OpenMed/multicare-articles huggingface dataset:
Rapid destruction of shoulder joint by pigmented villonodular synovitis treated by hemiarthroplasty: A case report
Authors: Minsung Kwon, Jin-Young Bang, Kyung Han Nam
Journal: Int J Surg Case Rep (2020)
Keywords: arthritis, arthroplasty, giant cell, hemiarthroplasty, pigmented villous nodular synovitis, shoulder
MeSH terms: Case Reports

Introduction:
Pigmented villous nodular synovitis is an uncommon proliferative disease of the joints, and rarely reported in the shoulder. It can become symptomatic when proliferating soft tissue infiltrates a joint, causing arthritic changes including bone erosion. The literature has described the disease progression as indolent. Here we report on a case of PVNS of the shoulder joint which rapidly lead to significant bone damage and was subsequently treated by shoulder arthroplasty.
Presentation of case:
We report here on a 71-year old female patient who presented with a 6 month history of aggravating shoulder pain. Radiography imaging over a one month period indicated rapid joint destruction. Magnetic resonance imaging suggested the presence of PVNS of the shoulder joint and significant bone erosion. The patient was subsequently treated by shoulder arthroplasty performed by authors. Histological examination confirmed the PVNS diagnosis. Shoulder pain significantly decreased during the follow up period, and the patient was able to resume daily activities.
Discussion:
Comparing to Milwaukee shoulder syndrome or a joint infection, PVNS is known to progress indolently. However, our case clearly showed that PVNS could also cause radical destruction of the joint. Previous reports showed the high recurrence rate of PVNS after joint preserving surgery. In our experience hemiarthroplasty could be the choice of treatment with the low recurrence rate and high functional outcome.
Conclusions:
Physicians should include PVNS in the differential diagnosis when they are presented with evidence of rapid destruction of the shoulder joint. Hemiarthroplasty could be treatment option for PVNS of shoulder joint.
Oesophageal perforation as a complication of ingested partial denture
Authors: Himansu Shekhar Mohanty, Kapil Shirodkar, Aruna R Patil, Govindrajan Mallarajapatna, Sharath Kumar, K Chinmay Deepak, Shrivalli Nandikoor
Journal: BJR Case Rep (2016)
MeSH terms: Case Reports

We report herein the case of a 53-year-old female who came to the emergency room with the chief complaints of severe dysphagia and chest pain following accidental swallowing of her denture. The patient had swelling of the face, neck and eyelids with difficulty in breathing. A skull radiograph was taken, which revealed a missing partial denture from the right lower jaw. Anteroposterior radiograph of the chest showed two metallic objects in the mid-thorax, adjacent to the descending aorta. CT scan of the neck and chest revealed two metallic objects (measuring approximately 17mm each) in the middle one-third of the oesophagus (right posterolateral aspect), causing perforation of the oesophagus and leading to pneumomediastinum, and left pneumothorax with subcutaneous emphysema of the neck and chest. An emergency thoracoscopic removal of the foreign body (partial denture) was performed with subsequent repair of the oesophageal tear in the same sitting. Post surgery, the patient was shifted to intensive care unit and she recovered well over a course of time. In summary, accidental ingestion of a partial denture can lead to grave complications such as oesophageal perforation, which should be managed on an emergency basis with thoracoscopic removal of the foreign body.
Rituximab in Antimelanoma Differentiation-Associated Protein-5 Dermatomyositis with Interstitial Lung Disease
Authors: Chiara Scirocco, Andreina Gubbiotti, Alfredo Sebastiani, Gian Domenico Sebastiani
Journal: Case Rep Rheumatol (2020)
MeSH terms: Case Reports

In this paper, we report a challenging case of a middle-age woman who developed antimelanoma differentiation-associated protein-5 dermatomyositis (anti-MDA5 DM) with interstitial lung disease (ILD) and was successfully treated with rituximab (RTX), after failure of a first-line therapy.
Case Report: Immune Mediated Necrotizing Myopathy With IgG Antibodies to 3-Hydroxy-3-Methylglutaryl-Coenzyme a Reductase (HMGCR) May Present With Acute Systolic Heart Failure
Authors: Malik Ghannam, Georgios Manousakis
Journal: Front Neurol (2020)
Keywords: anti-hmgcr antibodies, critical diagnosis, inflammation, myopathic, systolic heart failure
MeSH terms: Case Reports

Involvement of cardiac muscle is felt to be very uncommon in anti-HMGCR myopathy, and therefore early cardiac evaluation is not considered a high priority for this condition. We herein present the case of a 72 year-old woman admitted due to dyspnea and orthopnea, who, in retrospect, suffered from proximal more than distal muscle weakness for 3 months prior to admission. She was found to have acute systolic heart failure. Serologic testing showed positive 3-hydroxy-3-methylglutaryl-coenzyme A reductase (HMGCR) IgG antibodies, and muscle biopsy showed necrotizing myopathy. No alternative explanation for heart failure was found. Despite immunotherapy and symptomatic treatment, she died from multiorgan failure. Our study suggests that heart failure in anti HMGCR myopathy may not be as rare as previously thought, and therefore early cardiac evaluation should be considered in patients with this diagnosis, to minimize morbidity and mortality.
Acute limb ischemia caused by ruptured cardiac hydatid cyst - A case report
Authors: Samer Makki Mohamed Al-Hakkak, Firas Shaker Mahmoud Al-Faham, Ali Najeh Al-Awwady
Journal: Int J Surg Case Rep (2019)
Keywords: cardiac, embolectomy, hydatid cyst, left ventricle, limb ischemia, rupture

Introduction:
Acute limb ischemia is a sudden decrease in limb perfusion that threatens the viability of the limb. Complete or even partial occlusion of the arterial supply to a limb can lead to rapid ischemia and poor functional outcomes within hours. In human echinococcosis cardiac involvement is a rare presentation, it may lead to life-threatening complications including cyst rupture; anaphylactic shock; tamponade; pulmonary, cerebral or peripheral arterial embolism. Cardiac hydatid cyst (CHC) may have different presentation include acute lower limb ischemia secondary to embolization from a ruptured cyst.
Case presentation:
We report an 18-year old male healthy building worker while he was working who presented with sudden onset acute right lower limb pain and paresthesia caused by rupture of primary CHC which managed as a surgical emergency.
Discussion:
Clinical presentation of ruptured of CHC depends on the specific location of the ruptured cyst that interferes and mobilization of daughter cyst that logged in vascular system with the function of the surrounding cardiac structures like our case that present with embolization of daughter cyst into the right external iliac artery which leads to acute limb ischemia.
Conclusion:
Cardiac hydatid cyst is a rare finding with a wide range of signs and symptoms. We are reporting this case to underline that cardiac hydatidosis should be considered as a differential diagnosis in young patients who suddenly develop acute limb ischaemic without a history of both cardiac diseases and trauma that lives in endemic regions of hydatidosis.
Case report: Treatment-resistant depression, multiple trauma exposure and suicidality in an adolescent female with previously undiagnosed Autism Spectrum Disorder
Authors: Ilaria Secci, Lucie Petigas, Alexandra Cuenod, Paul Klauser, Carole Kapp, Audrey Novatti, Marco Armando
Journal: Front Psychiatry (2023)
Keywords: autism spectrum disorder, covid-19, adolescent, case report, depression, female, lithium, suicidality
MeSH terms: Case Reports

High rates of co-occurring depression are commonly reported in youth with Autism Spectrum Disorder (ASD), especially in individuals without intellectual disability (ID). Depression in ASD undermines adaptive behavior and is associated with a higher risk of suicidality. Females with ASD may be particularly vulnerable due to their greater use of camouflaging strategies. Indeed, in comparison to males, ASD is underdiagnosed in females, despite higher rates of internalizing symptoms and suicidality. Trauma exposure may also play a role in the development of depressive symptoms in this population. Moreover, evidence for effective treatments of depression in autistic youth are lacking, with ASD individuals frequently experiencing low efficacy and side effects. We present the case of an adolescent female with previously undiagnosed ASD without ID, admitted for active suicidal plans and a treatment-resistant depression (TRD), occurred after a COVID-19 lockdown in the context of cumulative exposure to stressful life events. Comprehensive clinical assessments performed at intake confirmed severe depression with suicidality. Intensive psychotherapy and different changes in medications were carried out (SSRI, SNRI, SNRI + NaSSA, SNRI + aripiprazole), all of which were ineffective, with persistent suicidal thoughts, often requiring intensive individual monitoring. The patient was finally successfully treated with lithium augmentation of fluoxetine, with no side effects. During hospitalization she was also evaluated by an ASD specialized center, where a diagnosis of ASD was made according to the Autism Diagnostic Observation Schedule (ADOS) and the Autism Diagnostic Interview-Revised (ADI-R) scores, as well as to clinical judgment of a senior psychiatrist. The present case report shows that clinicians should not overlook undiagnosed autism as a possible cause of TRD, especially in females without ID, where higher rates of under diagnosis may be in part related to their greater use of camouflage. It also suggests that ASD underdiagnosis and resulting unmet needs may be involved in vulnerability to stressful experiences, depression, and suicidality. Furthermore, it shows the complexity of providing care to TRD in youth with autism, suggesting that an augmentation therapy with lithium, a commonly recommended therapeutic strategy for refractory depression in typically developing samples, may also be effective in this population.
Behçet's Syndrome in a Chinese Pedigree of NLRP3-Associated Autoinflammatory Disease: A Coexistence or Novel Presentation?
Authors: Jinjing Liu, Xin Yu, Chaoran Li, Yi Wang, Weihong Yu, Min Shen, Wenjie Zheng
Journal: Front Med (Lausanne) (2021)
Keywords: behçet's syndrome, nlrp3-associated autoinflammatory disease, pedigree analysis, vasculitis, whole-exome sequencing

Objectives: NLRP3-associated autoinflammatory disease (NLRP3-AID) and Behçet's syndrome (BS) both belong to autoinflammatory diseases and rarely co-occur. Here we reported a Chinese pedigree of NLRP3-AID presented with BS. Methods: We recorded a Chinese pedigree of NLRP3-AID presented with BS. Whole-exome sequencing was performed to find the hereditary susceptibility gene, and Sanger sequencing was performed on a consecutive cohort of 30 BS patients. We also reviewed the English literature on vasculitis associated with NLRP3-AID. Results: The proband was a 45-year-old Chinese Han woman. She and her 12-year-old daughter presented with recurrent fevers, cold-induced urticaria, oral, and genital ulcers, conjunctivitis, uveitis, optic atrophy, erythema nodosum, headache, and hearing loss. They were initially suspected of having BS, and both responded poorly to corticosteroids and immunosuppressants, while anti-TNF therapy was moderately effective. Pedigree analysis revealed another four relatives with similar symptoms, and a heterozygous NLRP3 gene mutation c.1316C>T, p.Ala439Val was identified by whole-exome sequencing and Sanger sequencing. However, we did not discover NLRP3 gene mutation by Sanger sequencing in a confirmative cohort of 30 BS cases. A few case reports of vasculitis coexisting with NLRP3-AID, including a case of glomerulonephritis, and five cases of retinal vasculitis, were summarized through literature review. Conclusions: Our study is the first report of NLRP3-AID associated with BS. The coexistence of NLRP3-AID and BS reveals the extensive heterogeneity of the pathogenesis of systemic autoinflammatory diseases and calls for specific therapeutics.
Spontaneous Complete Remission in a Patient with Acute Myeloid Leukemia and Severe Sepsis
Authors: Rambod Mozafari, Mahsa Moeinian, Ali Asadollahi-Amin
Journal: Case Rep Hematol (2017)
MeSH terms: Case Reports

Without treatment, acute myeloid leukemia (AML) is almost always fatal. Spontaneous remission of AML is a rare phenomenon and usually with a short duration. The exact mechanisms are unknown. However, its association with infection and blood transfusions has been described. We report a 53-year-old male who presented with severe sepsis and who was diagnosed with AML (M4). He has experienced complete spontaneous remission with relatively long duration. To the best of our knowledge, it is the first case of spontaneous remission described in Iran.
Cellulosimicrobium cellulans aortic prosthetic valve endocarditis
Authors: Jacopo Monticelli, Riccardo Gerloni, Claudio Farina, Anna Knezevich, Franca Dore, Roberto Luzzati
Journal: Access Microbiol (2019)
Keywords: cellulosimicrobium, endocarditis, single photon emission computed tomography
MeSH terms: Case Reports

Introduction:
Invasive infections due to Cellulosimicrobium spp. (a Gram-positive coryneform) are extremely rare. Only a few cases of bloodstream infections and endocarditis have been described, as bacteraemia due to coryneforms is usually discarded as blood culture contamination.
Case presentation:
A 66-year-old female, with a history of aortic valve replacement, presented with fever, left leg purpura and acute kidney injury. Multiple repeated blood cultures were positive for Cellulosimicrobium cellulans , and targeted therapy was started. At first, endocarditis was excluded by echocardiograms, and the acute nephritis was interpreted as an atypical presentation of Henoch-Shönlein purpura. High-dose prednisone was started, and after 10 weeks the patient presented again with fever, mental confusion and acute left arm ischaemia. A subsequent echocardiogram and radiolabelled leukocyte scintigraphic evaluation revealed aortic prosthetic valve endocarditis with periprosthetic abscess and arterial brachial thrombosis. The patient deceased, and the autoptic examination confirmed an aortic valve periprosthetic abscess and revealed multiple arterial thromboses and septic embolisms in the kidneys, brain, spleen and myocardium.
Conclusion:
Isolation of coryneform bacteria on blood culture should not always be discarded as blood culture contamination. In the case of endocarditis due to Cellulosimicrobium spp., the removal of any prosthetic material, along with prolonged in vitro active antimicrobial therapy, should be pursued in order to reduce persistence or relapses of infection.
Severe Pulmonary Hypertension as Initial Presentation of SLE: A Case Report and Literature Review
Authors: Rabia Kiani, Muhammad Danial Siddiqui, Hamza Tantoush
Journal: Case Rep Rheumatol (2020)
MeSH terms: Case Reports

Severe pulmonary artery hypertension (PAH) is a rare initial presentation of systemic lupus erythematosus (SLE). SLE associated with PAH carries worse prognosis that isolated SLE. However, there has been improvement in mortality of the patients in the recent years owing to newer treatment options available. Early recognition remains of prime importance. We present here a case of young female who presented with severe pulmonary hypertension with right heart failure leading to cardiogenic shock and was found to have SLE. She was started on appropriate treatment; however, given the severity of her illness, the patient did not survive. This case highlights the importance of early recognition and prompt treatment of SLE-associated PAH, which might improve the survival rate in the patients.
A large ectopic hepatocellular carcinoma with adrenal infiltration: a rare case report
Authors: Yongjun Yang, Qiang Lu, Zonglin Li, Chen Wang, Yuanwei Li
Journal: Front Oncol (2023)
Keywords: adrenal infiltration, case report, ectopic hepatocellular carcinoma, endocrine function, retroperitoneal tumor
MeSH terms: Case Reports

Ectopic hepatocellular carcinoma (EHCC) originates from the ectopic liver, which refers to a liver organ or tissue unrelated to surrounding tissues. EHCC is a rare disease that lacks specific clinical signs, and preoperative diagnosis is often difficult. In a 61-year-old male patient with positive hepatitis B virus antibody, abdominal contrast-enhanced computed tomography scan showed a large heterogenously enhancing mass both on arterial and portal venous phase imaging arising from the right adrenal gland. Similar enhancement features were seen on magnetic resonance imaging. Serum potassium, aldosterone, cortisol, and plasma metanephrines were normal. The tumor markers of serum alpha-fetoprotein and alpha-fetoprotein-L3% were increased to 23.69 ng/mL and 82.1%, respectively. Exploratory laparotomy was performed and operative findings showed that the retroperitoneal tumor was disconnected from the right kidney and the liver, but invaded the right adrenal gland. Immunohistochemical examination showed that Arginase-1 was positive expression, and the retroperitoneal tumor was finally diagnosed as EHCC. We report a rare EHCC with adrenal infiltration that is difficult to diagnose preoperatively and mimics a retroperitoneal tumor or adrenal tumor, and we present a review of the literature on EHCC case reports.
Dopamine neurons in the ventral tegmental area: an autopsy case of disorganized type of schizophrenia
Authors: Keiko Ikemoto, Tatsuro Oda, Akiyoshi Nishimura, Katsuji Nishi
Journal: Case Rep Neurol Med (2011)
MeSH terms: Case Reports

The mesolimbic dopamine (DA) system has been associated with the pathogenesis of schizophrenia. Here, we examined DA-containing neuronal structures of the ventral tegmental area (VTA) of an autopsy case of disorganized type of schizophrenia (75-year-old female), using tyrosine hydroxylase (TH) immunohistochemistry. A free floating method using 50-μm cryostat sections and three-dimensional imaging analyzer AxioVision were applied to observe the wide range structures of TH-immunoreactive (-ir) neurons. TH-ir neuronal cell bodies in the VTA of the present case had irregular shape and various size, and TH-ir neuronal processes had irregular thickness and straightened shape or curved shape having many corners, when compared to a control autopsy case with no detectable neurological and psychiatric diseases (64-year-old male). The mechanisms underlying the morphological characteristics of DA neurons of the brains with schizophrenia should be elucidated epigenetically as well as genetically.
Pembrolizumab Induced Calcitriol-Mediated Hypercalcemia
Authors: Tiffany T Oommen, Kelly Sun, Javier Barranco-Trabi, Jeffrey Berenberg, Myungjin Kim
Journal: Case Rep Nephrol (2023)
MeSH terms: Case Reports

PD-1/PD-L1 inhibitors such as pembrolizumab have radically improved the prognosis for many patients with advanced malignancies. Although revolutionary, its use can be complicated and limited by various immune-related adverse effects. Effective management depends on early recognition and prompt intervention. Herein, we describe a unique syndrome of hypercalcemia, with associated acute renal injury and hypoxic respiratory failure that was responsive to corticosteroids suggestive of immunotoxicity from pembrolizumab.
Interpretation of the post-surgical Somatostatin Receptor Scintigram of a Primary Neuroendocrine Tumor of the Thymus: a case report and literature review
Authors: Anastasia Leondi, John Koutsikos, Cherry Zerva
Journal: Int Semin Surg Oncol (2005)

A case of a thymic neuroendocrine tumor and the interpretation problems in a post-surgical Somatostatin Receptor Scintigraphy are presented. In a 53-year-old man with superior vena cava obstruction syndrome an atypical carcinoid of the thymus (neuroendocrine carcinoma of intermediate grade 2), was found at surgery.During his first year of follow-up a Somatostatin Receptor Scintigraphy was recommended. An area of abnormal concentration of the radiopharmaceutical was revealed in the mediastinum at this time.A thorough understanding of the mechanisms of the radiopharmaceutical uptake and of the various clinical settings in which uptake can occur are essential for a proper evaluation of the scintigraphic findings and result in the optimal use of this valuable modality.The literature review provides an overview of this rare type of tumor and insight into the clinical significance of Somatostatin Receptor Scintigraphy.
Intra-articular knee arborescent lipoma: a case treated with arthroscopic synoviectomy
Authors: Aymen Saidi, Lassaad Hassini, Aymen Fekih, Monia Ben Othmen, Mohamed Allagui, Abderrazek Abid, Issam Aloui
Journal: Pan Afr Med J (2017)
Keywords: arborescent lipoma, arthroscopic, knee, synoviectomy
MeSH terms: Arthroscopy / methods, Knee Joint / surgery, Lipoma / surgery, Synovectomy / methods, Case Reports, Humans, Knee Joint / pathology, Lipoma / pathology, Middle Aged, Treatment Outcome

Arborescent lipoma is an unusual intra-articular lesion that typically develops in the knee and has to be evoked before chronic effusion. It corresponds to hyperplasia of mature fatty tissue and hypertrophy of synovial villi, developing within a joint. The reference treatment is synovectomy by arthrotomy. The rare forms localized to the anterior compartment of the knee can benefit from an arthroscopic synovectomy. The authors report a case of arborescent knee lipoma in a 47-year-old patient who received arthroscopic synoviectomy. To our knowledge, only a few cases of arborescent lipoma treated by arthroscopic synoviectomy have been reported in the literature.
New technique for the treatment of fresh bony mallet finger: A retrospective case series study
Authors: Zhenshuang Yue, Yafeng Mo, Zhenfei Xiong, Yanghua Tang
Journal: Front Surg (2023)
Keywords: bony mallet finger, fracture, kirschner wire, phalanx, tendon

Background:
The bony mallet finger is a tear fracture of the extensor tendon, resulting in a flexion deformity of the finger, which affects both the function of the finger. The classical Ishiguro's method is associated with damage to the cartilage of the distal interphalangeal (DIP) joint and always lead to the joint stiffness. This paper explores a new technique to overcome the shortcomings of the classical Ishiguro's method and achieve better clinical efficacy.
Methods:
We examined 15 patients with bony mallet fingers, 9 males and 6 females, from February 2020 to June 2022, ranged from 23 to 58 years, including 1 case of index finger, 5 cases of middle finger, 3 cases of ring finger and 6 cases of little finger. The median course of the injury to surgery was 2 days (range, 1∼7 days). All had fresh closed injuries, according to the Wehbe and Schneider classification: 4 cases of type IA, 6 cases of type IB, 3 cases of type IIA and 2 cases of type IIB. All patients were treated surgically by the new technique. Post-operative follow-up was conducted to record the healing of the fracture, the pain of the affected finger and the function of joint movement.
Results:
The 15 cases were followed up after surgery. The median active range of motion was 65° (range, 55∼75°). The median extension deficit of DIP joint was 0° (range, 0∼11°). The median clinical healing time of the fracture was 6 weeks (range, 6∼10 weeks). None of the patients experienced significant pain. The patients were assessed according to the Crawford criteria at the final follow-up: 11 cases were assessed as excellent, 3 cases were assessed as good and 1 case was assessed as fair. No loss of fracture repositioning, loosening of internal fixation, skin necrosis or infection was observed.
Conclusion:
The application of the new technique for the treatment of bony mallet fingers has the advantages of good stability, fracture healing and functional recovery of the DIP joint, making it an ideal surgical procedure for the treatment of fresh bony mallet fingers.