OpenMed/multicare-articles huggingface dataset:
Herpes simplex virus-1 triggered hemophagocytic lymphohistiocytosis in a patient with granulomatosis with polyangiitis
Authors: Vanessa A States, Meghan E Kapp
Journal: Autops Case Rep (2022)
Keywords: herpes simplex, lymphocytes, lymphohistiocytosis, hemophagocytic, macrophages, vasculitis, central nervous system
MeSH terms: Case Reports

Hemophagocytic lymphohistiocytosis (HLH) is a rare, aggressive hyperinflammatory syndrome in which an inciting event triggers massive, uninhibited activation of T lymphocytes and macrophages. Although viral infections are the most common trigger of HLH, cases of HSV-1 induced HLH are rare in adults. We present the case and postmortem findings of a 27-year-old woman diagnosed with HLH in the setting of immunosuppression for the treatment of granulomatosis with polyangiitis (GPA). Autopsy revealed evidence of herpes simplex virus-1 (HSV-1) infection and no findings suggestive of GPA recurrence.
Catastrophic hemorrhage of adrenal pheochromocytoma following thrombolysis for acute myocardial infarction: case report and literature review
Authors: Tarik Souiki, Zoheir Tekni, Hind Laachach, Amal Bennani, Youssef Zrihni, Azeddine Tadmori, Farida Ajdi, Abderahim Bouazzaoui, Laila Chbani, Hafid Akoudad, Khalid Mazaz, Said Aitlaalim
Journal: World J Emerg Surg (2014)
Keywords: hemorrhage, pheochromocytoma, thrombolysis
MeSH terms: Review

We describe here the case of a 62-year-old man with acute abdominal syndrome and severe hemorrhagic shock following successful thrombolysis for acute cardiac infarction. Emergency surgical exploration revealed extensive intraperitoneal and retroperitoneal hemorrhage resulting from the rupture of a large adrenal tumor. The diagnosis of pheochromocytoma was confirmed by histological findings. The patient died a few hours after surgery from multiorgan failure despite resuscitation attempts. This report discusses the diagnosis difficulties, treatment approach, and relevant literature.
Metachronous Liver Metastasis from Alpha-Fetoprotein-Producing Gastric Cancer Successfully Treated with Capecitabine/Oxaliplatin Combination Chemotherapy
Authors: Soichi Furukawa, Takashi Kobayashi, Saori Shiono, Shuta Nishinakagawa
Journal: Case Rep Surg (2022)
MeSH terms: Case Reports

A consensus regarding the treatment of recurrent alpha-fetoprotein-producing gastric carcinoma due to its rarity is lacking. We herein describe a case of such a carcinoma that was associated with metachronous liver metastasis. A 73-year-old man was referred for the surgical treatment of a type 2 gastric tumor that extended from the greater curvature of the gastric corpus to the pylorus. As no remote metastases were detected, the patient underwent open total gastrectomy with lymphadenectomy and Roux-en-Y reconstruction. Histopathological examination revealed regional lymph node metastasis and the invasion of the muscularis propria by a moderately differentiated adenocarcinoma. Immunostaining of the primary tumor was positive for alpha-fetoprotein and negative for human epidermal growth factor receptor 2. Serum alpha-fetoprotein levels decreased to within normal limits after eight courses of S-1 monotherapy; however, levels started to increase, and a hypovascular nodule in segment 5/6 of the liver was detected 3.5 years later. Serum alpha-fetoprotein levels returned to the normal range, and the tumor was undetectable after four courses of capecitabine and oxaliplatin therapy. No recurrence was detected at 1.5-year follow-up. This case demonstrates that combined capecitabine and oxaliplatin therapy can successfully treat metachronous liver metastasis from alpha-fetoprotein-producing gastric carcinoma.
Teenage Neurologic Manifestation of a Complex Brain Malformation Consisting of Gray Matter Heterotopia, Schizencephaly and Absent Septum Pellucidum
Authors: Nico Hustings, Marc Lemmerling
Journal: J Belg Soc Radiol (2021)
Keywords: congenital brain malformation, dystonia, upper motor neuron syndrome
MeSH terms: Case Reports

Teaching point: Since some minor congenital brain defects manifest long after birth, even in adults they should be kept in the differential of new epileptic seizures or focal neurologic deficits.
Paroxysmal Nocturnal Hemoglobinuria: Diagnostic Challenges in Pediatric Patient
Authors: Dharshana Krishnaprasadh, Inna Kaminecki, Anna Sechser Perl, Jonathan Teitelbaum
Journal: Case Rep Pediatr (2019)
MeSH terms: Case Reports

Paroxysmal nocturnal hemoglobinuria (PNH) is a rare, life-threatening hematologic stem cell disorder characterized by hemoglobinuria, thrombosis, and tendency for bone marrow failure. The rare incidence of PNH in children, its nonspecific clinical presentation, and occasional absence of hemoglobinuria make the diagnosis challenging. We present a case of a 17-year-old boy who was hospitalized with a history of recurrent abdominal pain, fever, and dark-colored urine. Laboratory tests revealed anemia, thrombocytopenia, and elevated inflammatory markers. Urinalysis was positive for protein and red blood cells, too many to be counted. Complement studies were within normal limits. Abdominal computed tomography showed a segment of the small bowel with wall thickening and signs of possible microperforation. Exploratory laparotomy revealed necrosis of the small bowel, and histological evaluation was suggestive of an autoimmune process with small vessel vasculitis. Bone marrow biopsy showed hypocellular marrow with a decreased number of myeloid cells, normal number of megakaryocytes, and signs of erythroid hyperplasia. Flow cytometry detected deficiency of CD59 leading to the diagnosis of PNH. The patient was treated with eculizumab infusions resulting in significant improvement. This case highlights the need for high clinical suspicion for rare entities such as PNH in patients presenting without hemoglobinuria.
A Child as a Donor for Hematopoietic Stem Cell Transplantation: Bioethical Justification-A Case Study on Sickle Cell Disease
Authors: Andrea Z Pereira, Ricardo Hellman, Nelson Hamerschlak, Andrea Kondo, Polianna Mara Rodrigues de Souza, Wilson Leite Pedreira Jr, Luiz Fernando Alves Lima Mantovani, Eduardo Juan Troster, Henrique Grunspun, Marco Aurélio Scarpinella Bueno
Journal: Case Rep Hematol (2017)
MeSH terms: Case Reports

Hematopoietic stem cell transplantation (HSCT) is an important treatment option for children with severe and refractory sickle cell disease (SCD) with debilitating clinical complications. HSCT with cells from the bone marrow of a HLA-identical sibling used in SCD has a low mortality risk, high cure rate, and high event-free survival rate after a median follow-up of 5-6 years. However, matched donors are found in only about 20% of the patients. A boy aged 8 years with SCD had a sister, <2 years old, a fully compatible donor. The boy met all eligibility criteria to undergo HSCT, and he was suffering from cognitive and neurologic impairment due to ischemic events. A Bioethical Committee jointly discussed the ethical issues on this case after a pediatric evaluation released the very young sister for donation. The justification was that the sister would benefit from the donation too because of the greater likelihood of survival and cure and less suffering of her brother. The parents were informed about the risks and benefits for both children, and the family was psychologically evaluated. After their consent, HSCT was performed and the patient is cured from SCD. The complication for the donor was the need for blood transfusion.
Paraneoplastic Neuromyelitis Optica Spectrum Disorder Associated With Lung Adenocarcinoma: A Case Report
Authors: Carlo Maiorca, Federica Moret, Valentina Martines, Daniele Tramontano, Maria Alessia Papassifachis, Simone Bini, Claudia Caramazza, Mario Fontana, Piernatale Lucia, Maurizio Inghilleri
Journal: Front Med (Lausanne) (2022)
Keywords: nmosd, adenocarcinoma, aquaporin-4, demyelination, neuro-oncology
MeSH terms: Case Reports

Neuromyelitis Optica spectrum disorder is an inflammatory demyelinating disease affecting the central nervous system (CNS), characterized by triad optic neuritis, transverse myelitis, and area postrema syndrome. Antibodies directed against aquaporin-4 (AQP-4), a water channel expressed on the astrocytic membrane, are supposed to play a pathogenic role and are detected in ~80% of cases. Clinical signs of Neuromyelitis Optica spectrum disorder (NMOSD) in elderly patients should arouse the suspicion of paraneoplastic etiology. In this article, we discussed a case of a 76-year-old woman with a 2-month history of confusion, dysarthria, and progressive bilateral leg weakness. A whole-body CT scan showed a neoformation of 5 cm in diameter in the median lobe infiltrating the mediastinal pleura. The tumor had already spread to both the upper and lower right lobes, parietal pleura, and multiple lymph nodes. Pleural cytology revealed adenocarcinoma cells. The brain MRI documented hyperintense alteration in fluid-attenuated inversion recovery (FLAIR) images, involving the anterior portion of the corpus callosum and the periependymal white matter surrounding the lateral ventricles, with mild contrast enhancement on the same areas and meningeal tissue. T2-weighted spinal cord MRI sequences showed extended signal hyperintensity from bulbo-cervical junction to D7 metamer, mainly interesting the central component and the gray matter. Cerebrospinal fluid analysis revealed no neoplastic cells. Serum AQP-4 immunoglobulin (IgG) antibodies were found. Meanwhile, the patient rapidly developed progressive paraparesis and decreased level of consciousness. High-dose intravenous methylprednisolone therapy was started but her conditions rapidly deteriorated. No other treatment was possible.
Successful Treatment of Refractory Palmoplantar Pustular Psoriasis With Apremilast: A Case Series
Authors: Sascha Ständer, Felicia Syring, Ralf J Ludwig, Diamant Thaçi
Journal: Front Med (Lausanne) (2020)
Keywords: apremilast, case series, psoriasis, pustular palmoplantar psoriasis, treatment

Introduction: Palmoplantar pustular psoriasis (PPPP) is a debilitating inflammatory skin disorder of the palms and soles that poses a high burden on affected patients. Satisfactory treatment response is rarely achieved using current treatment options, little is known about the potential benefit of the PDE4 inhibitor apremilast in the treatment of refractory PPPP patients. We aimed to evaluate the use of apremilast in PPPP patients. Patients and Methods: Six patients, four with severe physician global assessment (PGA) = 3 on a scale of 0-4 and two with very severe (PGA = 4) treatment-refractory PPPP [mean age (years ± SD): 56.2 ± 15.6], were included in this study. Five patients had concomitant psoriatic arthritis (PsA). Prior to apremilast administration, topical corticosteroids, psoralen-UVA and multiple systemic oral and biologic anti-inflammatory treatments were insufficient to improve their skin condition or had to be discontinued due to adverse events. Apremilast (titrated to a maintenance dose of 30 mg 2x/d) was commenced in all patients with clinical follow-up over 18 months. Results: Within the first 4 weeks of treatment, each patient's symptoms improved as assessed by PGA score. At 3 months, four patients had a mild PGA score and two were cleared from PPPP. After 18 months of follow-up, three patients improved from PGA = 3 to PGA = 1 and one patient from PGA = 4 to PGA = 1. Two patients discontinued treatment, one due to a lack of efficacy against PsA and the other to a desire to have a child. However, both patients recorded improvements before discontinuing treatment. Conclusion: Apremilast may be a promising treatment option for refractory and severely affected PPPP patients. Our observation, however, requires further validation.
Quantification of a secondary task-specific tremor in a violinist after a temporal lobectomy
Authors: André Lee, Kenta Tominaga, Shinichi Furuya, Fumio Miyazaki, Eckart Altenmüller
Journal: Front Hum Neurosci (2014)
Keywords: coactivation, coherence, dystonia, epilepsy, musicians, task specificity, tremor

Task-specific tremors (TSTs) occur mainly during certain tasks and may be highly disabling. In this case study, we report on a 66-year-old violinist who developed a TST of the right arm only while playing the violin 4 weeks after a temporal lobectomy, which had been performed as a result of his temporal lobe epilepsy. Since a similar case, to our knowledge, has not been reported so far, our aim was to quantitatively assess and describe the tremor by measuring (a) the electromyography (EMG) activity of the wrist flexor and extensor as well as (b) an accelerometer signal of the hand. We found a tremor-related frequency of about 7 Hz. Furthermore, at a similar frequency of about 7 Hz, there was coherence between the tremor acceleration and EMG-activity of the wrist flexor and extensor as well as between the tremor acceleration and coactivation. The tremorgenesis remains unclear, and possible explanations can only be speculative.
Persistent dyselectrolytemia in a neonate induced by liposomal amphotericin B. A case report
Authors: Adrian Puertas Sanjuan, Carlos Javier Parramón-Teixidó, Susana Hernandez-Perez, Marie Antoinette Frick, Maria Jose Cabañas Poy
Journal: Front Pediatr (2023)
Keywords: dyselectrolytemia, hypokalaemia, liposomal amphotericin b [ambisome(®)], neonate, tubulopathy
MeSH terms: Case Reports

Background:
Nephrotoxicity is the most frequent serious adverse effect associated with amphotericin B deoxycholate treatment, for this reason, in recent years it has been relegated from routine clinical practice and replaced by the new liposomal formulations that have less nephrotoxicity. Nevertheless, dyselectrolytemia are a frequent adverse effect of the use of liposomal amphotericin B that usually are resolved with the withdrawal of the drug.
Case presentation:
We present a preterm neonate of 25 weeks gestation, with preserved renal function and most electrolytes within normal limits for gestational age except for mild hyponatremia in the first month of life. Due to an infection of the central nervous system and growth of Candida albicans, he required treatment with endovenous liposomal amphotericin B as well as intrathecal amphotericin B deoxycholate showing severe hydroelectrolyte disturbances and clinical worsening compatible with possible tubulopathy showing hypokalemia and severe hyponatremia a few days after starting treatment that persisted over time even after withdrawal of both drugs. Subsequently to the main alterations described, hypomagnesemia, hypophosphatemia, glycosuria and tubular proteinuria were also observed. Calcium levels remained stable after amphotericin B administration and did not require supplementation. In preterm or low birth weight newborns who present unjustified, severe and difficult to correct hydroelectrolyte disturbances despite the usual treatment, a possible tubulopathy should be considered, whether hereditary, primary or secondary to toxins or drugs.
What is new and conclusion:
We present the first case reported in a neonate in whom dyselectrolithemia has been maintained over time after withdrawal of liposomal amphotericin B.
Bilateral Birdshot Retinochoroiditis and Retinal Astrocytoma
Authors: Sunil Mamtora, Yun Wong, Dugald Bell, Teresa Sandinha
Journal: Case Rep Ophthalmol Med (2017)
MeSH terms: Case Reports

Background. This case highlights the importance of recognising multiple pathologies within the eye which may not necessarily be linked. Both birdshot retinochoroiditis and astrocytoma are rare conditions. The case underlines the need for early identification and treatment of birdshot retinochoroiditis with steroids and disease modifying drugs. Astrocytoma in the absence of tuberous sclerosis is also uncommon. Case Presentation. A 36-year-old male presented with 3-month history of bilateral progressive flashing lights and floaters. He was systemically well with no significant past medical history. Fundal examination revealed retinal vasculitis and active creamy lesions in the choroid radiating from the optic nerve. In the supranasal periphery of the right eye there was a raised white, jagged lesion protruding into the vitreous. Fluorescein angiogram and indocyanine green showed marked venous vasculitis, hypofluorescence, and disc leakage in keeping with birdshot retinochoroiditis. The supranasal lesion features were in keeping with astrocytoma and this was thought to be a coincidental finding. Conclusions. Retinal astrocytoma may be present as an isolated ocular finding; however, patients must still be investigated for tuberous sclerosis which is the most common association. Birdshot retinochoroiditis typically responds well to steroid therapy, and disease modifying drugs should be considered as soon as possible.
Intracerebral Bleeding and Massive Pericardial Effusion as Presenting Symptoms of Myxedema Crisis
Authors: M Kirsch, C Rimpau, C H Nickel, P Baier
Journal: Case Rep Emerg Med (2017)
MeSH terms: Case Reports

The endocrinological emergency of a fully blown myxedema crisis can present as a multicolored clinical picture. This can obscure the underlying pathology and easily lead to mistakes in clinical diagnosis, work-up, and treatment. We present a case of an unconscious 39-year-old patient with a medical history of weakness, lethargy, and findings of hyponatremia, intracerebral bleeding, and massive pericardial effusion. Finally, myxedema crisis was diagnosed as underlying cause. Replacement therapy of thyroid hormone and conservative management of the intracerebral bleeding resulted in patient's survival without significant neurological impairment. However, diagnostic pericardiocentesis resulted in life-threatening pericardial tamponade. It is of tremendous importance to diagnose myxoedema crisis early to avoid adverse health outcomes.
Pseudo-Meigs' syndrome secondary to breast cancer with ovarian metastasis: a case report and literature review
Authors: Xiang-Ying Lin, Xiao-Jun Zhou, Shi-Ping Yang, Jia-Xuan Zheng, Zhao-Jun Li
Journal: Front Oncol (2023)
Keywords: pseudo-meigs’ syndrome, breast cancer, endocrine therapy, oophorectomy, ovarian metastasis
MeSH terms: Case Reports

Ovarian metastasis of breast cancer with pseudo-Meigs' syndrome (PMS) is extremely rare. Only four cases of PMS secondary to breast cancer with ovarian metastasis have been reported to date. In this report, we present the fifth case of PMS caused by ovarian metastasis of breast cancer. On the 2nd of July 2019, a 53-year-old woman presented to our hospital with complaints of abdominal distension, irregular vaginal bleeding, and chest distress. Color Doppler ultrasound examination revealed a mass approximately 109×89 mm in size in the right adnexal area, accompanied by multiple uterine fibroids and a large amount of pelvic and peritoneal effusions. The patient had no common symptoms and showed no signs of breast cancer. The main manifestations were a right ovarian mass, massive hydrothorax, and ascites. Lab workup and imaging revealed raised CA125 (cancer antigen 125) levels and multiple bone metastases. At first the patient was misdiagnosed with ovarian carcinoma. After the rapid disappearance of oophorectomy hydrothorax and ascites, and decreased CA125 levels, from 1,831.8u/ml to normal range. According to the pathology report, breast cancer was finally diagnosed. The patient underwent endocrine therapy (Fulvestrant) and azole treatment after oophorectomy. At the 40-month follow-up, the patient was still alive and doing well.
The Socket-Shield Technique: Digital Planning, Guided Surgery, and Immediate Implant Loading-2-Year Follow-Up
Authors: Sušić Mato, Smojver Igor, Stojić Luka, Katalinić Ivan, Gerbl Dražena, Vuletić Marko
Journal: Case Rep Dent (2022)
MeSH terms: Case Reports

Anterior aesthetic zone implant placement and tooth restoration can be a quite challenging procedure. Modern technology offers new tools that can help maximize results in both aesthetic and functional ways. The socket-shield technique, as described by Hürzeler et al., boosted with the 3D diagnostics, digital planning, and 3D printing, could provide a valuable alternative to traditional approaches. This case report describes a clinical workflow for an efficient anterior implant-prosthetic restoration.
Microscopic Polyangiitis With Selective Involvement of Central and Peripheral Nervous System: A Case Report
Authors: Federica Arienti, Giulia Franco, Edoardo Monfrini, Alessandro Santaniello, Nereo Bresolin, Maria Cristina Saetti, Alessio Di Fonzo
Journal: Front Neurol (2020)
Keywords: allodynia, anti-neutrophil cytoplasmic antibodies specific for myeloperoxidase (mpo-anca), central pain, foot drop, microscopic polyangiitis, peripheral neuropathy, vasculitis
MeSH terms: Case Reports

Background: Microscopic polyangiitis (MPA) is a necrotizing vasculitis that affects predominantly small-sized vessels in many organ systems. The disease generally causes glomerulonephritis, pulmonary damage, arthritis, and neuropathy. An exclusive involvement of both central nervous system (CNS) and peripheral nervous system (PNS) is extremely rare. Case Presentation: A 62-year-old woman was admitted to our hospital with a 3 months history of right foot drop, recently complicated by intense myalgia, arthralgia, and allodynia to tactile, vibratory, and pressure stimuli. Since blood tests revealed elevated inflammatory indexes, we suspected either infectious or immune-mediated disorders. Chest radiograph, blood culture series, and echocardiogram revealed normal findings, while urinalysis showed a bacterial infection that was successfully treated. The neurophysiological findings were compatible with multiple mononeuritis, and a brain MRI evidenced ischemic lesions of both basal ganglia and thalamus. A wide-spectrum autoantibody assay revealed the presence of high-titer perinuclear anti-neutrophil cytoplasmic antibodies specific for myeloperoxidase (MPO-ANCA). According to these findings, the diagnosis of MPA was made, and the patient was successfully treated with intravenous (IV) methylprednisolone, followed by two doses of rituximab. Conclusions: An assessment of both CNS and PNS should be included in the diagnostic evaluation of MPA. The involvement of the PNS may raise the risk of a relapsing course and treatment failure, therefore it should be considered in the choice of induction and maintenance therapy.
Case report: Triple whammy: Synchronous radiotherapy induced glioblastoma multiforme and papillary thyroid cancer following nasopharyngeal carcinoma
Authors: Kamalanathan Palaniandy, Zalikha Kamarudin, Yin Ping Wong, Shahizon Azura Mohamed Mukari, Winnie Xiao Hui Jiau, Azizi Abu Bakar
Journal: Front Oncol (2022)
Keywords: glioblastoma multiforme, nasopharyngeal carcinoma, papillary thyroid carcinoma, radiotherapy - adverse effects, synchronous radiotherapy induced malignancy
MeSH terms: Case Reports

Secondary malignancies following radiotherapy are well documented, with an estimated incidence of 5%. These may manifest as carcinomas, gliomas, or sarcomas within the previous radiation field. Glioblastoma multiforme following radiotherapy for nasopharyngeal carcinoma is an uncommon occurrence and carries a poor prognosis, whereas papillary thyroid carcinoma following radiotherapy is well documented, though the exact incidence is not well documented. The occurrence of synchronous radiotherapy-induced malignancy over both sites has not been described in the literature before. We describe a middle-aged gentleman diagnosed with glioblastoma multiforme and papillary thyroid carcinoma 6 years after radiotherapy for nasopharyngeal carcinoma. Though our case is the first reported case of a synchronous tumour of its nature, it is likely that such cases are under-reported. Long-term vigilance for loco-regional radiotherapy-induced secondary malignancies is a must, and the presence of a second distinct secondary malignancy must be entertained.