OpenMed/multicare-articles huggingface dataset:
Unveiling an oral hemangiolymphangioma
Authors: Rívea Inês Ferreira-Santos, Kamilla Alves Santos, Alexandre Prado Scherma, Jorge Esquiche León, Estela Kaminagakura
Journal: Autops Case Rep (2023)
Keywords: angiokeratoma, covid-19, case reports, lymphangioma, tongue
MeSH terms: Case Reports

Hemangiolymphangioma is a very rare vascular malformation that develops as a combination of dilated venous and lymphatic vessels. We describe an unusual case of hemangiolymphangioma of the tongue affecting an adult man who complained of an uncomfortable, slowly progressing exophytic irregular dark red-violaceous nodular mass on his tongue that impaired speech and swallowing for two weeks. The clinical differential diagnoses were Kaposi's sarcoma and a COVID-19-related lesion. A complete blood count and serology for HIV-1 and 2 and RT-PCR for COVID-19 were requested and results were negative. An incisional biopsy was performed. Microscopically, the lesion exhibited several dilated vessels lined by normal-appearing endothelial cells, some filled with prominent intravascular erythrocytes and others containing proteinaceous eosinophilic material resembling lymphatic vessels, in close association with hyperkeratosis, papillomatosis, and acanthosis. From immunohistochemical analysis, most vessels were found to be CD34 positive, some highlighted by α-SMA, whereas D2-40 was focal. Positive staining for some lymphatic and blood vessel markers, i.e., D2-40 and CD34, respectively, indicates a mixed derivation of the lesion. HHV-8 was negative. Clinical features, the congested blood vessels with ectasia in intimate association with hyperplastic epithelium, and the immunohistochemical profile supported the final diagnosis of oral hemangiolymphangioma. The patient underwent minimally invasive surgical excision with no intercurrences. After 18 months of follow-up, there were no signs of relapse.
Case Report: Semantic Variant Primary Progressive Aphasia With Impaired Verbal Word Discrimination
Authors: Nobuko Kawakami, Ayumi Morita, Shigenori Kanno, Nanayo Ogawa, Kazuo Kakinuma, Yumiko Saito, Erena Kobayashi, Wataru Narita, Kyoko Suzuki
Journal: Front Neurol (2022)
Keywords: auditory agnosia, nonverbal sound discrimination, primary progressive aphasia, semantic variant, verbal word discrimination, verbal word recognition, word discrimination
MeSH terms: Case Reports

Some patients with primary progressive aphasia (PPA) present with various types of hearing deficits. Research on the auditory function and speech sounds in PPA, including temporal, phonemic, and prosodic processing, revealed impairment in some of these auditory processes. Many patients with PPA who present with impaired word recognition subsequently developed non-fluent variant PPA. Herein, we present a patient with semantic variant PPA (svPPA) who demonstrated impaired verbal word discrimination. Audiological examinations revealed normal auditory brainstem responses and slightly impaired pure-tone perception. By contrast, verbal word discrimination and monosyllable identification were impaired, and temporal auditory acuity deteriorated. Analyses of brain magnetic resonance images revealed a significant decrease in the gray matter volume in bilateral superior temporal areas, predominantly on the left, compared with those of patients with typical svPPA, which appeared to be associated with impaired word recognition in our patient.
Carbon monoxide poisoning-induced cardiomyopathy from charcoal at a barbecue restaurant: a case report
Authors: Hyun-Jun Kim, Yun Kyung Chung, Kyeong Min Kwak, Se-Jin Ahn, Yong-Hyun Kim, Young-Su Ju, Young-Jun Kwon, Eun-A Kim
Journal: Ann Occup Environ Med (2015)
Keywords: carbon monoxide poisoning, cardiomyopathy, charcoal, restaurant worker
MeSH terms: Case Reports

Objective:
Acute carbon monoxide poisoning has important clinical value because it can cause severe adverse cardiovascular effects and sudden death. Acute carbon monoxide poisoning due to charcoal is well reported worldwide, and increased use of charcoal in the restaurant industry raises concern for an increase in occupational health problems. We present a case of carbon monoxide poisoning induced cardiomyopathy in a 47-year-old restaurant worker.
Materials and methods:
A male patient was brought to the emergency department to syncope and complained of left chest pain. Cardiac angiography and electrocardiography were performed to rule out acute ischemic heart disease, and cardiac markers were checked. After relief of the symptoms and stabilization of the cardiac markers, the patient was discharged without any complications.
Results:
Electrocardiography was normal, but cardiac angiography showed up to a 40% midsegmental stenosis of the right coronary artery with thrombotic plaque. The level of cardiac markers was elevated at least 5 to 10 times higher than the normal value, and the carboxyhemoglobin concentration was 35% measured at one hour after syncope. Following the diagnosis of acute carbon monoxide poisoning induced cardiomyopathy, the patient's medical history and work exposure history were examined. He was found to have been exposed to burning charcoal constantly during his work hours.
Conclusions:
Severe exposure to carbon monoxide was evident in the patient because of high carboxyhemoglobin concentration and highly elevated cardiac enzymes. We concluded that this exposure led to subsequent cardiac injury. He was diagnosed with acute carbon monoxide poisoning-induced cardiomyopathy due to an unsafe working environment. According to the results, the risk of exposure to noxious chemicals such as carbon monoxide by workers in the food service industry is potentially high, and workers in this sector should be educated and monitored by the occupational health service to prevent adverse effects.
Nonhealing scrotal ulceration-an unusual manifestation of TB epididymo-orchitis: case report and review of literature
Authors: Umesh Jayarajah, Pamathy Gnanaselvam, Sivasuriya Sivaganesh
Journal: Clin Case Rep (2017)
Keywords: case report, chronic epididymo‐orchitis, genitourinary tuberculosis, scrotal ulcers
MeSH terms: Case Reports

The clinical presentation of genitourinary tuberculosis (TB) may be variable and a high index of suspicion is required for a timely diagnosis, especially in endemic areas. Recurrent scrotal ulcers associated with epididymo-orchitis even without other constitutional symptoms should alert the clinician of a possible diagnosis of TB.
Delayed spontaneous remission of acquired factor V inhibitor refractory to immunosuppressive therapy with pregnancy-associated improvement
Authors: Andrea Ceglédi, János Dolgos, Mónika Fekete, László Gopcsa, Andrea Várkonyi, Beáta Vilimi, Gábor Mikala, Imre Bodó
Journal: Pathol Oncol Res (2023)
Keywords: autoimmune, bleeding disorder, coagulopathy, factor v inhibitor, gestation
MeSH terms: Cesarean Section, Factor V, Case Reports, Adult, Child, Cyclophosphamide / therapeutic use, Cyclosporine, Dexamethasone, Female, Humans, Immunosuppression Therapy, Immunosuppressive Agents / therapeutic use, Pregnancy, Remission, Spontaneous, Retrospective Studies, Rituximab, Sirolimus, Factor V, Rituximab, Immunosuppressive Agents, Cyclophosphamide, Cyclosporine, Dexamethasone, Sirolimus

Introduction: Acquired factor V inhibitor (AFVI) is a rare autoimmune bleeding disorder. The treatment of AFVI is challenging, and patients often require both bleeding control and inhibitor eradication. Methods: We conducted a retrospective analysis of the medical records of a 35-year-old Caucasian woman who presented with severe AFVI-induced bleeding and subsequent immunosuppressive therapy. Results: To provide haemostasis, rFVIIa was given with good efficacy. The patient was treated with various combinations of immunosuppressive regimens over the course of 2.5 years, including plasmapheresis plus immunoglobulins, dexamethasone + rituximab, cyclophosphamide + dexamethasone + rituximab + cyclosporine, cyclosporin + sirolimus + cyclophosphamide + dexamethasone, bortezomib + sirolimus + methylprednisolone, and sirolimus + mycophenolate mofetil. Although these treatment modalities resulted in intermittent partial reversals of AFVI over 2.5 years, eventually the inhibitor became therapy-resistant. However, following the discontinuation of all immunosuppressive therapy, the patient experienced a partial spontaneous remission, which was followed by a pregnancy. During the pregnancy, the FV activity increased to 54% and the coagulation parameters returned to normal levels. The patient underwent Caesarean section without any bleeding complications and delivered a healthy child. Discussion: The use of an activated bypassing agent for bleeding control is effective in patients with severe AFVI. The presented case is unique because the treatment regimens included multiple combinations of immunosuppressive agents. This demonstrates that AFVI patients may undergo spontaneous remission even after multiple courses of ineffective immunosuppressive protocols. Additionally, pregnancy-associated improvement of AFVI is an important finding that warrants further investigation.
Drug-Drug Interaction of Ergotamine with a Combination of Darunavir, Abacavir, and Lamivudine Causing a Fatal Vasospastic Ischemia
Authors: Elisabeth Petit, Karen Schoonheydt, Philippe Meert, Marie Van Laer
Journal: Case Rep Emerg Med (2018)
MeSH terms: Case Reports

Ergotamine toxicity has become a rare condition which can be caused by, among others, drug-drug interaction. In this work we report a case with vasospastic ischemia induced by the wrongful combination of ergotamine with recently started Antiretroviral Therapy. Clinicians were not aware that patient was self-medicating for years with medication containing ergotamine and caffeine for migraines. This diagnosis was established after evaluating the evolving 'and spreading' ischemia and CT scans and thoroughly interviewing patient's family. Treatment was started with intravenous nimodipine and intra-arterial sodium nitroprusside on the affected limbs. The patient developed severe limb ischemia, cerebral ischemia, and metabolic encephalopathy. Unfortunately no improvements were noticeable and due to evolving cerebral edema as a result of the ischemia, the patient developed brain herniation and died shortly after.
Successful temporary resection of a ruptured hepatoblastoma without preoperative chemotherapy: A case report presenting a novel surgical strategy
Authors: Masaki Horiike, Maya Sogabe, Sinchul Jwa, Sadao Tokimasa, Shoji Kubo
Journal: Int J Surg Case Rep (2020)
Keywords: case report, emergency surgery, hepatoblastoma, lung metastases, temporary tumor resection, tumor rupture
MeSH terms: Case Reports

Introduction:
Although spontaneous tumor rupture is a serious complication of hepatoblastoma, there is no consensus regarding the treatment strategy in infants. We report a patient with lung metastases who had a ruptured hepatoblastoma prior to the start of the scheduled chemotherapy and was successfully treated with a combined treatment including liver resection, lung resection, and chemotherapy.
Presentation of case:
A 22-month-old boy with a ruptured hepatoblastoma and lung metastases underwent an emergency laparotomy with complete tumor resection, followed by chemotherapy. Moreover, a barely detectable metastatic lung lesion shown by a chest CT scan was resected after the fifth chemotherapy treatment. Both postoperative and chemotherapy courses were uneventful. The patient survived without any recurrent hepatoblastoma 2 years after the emergency surgery despite the poor prognosis indicated by distant metastases at the time of diagnosis.
Discussion:
Because rupture itself can be the main cause of death in patients with hepatoblastoma, emergency tumor hemostasis is essential. However, there are no reports comparing the prognosis of the treatment method performing tumor hemostasis alone, tumor resection after chemotherapy, and tumor hemostasis and resection at the same time. The clinical course of the patient indicates that performing tumor hemostasis and resection simultaneously and lung resection after chemotherapy is an effective option to treat a ruptured hepatoblastoma with disseminated tumors and lung metastases if the patient's condition is stable.
Conclusion:
Aggressive treatment with surgery and chemotherapy is an effective option for ruptured hepatoblastoma with disseminated tumors and lung metastases in infants.
Case report: High-grade endometrial stromal sarcoma with adrenal glands metastases: An unreported site of metastasis
Authors: Tao Zhang, Run-Lin Feng, Si-Fan Yin, Wen-Bo Feng, Zhi-Yuan Yin, Hao Wang, Chang-Xing Ke
Journal: Front Oncol (2022)
Keywords: adrenal glands, clinical features, diagnosis, endometrial stromal sarcoma, treatment
MeSH terms: Case Reports

Background:
Endometrial stromal sarcoma is a relatively rare malignant tumor that derives from the malignant transformation of primitive uterine mesenchymal cells. It can lead to distant metastases. High-grade endometrial stromal sarcoma is extremely rare. The adrenal glands are an unreported site of metastasis.
Case summary:
A 71-year-old woman with a diagnosis of endometrial stromal sarcoma 30 months ago. After receiving treatment with radiotherapy and chemotherapy, the patient was kept asymptomatic during the follow-up until 2 years later, when she complained of dyspnea. Pulmonary and right adrenal gland metastases were detected by 18F-FDG PET/CT. The right upper lobe mass was diagnosed as a high-grade endometrial stromal sarcoma metastasis after postoperative pathology. Due to the patient's high risk of surgery, as she had many underlying diseases, we performed adequate preoperative preparation. The physical examination revealed that a hard mass was palpable in the right renal area. The right adrenal mass was resected in our hospital. Immunohistochemistry showed ER (-), PR (-), CD10 (+), P16 (+), Ki-67 (50%). The final diagnosis on pathological examination was a high-grade ESS metastatic to the right adrenal gland. The patient continued treatment in other hospitals after surgical resection. After four months of postoperative follow-up, metastasis was detected again during a PET/CT examination at an outside hospital.
Conclusion:
Endometrial stromal sarcoma is rare, and the adrenal glands are an unreported site of metastasis. It has no specific clinical symptoms and mainly found for other reasons. The diagnosis still depends on pathology and immunohistochemistry. If there is no relevant past history, it is difficult to exclude a primary adrenal tumor.
Irreparable Radial Nerve Palsy Due to Delayed Diagnostic Management of a Giant Lipoma at the Proximal Forearm Resulting in a Triple Tendon Transfer Procedure: Case report and Brief Review of Literature
Authors: Ingo Schmidt
Journal: Open Orthop J (2017)
Keywords: elbow joint, functional recovery, giant lipoma, neurotmesis, radial nerve, radial nerve palsy, triple tendon transfer

Background:
Non-traumatic radial nerve palsy (RNP) caused by local tumors is a rare and uncommon entity.
Methods:
A 62-year-old female presented with a left non-traumatic RNP, initially starting with weakness only. It was caused by a benign giant lipoma at the proximal forearm that was misdiagnosed over a period of 2 years. The slowly growth of the tumor led to an irreparable overstretching-related partial nerve disruption. For functional recovery of the patient, a triple tendon transfer procedure had to be performed.
Results:
Four months after surgery, the patient was completely able to perform her activities of daily living again. At the 10-months follow-up, strength of wrist extension, thumb's extension and abduction, and long fingers II-V extension had all improved to grade 4 in Medical Research Council scale (0-5). In order to restore motion, the patient reported that she would undergo the same triple tendon transfer procedure a second time where necessary. Due to the initially misdiagnosed tumor, there was an overall delayed duration of time for functional recovery of the patient.
Conclusion:
The triple tendon transfer procedure offers a useful and reliable method to restore functionality for patients sustaining irreparable RNP. However, it must be noted critically with our patient that this procedure probably would have been avoided. Initially, there was weakness only by entrapment of the radial nerve. RNP caused by local tumors are uncommon but known from the literature, and so it should be considered generally in differential diagnosis of non-traumatic RNP.
A case report of Mycoplasma wenyonii associated immune-mediated haemolytic anaemia in a dairy cow
Authors: Nicola Gladden, Hayley Haining, Livia Henderson, Francesco Marchesi, Libby Graham, Michael McDonald, Fraser R Murdoch, Anna Bruguera Sala, Jayne Orr, Kathryn Ellis
Journal: Ir Vet J (2016)
Keywords: bovine, immune-mediated haemolytic anaemia, mycoplasma wenyonii

Background and case presentation:
A three year old, second lactation Holstein dairy cow presented to the Scottish Centre for Production Animal Health and Food Safety, Glasgow University Veterinary School in November 2014 with a history of post-calving vulval/vaginal bleeding nine days prior to presentation, followed by a sudden reduction in milk yield. Subsequent investigations resulted in a diagnosis of immune-mediated haemolytic anaemia secondary to infection with Mycoplasma wenyonii.
Conclusion:
This report of a novel presentation of Mycoplasma wenyonii in a dairy cow illustrates the need to consider M.wenyonii as a potential differential diagnosis when a cow presents with anaemia and will discuss the potential implications of the condition at herd-level.
An Unusual Hybrid Salivary Gland Tumor: Molecular Analysis Informs the Potential Pathogenesis of This Rare Neoplasm
Authors: Yan Zhou, Ernesto Martinez Duarte, David J Eleff, Laura J Tafe, Jason M Leibowitz, Darcy A Kerr
Journal: Case Rep Pathol (2019)
MeSH terms: Case Reports

Background:
The presence of two or more tumor entities growing in adjacent locations within the salivary gland is very rare, and pathologic studies on such lesions are limited, particularly those with molecular information. Since the clinical history and imaging studies are usually nonspecific, accurate diagnosis and clinical management largely depend on a thorough histological examination.
Methods and results:
We describe a 71-year-old man with an unusual case of hybrid salivary gland tumor composed of mucoepidermoid carcinoma and basal cell adenoma. Molecular analysis revealed differing driver genetic alterations in each component.
Conclusions:
Hybrid salivary gland tumors are rare, and their pathogenesis is controversial. The combination of mucoepidermoid carcinoma and basal cell adenoma has not been previously described. While malignant transformation of adenoma to carcinoma seems plausible, gene sequencing was more suggestive of their independent derivation. Key to appropriate surgical management is identifying the more aggressive component, ideally at the time of intraoperative consultation.
Heart Transplantation in Mustard Patients Bridged With Continuous Flow Systemic Ventricular Assist Device - A Case Report and Review of Literature
Authors: Rody G Bou Chaaya, Joel W Simon, Mark Turrentine, Jeremy L Herrmann, William Aaron Kay, Maya Guglin, Kashif Saleem, Roopa A Rao
Journal: Front Cardiovasc Med (2021)
Keywords: d-tga, heart transplantation, mustard procedure, pulmonary hypertension, pulmonary hypertension reversal, transposition of the great arteries, ventricular assist device
MeSH terms: Case Reports

Thirty four-year-old male with history of D-transposition of the great arteries (D-TGA) who underwent Mustard operation at 14 months of age presented in cardiogenic shock secondary to severe systemic right ventricular failure. Catheterization revealed significantly increased pulmonary pressures. Due to the patient's inotrope dependence and prohibitive pulmonary hypertension, he underwent implantation of a Heart Ware HVAD® for systemic RV support. Within 4 months of continuous flow ventricular assist device (VAD) implantation complete normalization of pulmonary vascular resistance (PVR) was achieved. He ultimately underwent orthotopic heart transplantation with favorable outcomes. This is the second report of complete normalization of PVR following VAD implantation into a systemic RV in <4 months. We conducted a thorough literature review to identify Mustard patients that received systemic RV VAD as a bridge to a successful heart transplantation. In this article, we summarize the outcomes and focus on pulmonary hypertension reversibility following VAD implant.
Osteogenic Sarcoma in an Adolescent With Cystic Fibrosis: Successful Treatment Despite Significant Obstacles
Authors: Thomas J C Ruffles, Ryan Black, Wayne Nicholls, Barbara Laing, Alan Isles
Journal: Front Pediatr (2018)
Keywords: chemotherapy, cystic fibrosis, non-tuberculous mycobacteria, osteogenic sarcoma, tibia
MeSH terms: Case Reports

Introduction: We describe the case of a 16-year old male with cystic fiborosis (CF) who presented with an osteosarcoma of his right distal tibia. Case Report: Treatment consisted of neoadjuvant chemotherapy of cisplatin, doxorubicin and high dose methotrexate followed by distal tibial resection and free fibula flap reconstruction and consolidation chemotherapy. Treatment was complicated by a pulmonary exacerbation, where Pseudomonas aeruginosa (PsA) and Staphylococcus aureus were grown on sputum culture which was treated with a 2-week course of intravenous piptazobactam and tobramycin. Mycobacterium intracellulare and Mycobacterium abscessus were also cultured following commencement of chemotherapy and successfully treated with a 6-month course of oral azithromycin, ethambutol, and moxifloxacin along with a 1-month course of inhaled amikacin. Pulmonary function improved during his treatment from baseline FEV1 of 3.8 l (93.9%) to 4.15 l (102.3% predicted) whilst nutritional status remained stable. Discussion: The combination of CF and osteosarcoma is rare with only one previous case reported (1). Our case is instructive as the patient faced the challenge of chronic PsA and the first reported culturing and successful treatment of non-tuberculous mycobacterium (NTM) during chemotherapy. Fatal outcomes have been reported previously for CF patients during immunosuppression (2). In concordance with our findings, a recent report noted an improvement in respiratory function in a child treated for leukemia (3). The anti-inflammatory nature of some chemotherapy agents could be responsible for the observed clinical improvement in CF with low dose methotrexate having been shown to increase FEV1 in adolescents with advanced CF (4). Whilst doxorubicin could improve pulmonary outcomes through increased total cellular CFTR protein expression and CFTR associated chloride secretion (5). It is hypothesized that the improved pulmonary function in patients with CF who require chemotherapy could be due to increased production of Multi-Drug Resistance Proteins (MDR) and Multi-Drug Resistant Associated Proteins (MRP) that may complement the depleted CFTR protein (6). Concluding Remarks: We report the well-tolerated management of osteosarcoma in a patient with CF including the first reported identification and eradication of NTM during chemotherapy. The observed positive pulmonary outcome following chemotherapy highlights several potential cellular mechanisms that deserve to be explored.