Showing papers in "Journal of Medical Case Reports in 2022"

Inflammatory myopathy occurring shortly after severe acute respiratory syndrome coronavirus 2 vaccination: two case reports

Abstract: Vaccination remains the cornerstone approach to exiting the current global coronavirus disease 2019 pandemic caused by severe acute respiratory syndrome coronavirus 2. The novel messenger ribonucleic acid vaccines offer a high level of protection and are widely used throughout the world. With more people receiving the vaccines, better understanding of their relative safety can be reached. In this report, we describe two patients who developed inflammatory myopathy within 48 hours of receiving the Pfizer BNT162b2 vaccine.Patient A, a 55-year-old South East Asian woman, presented with a 6-week history of pruritic facial and torso rash and a 1-week history of worsening proximal myopathy. Her rash first developed 2 days after receiving the first dose of BNT162b2 vaccine. Patient B, a 72-year-old Caucasian woman, presented with a 2-week history of proximal myopathy a day after receiving the second dose of BNT162b2 vaccine. Both patients had elevated creatine kinase on admission. Patient A tested positive for anti-Mi-2a antibody and anti-Ro-52 antibody, while Patient B was positive for anti-fibrillarin antibody. Magnetic resonance imaging subsequently confirmed generalized acute muscle inflammation and subcutaneous inflammation consistent with inflammatory myositis. Both patients did not have a previous history or family history of autoimmune disease. Patients A and B were diagnosed with dermatomyositis and inflammatory myositis, respectively. They were initially treated with pulsed intravenous methylprednisolone followed by oral prednisolone. However, as their conditions were resistant to corticosteroids, both eventually received and responded well to intravenous immunoglobulin therapy.There are previously reported cases of severe acute respiratory syndrome coronavirus 2-induced and other vaccine-related inflammatory myopathies. However, the precise mechanisms are not elucidated. Without more evidence and convincing pathophysiology, it is not possible to conclude that our patients developed inflammatory myopathy because of the vaccine. However, the timing of the disease onset and the lack of previous history raise an important question of this novel messenger ribonucleic acid therapy.

Hyperbaric oxygen treatment for long coronavirus disease-19: a case report

Abstract: The coronavirus disease 2019 pandemic has resulted in a growing population of individuals who experience a wide range of persistent symptoms referred to as "long COVID." Symptoms include neurocognitive impairment and fatigue. Two potential mechanisms could be responsible for these long-term unremitting symptoms: hypercoagulability, which increases the risk of blood vessel occlusion, and an uncontrolled continuous inflammatory response. Currently, no known treatment is available for long COVID. One of the options to reverse hypoxia, reduce neuroinflammation, and induce neuroplasticity is hyperbaric oxygen therapy. In this article, we present the first case report of a previously healthy athletic individual who suffered from long COVID syndrome treated successfully with hyperbaric oxygen therapy.A previously healthy 55-year-old Caucasian man presented 3 months after severe coronavirus disease 2019 infection with long COVID syndrome. His symptoms included a decline in memory, multitasking abilities, energy, breathing, and physical fitness. After evaluation that included brain perfusion magnetic resonance imaging, diffusion tensor imaging, computerized cognitive tests, and cardiopulmonary test, he was treated with hyperbaric oxygen therapy. Each session included exposure to 90 minutes of 100% oxygen at 2 atmosphere absolute pressure with 5-minute air breaks every 20 minutes for 60 sessions, 5 days per week. Evaluation after completing the treatment showed significant improvements in brain perfusion and microstructure by magnetic resonance imaging and significant improvement in memory with the most dominant effect being on nonverbal memory, executive functions, attention, information procession speed, cognitive flexibility, and multitasking. The improved cognitive functions correlated with the increased cerebral blood flow in brain regions as measured by perfusion magnetic resonance imaging. With regard to physical capacity, there was a 34% increase in the maximum rate of oxygen consumed during exercise and a 44% improvement in forced vital capacity. The improved physical measurements correlated with the regain of his pre-COVID physical capacity.We report the first case of successfully treated long COVID symptoms with hyperbaric oxygen therapy with improvements in cognition and cardiopulmonary function. The beneficial effects of hyperbaric oxygen shed additional light on the pathophysiology of long COVID. As this is a single case report, further prospective randomized control studies are needed.

Vomiting, electrolyte disturbance, and medications; the perfect storm for acquired long QT syndrome and cardiac arrest: a case report

Abstract: Acquired long QT syndrome is an important and preventable cause of cardiac arrest. Certain medications and electrolyte disturbance are common contributors, and often coexist. In this case, we report five contributors to cardiac arrest.This case is of a 51-year-old Caucasian female patient who presented with vomiting associated with hypokalemia and hypomagnesemia. She subsequently received ondansetron and metoclopramide, on the background of chronic treatment with fluoxetine. She then suffered an in-hospital monitored cardiac arrest, with features of long QT and torsades de pointes retrospectively noted on her prearrest electrocardiogram. She was diagnosed with acquired long QT syndrome, and her QT interval later normalized after removal of offending causes.This case highlights the importance of proper consideration prior to prescribing QT prolonging medications, especially in patients who have other risk factors for prolonged QT, such as electrolyte disturbances and pretreatment with QT prolonging medications.

Aggressive (deep) angiomyxoma of the vulva: a case report

Abstract: Aggressive angiomyxoma of the vulva is a benign, slow-growing tumor originating from myxoid cells of connective tissue. The tumor is known for multiple local recurrences with a low tendency to metastasize. Only around 350 cases have been documented in the scientific literature so far.We report a case of a 40-year-old North Indian, unmarried woman with a swelling on the left labium majora for 7 years, along with surface ulceration over the mass. Local examination showed a well-circumscribed, 8 × 8 cm pedunculated mass arising from the left labium majora with an overlying ulcer measuring 6 cm × 4 cm. After taking informed written consent, wide local excision of the mass and surrounding margins was done under anesthesia. Histopathology was suggestive of aggressive angiomyxoma. Immunohistochemistry was done, which was positive for estrogen and progesterone receptors. Her postoperative recovery was uneventful. The patient was given three doses of gonadotropin-releasing hormone agonist (injection, leuprolide 3.75 mg) once a month. No recurrence has been reported so far on follow-up visits for 1 year.Aggressive angiomyxoma is one of the differential diagnoses for vulvovaginal growth in a female. As the tumor is well known for local recurrences, correct diagnosis and appropriate management using a multidisciplinary approach are crucial to managing such patients.

Herpetic anterior uveitis following Pfizer–BioNTech coronavirus disease 2019 vaccine: two case reports

Abstract: To describe two cases of herpetic anterior uveitis after inoculation of the first dose of Pfizer-BioNTech coronavirus disease 2019 vaccine.Case 1: a healthy 92-year-old Caucasian woman developed symptomatic unilateral anterior uveitis for 3 days after Pfizer-BioNTech coronavirus disease 2019 vaccination (Pfizer Inc.). The episode fully resolved with topical and oral antiviral treatment. Case 2: a previously healthy 85-year-old Caucasian woman with left hemicranial signs of herpes zoster infection, associated with herpetic keratouveitis for 3 days after Pfizer-BioNTech coronavirus disease 2019 vaccination. Treatment with topical antibiotics and both oral and topical antiherpetic medication was administered, and she recovered successfully in 5 weeks.Clinicians should be aware of the possibility of eye inflammation in the form of herpetic reactivation after coronavirus disease 2019 vaccination.

Mononeuritis multiplex as a rare and severe neurological complication of immune checkpoint inhibitors: a case report

Abstract: Mononeuritis multiplex is a rare autoimmune peripheral neuropathy that typically presents in the context of vasculitis, diabetes, infection, or as a paraneoplastic syndrome. Adverse immune-related neurological conditions have been increasingly reported with the use of immune checkpoint inhibitors against cytotoxic T-lymphocyte antigen-4 and/or the programmed cell death protein 1/programmed death ligand-1 axis. Mononeuritis multiplex has only been reported twice from treatment of cancers with immunotherapy.Here we report a case of mononeuritis multiplex as a complication of immune checkpoint inhibitor therapy for melanoma. An 80-year-old non-Hispanic white female with recurrent melanoma was treated with combination ipilimumab and nivolumab and subsequently presented with progressive leg weakness, back pain, and difficulty ambulating. The diagnosis of mononeuritis multiplex was made, which was resistant to steroid pulses, chronic steroids, intravenous immunoglobulin, and rituximab. She developed progressive neurologic dysfunction and elected for hospice care. We found only two other cases reported in the literature.Increased awareness, prompt recognition, and aggressive treatments are likely the best opportunity for improved outcomes in this severe side effect.

Multisystem inflammatory syndrome in adults: a case report and review of the literature

Abstract: The current coronavirus disease pandemic has brought recognition of multisystem inflammatory syndrome in adults as a de novo entity, temporally associated with severe acute respiratory syndrome coronavirus 2 viral infection in adults. Hypothesis about its true pathophysiology remains controversial.The patient was a 22-year-old African American female presenting to the emergency department with fever, sore throat, and neck swelling for the past 3 days. During her initial emergency department visit, her blood pressure was stable at 110/57 mmHg, temperature of 39.4 °C, and heart rate of 150 beats per minute. While in the emergency department, she received broad-spectrum antibiotics (vancomycin and ceftriaxone) and 30 cc/kg bolus of normal saline. Originally, she was admitted to a telemetry floor. The following night, a rapid response code was called due to hypotension. At that time, her blood pressure was 80/57 mmHg. She appeared comfortable without signs of respiratory distress. She received intravenous fluids and vasopressors, and was transferred to the intensive care unit. The patient had reported a previous coronavirus disease infection a few weeks prior. She was diagnosed and treated for multisystem inflammatory syndrome in adults. Intravenous immunoglobulin infusion was initiated and completed on hospital day 5. She was weaned off vasopressors by day 6, and discharged home on day 11.Our case report is an example of the presentation, diagnosis, and management of multisystem inflammatory syndrome. Our research into previous case reports illustrates the wide range of presentations, degree of end organ damage, and treatment modalities. This diagnosis needs to be considered in the presence of recent coronavirus disease infection with new-onset end organ failure, as prompt diagnosis and treatment is crucial for better outcomes.

Nivolumab-related myasthenia gravis with myositis requiring prolonged mechanical ventilation: a case report

Abstract: Nivolumab is an immune checkpoint inhibitor that blocks inhibitors of T-cell activation and blunts antitumor immunity and is used in the treatment of various cancers. However, immune checkpoint inhibitors have immune-related adverse effects on various organs due to promoting T-cell activity against host tissues by blocking inhibition of T-cell function. Although immune-related adverse effects including hepatitis, colitis, pneumonitis, dermatitis, nephritis, endocrinopathies, and hypophysitis are well recognized with established treatment guidelines, neuromuscular immune-related adverse effects are rare phenomena.A 55-year-old Asian (Japanese) woman was diagnosed with nivolumab-related myasthenia gravis with myositis and myocarditis. She had a past history of thymectomy for large thymoma with a high anti-acetylcholine receptor antibody level without any symptoms. Nivolumab was administered for the treatment of malignant melanoma. Creatine kinase levels began to rise 2 weeks after the administration, and abnormal neurological findings appeared 3 weeks after the administration. Ventricular arrhythmia, wide QRS complex, and dyssynchrony of the left ventricle also appeared. Intravenous immunoglobulin and corticosteroids were administered, and plasma exchange was performed. The patient required intensive care and prolonged mechanical ventilation with tracheostomy owing to weakness of the diaphragm; she was eventually weaned from the ventilator and discharged. Diaphragm ultrasound was used for the decision-making of the weaning strategy and evaluation of the diaphragmatic function.Nivolumab-induced severe myasthenia gravis with myositis and myocarditis required intensive care and prolonged mechanical ventilation. Although immune checkpoint inhibitor-related myasthenia gravis is a rare adverse event, appropriate and prompt treatment is required because of its severity and rapid progression. Diaphragm ultrasound was useful not only in diagnosing diaphragm dysfunction and deciding the strategy for weaning from mechanical ventilation but also in evaluating the recovery of the diaphragmatic function.

Spontaneous tumor lysis syndrome in adrenal adenocarcinoma: a case report and review of the literature

Abstract: Tumor lysis syndrome is an oncologic emergency that classically occurs following cancer therapy, although spontaneous tumor lysis syndrome can also occur in malignancies, albeit rarely. Spontaneous tumor lysis syndrome has previously been reported in some hematologic malignancies, but it rarely happens in solid tumors and seems to be associated with a higher mortality rate. This is the first case of adrenal adenocarcinoma that developed spontaneous tumor lysis syndrome.We present a rare case of spontaneous tumor lysis syndrome occurring in a patient previously diagnosed with adrenal adenocarcinoma. The patient was a 64-year-old Persian man with abdominal pain, hypersomnia, and fatigue who was previously diagnosed with right adrenocortical carcinoma and had undergone right adrenalectomy with regional lymph nodes resection 5 months previously. On physical examination, the patient had abdominal distension and mild tenderness at the right upper quadrant. Pitting edema was detected bilaterally in the lower extremities. Initial imaging revealed multiple and large lesions suggestive of liver metastases. The laboratory data showed hyperkalemia, hyperuricemia, hyperphosphatemia, and elevated serum creatinine level indicative of spontaneous tumor lysis syndrome in the patient. Despite immediate and intensive care with antibiotics, hydration, treatment with a hypouricemic agent, and renal replacement therapy, the patient ultimately died from multiorgan failure.Tumor lysis syndrome in solid tumors has high mortality. Patients susceptible to spontaneous tumor lysis syndrome must receive aggressive treatment immediately, which is crucial for preventing morbidity and mortality. Spontaneous tumor lysis syndrome may be underdiagnosed, and a high degree of clinical suspicion is needed to make the diagnosis and proceed with required interventions. Therefore, clinicians should be aware of this rare phenomenon.

Malignancies in patients with cystic fibrosis: a case series

Abstract: Previous reports have shown an increased number of colorectal cancers in patients with cystic fibrosis. We assessed the database of our cystic fibrosis center to identify patients with all kinds of cancer retrospectively. All patients visiting the Cystic Fibrosis Centre Innsbruck between 1995 and 2019 were included.Among 229 patients with cystic fibrosis treated at the Cystic Fibrosis Centre in Innsbruck between 1995 and 2019, 11 subjects were diagnosed with a malignant disease. The median age at diagnosis was 25.2 years (mean 24.3 years). There were four gynecological malignancies (cervical intraepithelial neoplasia and cervical cancer), two hematological malignancies (acute lymphocytic leukemia), one gastrointestinal malignancy (peritoneal mesothelioma), and four malignancies from other origins (malignant melanoma, neuroblastoma, adrenocortical carcinoma, and thyroid cancer). One malignancy occurred after lung transplantation. There was a strong preponderance of females, with 10 of the 11 cases occurring in women. Six deaths were attributed to cancer.Most diagnoses were made below 30 years of age, and half of the subjects died from the malignant disease. Awareness of a possible malignancy is needed in patients with atypical symptoms. Regular screenings for cancer should also be considered, not only for gastrointestinal tumors.

Differential abscopal effect in extracranial and intracranial lesions after radiotherapy alone for vertebral bone metastasis of unknown primary: a case report

Abstract: The abscopal effect is a phenomenon in which a tumor located far from irradiated lesions regresses. We have experienced a case in which both intracranial and extracranial lesions showed an abscopal effect after radiotherapy for spinal metastases of unknown primary. We report the differential abscopal effect in extracranial and intracranial lesions.A 57-year-old Japanese man was diagnosed with multiple lung nodules, bone metastases, and brain metastases. The results of pathological examination at the previous hospital he visited suggested adenocarcinoma of the lung. However, there was a possibility that the biopsy specimen was inadequate. Radiation therapy was performed on the ninth thoracic vertebra for a total dose of 39 Gy in 13 fractions because the lesion in the ninth thoracic vertebra was destructively extending. After thorough examination, the primary lesion could not be identified, and we made diagnosis of cancer of unknown primary. The patient did not want to receive systemic chemotherapy; however, all of the lesions except for the brain metastases had spontaneously shrunk 2 months after radiation therapy. Although the brain metastases had partially shrunk, whole-brain radiotherapy for a total dose of 36 Gy in 12 fractions was performed. Fifteen months after initial radiation therapy, the brain metastasis recurred, and Gamma Knife radiosurgery was additionally performed. The brain metastases disappeared after the radiosurgery. During a period of 30 months after radiation therapy for the ninth vertebra, the lesions of the trunk all maintained their shrinkage without systemic chemotherapy. Right cervical lymph node metastasis and brain metastases occurred 30 months after the initial radiation therapy. A biopsy of the right cervical lymph node led to the diagnosis of clear cell carcinoma. Although we considered additional radiation therapy or chemotherapy, the patient died 3 months after the progression of recurrence lesions.We report a rare case in which radiotherapy alone for an extracranial metastatic lesion of a vertebra resulted in an abscopal effect on both extracranial and intracranial lesions. Notably, the abscopal effect in the intracranial lesions was weaker than that in the extracranial lesions.

Complete response to combination therapy using nivolumab and ipilimumab for metastatic, sarcomatoid collecting duct carcinoma presenting with high expression of programmed death-ligand 1: a case report

Abstract: Collecting duct carcinoma and sarcomatoid renal cell carcinoma are tumors with poor prognosis. Immune checkpoint inhibitors have been established as the standard treatment for advanced renal cell carcinoma. Some cases of remission of collecting duct carcinoma and sarcomatoid renal cell carcinoma have been reported using immune checkpoint inhibitor interventions. Specifically, sarcomatoid renal cell carcinoma expresses high levels of programmed death-ligand 1, an immune checkpoint protein, and immune checkpoint inhibitors have been reported to be highly effective for treating sarcomatoid renal cell carcinoma.We describe the case of a 70-year-old Japanese male who underwent radical right nephrectomy for a right renal mass identified on computed tomography. The pathological examination demonstrated that the renal mass was urothelial carcinoma and collecting duct carcinoma with sarcomatoid changes, and programmed death-ligand 1 was highly expressed with a tumor proportion score of more than 10%. There was no evident submucosal connective tissue invasion in the urothelial carcinoma component, and collecting duct carcinoma was diagnosed as primary cancer. The tumor-node-metastasis classification was pT3aN0, venous invasion 1, lymphovascular invasion 0, and Fuhrman nuclear grade 4. Two months after the nephrectomy, multiple metastases were observed in both lungs, the right hilar lymph node, and the S6 segment of the right liver lobe. We initiated first-line combination therapy with nivolumab (240 mg, fixed dose) and ipilimumab (1 mg/kg). One day after administration, the patient developed drug-induced interstitial pneumonia, thus we applied steroid injections. After one administration of immunotherapy, the metastatic lesion showed complete response within 6 months, which was maintained after 3 years.We report the first case of complete response to a single dose of combination therapy with nivolumab and ipilimumab for metastatic collecting duct carcinoma with sarcomatoid changes and high expression of programmed death-ligand 1. This case suggests high expectations for immune checkpoint inhibitors as treatment for sarcomatoid-transformed renal carcinoma tumors that express high levels of programmed death-ligand 1.

Cytomegalovirus pneumonitis amid COVID-19 chaos: the hidden enemy—a case report

Abstract: The effect of coronavirus disease 2019 on the immune system is increasingly recognized. When severe, it causes immune dysregulation that may favor other infections, including Herpesviridae. Cytomegalovirus shares many innate immune pathways with severe acute respiratory syndrome coronavirus 2, which may potentiate each other. We describe a case of cytomegalovirus pneumonitis complicating the course of coronavirus disease 2019 in a patient with systemic lupus erythematosus/systemic sclerosis overlap and usual interstitial pneumonia, mimicking interstitial lung disease exacerbation. To the best of the authors' knowledge, this is the first case to be reported worldwide in the setting of connective tissue disease-associated interstitial lung disease.We describe the case of a 47-year-old white/Yemeni female who is known to have systemic lupus erythematosus/scleroderma overlap and usual interstitial pneumonia who was initially admitted with severe coronavirus disease 2019 pneumonia mandating intensive care. After initial improvement, it was later complicated with cytomegalovirus pneumonitis, mimicking interstitial lung disease exacerbation. The case was successfully treated with ganciclovir.Intriguingly, severe acute respiratory syndrome coronavirus 2 and cytomegalovirus may potentiate each other, since they share some innate immune pathways. Subjects with severe coronavirus disease 2019 and underlying connective tissue diseases and those who are immunosuppressed carry higher risk compared with other cohorts, which may mandate active surveillance for cytomegalovirus coinfection or reactivation. Among various immunosuppressive therapies that has been tried for cytokine storm, use of anti-interleukin-6 inhibitors in the aforementioned population may carry more harm than previously thought, which may suggest that is reasonable to omit its use in treating this group with coronavirus disease 2019. This case underlines an underrecognized and underreported cause of morbidity and mortality during the course of severe coronavirus disease 2019 and will help to alert clinicians of its occurrence.

Pseudoprogression in advanced non-small cell lung cancer treated with combination chemoimmunotherapy: a case report

Abstract: Pseudoprogression, the initial apparent worsening of cancer prior to eventual improvement, is a documented feature of immune checkpoint inhibitor administration and presents a challenge to clinicians distinguishing true progression from pseudoprogression. This phenomenon does not typically occur with traditional cytotoxic chemotherapy. We present a case in which a patient treated with combination carboplatin-pemetrexed plus pembrolizumab experienced transient radiographic worsening of disease with subsequent regression.A 65-year-old never-smoking white male with advanced sarcomatoid non-small cell lung cancer (NSCLC) harboring a MET exon 14 skipping mutation and with PD-L1 tumor proportion score of 80% was initiated on combination chemotherapy plus immune checkpoint inhibitor (ICI) therapy after progression on a MET inhibitor. After two cycles of carboplatin-pemetrexed plus pembrolizumab, repeat imaging suggested disease progression. Following discontinuation of the carboplatin-pemetrexed plus pembrolizumab regimen, the patient reported improved symptoms and energy levels, which were attributed to the waning of treatment-associated toxicities. On the day prior to initiation of the next planned line of therapy, repeat imaging was preformed to provide a baseline for treatment efficacy. Imaging revealed improvement compared to the prior imaging. Chemotherapy with carboplatin-pemetrexed plus pembrolizumab was resumed, with response ongoing 8 months later.Pseudoprogression is a documented feature of ICI administration. Pseudoprogression is not typically observed in patients treated with traditional cytotoxic chemotherapy and has not yet been documented in patients treated with combination cytotoxic chemotherapy plus immunotherapy. At this time, there are no reliable means to predict or diagnose these rare events; therefore, more studies should be conducted to understand which patients are predisposed to developing this phenomenon and to increase clinical recognition.

Acute pancreatitis in pregnancy following COVID-19 vaccine: a case report

Abstract: Since the approval of the Pfizer-BioNTech (BNT162b2) mRNA vaccine for COVID-19 infection, a few adverse effects have been reported. Acute pancreatitis has been reported in a few patients. However, there is currently no research showing a direct relationship between the vaccine and acute pancreatitis. Here, we report a case of acute pancreatitis following Pfizer vaccination in a young healthy pregnant woman without any known risk factors. To our knowledge, this is the first case report of possible vaccine-induced pancreatitis in a pregnant woman.The patient, a 24-year-old South-Asian female, at 31 weeks of gestation, presented with severe epigastric pain radiating to the back and worsening on lying supine, associated with nausea and vomiting. She was diagnosed with acute pancreatitis with a serum lipase level of 4376 U/L and an ultrasound showing features of pancreatitis. The patient received her first dose of the Pfizer vaccine 1 week prior to these symptoms. Detailed evaluation did not show any etiological cause of pancreatitis. The patient had a spontaneous vaginal delivery and the baby was shifted to the neonatal intensive care unit in a stable condition. A computed tomography scan postpartum (day 2) demonstrated acute interstitial edematous pancreatitis. The patient was managed conservatively in the intensive care unit and discharged home in a stable condition.This report highlights the importance of a detailed history and evaluation, and the close monitoring of any patient presenting with abdominal pain after vaccination. Acute pancreatitis can be fatal if not picked up early.

Gross hematuria can be an impact of severe acute respiratory syndrome coronavirus 2 vaccination on immunoglobulin A nephropathy: a case report

Abstract: Immunoglobulin A nephropathy is typically accelerated by upper respiratory tract infections and can relapse following vaccination. There have been reports of patients who presented with immunoglobulin A nephropathy flares with or without gross hematuria following coronavirus disease 2019 vaccination. However, this relationship remains to be elucidated.Herein, we present the case of a patient with newly diagnosed immunoglobulin A nephropathy who presented with gross hematuria following the second dose of coronavirus disease 2019 vaccine. A 21-year-old Japanese woman presented with fever and new-onset gross hematuria 1 day after receiving the second dose of the coronavirus disease 2019 vaccine (Pfizer). She had microhematuria without proteinuria for 2 years at the time of her medical check-up. Gross hematuria resolved 6 days after the second dose of the coronavirus disease 2019 vaccine; however, microhematuria (> 100 per high-power field) and mild proteinuria were observed. She was admitted to our hospital 4 weeks after the second vaccination because of persistent urinary abnormalities. She was well before the vaccination and did not have any pulmonary involvement on chest radiography or any symptoms suggestive of coronavirus disease 2019. Renal biopsy revealed an immunoglobulin A nephropathy. The Oxford MEST-C score was M0E0S0T0C0. Our patient's urinary abnormalities implied exacerbation of immunoglobulin A nephropathy after coronavirus disease 2019 vaccination.In our case, gross hematuria served as a trigger for immunoglobulin A nephropathy diagnosis, suggesting that nephrologists should pay close attention to gross hematuria and urinalysis after coronavirus disease 2019 vaccination.

Long-term survival after definitive proton beam therapy for oligorecurrent esophageal squamous cell carcinoma: a case report

Abstract: Abstract Background Radical esophagectomy for esophageal squamous cell carcinoma has improved survival, but the rate of recurrence is high. Patients of recurrent esophageal squamous cell carcinoma after failure of chemotherapy have a poor prognosis. We herein report the achievement of long-term survival after definitive proton beam therapy for oligorecurrent esophageal squamous cell carcinoma after failure of chemotherapy. Case presentation A 60-year-old Japanese man was diagnosed as having squamous cell carcinoma of the lower thoracic esophagus (cT2N0M0, stage IIA). He underwent two courses of neoadjuvant chemotherapy with cisplatin and 5-fluorouracil, and esophagectomy with three-field lymphadenectomy was performed. Microscopic findings after resection showed two lymph node metastases (ypT2N1M0, stage IIB). Five months after resection, a computed tomography scan revealed a solitary liver metastasis in the S4 area. He underwent three courses of chemotherapy with cisplatin and 5-fluorouracil; however, positron emission tomography revealed two lymph node metastases. Surgeons recommended second-line chemotherapy, but the patient refused chemotherapy and requested proton beam therapy. We performed proton beam therapy without chemotherapy for the liver metastasis and lymph node metastases, with total doses of 79.2 and 60 Gy relative biological effectiveness, respectively, according to the tumor location. An acute side effect of grade 1 dermatitis occurred after proton beam therapy, but there was no acute or late complication of more than grade 2. The patient remains in complete remission 5 years after treatment without surgery or chemotherapy. Discussion and conclusions Proton beam therapy exerted a curative effect on oligorecurrent esophageal squamous cell carcinoma. This is the first report on the achievement of long-term survival after definitive proton beam therapy for oligorecurrent esophageal squamous cell carcinoma.

Guillain–Barré/Miller Fisher overlap syndrome in a patient after coronavirus disease-2019 infection: a case report

Abstract: Beyond the typical respiratory symptoms associated with novel coronavirus, increasing evidence has been reported of the neurological manifestations affecting both the central and peripheral nervous systems.We observed a 30-year-old Persian woman developing acute motor sensory axonal neuropathy, a variant of Guillain-Barré syndrome that overlaps Miller Fisher syndrome, 30 days after confirmed coronavirus disease-2019 infection. Our case highlight the rare occurrence of Guillain-Barré syndrome overlapping with Miller Fisher during the coronavirus disease-2019 pandemic. These neurologic manifestations may occur because of an aberrant immune response to coronavirus disease-2019.The early recognition of Guillain-Barré syndrome symptoms is critical, given the associated severe motor disabilities that may seriously limit the quality of life of these patients. We may still have much to learn about the co-occurrence of Guillain-Barré syndrome and Miller Fisher to improve the quality of life of these patients requiring an accurate evaluation by neurologists.

Iatrogenic ischiofemoral impingement due to high tibial osteotomy with overvalgization: a case report

Abstract: Open wedge high tibial osteotomy is a standard procedure for frontal realignment. It is indicated in varus knee with reduced mechanical medial proximal tibia angle. Overcorrection producing a mechanical medial proximal tibia angle out of the normal range (85-90°) is not recommended because this would lead to unphysiological joint-line orientation. Osteotomies around the knee also influence the adjacent ankle and hip joints. For the hip, it is known that frontal alignment of the leg influences the ischiofemoral space. A decreased ischiofemoral space can lead to painful impingement between the ischial bone and the lesser trochanter.A 53-year-old German woman presented with severe ischiofemoral impingement symptoms and valgus malalignment of the left leg after open wedge high tibial osteotomy, which was indicated and performed by an orthopedic surgeon with intention to treat medial knee pain due to degenerative arthritis of the medial compartment years after medial meniscectomy. The mechanical medial proximal tibia angle was 100.5°. We performed closed wedge high tibial osteotomy producing a mechanical medial proximal tibia angle of 90.0° and normal joint-line orientation. The hip pain was gone immediately after the surgery, and the patient had no signs of ischiofemoral impingement or hip pain at last follow-up 12 months after closed wedge high tibial osteotomy.Frontal realignment osteotomy around the knee can create problems at adjacent joints. Overvalgization of the proximal tibia made the patient compensate by hyperadduction of the hip to enable full foot sole contact with the floor. Hyperadduction of the hip decreased the ischiofemoral space, leading to severe impingement. Therefore, meticulous planning of osteotomies is important not to produce unphysiological situations or unwanted negative effects at the level of an adjacent joint.

Concomitant myocarditis and painless thyroiditis after AstraZeneca coronavirus disease 2019 vaccination: a case report

Abstract: Incidence of myocarditis following messenger RNA coronavirus disease 2019 vaccination has been widely described, but this clinical scenario after adenoviral vector coronavirus disease 2019 vaccination has only been rarely reported. In addition, a few case reports of thyroiditis after adenoviral vector coronavirus disease 2019 vaccination have been published.A 55-year-old Thai woman presented with palpitation without neck pain 14 days after receiving AstraZeneca coronavirus disease 2019 vaccination. Electrocardiography revealed sinus tachycardia. Her blood tests showed elevation of cardiac troponin and free triiodothyronine with suppressed serum thyroid stimulating hormone, reflecting a hyperthyroid status. Evidence of myocardial inflammation and necrosis from cardiac magnetic resonance imaging supported the diagnosis of recent myocarditis. Laboratory results and imaging findings were consistent with thyroiditis. After 3 weeks of symptomatic treatment, her symptom and blood tests had returned to normal.This case demonstrates that the adenoviral vector coronavirus disease 2019 vaccine could possibly cause myocarditis and painless thyroiditis. Clinicians should have a high index of suspicion and promptly evaluate these conditions, despite minimal symptoms.

Two different presentations of de novo variants of CSNK2B: two case reports

Abstract: Poirier-Bienvenu neurodevelopmental syndrome is a neurologic disorder caused by mutations in the CSNK2B gene. It is mostly characterized by early-onset seizures, hypotonia, and mild dysmorphic features. Craniodigital syndrome is a recently described disorder also related to CSNK2B, with a single report in the literature.To report two unrelated cases of children harboring CSNK2B variants (NM_001320.6) who presented with distinct diseases.Case 1 is a 7-month-old, Caucasian, female patient with chief complaints of severe hypotonia and drug-refractory myoclonic epilepsy, with a likely pathogenic de novo variant c.494A>G (p.His165Arg). Case 2 is a 5-year-old male, Latino patient with craniodigital intellectual disability syndrome subjacent to a de novo, likely pathogenic variant c.94G>T (p.Asp32Tyr). His dysmorphic features included facial dysmorphisms, supernumerary nipples, and left-hand postaxial polydactyly.This report suggest that the CSNK2B gene may be involved in the physiopathology of neurodevelopmental disorders and variable dysmorphic features.

Methanol poisoning outbreak in Saudi Arabia: a case series

Abstract: Outbreaks of methanol poisoning have been described in the medical literature in different regions around the world. Even though in Saudi Arabia a few outbreaks of methanol poisoning have occurred, they remain undocumented. Herein, we describe several cases of methanol poisoning in Saudi Arabia with the goal of increasing awareness about the dangers of methanol poisoning among healthcare staff.Nine middle-aged Saudi patients (five men aged 24, 26, 27, 36, and 49 years and four females aged 19, 20, 24, and 25 years) were admitted to our emergency department after alcohol consumption. All patients presented with severe metabolic acidosis and some visual impairment. Treatment was initiated based on the clinical suspicion of methanol intoxication because of laboratory test limitations and time constraints. Patients showed improvement and favorable hospital outcomes after aggressive empirical treatment.Many social and cultural factors influence the lack of reporting of methanol poisoning cases in Saudi Arabia. We believe it is important to document these outbreaks to increase the knowledge among healthcare providers and promote public health awareness. A high index of suspicion and the development of local public health networks to monitor, survey, follow-up, and facilitate data exchange can help healthcare providers recognize and aggressively treat affected individuals. Early empiric and aggressive management can greatly decrease morbidity and mortality despite challenges and limited resources.

Successful treatment with proton beam therapy for a solitary sternal metastasis of breast cancer: a case report

Abstract: Breast cancer infrequently metastasizes to the sternum as solitary metastasis. We experienced successful treatment with proton beam therapy for a case of sternal metastasis of breast cancer. This case demonstrates for the first time the role of proton therapy in the treatment of oligometastatic sternal metastasis with limited tolerance of normal tissue due to previous photon irradiation.A 40-year-old Japanese female presented with lumpiness in her left breast. The patient was diagnosed with breast cancer (cT1N0M0, cStage IA) and underwent partial mastectomy with axillary lymph node dissection. After the mastectomy, the patient received radiation therapy with 50 Gy in 25 fractions for initial irradiation of the left breast. After the initial irradiation of 50 Gy, the patient received 10 Gy in five fractions of a sequential boost for the tumor bed to a total dose of 60 Gy. Although the patient was administered tamoxifen after radiation therapy, solitary sternal metastasis occurred 6 months after radiation therapy. She refused chemotherapy and requested proton beam therapy for her sternal metastasis. The daily proton beam therapy fractions were 2.5 relative biological effectiveness, receiving a total dose of 70 Gy relative biological effectiveness. An acute side effect of grade 2 dermatitis according to the National Cancer Institute Common Terminology Criteria for Adverse Events version 4.0. occurred during proton beam therapy, but there was no acute or late complication of more than grade 3. At 3 years after proton beam therapy, the patient remains in complete remission without surgery or chemotherapy.Proton beam therapy for solitary sternal metastasis of breast cancer is considered to be a therapeutic option.

Chronic expanding hematoma of the left erector spinae muscle after stereotactic body radiotherapy for renal cell carcinoma: a case report

Abstract: Hematomas that slowly increase in size for more than 1 month after the initial hemorrhage are referred to as chronic expanding hematomas. Chronic expanding hematoma can also occur after radiosurgery; however, there have been no reports about chronic expanding hematoma in the trunk after stereotactic body radiotherapy. We report a case of chronic expanding hematoma of the left erector spinae muscle after stereotactic body radiotherapy for renal cell carcinoma.A 74-year-old Japanese male complained of back pain 7 years after stereotactic body radiotherapy for renal cell carcinoma of the left kidney. There was no history of surgery or trauma to his back. After stereotactic body radiotherapy, there was no acute or late complication of more than grade 2. The renal cell carcinoma did not show shrinkage or progression, and he was diagnosed with stable disease on computed tomography. The patient remains in a stable disease condition 7 years after treatment without surgery or chemotherapy; however, he came to the hospital with gradually worsening back pain for several months. Computed tomography revealed the left erector spinae muscle to be swollen compared with the contralateral side at the third lumbar level. Ultrasonography showed a tumor of 30 mm in size without blood flow in the left paraspinal muscle. Positron emission tomography-computed tomography revealed uptake in the left paraspinal muscle. Pathological examination showed radiation-induced chronic expanding hematoma.We present the first case report of chronic expanding hematoma of the left erector spinae muscle after stereotactic body radiotherapy for renal cell carcinoma. Usually, stereotactic body radiotherapy for renal cell carcinoma would be considered unlikely to cause chronic expanding hematoma, but the introduction of dialysis and antiplatelet drugs may have increased the risk.

Whole-exome sequencing in a subject with fluctuating neuropsychiatric symptoms, immunoglobulin G1 deficiency, and subsequent development of Crohn’s disease: a case report

Abstract: Mutations or polymorphisms of genes that are associated with inflammasome functions are known to predispose individuals to Crohn's disease and likely affect clinical presentations and responses to therapeutic agents in patients with Crohn's disease. The presence of additional gene mutations/polymorphisms that can modify immune responses may further affect clinical features, making diagnosis and management of Crohn's disease even more challenging. Whole-exome sequencing is expected to be instrumental in understanding atypical presentations of Crohn's disease and the selection of therapeutic measures, especially when multiple gene mutations/polymorphisms affect patients with Crohn's disease. We report the case of a non-Hispanic Caucasian female patient with Crohn's disease who was initially diagnosed with pediatric acute-onset neuropsychiatric syndrome with fluctuating anxiety symptoms at 9 years of age. This patient was initially managed with pulse oral corticosteroid treatment and then intravenous immunoglobulin due to her immunoglobulin G1 deficiency. At 15 years of age, she was diagnosed with Crohn's disease, following onset of acute abdomen. Treatment with oral corticosteroid and then tumor necrosis factor-α blockers (adalimumab and infliximab) led to remission of Crohn's disease. However, she continued to suffer from chronic abdominal pain, persistent headache, general fatigue, and joint ache involving multiple joints. Extensive gastrointestinal workup was unrevealing, but whole-exome sequencing identified two autosomal dominant gene variants: NLRP12 (loss of function) and IRF2BP2 (gain of function). Based on whole-exome sequencing findings, infliximab was discontinued and anakinra, an interleukin-1β blocker, was started, rendering marked improvement of her clinical symptoms. However, Crohn's disease lesions recurred following Yersinia enterocolitis. The patient was successfully treated with a blocker of interleukin-12p40 (ustekinumab), and anakinra was discontinued following remission of her Crohn's disease lesions.Loss-of-function mutation of NRLRP12 gene augments production of interleukin-1β and tumor necrosis factor-α, while gain-of-function mutation of IRF2BP2 impairs cytokine production and B cell differentiation. We propose that the presence of these two autosomal dominant variants caused an atypical clinical presentation of Crohn's disease.

Acute right-sided ischemic colitis in a COVID-19 patient: a case report and review of the literature

Abstract: In addition to attacking the respiratory system, the coronavirus disease may attack the gastrointestinal tract in various ways, one of which is by creating a coagulopathy that may lead to acute ischemia of the bowel, increasing morbidity and mortality rates in these patients.We present a case of a white 72-year-old European male, who was admitted to the intensive care unit after developing COVID-19-induced acute respiratory distress syndrome. On the third week, despite a favorable evolution of his respiratory symptoms, the patient became clinically septic; laboratory findings showed an augmentation of his D-dimer, fibrinogen, C-reactive protein, and procalcitonin levels. Imaging showed signs of ischemia of the right colon. The patient was taken to the operating room; only the right side of his colon was ischemic, with a well demarcated cut-off. A laparoscopic right hemicolectomy with a terminal ileostomy was performed. The patient was able to go home 2 weeks after surgery.Ischemic colitis is an uncommon pathology in the general population, and is rare in COVID-19 patients. Most cases of ischemic colitis in COVID-19 patients in the literature were limited to the left colon, with < 10 cases involving the right colon. Accurate and quick diagnosis with appropriate management is the key to avoid any mortality in those patients who are already weakened by the coronavirus.

Superficial venous thrombosisas a possible consequence of ChAdOx1 nCoV-19 vaccine: two case reports

Abstract: Many scientists across the world got involved in the race to develop successful anti-SARS-CoV-2 vaccines to overcome COVID-19 pandemic. Among the different vaccines developed against SARS-CoV-2, Covishield was the first vaccine approved for emergency use in Nepal. We report two cases of Superficial Vein Thrombosis (SVT) for the first time in the literature after vaccination with the Chimpanzee Adenovirus-vectored Vaccine (ChAdOx1 nCoV-19 vaccine).Two cases, a 24-year-old young Chhetri male and a 62-year-old Chhetri female who have received Covishield (ChAdOx1 nCoV-19) vaccine, developed pain in left calf after 2 weeks and 10 weeks of vaccination, respectively. Both the case belongs to the Chhetri ethnic group of Nepal. The pain became severe on the fourth week of immunization in the first case while the pain was acute and severe on the 10th week of vaccination in the second case. The first presented to emergency room and second case was referred to the emergency room from Orthopedic Clinic. On evaluation the first patient had normal vitals with no history of fever and swelling yet displayed non-radiating mild to moderate intensity pain localized to left leg below the knee which became aggravated by movements. In the second case however pain was more intense with other characteristics as first case. Both cases had low wells score (< 4). On local examination tenderness was noted on squeezing but other systemic examination findings of the patient were within normal limits in both cases. Among the numerous vaccines used to fight the battle against COVID-19 disease, the ChAdOx1 nCoV-19 vaccine, Covishield, has been widely used in Nepal and India. Apart from other minor side effects, in few cases thromboses have been reported after vaccination of ChAdOx1 nCoV-19, Covishield, vaccine.These cases reporting Superficial Vein Thrombosis may be an additional adverse effect to the list of adverse events associated with ChAdOx1 nCoV-19, Covishield, vaccine. However, the benefits of the vaccine in breaking the chain of COVID 19 spread are certainly greater than the risk of thromboses.

Spontaneous regression of breast lymphoproliferative disorders after withdrawal of methotrexate in rheumatoid arthritis patients with Epstein–Barr virus infection: a case report and review of the literature

Abstract: Lymphoproliferative disorder (LPD) has been shown to occur after treatment with methotrexate (MTX). Currently, MTX-LPD has become widely recognized, but its mechanism and prognostic factors remain unclear.We report the first case of Epstein-Barr virus (EBV)-associated MTX-LPD of the breast. A 63-year-old Asian woman with long-term rheumatoid arthritis presented to our facility with intermittent fever. A physical examination revealed a 3-cm lump in her left breast. She had been taking MTX for the past 15 years. Laboratory studies revealed slightly elevated levels of EBV-viral capsid antigen antibody immunoglobulin G and EBV nuclear antibody. Contrast-enhanced computer tomography revealed a mass in the left breast, a subcutaneous nodule in the abdomen, a mass in the left lung, and a nodule in the left retroperitoneum. The definitive diagnosis was consistent with MTX-LPD merging into an EBV-positive, diffuse large B-cell lymphoma. Six months following the withdrawal of MTX, the breast mass had markedly shrunk and the patient remained in good health for 1 year with no evidence of relapse of LPD.MTX-LPD rarely occurs in the breast, and it is difficult to diagnose because there have only been six reported cases of breast MTX-LPD reported in the literature. EBV-positive MTX-LPD tends to regress spontaneously after MTX withdrawal, and our case also had similar results. It is important to make an appropriate diagnosis of MTX-LPD of the breast based on imaging and pathology to determine the appropriate treatment protocol for this rare disorder.

Ruptured endometrioma in a nonpregnant patient: a case report

Abstract: Endometriomas are a type of ovarian cyst composed of degenerated blood products from hemorrhage of ectopic endometrial tissue. Endometriomas can rupture, causing hemoperitoneum, and present with signs and symptoms similar to other, more common abdominal emergencies. Therefore, they are not often diagnosed preoperatively. Ultrasound and cross-sectional imaging can assist in diagnosis of endometriomas. We present a case of ruptured endometrioma causing massive hemoperitoneum that was initially suspected to represent malignancy with carcinomatosis.A 32-year-old Hispanic woman presented with sharp abdominal pain and 15-pound unintentional weight loss over 6 months. Laboratory work was significant for a negative pregnancy test and elevated cancer antigen-125. Computed tomography of the abdomen and pelvis demonstrated a 13-cm complex cystic mass in the left adnexa with moderate hyperdense ascites and omental nodularity. Ultrasound demonstrated a large left adnexal complex cystic structure with internal echoes, and chest computed tomography showed no signs of intrathoracic neoplastic or infectious processes. Her presentation was concerning for malignancy with carcinomatosis. Fluid from a paracentesis was sent for culture and cytology. Diagnostic laparoscopy revealed that the left ovary had been completely replaced by an endometrioma, which had a small ruptured area superiorly. Brown deposits of endometriosis were present on the cyst, omentum, and various peritoneal linings. Tissue samples of the endometrium, myometrium, cervix, ovaries, fallopian tubes, peritoneum, omentum, and paracolic spaces were taken and showed no hyperplastic, dysplastic, or malignant cells on pathology.Ruptured endometrioma and ruptured hemorrhagic cyst should be included in the differential diagnosis when a premenopausal female presents with hemoperitoneum in combination with complex adnexal cystic masses in the absence of trauma. Cancer antigen-125 and cancer antigen 19-9 can be falsely elevated in the setting of ruptured endometrioma.

Giant parathyroid adenoma: a case report

Abstract: Giant parathyroid adenoma is a type of parathyroid adenoma weighing > 3.5 g and having a size of more than 2 cm.This report describes giant primary parathyroid adenoma with reference to the literature. We report the case of a 48-year-old Persian man referred to the clinic with knee and lower back pain. He had a history of mitral valve replacement and several episodes of bilateral nephrolithiasis. After a thorough assessment, a neck mass with a possible thyroid origin was detected, but further assessment showed it was of parathyroid origin. The resected mass was 9 × 6× 4 cm and weighed 122 g, and histopathology showed a giant parathyroid adenoma.Giant parathyroid adenomas that weigh more than 110 g and are larger than 8 cm can lead to significant hypercalcemia. Despite giant parathyroid adenomas and high parathyroid hormone levels, a calcium crisis may not always occur in these patients, and the masses may be initially misdiagnosed as a thyroid mass.