OpenMed/multicare-articles huggingface dataset:
Legionella longbeachae wound infection: case report and review of reported Legionella wound infections
Authors: Drifa Frostadottir, Lisa Wasserstrom, Karolin Lundén, Lars B Dahlin
Journal: Front Cell Infect Microbiol (2023)
Keywords: legionella longbeachae, bacteria, cutaneous, extrapulmonary manifestation, infection, legionella
MeSH terms: Legionella longbeachae / genetics, Legionella / genetics, Legionellosis / diagnosis, Wound Infection, Review, Case Reports, Female, Humans, Middle Aged, RNA, Ribosomal, 16S / genetics, RNA, Ribosomal, 16S

Extrapulmonary manifestations of infection with Legionella species, of which 24 may cause disease in humans, are very rare. Here, we describe a case of a 61-year-old woman with no history of immunosuppression presenting with pain and swelling of her index finger after a prick by rose thorns during gardening. Clinical examination showed fusiform swelling of the finger with mild redness, warmth, and fever. The blood sample revealed a normal white blood cell count and a slight increase in C-reactive protein. Intraoperative observation showed extensive infectious destruction of the tendon sheath, while the flexor tendons were spared. Conventional cultures were negative, while 16S rRNA PCR analysis identified Legionella longbeachae that also could be isolated on buffered charcoal yeast extract media. The patient was treated with oral levofloxacin for 13 days, and the infection healed quickly. The present case report, with a review of the literature, indicates that Legionella species wound infections may be underdiagnosed due to the requirement for specific media and diagnostic methods. It emphasizes the need for heightened awareness of these infections during history taking and clinical examination of patients presenting with cutaneous infections.
Imperforate hymen causing bilateral hydroureteronephrosis in an infant with bicornuate uterus
Authors: Ayse Secil Eksioglu, Hasim Ata Maden, Gokce Cinar, Yasemin Tasci Yildiz
Journal: Case Rep Urol (2012)
MeSH terms: Case Reports

A rare case of imperforate hymen associated with bicornuate uterus in an infant is presented as a cause of bilateral hydroureteronephrosis and pelvic mass in infancy. The importance of postoperative radiologic evaluation for diagnosis of accompanying uterine abnormalities is introduced. A 8-month-old girl with restlessness and intermittent fever was brought to the daily outpatient clinic by her parents. Ultrasound exam showed bilateral grade 4 hydroureteronephrosis and a large cystic pelvic mass. Magnetic resonance scan of the pelvis revealed marked hematocolpos. A cruciate incision was made over the hymen under general anesthesia. During a 6-month followup gradual resolution of bilateral hydroureteronephrosis was documented. Although the details of the uterine anomaly were obscured in preoperative imaging, postoperative US and MR demonstrated bicornuate uterus. Postoperative pelvic radiologic examination is highly recommended to verify the resolution of hematocolpos and to screen for any concomitant anomalies that can have long-term clinical significance.
14q32.3-qter trisomic segment: a case report and literature review
Authors: Nicoletta Villa, Agnese Scatigno, Serena Redaelli, Donatella Conconi, Paola Cianci, Clotilde Farina, Chiara Fossati, Leda Dalprà, Silvia Maitz, Angelo Selicorni
Journal: Mol Cytogenet (2016)
Keywords: 14q323-qter duplication, array-cgh, translocation (14, 21)
MeSH terms: Case Reports

Background:
Segmental duplication of the long arm of chromosome 14 (14q) has commonly been reported to affect the proximal segment of 14q, while distal duplication is a rare condition and often associated with segmental monosomy of other chromosomes.
Case presentation:
We report the clinical and genetic characterization of a 4-year-old male patient with 14q32.3-qter trisomy resulting from an adjacent segregation of a paternal reciprocal translocation (14;21)(q32.1;p12). The child shows minor facial anomalies, severe developmental delay, growth retardation, and a history of congenital hypothyroidism and neonatal transitory hyperglycemic crises.
Conclusions:
To the best of our knowledge, only 15 other cases of segmental 14q trisomy were documented. We compared molecularly defined cases to identify a minimal common duplicated region and to find genes with a hypothetical role in the phenotype. The presented case supports the previous suggestion of a pure "distal 14q partial duplication" and underlines the clinical variability.
Factors influencing the choice of facility-based delivery in the ethnic minority villages of Lao PDR: a qualitative case study
Authors: Chika Sato, Khampheng Phongluxa, Noriko Toyama, Ernesto R Gregorio Jr, Chiaki Miyoshi, Futoshi Nishimoto, Tomomi Takayama, Tiengkham Pongvongsa, Kenzo Takahashi, Sengchanh Kounnavong, Jun Kobayashi
Journal: Trop Med Health (2019)
Keywords: decision-making, ethnic minority, facility-based delivery, qualitative case study, thematic analysis

Background:
Facility-based delivery has been promoted to improve maternal and child health care in Lao PDR and a free delivery policy was introduced at designated health care facilities (HCF) in 2013. However, according to birth records of HCFs in the impoverished and remote district, only a few women utilized the HCFs despite good physical accessibility. The aim of this study was to analyze the factors influencing the choice of facility-based delivery in the impoverished and remote district after a free delivery policy was introduced.
Methods:
Qualitative case study was employed. Focus group discussions (FGDs) and in-depth interviews (IDIs) were conducted from August to October 2015. Five hamlets (or small village) located along the main road where only a few women delivered at HCFs were selected for the study based on birth records. The participants of the FGDs and IDIs were the village heads, village health volunteers, women who delivered at home or at a health facility within the past 2 years, their husbands, and mothers or mothers-in-law. Thematic analysis was used to analyze the data.
Results:
A total of 12 FGDs and 27 IDIs were conducted, and the number of participants was 105. The factors influencing the choice of facility-based delivery were classified into nine categories and 19 subcategories. The categories were labeled, "perception of childbirth," "traditional health concept: sabaai (a condition of health, ease, and comfort)," "perception of health care facilities and staff," "previous pregnancy and childbirth experience," "mode of available transportation," "financial burden of childbirth at health care facility," "family and community context," "institutional context," and "government policy on delivery."
Conclusion:
Our study demonstrated that five major factors negatively influenced the choice of facility-based delivery: (1) perception of childbirth, (2) preference for sabaai, (3) financial burden, (4) family decision-making, and (5) institutional context. To promote facility-based delivery in the impoverished and remote district, three strategies are recommended: (1) promoting community-based health education involving women and strengthening community-based mutual support, (2) clarifying items essential for delivery at HCFs, and (3) making HCFs more comfortable in terms of "sabaai."
A Case of Adenocarcinoma of Uterus Masculinus in a Pomeranian Dog
Authors: Massimo Vignoli, Ippolito De Amicis, Roberto Tamburro, Gina Quaglione, Nicoletta Salviato, Francesco Collivignarelli, Rossella Terragni, Stefano Pastrolin, Giuseppe Marruchella
Journal: Front Vet Sci (2020)
Keywords: müllerian duct, adenocarcinoma, computed tomography, dog, uterus masculinus
MeSH terms: Case Reports

Introduction: Persistent Müllerian duct syndrome (PMDS), or uterus masculinus, is a rare autosomal recessive form of male pseudohermaphroditism due to the failure of paracrine anti-Müllerian hormone (AMH) secretion by Sertoli cells or failure of the Müllerian ducts to respond to AMH secretion. The malignant degeneration of persistent Müllerian remnants is rare. In human medicine, few related reports exist. In veterinary medicine, this is the first report describing adenocarcinoma of the uterus masculinus involving the prostate in a dog. Clinical history: An 11-year-old, male, neutered Pomeranian dog was referred for computed tomography due to the suspicion of prostatic carcinoma based on ultrasound and cytological examinations. The computed tomography findings were consistent with a uterus masculinus mass with possible prostatic infiltration. Uterus masculinus removal and total prostatectomy were performed; termino-terminal urethral anastomosis was carried out. Dehiscence of the anastomosis was observed 3 days after surgery. The owner declined any further procedures, and the dog was euthanized 5 days after surgery. Histopathological evaluation revealed adenocarcinoma of the uterus masculinus. Conclusion: Adenocarcinoma of the uterus masculinus may occur, suggesting that patients with PMDS should be evaluated for malignant changes of Müllerian remnants.
Pediatric Multisystem Inflammatory Syndrome in Children as a Challenging Problem for Pediatric Surgeons in the COVID 19 Pandemic-A Case Report
Authors: Beata Jurkiewicz, Magdalena Szymanek-Szwed, Piotr Hartmann, Joanna Samotyjek, Eliza Brędowska, Joanna Kaczorowska, Ewa Wajszczuk, Martyna Twardowska-Merecka, Joanna Cybulska
Journal: Front Pediatr (2021)
Keywords: appendectomy, case report, children, coronavirus disease-19, pediatric inflammatory multisystem syndrome
MeSH terms: Case Reports

The first cases of severe acute respiratory syndrome coronavirus-2 (SARS-CoV-2) infection were identified at the end of 2019 and, in the next few months, coronavirus disease (COVID-19) spread throughout the world. Initially, it was believed that this disease mainly affected elderly individuals with comorbidities, in whom respiratory failure often occurs. It was believed that children fell ill from the infection more often, although the course of infection in the vast majority of pediatric cases has been asymptomatic or mildly symptomatic. In April and May 2020, the first report of a rapidly progressing disease, similar to Kawasaki syndrome, was found in children who had been infected with SARS-CoV-2. Shortly thereafter, children with symptoms of pediatric inflammatory multisystem syndrome (PIMS-ST [temporally associated with SARS-CoV-2 infection]) began presenting to pediatric hospitals around the world. The syndrome has a mortality rate of up to 2%. Symptoms of PIMS-TS include those that may suggest the need for surgical treatment (severe abdominal pain with the presence of peritoneal symptoms, ascites, high levels of inflammatory markers, intestinal inflammation, and appendages revealed on ultrasound examination). However, there are few reports addressing surgical cases associated with this condition. The authors present a case involving an 11-year-old boy who was admitted to hospital with severe abdominal pain and underwent surgery for symptoms of peritonitis and was diagnosed with PIMS in the post-operative period. Due to the large number of illnesses caused by SARS-CoV-2 infection in recent months, the diagnosis of PIMS-TS/MISC should be considered in the differential diagnosis of acute abdominal symptoms, especially in atypical courses and interviews indicating exposure to SARS-CoV-2.
Pilomyxoid astrocytoma of the corpus callosum presenting with primary haemorrhage in an adolescent
Authors: Maria Longo, Juliano Adams Perez, Francine Oliveira, Apio Antunes, Leonardo Vedolin, Juliana Avila Duarte
Journal: BJR Case Rep (2017)
MeSH terms: Case Reports

A 17-year-old male patient with history of intraventricular haemorrhage in 2007 underwent a brain MRI scan in 2013 owing to headache. Brain MRI scan showed an expansive lesion adjacent to the left lateral ventricle infiltrating the anterior portion of the corpus callosum. After surgery, pathology confirmed a pilomyxoid astrocytoma (PMA), an aggressive subtype of astrocytoma that occurs predominantly in the hypothalamic-chiasmatic region. On imaging, PMA presents as a tumour isointense on T 1, hyperintense on T 2 that enhanced heterogeneously with contrast. The T 2 signal is higher than pilocytic astrocytoma, which indicates the presence of myxoid matrix. These findings on MRI scan have a direct correlation with a specific pathological finding-monomorphic proliferation of piloid cells in a mucopolysaccharide-rich matrix. These characteristics associated with the absence of Rosenthal fibres or eosinophilic granules indicated the diagnosis of PMA. To our knowledge, this is the first case report of PMA affecting the corpus callosum in an adolescent.
Retroperitoneal extrarenal angiomyolipomas: an evidence-based approach to a rare clinical entity
Authors: Emmanuel J Minja, Megan Pellerin, Nicole Saviano, Ronald S Chamberlain
Journal: Case Rep Nephrol (2012)

Extrarenal angiomyolipomas (ERAMLs) are rare tumors that present as incidentalomas upon imaging for other conditions. Retroperitoneal ERAMLs present a unique challenge from a diagnostic and treatment standpoint as they can mimic other benign and malignant retroperitoneal tumors. We present a case of a 39-year-old female with a 19.3 cm × 13.5 cm × 10.7 cm left extrarenal retroperitoneal mass. Histopathologic examination and HMB-45 staining revealed the mass to be a retroperitoneal ERAML. Our case report provides a comprehensive literature review and an evidence-based algorithm for taking care of patients with ERAMLs.
Reduced vitamin D-induced cathelicidin production and killing of Mycobacterium tuberculosis in macrophages from a patient with a non-functional vitamin D receptor: A case report
Authors: Fatima A H Al-Jaberi, Cornelia Geisler Crone, Thomas Lindenstrøm, Nicolai Skovbjerg Arildsen, Emilia Sæderup Lindeløv, Louise Aagaard, Eva Gravesen, Rasmus Mortensen, Aase Bengaard Andersen, Klaus Olgaard, Jessica Xin Hjaltelin, Søren Brunak, Charlotte Menné Bonefeld, Martin Kongsbak-Wismann, Carsten Geisler
Journal: Front Immunol (2022)
Keywords: cathelicidin, hereditary vitamin d-resistant rickets (hvdrr), macrophage, tuberculosis, vitamin d
MeSH terms: Familial Hypophosphatemic Rickets / metabolism, Mycobacterium tuberculosis / metabolism, Tuberculosis, Lymph Node, Case Reports, Research Support, Non-U.S. Gov't, Adolescent, Cathelicidins, Female, Humans, Macrophages / metabolism, Receptors, Calcitriol / genetics, Receptors, Calcitriol / metabolism, Vitamin D / metabolism, Vitamin D / pharmacology, Vitamins / metabolism, Receptors, Calcitriol, Vitamin D, Vitamins, Cathelicidins

Tuberculosis (TB) presents a serious health problem with approximately a quarter of the world's population infected with Mycobacterium tuberculosis (M. tuberculosis) in an asymptomatic latent state of which 5-10% develops active TB at some point in their lives. The antimicrobial protein cathelicidin has broad antimicrobial activity towards viruses and bacteria including M. tuberculosis. Vitamin D increases the expression of cathelicidin in many cell types including macrophages, and it has been suggested that the vitamin D-mediated antimicrobial activity against M. tuberculosis is dependent on the induction of cathelicidin. However, unraveling the immunoregulatory effects of vitamin D in humans is hampered by the lack of suitable experimental models. We have previously described a family in which members suffer from hereditary vitamin D-resistant rickets (HVDRR). The family carry a mutation in the DNA-binding domain of the vitamin D receptor (VDR). This mutation leads to a non-functional VDR, meaning that vitamin D cannot exert its effect in family members homozygous for the mutation. Studies of HVDRR patients open unique possibilities to gain insight in the immunoregulatory roles of vitamin D in humans. Here we describe the impaired ability of macrophages to produce cathelicidin in a HVDRR patient, who in her adolescence suffered from extrapulmonary TB. The present case is a rare experiment of nature, which illustrates the importance of vitamin D in the pathophysiology of combating M. tuberculosis.
Spontaneous resolution of massive spontaneous tubercular pneumothorax
Authors: Surya Kant, S Saheer, G Hassan, Jabeed Parengal
Journal: Case Rep Pulmonol (2011)
MeSH terms: Case Reports

A 29-year-old female presented with complaints of fever and productive cough of three weeks duration. Pulmonary tuberculosis was diagnosed bacteriologically and she was prescribed antituberculosis drugs. During follow-up she developed massive pneumothorax, for which patient refused surgical management and was managed conservatively. After six months there was complete spontaneous resolution of pneumothorax. The unusual presentation and unexpected outcome prompted us to report this case.
Peripheral Odontogenic Fibroma: A Rare Tumor mimicking a Gingival Reactive Lesion
Authors: Komal Khot, Swati Deshmane, Kriti Bagri-Manjrekar, Paresh Khot
Journal: Int J Clin Pediatr Dent (2017)
Keywords: fibroma, pediatric, peripheral odontogenic fibroma, peripheral odontogenic tumor
MeSH terms: Case Reports

Gingival growths are one of the most frequently encountered lesions in the oral cavity. A plethora of lesions can be seen having similar clinical presentation, making diagnosis a dilemma. Peripheral odontogenic tumors are rare neoplasms to occur on gingiva, the most common among them being the peripheral odontogenic fibroma (POdF). The POdF is a benign, slow-growing, exophytic lesion. Although considered to have a recurrence potential after excision, the actual recurrence rate is not known due to paucity of reported cases. Presented here is a case of a rare neoplasm mimicking an inflammatory gingival lesion with review of the available literature.
How to cite this article:
Khot K, Deshmane S, Bagri-Manjrekar K, Khot P. Peripheral Odontogenic Fibroma: A Rare Tumor mimicking a Gingival Reactive Lesion. Int J Clin Pediatr Dent 2017;10(1):103-106.
Case Report: A Case of Intestinal Behçet's Disease Exhibiting Enhanced Expression of IL-6 and Forkhead Box P3 mRNA After Treatment With Infliximab
Authors: Keisuke Yoshikawa, Tomohiro Watanabe, Ikue Sekai, Ryutaro Takada, Akane Hara, Masayuki Kurimoto, Yasuhiro Masuta, Yasuo Otsuka, Tomoe Yoshikawa, Sho Masaki, Ken Kamata, Kosuke Minaga, Yoriaki Komeda, Takaaki Chikugo, Masatoshi Kudo
Journal: Front Med (Lausanne) (2021)
Keywords: foxp3, il-6, infliximab, intestinal behçet's disease, regulatory t cells
MeSH terms: Case Reports

Behçet's disease (BD) is a rare inflammatory condition characterized by oral and genital ulcers, skin lesions, as well as ophthalmological, neurological, and gastrointestinal manifestations. BD involving the gastrointestinal tract is known as intestinal BD. The mucosa of the gastrointestinal tract of patients with intestinal BD exhibits enhanced levels of proinflammatory cytokines, such as IL-1β, IL-6, and TNF-α. These proinflammatory cytokines play pathogenic roles in the development of BD, as evidenced by the fact that biologics targeting these cytokines effectively induce BD remission. It should be noted, however, that the molecular mechanisms by which the blockade of these cytokines suppresses chronic inflammatory responses in BD are poorly understood. Herein, we report a case of intestinal BD resistant to prednisolone that was successfully treated with infliximab (IFX). The induction of remission by IFX was accompanied by a marked elevation of IL-6 and forkhead box P3 (FOXP3) at mRNA level. This case suggests that induction of remission by IFX is mediated not only by the suppression of TNF-α-mediated signaling pathways, but also by the promotion of IL-6 expression and accumulation of regulatory T cells expressing FOXP3.
Acquired generalized lipodystrophy associated with autoimmune hepatitis and low serum C4 level
Authors: Erdal Eren, Tanju Başarır Özkan, Esra D Papatya Çakır, Halil Sağlam, Ömer Tarım
Journal: J Clin Res Pediatr Endocrinol (2010)
Keywords: lipodystrophy, autoimmune hepatitis, complement c4
MeSH terms: Complement C4 / deficiency, Hepatitis, Autoimmune / complications, Lipodystrophy / complications, Case Reports, Child, Female, Hepatitis, Autoimmune / blood, Hepatitis, Autoimmune / physiopathology, Humans, Lipodystrophy / blood, Lipodystrophy / physiopathology, Complement C4

Lipodystrophies are a group of diseases characterized by loss of fat tissue and are associated with insulin resistance. A six-year-old girl followed with the diagnosis of autoimmune hepatitis showed a severe loss of fat tissue, hyperinsulinemia, impaired glucose tolerance, hypertriglyceridemia and low serum complement 4 (C4) levels. She had coarse facial features with generalized loss of subcutaneous fat and prominent muscularity. Remarkable acanthosis nigricans was present over the neck, axilla, and umbilicus. Two hours after glucose loading, the glucose tolerance test revealed a glucose level of 258 mg/dL, a HbA1c value of 6.8%, and an insulin level of 642.9 mIU/mL, documenting a state of insulin resistance and type 2 diabetes mellitus. Acquired generalized lipodystrophy was diagnosed and metformin with dietary intervention was initiated. Low serum complement levels proved the autoimmune nature of the process. We conclude that the serum complement levels must be investigated in patients with acquired lipodystrophy, particularly when it is associated with autoimmune hepatitis.
Recurrent Cervical Cancer Treated Successfully with Single-Agent PARP-Inhibitor, Olaparib
Authors: Maya Gross, Ryan J Spencer
Journal: Case Rep Obstet Gynecol (2022)
MeSH terms: Case Reports

Recurrent cervical cancer has a grim prognosis with 5-year survival <5%. Current treatment options are limited; standards of care such as palliative chemotherapy and surgical resection often provide a small survival advantage. To date, only one targeted agent has FDA approval for the treatment of recurrent cervical cancer. We present the case of a novel application of olaparib, a poly (adenosine diphosphate-ribose) polymerase (PARP) inhibitor, as single-agent therapy for recurrent metastatic clear cell cervical cancer in a patient with a somatic BRCA2 mutation. The patient had excellent response to therapy with stable disease without evidence of progression until 14 months of therapy, at which time she was switched to an alternative regimen.
Granulosis Rubra Nasi Response to Topical Tacrolimus
Authors: Farhana Tahseen Taj, Divya Vupperla, Prarthana B Desai
Journal: Case Rep Dermatol Med (2017)
MeSH terms: Case Reports

Granulosis Rubra Nasi (GRN) is a rare disorder of the eccrine glands. It is clinically characterized by hyperhidrosis of the central part of the face, most commonly on the tip of the nose, followed by appearance of diffuse erythema over the nose, cheeks, chin, and upper lip. It is commonly seen in childhood but it can present in adults. Here we report a case of GRN in an adult patient with very unusual histopathological presentation.
Bedside Carotid Sinus Massage for Syncope Evaluation With Bifascicular Block and First-Degree Atrioventricular Block
Authors: Mashood B Badshah, Muhammad Hamza Saad Shaukat, Azar Birlas, Scott Pham
Journal: Cureus (2023)
Keywords: atrioventricular block, bifascicular block, cardiac syncope, carotid sinus massage, permanent pacemaker
MeSH terms: Case Reports

Symptomatic bifascicular block (BFB) with a reversible high-grade atrioventricular block (AVB) is an overlooked cause of syncope with differing diagnostic and therapeutic approaches. We present a case of a 79-year-old gentleman with multiple episodes of cardiac syncope. Initial electrocardiogram revealed a left bundle branch block and first-degree AVB worsened by bedside carotid sinus massage (CSM) obviating the need for electrophysiologic (EP) studies or continuous electrocardiographic monitoring for further evaluation. This case highlights the importance of CSM as a useful clinical tool in addition to EP studies and internal loop recorder (ILR) placement for assessment and appropriateness of permanent pacemaker (PPM) implantation. It also sheds light on the differing management protocols between EP studies and ILR evaluation versus empiric PPM implantation for patients with cardiac syncope secondary to BFB and AVB.