Journal of Medical Case Reports

Journal Information
ISSN / EISSN : 1752-1947 / 1752-1947
Current Publisher: Springer Science and Business Media LLC (10.1186)
Former Publisher: Cases Network, Ltd. (10.4076)
Total articles ≅ 2,993
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, Alina Paraschiv, Petrișor Tudorașcu, Diana Tudorașcu, Constantin Bataiosu, Adrian Balșeanu
Journal of Medical Case Reports, Volume 15, pp 1-9; doi:10.1186/s13256-021-02862-x

Background Left ventricular noncompaction is a rare cardiomyopathy characterized by a thin, compacted epicardial layer and a noncompacted endocardial layer, with trabeculations and recesses that communicate with the left ventricular cavity. In the advanced stage of the disease, the classical triad of heart failure, ventricular arrhythmia, and systemic embolization is common. Segments involved are the apex and mid inferior and lateral walls. The right ventricular apex may be affected as well. Case presentation A 29-year-old Caucasian male was hospitalized with dyspnea and fatigue at minimal exertion during the last months before admission. He also described a history of edema of the legs and abdominal pain in the last weeks. Physical examination revealed dyspnea, pulmonary rales, cardiomegaly, hepatomegaly, and splenomegaly. Electrocardiography showed sinus rhythm with nonspecific repolarization changes. Twenty-four-hour Holter monitoring identified ventricular tachycardia episodes with right bundle branch block morphology. Transthoracic echocardiography at admission revealed dilated left ventricle with trabeculations located predominantly at the apex but also in the apical and mid portion of lateral and inferior wall; end-systolic ratio of noncompacted to compacted layers > 2; moderate mitral regurgitation; and reduced left ventricular ejection fraction. Between apical trabeculations, multiple thrombi were found. The right ventricle had normal morphology and function. Speckle-tracking echocardiography also revealed systolic left ventricle dysfunction and solid body rotation. Abdominal echocardiography showed hepatomegaly and splenomegaly. Abdominal computed tomography was suggestive for hepatic and renal infarctions. Laboratory tests revealed high levels of N-terminal pro-brain natriuretic peptide and liver enzymes. Cardiac magnetic resonance evaluation at 1 month after discharge confirmed the diagnosis. The patient received anticoagulants, antiarrhythmics, and heart failure treatment. After 2 months, before device implantation, he presented clinical improvement, and echocardiographic evaluation did not detect thrombi in the left ventricle. Coronary angiography was within normal range. A cardioverter defibrillator was implanted for prevention of sudden cardiac death. Conclusions Left ventricular noncompaction is rare cardiomyopathy, but it should always be considered as a possible diagnosis in a patient hospitalized with heart failure, ventricular arrhythmias, and systemic embolic events. Echocardiography and cardiac magnetic resonance are essential imaging tools for diagnosis and follow-up.
, A. Di Giacomo, E. Lacotte, F. Porcheret, N. Letouzé, B. Lauzier, I. Goyer, D. Brossier
Journal of Medical Case Reports, Volume 15, pp 1-8; doi:10.1186/s13256-021-02889-0

Background The principles for maintenance intravenous fluid prescription in children were developed in the 1950s. These guidelines based on the use of hypotonic solutions have been challenged regularly for they seem to be associated with an increased risk of hospital-acquired hyponatremia. Case presentation We report the case of a 4-week-old Caucasian child admitted for acute bronchiolitis who received hypotonic maintenance fluids and developed severe hyponatremia (94 mmol/L) with hyponatremic encephalopathy. Conclusion This clinical situation can serve as a reminder of the latest recommendations from the American Academy of Pediatrics regarding the use of intravenous fluids that promote the use of isotonic fluids in children.
Tatsuo Horiuchi, , Shinya Sakamoto, Masaki Orihara, Akihiko Yokohama, Mutsumi Uchiyama, Shigeru Saito
Journal of Medical Case Reports, Volume 15, pp 1-4; doi:10.1186/s13256-021-02894-3

Background Sugammadex is a synthetic γ-cyclodextrin derivative designed to selectively bind to steroidal neuromuscular blocking agents and reverse their effects. Although many cases of sugammadex-induced anaphylaxis have been reported, few studies have investigated the underlying mechanism. Case presentation A 55-year-old Japanese man underwent a laryngectomy under general anesthesia. One month before laryngectomy, he had undergone laryngoscopy under general anesthesia and received sugammadex administration without causing hypersensitivity. He had no history of allergies. The operation was finished without complications. Shortly after sugammadex administration, his blood pressure dropped to approximately 70 mmHg, and his heart rate increased to 110 beats/minute with systemic erythema. Suspecting anaphylaxis, he was treated with the intravenous injection of phenylephrine, d-chlorpheniramine, and hydrocortisone. After these treatments, his cardiovascular condition stabilized. Eight months after the event, skin prick tests and intradermal tests with all agents used during general anesthesia were performed. Intradermal tests showed positive results only for sugammadex. Subsequently, basophil activation tests with CD203c were performed using sugammadex, γ-cyclodextrin, and positive controls (anti-immunoglobulin-E and formyl-methionyl-leucyl-phenylalanine). In addition to both controls, sugammadex, but not γ-cyclodextrin, induced significant upregulation of CD203c expression. We performed additional basophil activation tests with wortmannin, an inhibitor of phosphoinositide 3-kinase, to investigate the mechanism underlying sugammadex-induced basophil activation. The inhibitory effect of wortmannin on basophil activation due to sugammadex was similar to that of anti-immunoglobulin-E, suggesting an immunoglobulin-E-dependent mechanism. Although the patient showed no hypersensitivity after the first exposure of sugammadex, anaphylaxis appeared after the second administration. Because most cases of sugammadex-induced anaphylaxis reportedly appeared after first administration, this seems to be a rare case. Conclusions In the present case, sugammadex-induced anaphylaxis might have occurred through an immunoglobulin-E-dependent mechanism and not involve γ-cyclodextrin as an epitope. Physicians should pay attention to the occurrence of sugammadex-induced anaphylaxis even when the patient has a history of safe administration of sugammadex.
Journal of Medical Case Reports, Volume 15, pp 1-5; doi:10.1186/s13256-021-02897-0

Background Intercostal neuralgia is most common in patients with herpes zoster, but it might be the initial symptom of serious diseases, such as atraumatic spinal fracture, which may lead to serious consequences if not diagnosed and treated early. Severe intercostal neuralgia is rarely reported as the first symptom of ankylosing spondylitis with atraumatic vertebral fractures. Case presentation A 70-year-old Chinese Han man previously diagnosed with ankylosing spondylitis presented to the hospital with intense intercostal pain without trauma. The patient was initially suspected of having herpes zoster neuralgia; however, he subsequently experienced numbness and weakness of both lower limbs as well as constipation. Thoracic vertebral fracture and compression of the spinal cord were detected with magnetic resonance imaging, and he underwent emergency posterior thoracic spinal canal decompression, and intercostal neuralgia was relieved after surgery. Spinal tuberculosis and tumors were later excluded by pathological examination and follow-up results. A 6-month postoperative follow-up showed that the weakness and numbness of the left lower limb had significantly improved, and his urinary function had recovered. Conclusions Patients with ankylosing spondylitis could develop atraumatic spinal fractures. Severe intercostal neuralgia is an early indicator of spinal fractures, and spinal magnetic resonance imaging is crucial for the diagnosis.
, Hiroshi Nakayama, Tomoya Iseki, Shintaro Onishi, Ryo Iwakura, Shinichi Yoshiya, Toshiya Tachibana
Journal of Medical Case Reports, Volume 15, pp 1-6; doi:10.1186/s13256-021-02893-4

Background To the best of our knowledge, arthroscopic treatment for symptomatic mucoid degeneration of the posterior cruciate ligament in young athletes has not been reported before. Case presentation An 18-year-old Asian male college soccer player presented with a 3-month history of right knee pain without episodes of trauma. Despite conservative treatment over the preceding 3 months, his symptoms persisted. Physical examination of the right knee revealed full range of motion, though posterior knee pain was induced when the knee approached full flexion. On ligament examination, posterior sagging and Lachman test were negative, and no clinical finding indicative of ligament insufficiency was noted. Magnetic resonance imaging showed a diffusely thickened posterior cruciate ligament with increased signal intensity on the T2-weighted sequence. A few intact fibers were observed with continuous margin from origin to insertion. Based on the patient's history and the magnetic resonance imaging findings, we suspected mucoid degeneration of the posterior cruciate ligament as the cause of the patient’s symptoms. Since conservative treatment had failed to relieve the symptoms, arthroscopic treatment was indicated. Arthroscopic examination revealed yellowish crumbly tissues along the thickened posterior cruciate ligament. Tension and bulk of the posterior cruciate ligament were well preserved. Curettage of degenerative tissue and decompression of the posterior cruciate ligament resulted in symptom relief without instability of the knee joint. The patient returned to play at 3 months. At 12 months, postoperative magnetic resonance imaging showed no evidence of recurrence and indicated that the remaining posterior cruciate ligament was thicker than before the surgery. At 2 years follow-up, the patient remained asymptomatic and could play soccer at the same level as before the onset of pain. Conclusions Arthroscopic decompression of the posterior cruciate ligament may relieve knee pain and facilitate early return to play with good functional results.
Omar M. Sharaf, Tomas D. Martin,
Journal of Medical Case Reports, Volume 15, pp 1-4; doi:10.1186/s13256-021-02886-3

Background Acute DeBakey type I and type II aortic dissections are indications for emergent surgical repair; however, there are currently no standard protocols in the management of isolated supra-aortic dissections. Prompt diagnosis and management of an isolated innominate artery dissection are necessary to prevent distal malperfusion and thromboembolic sequelae. Case presentation A 50-year-old Caucasian gentleman presented with chest pain radiating to his jaw and right arm. He had no recent history of trauma. On physical exam, he was neurologically intact and malignantly hypertensive. Computed tomographic angiography of the chest and neck confirmed a spontaneous isolated innominate artery dissection without ascending aorta involvement. Given the lack of evidence for rupture, distal emboli, and/or end-organ malperfusion, the decision was made for initial non-operative management—anti-impulse regimen, antiplatelet therapy, and close follow-up. Conclusions Medical management of a spontaneous isolated innominate artery dissection is appropriate for short-term and potentially long-term therapy. This not only spares the patient from a potentially unnecessary surgical operation but also provides the surgeon and the patient the time to plan for a surgical approach if it becomes necessary.
Esmatullah Esmat, Haider Ali Malakzai, Mujtaba Haidari, Ahmed Maseh Haidary, Merwaise Baha,
Journal of Medical Case Reports, Volume 15, pp 1-4; doi:10.1186/s13256-021-02878-3

Background Polyps of the uterine cervix are one of the most common benign hyperplastic lesions occurring in the female genital tract that usually arise from the endocervical canal and are believed to be the result of reactive changes due to long-standing chronic inflammation, multiparty, and foreign bodies. Cervical polyps are usually small in size (less than 4 cm) that are commonly found in adult women; however, a few cases of giant polyps and the rare occurrence of these lesions in children have also been reported. Heterotopias and malignant transformation in cervical polyps are considered to be very rare. Case presentation We present the case of a 10-year-old Afghan girl with a giant pedunculated mass protruding out of the uterine cervix that was accompanied by abdominal pain and mass sensation. The mass was completely excised by surgical intervention and the specimen was submitted for histopathological evaluation. Upon gross and microscopic examination, the characteristic findings of a hamartomatous polyp with heterologous mesenchymal tissue in the form of mature cartilage were seen. To the best of our knowledge, this is the first case of a giant (17.5 cm) hamartomatous polyp of the uterine cervix in this age group. Conclusion Giant hamartomatous cervical polyps rarely occur in patients below 10 years of age. The majority of these lesions are benign; however, a few cases with malignant transformation are also reported, which demands elaborate investigations into the etiopathogenesis and nature of the lesions.
D. Baur, C. Pfeifle, C. E. Heyde
Journal of Medical Case Reports, Volume 15, pp 1-4; doi:10.1186/s13256-021-02880-9

Background We report a patient who fractured the seventh cervical vertebra while playing a virtual reality (VR) game, without any other trauma. This case report aims to describe the spinal trauma incurred during the use of a VR headset in a video game. Case presentation The Caucasian patient presented with pain and swelling in the lower cervical spine at our clinic after playing a video game involving a combination of shoulder, arm and head movements while wearing a VR headset. Preexisting comorbidities were not present in the 31-year-old male. No history of regular medication use or drug abuse was recorded. After performing a clinical examination and radiological diagnostics, we found a dislocated traumatic fracture of the spinous process of the seventh cervical vertebra. After a soft tissue defect was excluded through magnetic resonance imaging (MRI) diagnostics, a conservative therapy regimen with pain therapy and immobilization was started. After hospitalization, outpatient controls were conducted at 4, 6 and 12 weeks. At 6 weeks after hospitalization, the patient had recovered from the injury without complications. Conclusions Rapid movements during VR gaming can lead to injuries of the cervical spine. In addition to rapid movements, the additional weight of the VR headset as well as the decoupling of audiovisual stimuli from the perceived proprioceptive information should be considered. Determining whether this is an isolated incident induced by unknown preexisting factors or whether the trauma mechanism alone can lead to severe spinal trauma needs to be studied further with additional cases.
Sara Wallengren, Björn Axel Johansson,
Journal of Medical Case Reports, Volume 15, pp 1-5; doi:10.1186/s13256-021-02879-2

Introduction Autoimmune disorders have become increasingly acknowledged as having a more causative role in encephalitis than previously assumed. Anti-N-methyl-d-aspartate receptor encephalitis seems to be the most prevalent disorder. Symptoms of the neuropsychiatric phase in children and adolescents include abnormal behavior, seizures, and neurologic symptoms. We present a report on a teenage girl with predominantly psychiatric symptoms, highlighting the need for awareness of the disease and multidisciplinary collaboration. Case presentation Our patient, a 17-year-old girl of Middle Eastern origin, had no earlier medical history, but a family history of autoimmune disease. One morning, she could not recognize her mother and soon developed increased energy and pressured speech. The condition worsened, with paranoid delusions. In the emergency unit, she ran around speaking incoherently. The condition was interpreted as a full-scale mania. After pediatric clearance, the patient was admitted to the Department of Child and Adolescent Psychiatry. Mood-stabilizing treatment was initiated with second-generation psychotics and lithium, but this brought no improvement. A multidisciplinary discussion was held with physicians from psychiatry and neurology. A lumbar puncture showed N-methyl-d-aspartate receptor antibodies, and autoimmune treatment was initiated. Computed tomography thorax/abdomen revealed a right-sided ovarian tumor. After salpingo-oophorectomy, our patient’s mental status gradually improved, as demonstrated by repeated testing. Seven months post discharge she was in a stable relationship and performing well in school. Conclusion This case underlines the importance of collaboration between child and adolescent psychiatry and pediatrics, and gives pointers for timely diagnosis. Manic adolescents who do not respond to mood-stabilizing treatment should be subjected to further consultations and investigations. Psychiatrists and neurologists should develop an integrated approach to the management of brain disorders.
, Daniel Ng, Laura Nicholson
Journal of Medical Case Reports, Volume 15, pp 1-4; doi:10.1186/s13256-021-02858-7

Background Immune checkpoint inhibitors are effective therapies for a wide range of malignancies. Their increased use has led to a wide range of immune-related adverse effects including skin, gastrointestinal, pulmonary, endocrine, cardiac, and neurologic complications. Case presentation We present the case of a 72-year-old Caucasian man with non-small cell lung cancer who was admitted for dyspnea after two cycles of durvalumab. He was found to have significantly elevated levels of serum creatinine kinase and troponin with a negative cardiac catheterization. During his hospitalization, he developed progressive dyspnea and new-onset axial weakness, ultimately leading to the diagnosis of durvalumab-induced myocarditis, myasthenia gravis, and myositis. Conclusion This is, to our knowledge, the first reported case of anti-programmed cell death ligand 1-induced combination of myocarditis, myasthenia gravis, and myositis. While the use of immunologic agents has resulted in overall improved cancer outcomes, their increased use has led to a vast spectrum of immune-related adverse effects. We review the diagnostic workup and management of patients with these immune-related adverse effects, underscoring the importance of early identification given the potential for rapid deterioration.
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