American Journal of Case Reports最新文献

BACKGROUND A single coronary artery is a rare congenital anomaly of the coronary arteries distribution, with an incidence of only 0.03% in the general population undergoing coronary angiography. RCA arising from the distal circumflex artery is an extremely rare variety. CASE REPORT We report the case of a 63-year-old man who was admitted in our hospital after an inconclusive treadmill stress test. He had no cardiovascular risk factors. He was initially evaluated for palpitations. A subsequent Holter monitor showed monomorphic premature ventricular beats. His coronary artery disease (CAD) pre-test probability was low. Non-invasive exploration with 64-detector-row multi-slice computed tomography (MSCT) was performed to visualize the coronary arteries. It showed a single left coronary artery (LCA) with no significant stenosis. There was no artery emerging from the right sinus of Valsalva. The right coronary artery (RCA) had an abnormal origin, arising from the distal circumflex artery. The patient remained asymptomatic. CONCLUSIONS This case report describes an accidental discovery of an unusual variety of single coronary artery. Computed tomography angiography, very useful in patients with low pre-test probability of coronary artery disease, showed a right coronary artery from the distal circumflex artery.

BACKGROUND Primary cardiac malignancies are extremely rare, with an incidence of 0.07% on autopsy series. Primary sarcomas represent up to 95% of malignant neoplasms, with myxofibrosarcomas accounting for only 10%. Around 90% of patients present with unspecific symptoms and a tumor with polypoid appearance on imaging, thus frequently receiving a misdiagnosis of myxoma. CASE REPORT A 65-year-old man presented with abrupt chest pain, blood pressure of 130/80 mmHg, and heart rate of 180 beats/min. Electrocardiogram showed atrial fibrillation, and imaging revealed a polypoid tumor on the atrioventricular septum obstructing the mitral valve. The tumor was removed and sent for histopathological evaluation, revealing a multinodular pattern with spindled hypocellular areas and hypercellular areas featuring pleomorphic cells. The mitotic count was 11 in 10 high-power fields, and necrosis was present in less than 50% of the tumor area. Tumor cells were calretinin and MDM2 negative and CD34 positive. Heterologous elements, necrosis and hemorrhage, were noted. Considering these findings, this tumor was classified as intermediate-grade myxofibrosarcoma. CONCLUSIONS Due to the rarity of myxofibrosarcomas, evidence for optimal diagnostic and therapeutic management is limited. Despite being frequently polypoid, seemingly benign tumors on imaging, the extent of infiltration at their base is usually deep. Their innocent appearance can hinder adequate presurgical planning, leading to suboptimal resections. We present the example of a seemingly benign tumor as a potential pitfall in evaluating cardiac neoplasms, highlighting the importance of histopathological and immunohistochemical evaluation in their correct characterization, in order to offer the best follow-up and adjuvant treatment, when needed.

BACKGROUND Pembrolizumab, a programmed cell-death protein-1 (PD-1)-targeting antibody, extends survival in cancer patients but may cause lung injury as a side effect. This immunotherapy enhances the immune system's ability to recognize and eliminate cancer cells. However, its immunomodulatory action can sometimes lead to immune-related adverse events, including lung injury. CASE REPORT A 40-year-old female patient, previously managed for malignant melanoma of the left gluteal region with surgery and immune checkpoint inhibitors, was transitioned to pembrolizumab for ongoing cancer treatment. Subsequently, she was referred to our department for further evaluation due to findings on chest imaging revealing multiple nodules in the bilateral lung fields. The patient exhibited neither cough, fever, nor breathlessness. Bronchoscopic examination yielded no abnormalities. Cytological assessments were negative, as were cultures for bacteria, fungi, and acid-fast bacilli. Bronchoalveolar lavage and endobronchial ultrasound-guided transbronchial needle aspiration biopsy of the right lower lobe bronchus B9a were conducted. Pathological analysis identified a combination of acute inflammatory and chronic fibrotic lesions, primarily histiocytic, leading to a diagnosis of pembrolizumab-induced lung injury. Steroid pulse therapy followed by tapering resulted in improvement of the pulmonary shadows. The patient is currently under observation without requiring steroid therapy. CONCLUSIONS This case underscores the importance of vigilance for potential pembrolizumab-induced lung injury in patients undergoing immunotherapy for cancer treatment. Prompt recognition and appropriate management are essential for optimizing patient outcomes. Additionally, this case highlights the challenge of diagnosing lung lesions based solely on imaging, necessitating bronchoscopy with tissue sampling as a critical diagnostic tool.

BACKGROUND Decompressive craniectomy is a common life-saving intervention in the setting of elevated intracranial pressure. Cranioplasty restores the calvarium and intracranial physiology once swelling recedes. Cranioplasty is often thought of as a low-risk intervention. However, numerous reports indicate that malignant cerebral edema (MCE) is an often-fatal complication of an otherwise uneventful cranioplasty. A careful review of the literature is needed to better understand this devastating condition. CASE REPORT A 41-year-old man presented after suffering a gunshot wound to the right frontal lobe. Upon initial evaluation, the patient had grossly visible brain matter, left-sided hemiparesis with a Glascow Coma Score (GCS) of 11, and vital signs concerning for elevated intracranial pressure. Computed tomography (CT) showed right-sided intraparenchymal and subarachnoid hemorrhage with a 5 mm leftward midline shift. The patient was taken to the operating room (OR) for right fronto-parietal craniectomy. Over the next 3 months, he recovered steadily and underwent PEEK cranioplasty on post-operative day 83. Pre-operative CT showed sunken skin flap syndrome with an 8-mm midline shift. Following an uneventful cranioplasty, he failed to regain consciousness. Examination revealed absent brainstem reflexes. CT showed global diffuse cerebral edema. The patient was declared brain dead. CONCLUSIONS Continued research is needed to better understand the pathophysiology of malignant cerebral edema so that future incidences may be prevented. A combination of negative-pressure suction drainage, sunken skin flap syndrome, and delayed time to cranioplasty likely play a significant role in the evolution of MCE. We urge neurosurgeons to consider the likelihood of MCE and adapt surgical planning accordingly.

BACKGROUND Primary cutaneous lymphomas (PCL) are a multifaceted spectrum of cutaneous T cell lymphoma (CTCL) and cutaneous B cell lymphomas (CBCL). Mycosis fungoides (MF) is a rare subset of CTCL that primarily affects adults, and its occurrence in children is exceedingly rare. Most pediatric MF manifests as hypopigmented patches resembling other benign dermatoses, causing diagnostic challenges. This report outlines a case of pediatric MF in a 7-year-old Malaysian boy. CASE REPORT A 7-year-old boy exhibited progressing skin lesions characterized initially by erythematous, papular rashes over the face and upper limbs, then to the whole body, becoming hypopigmented, with pruritus and scaling for 1 year. Multiple clinics treated him for eczema and pityriasis alba but he responded poorly to courses of various topical steroids and emollient treatment. Due to the refractory nature of the lesions, he was subsequently referred to a dermatology clinic, where 2 skin biopsies were performed. The first biopsy revealed epidermotropism of atypical lymphocytes, consistent with MF. Immunohistochemical analysis revealed positive CD3+ expression with slightly reduced CD4+, CD7+, and CD8+ expression, and normal CD2+ and CD5+ expression at the epidermis level. Nevertheless, due to the rarity of MF in children, a second biopsy was performed, validating the diagnosis. CONCLUSIONS Pediatric MF is a rare and challenging diagnosis. This case report highlights the importance of close monitoring of unresolved hypopigmented lesions and increased vigilance on lesions not responding to standard treatment. Timely diagnosis with support of skin biopsy is crucial to avoid potentially serious disease progression and helps provide appropriate management leading to improved outcomes.

BACKGROUND Mondor's disease (MD), or sclerosing superficial thrombophlebitis of the veins of the anterior thoracic wall, is a rare condition of unknown cause that usually involves the superior epigastric vein, producing a visible and palpable Mondor cord. This report describes a 27-year-old Japanese woman presenting with left chest wall pain due to palpable and visible sclerosing superficial thrombophlebitis. CASE REPORT We present the case of a 27-year-old Japanese woman who presented with 8 days of left chest wall and upper abdominal pain. Physical examination revealed a firm, palpable cord in the painful area. Chest wall ultrasound revealed a tubular and anechoic superficial cord. Doppler imaging demonstrated normal blood flow surrounding the cord, with no blood flow within. Computed tomography (CT) revealed a subtle structure beneath the lower left breast skin and without breast or lung involvement. We diagnosed her as having MD, and she was treated with non-steroidal anti-inflammatory drugs (NSAIDs). Her pain gradually improved in 3 weeks and the cord disappeared after approximately 2 months. CONCLUSIONS Mondor's disease can be distressing and painful for patients. Clinicians should be aware of this rare and benign disease when a longitudinal painful cord is found in the torso wall. Pain relief and reassurance are typically adequate until resolution. In addition to ultrasonography, CT is also important for diagnosing MD. This report of a rare diagnosis of MD highlights the importance of accurate and timely diagnosis and investigating the patient to exclude superficial and deep venous thrombotic disease.

BACKGROUND Carotid artery injury has an incidence of 0.2% in the National Trauma Data Bank. The true incidence of intracranial carotid injury is unknown but can be estimated at less than one in 1000 trauma-related inpatient admissions in America. Operatively managed penetrating carotid trauma has a mortality rate approaching 20%, and the selection of the appropriate operative approach is not straightforward. We present a case of penetrating carotid trauma successfully managed via combined approach by neurosurgery and otolaryngology teams. CASE REPORT A 74-year-old woman fell into a honeysuckle bush. She presented with a branch embedded in the left cheek and blindness of the right eye. Further workup revealed the branch had penetrated the maxillary bone, pierced the right optic nerve, and lodged near the intracranial portion of the right internal carotid artery. She underwent emergent operative intervention via right pterional craniotomy with microsurgery and endoscopic transsphenoidal surgery with repair of the skull base. The foreign body was removed and the traumatic carotid laceration was repaired. The patient recovered successfully and was discharged on postoperative day 14. CONCLUSIONS The management of facially penetrating foreign bodies begins with assessment for neurologic deficits and vascular injury. We recommend leaving such objects in place and not removing them until definitive imaging is obtained. We present an interesting case of penetrating trauma to the intracranial carotid artery in which a retained foreign body was removed with satisfactory patient outcome using a combined endoscopic and open surgical approach.

BACKGROUND Although hypomagnesemia is common in type 2 diabetes, clinical presentations with severe hypomagnesemia are rare. A number of oral blood glucose-lowering medications can reduce serum magnesium concentrations, and several severe cases have been reported in the presence of marked glucagon-like peptide-1 receptor agonist (GLP-1RA)-associated gastrointestinal adverse effects. In the present case, an acute presentation with severe hypomagnesemia was likely due to polypharmacy including semaglutide, albeit with a delayed relationship to discontinuation of this GLP-1RA, due to nausea and vomiting. CASE REPORT A 73-year-old woman with type 2 diabetes treated with several oral medications known to reduce serum magnesium (metformin, gliclazide, sitagliptin, esomeprazole) presented after an unwitnessed collapse at home without premonitory symptoms. She had discontinued low-dose semaglutide (0.25 mg subcutaneous weekly) 2 weeks beforehand, and her gastrointestinal adverse effects had largely resolved. She was found to have an undetectable serum magnesium (<0.3 mmol/L) and hypocalcemia. She responded to electrolyte replacement and was discharged well 2 days later. Three weeks after discharge, her serum magnesium and calcium concentrations were within the reference range, on regular oral supplements of both minerals. She spontaneously reported her longstanding muscle cramps had resolved after discharge. Her clinical features and course suggested she had chronic unrecognized hypomagnesemia associated with polypharmacy that progressed to a clinically severe level, with a likely contribution from recent antecedent semaglutide use. CONCLUSIONS Periodic monitoring of serum magnesium concentrations in at-risk individuals with type 2 diabetes is recommended, since the clinical presentation of severe hypomagnesemia can be sudden and without indicative warning symptoms.

BACKGROUND Perineal injuries affecting the scrotum and penis are rare in pediatric patients, owing to the protective anatomy of the male genitalia. However, when such injuries do occur, timely surgical intervention is crucial. This kind of damage might not be life-threatening but could cause functional disorders and have a huge impact on the patients' psychological condition if not treated appropriately, especially as they enter puberty. This case illustrates the successful management of a severe genital injury in a young child. CASE REPORT We present the case of an 8-year-old boy who sustained a penile degloving injury extending from the glans to the penile base, exposing the suspensory ligament, both spermatic cords, and ruptured scrotal skin, due to sliding of an agricultural machine. Immediate surgical intervention involved replantation of the penile skin and closure of the scrotal wound with absorbable sutures. Postoperative care focused on infection prevention with appropriate antibiotics and daily dressing changes. The monitoring of the graft was essential to detect any signs of necrosis. CONCLUSIONS The 6-month follow-up of our patient showed that the performed procedure and appropriate medical approach led to full recovery and satisfactory esthetic results, without dysfunction of the male genitals or urethra. Through this case, we emphasize that, with rapid surgical intervention and proper postoperative care, pediatric patients with similar injuries can achieve full recovery. However, long-term follow-up until adulthood is recommended to monitor for potential complications that can arise later in the patient's life.

BACKGROUND Terminal ileum (TI) anastomoses present challenges due to anatomical features and pressure from the ileocecal valve (ICV). The use of negative-pressure wound therapy (NPWT) is commonly used to treat chronic skin ulcers. Its use for temporary abdominal closure following anastomosis is controversial but has shown promise in patients with inflammatory or vascular disease. This report presents 3 successful cases in which NPWT was used after TI anastomosis surgery. CASE REPORT Case 1: A 65-year-old woman with chronic kidney disease and paroxysmal atrial fibrillation was diagnosed with mesenteric ischemia. Surgical resection removed a segment of the jejunum and ileum, with an end-to-side anastomosis 10 cm from the ICV. NPWT was initiated, and abdominal wall closure was achieved. Case 2: A 73-year-old man with a history of an incarcerated left inguinal hernia, previously treated with herniorrhaphy and intestinal resection, presented with generalized peritonitis and anastomotic dehiscence 70 cm from the ICV. Resection was performed, followed by ileectomy for ischemia 15 cm from the ICV and a 2-layer isoperistaltic side-to-side anastomosis. NPWT was initiated to reduce edema and achieve primary abdominal closure. Case 3: A 69-year-old man diagnosed with mesenteric ischemia underwent resection of 40 cm of ischemic intestine. Follow-up laparotomy revealed the need for manual end-to-end anastomosis 12 cm from the ICV. NPWT was applied due to the inability to achieve primary closure. CONCLUSIONS Despite concerns regarding vascularization and pressure near the ICV, these cases demonstrate that NPWT can be safely utilized following TI anastomosis. This challenges conventional guidelines and supports its use, even in high-risk anastomoses.