcome; Retrospective Studies; Laparotomy
PubMed: 38877409
DOI: 10.1186/s12893-024-02475-3 -
International Journal of Surgery Case... Jun 2024Fewer than 200 cases of Perforated Duodenal Diverticulum ("PDD") have been reported in the literature. The percentage of caused by trauma is generally very low and...
INTRODUCTION
Fewer than 200 cases of Perforated Duodenal Diverticulum ("PDD") have been reported in the literature. The percentage of caused by trauma is generally very low and similar to rates recorded for duodenal injuries caused by trauma in the absence of diverticula (3 %-5 %). As a rare cause of abdominal pain after trauma, perforated duodenal diverticula are seldom diagnosed preoperatively. Despite preoperative CT scan demonstrating duodenal perforation, a diverticular origin is often only identified intraoperatively.
PRESENTATION OF CASE
A 36-year-old man was admitted to the emergency department with severe upper abdominal pain that began after blunt trauma by a kick to his upper abdomen during a fight. A duodenal injury was identified by CT. Exploratory laparotomy then revealed a retroperitoneal perforation of a diverticulum located at the second segment of the duodenum.
DISCUSSION
A review of the literature found few cases reporting perforated duodenal diverticulum secondary to trauma. Most cases identified the diverticulum at intraoperative exploration, with most approaches being through open surgery.
CONCLUSION
Perforation of a duodenal diverticulum secondary to trauma is an extremely rare event, which is why it is often overlooked in the differential diagnosis of acute abdomen. As the presenting signs are often suggestive of duodenal perforation without a clear notion of duodenal diverticula at CT scan, a surgical approach and exploration is most frequently described. In our experience, the management of traumatic PDD aligns with the literature favoring the open surgical approach.
PubMed: 38875829
DOI: 10.1016/j.ijscr.2024.109882 -
International Journal of Surgery Case... Jun 2024Purtscher retinopathy is the rare form of occlusive microvasculopathy, characterized by multiple retinal white areas around the optic nerve head and fovea with...
INTRODUCTION AND IMPORTANCE
Purtscher retinopathy is the rare form of occlusive microvasculopathy, characterized by multiple retinal white areas around the optic nerve head and fovea with paravascular clearing and may be related to intraretinal hemorrhages. Acute Pancreatitis (AP) is one of the most common gastrointestinal reasons for hospital admissions globally. The complications of Acute Pancreatitis may include Purtscher's-like retinopathy, which has a low incidence rate of less than 0.24 instances per million cases. This case report highlights the value of thorough medical history taking and examination, and it apprises the consideration of ophthalmological manifestation in patients of Acute Pancreatitis.
CASE PRESENTATION
A 34-year-old female came to the emergency room due to intense abdominal pain associated with nausea and vomiting, which worsened over the last 24 h. The pain was described as continuous, sharp, and cramping-like in the upper abdomen, radiating to the back. Lab tests revealed elevated serum amylase and lipase levels, indicating pancreatitis, along with slight leukocytosis. A contrast-enhanced CT scan confirmed acute pancreatitis with mild inflammation and enlargement of the pancreas. Two days after admission, the patient experienced a sudden and painless loss of central vision in both eyes. There was no history of trauma or any other significant relevant history, other than pancreatitis. The ophthalmologist's examination found reduced visual acuity (6/60 in the right eye, 3/60 in the left eye), normal corneas, and anterior chambers.
DISCUSSION
Inkeles and Walsh established the first link between acute pancreatitis and Purtscher-like retinopathy when they reported three cases of the distinctive retinal appearance in individuals with acute pancreatitis in 1975.
CONCLUSION
The recovery and prognosis in cases of Purtscher-like retinopathy is variable and further research is required to ascertain the usage of corticosteroids and pentoxifylline in improving the course of a patient's with Purtscher's-like retinopathy.
PubMed: 38875828
DOI: 10.1016/j.ijscr.2024.109881 -
Medicine Jun 2024Most adrenal tumors are benign and primary adrenal malignancies are relatively rare. Primary adrenal lymphoma (PAL) is a very rare and highly aggressive malignant tumor...
INTRODUCTION
Most adrenal tumors are benign and primary adrenal malignancies are relatively rare. Primary adrenal lymphoma (PAL) is a very rare and highly aggressive malignant tumor with unknown etiology, atypical clinical symptoms, nonspecific imaging manifestations, difficult disease diagnosis and poor prognosis.
CASE REPORT
This case report details a 42-year-old woman who was admitted to the hospital with a 1-year-old bilateral adrenal mass and 1-month-old left upper abdominal pain. Enhanced CT of the abdomen showed a right adrenal nodule and a large occupying lesion in the left adrenal region, with a high probability of pheochromocytoma. Intraoperatively, a huge tumor measuring about 12*12*10 cm was found in the left adrenal region, infiltrating the left kidney, spleen and pancreatic tail. Postoperative pathology: lymphocytes were found in the renal capsule and subcapsule, lymphocytes were found in the pancreas; lymphocytes were found in the spleen. Consider a tumor of the lymphohematopoietic system, possibly lymphoma.
CONCLUSION
This case demonstrates that primary adrenal diffuse large B-cell lymphoma (PADLBCL) is highly aggressive, has a poor prognosis, is prone to recurrence, has poor therapeutic outcomes, and is difficult to diagnose. Clinicians should consider the possibility of PADLBCL when encountering huge adrenal-occupying lesions and consider chemotherapy before surgery. Reducing the tumor size before surgery is a more favorable therapeutic approach, thus prolonging the patient life and improving the quality of survival.
Topics: Humans; Female; Lymphoma, Large B-Cell, Diffuse; Adrenal Gland Neoplasms; Adult; Tomography, X-Ray Computed; Adrenal Glands
PubMed: 38875421
DOI: 10.1097/MD.0000000000038298 -
Medicine Jun 2024Urachal anomalies are rare and can present with various clinical manifestations. Urachal remnants, in particular, can be difficult to diagnose because of atypical...
RATIONALE
Urachal anomalies are rare and can present with various clinical manifestations. Urachal remnants, in particular, can be difficult to diagnose because of atypical symptoms at presentation. This study reports a case of intestinal obstruction in an infant secondary to an infected urachal cyst.
PATIENTS CONCERNS
A 3-month-old boy with a known febrile urinary tract infection developed acute abdominal distension.
DIAGNOSES
Abdominal ultrasound (US) and computed tomography (CT) revealed a nonspecific, ill-defined soft tissue density at the mid-abdomen, associated with intestinal obstruction.
INTERVENTIONS
Emergency exploratory laparotomy was performed. The site of the obstruction was found to be at the mid-small bowel; the proximal small bowel was markedly distended, and the small bowel and sigmoid colon were adherent to urachal remnant. The urachal remnant was excised, and the peritoneal adhesions were lysed.
OUTCOMES
The day after surgery, the patient was discharged without any complications.
LESSONS
Intestinal obstruction is an exceedingly rare presentation of urachal remnants. This case highlights that urachal anomalies should be considered in the differential diagnosis in patients with intestinal obstruction and a concurrent febrile urinary tract infection.
Topics: Humans; Male; Urinary Tract Infections; Urachal Cyst; Infant; Intestinal Obstruction; Fever; Diagnosis, Differential; Ultrasonography
PubMed: 38875366
DOI: 10.1097/MD.0000000000038615