OpenMed/multicare-articles huggingface dataset:
The first rare and fatal case of invasive aspergillosis of spinal cord due to Aspergillus nidulans in an Iranian child with chronic granulomatosis disease: review of literature
Authors: Mahin Tavakoli, Mohammad Taghi Hedayati, Hossein Mirhendi, Sadegh Nouripour-Sisakht, Newsha Hedayati, Fatemeh Saghafi, Setareh Mamishi
Journal: Curr Med Mycol (2020)
Keywords: aspergillus nidulans, chronic granulomatosis disease, invasive aspergillosis, spinal cord
MeSH terms: Case Reports

Background and purpose:
Invasive aspergillosis (IA) of the central nervous system (CNS) is a devastating complication which is rarely reported in immunocompromised children. In this case presentation, we reported a rare and fatal IA with spinal cord involvement in a 10-year-old child with X-linked chronic granulomatosis disease (CGD).
Case report:
The child had a previous history of pulmonary tuberculosis. A cervical spine X-ray revealed the involvement of cervical vertebrae (T4/T5) and ribs causing spinal cord compression and epidural abscess. The patient underwent a decompressive laminectomy and mass removal. The histopathology and culture results suggested IA. Despite the aggressive and prolonged therapy, he died within one year. Aspergillus nidulans was identified as the causative agent based on morphological and molecular studies.
Conclusion:
This synopsis represents the aggressive behavior of infection caused by A. nidulans in the CGD patient.
Francisella tularensis Bacteremia: A Case Report from Sudan
Authors: Salma E R Mohamed, Aymun I Mubarak, Lamia O Alfarooq
Journal: Case Rep Infect Dis (2012)

Francisella tularensis is a highly virulent intracellular gram-negative bacterium. The organism is usually isolated from wild and domestic animals and invertebrate. Man gets infection by direct contact with those animals or their products but the most common mode of transmission is via arthropod vectors. The disease is endemic in North America, parts of Europe, and Asia but has never been reported in Africa. A 29-year old male living in a rural area of Southern Sudan has been maintained on continuous ambulatory peritoneal dialysis for two years. He presented to our center in May 2010 complaining of fever, dry cough, shortness of breath, and abdominal discomfort for four days. He was very ill, pale, and dehydrated. There were enlarged tender submandibular lymph nodes, but no mouth ulcers or other palpable lymph nodes. Peritonitis was excluded by effluent white blood cell count and culture. Empiric antibiotic treatment with ceftriaxon, and ciprofloxacin was started. Gram-negative coccobacilli were isolated by blood culture. The organism was identified as Francisella tularensis. We started him on a ten-day course of gentamicin after which he improved. This is, to the best of our knowledge, the first reported case of bacteremia caused by Francisella tularensis in Sudan.
Asymptomatic Brain Edema after Hemodialysis Initiation in a Patient with Severe Uremia
Authors: Kiichiro Fujisaki, Kaneyasu Nakagawa, Hiroshi Nagae, Toshiaki Nakano, Masatomo Taniguchi, Kosuke Masutani, Takanari Kitazono, Kazuhiko Tsuruya
Journal: Case Rep Med (2017)
MeSH terms: Case Reports

A 66-year-old man with severe renal insufficiency presented with mild confusion associated with uremia. Cranial magnetic resonance imaging (MRI) showed no remarkable changes. The patient was placed on short-duration hemodialysis (2 hours) with smaller surface area and low blood flow (100 mL/min) to avoid dialysis disequilibrium syndrome (DDS). His consciousness gradually improved and he did not develop apparent DDS symptoms. However, T2-weighted FLAIR MRI showed increased signal intensities bilaterally in the cortical and subcortical areas of the occipital lobe on day 15. In other words, cranial MRI showed cerebral edema, indicating asymptomatic DDS. On day 29, cranial MRI showed a return to findings on admission. In this case, because the patient did not have apparent DDS symptoms despite MRI changes, we diagnosed asymptomatic cerebral edema. The patient was discharged on regular intermittent HD without any neurological deficits. No further neurological disturbances were noted during 1-year follow-up. MRI findings in ESKD patients without DDS symptoms help to clarify the diagnosis of cerebral edema. In this case, the patient did not have apparent DDS symptoms and was therefore diagnosed with asymptomatic cerebral edema.
Florid cystitis glandularis (intestinal type) with mucus extravasation: Two case reports and literature review
Authors: Tao Zhang, Si-Fan Yin, Wen-Bo Feng, Chang-Xing Ke
Journal: Front Surg (2023)
Keywords: clinical features, cystitis glandularis, diagnosis, intestinal type, treatment
MeSH terms: Case Reports

Background:
Cystitis glandularis is a common bladder epithelial lesion characterized by hyperplasia and metaplasia of the bladder mucosa epithelium. The pathogenesis of cystitis glandularis of the intestinal type is unknown and less common. When cystitis glandularis (intestinal type) is extremely severely differentiated, it is called florid cystitis glandularis (the occurrence is extremely rare).
Case summary:
Both patients were middle-aged men. In patient 1, the lesion was also seen in the posterior wall and was diagnosed more than 1 year ago as cystitis glandularis with urethral stricture. Patient 2 was examined for symptoms such as hematuria and was found to have an occupied bladder; both were treated surgically, and the postoperative pathology was diagnosed as florid cystitis glandularis (intestinal type), with mucus extravasation.
Conclusion:
The pathogenesis of cystitis glandularis (intestinal type) is unknown and less common. When cystitis glandularis of the intestinal type is extremely severely differentiated, we call it florid cystitis glandularis. It is more common in the bladder neck and trigone. The clinical manifestations are mainly symptoms of bladder irritation, or hematuria as the main complaint, which rarely leads to hydronephrosis. Imaging is nonspecific and the diagnosis depends on pathology. Surgical excision of the lesion is possible. Due to the malignant potential of cystitis glandularis of intestinal type, postoperative follow-up is required.
Bilio-thorax: an unrecognized complication of liver surgery
Authors: Asad Ali Kerawala, Abid Jamal
Journal: Int J Surg Case Rep (2020)
Keywords: biliothorax, liver resection, oncology, pleural effusion, surgery, tube thoracostomy
MeSH terms: Case Reports

Introduction:
Although a rare complication, Biliothorax, or chole-thorax as it has been called in literature, is a devastating entity if not recognized early. Various causes have been reported for biliothorax like inflammatory, abdominal trauma, neoplastic, the most frequent being percutaneous hepatic intervention.
Case:
We present the case of a 60 year old male, treated for colon cancer, who developed this complication after liver surgery. He was promptly diagnosed, managed conservatively and responded to our treatment. This has been infrequently reported.
Discussion:
Although a rare complication, Biliothorax, is a fatal complication if missed. The presence of bile in the pleural cavity is damaging and can lead to empyema, entrapped lung and even ARDS. Hence it is imperative to recognize early, find the cause and treat appropriately.
Conclusion:
Biliothorax is an under reported complication of liver surgery which can have disastrous effects on the patient leading to morbidity and even mortality. It is vital to recognize it early and treat accordingly.
Successful management of neonatal alloimmune thrombocytopenia in the second pregnancy: a case report
Authors: Fabiana Mendes Conti, Sergio Hibner, Thiago Henrique Costa, Marcia Regina Dezan, Maria Giselda Aravechia, Ricardo Antonio D Almeida Pereira, Andrea Tiemi Kondo, Élbio Antônio D'Amico, Mariza Mota, José Mauro Kutner
Journal: Einstein (Sao Paulo) (2014)
MeSH terms: Pregnancy, High-Risk, Thrombocytopenia, Neonatal Alloimmune / therapy, Case Reports, Adult, Antigens, Human Platelet / genetics, Female, Humans, Immunoglobulins, Intravenous / administration & dosage, Infant, Newborn, Intracranial Hemorrhages / diagnostic imaging, Intracranial Hemorrhages / prevention & control, Male, Platelet Count, Pregnancy, Risk Assessment, Thrombocytopenia, Neonatal Alloimmune / diagnostic imaging, Treatment Outcome, Ultrasonography, Prenatal, Antigens, Human Platelet, Immunoglobulins, Intravenous

Neonatal alloimmune thrombocytopenia is a serious disease, in which the mother produces antibodies against fetal platelet antigens inherited from the father; it is still an underdiagnosed disease. This disease is considered the platelet counterpart of the RhD hemolytic disease of the fetus and newborn, yet in neonatal alloimmune thrombocytopenia the first child is affected with fetal and/or neonatal thrombocytopenia. There is a significant risk of intracranial hemorrhage and severe neurological impairment, with a tendency for earlier and more severe thrombocytopenia in subsequent pregnancies. This article reports a case of neonatal alloimmune thrombocytopenia in the second pregnancy affected and discusses diagnosis, management and the clinical importance of this disease.
A case of recalcitrant silicone granuloma treated with adalimumab: A case report
Authors: Orli M Silverberg, Benoit M Cyrenne, David Croitoru, Matthew K Sandre, Kucy Pon
Journal: SAGE Open Med Case Rep (2022)
Keywords: silicone granuloma, adalimumab, granulomatous inflammation, recalcitrant silicone granuloma, tumour necrosis factor-α inhibitor
MeSH terms: Case Reports

Liquid silicone is a relatively inexpensive injectable used for soft tissue augmentation. Injection of silicone is associated with a risk of delayed granuloma formation associated with elevated levels of tumour necrosis factor-α. We report a case of recalcitrant silicone granulomas following facial injections of silicone successfully treated with tumour necrosis factor-α blockade. Our case, as well as previous reports, demonstrates the effectiveness of this therapy for the treatment of foreign body granulomas from due to silicone.
Case report: Analysis of phage therapy failure in a patient with a Pseudomonas aeruginosa prosthetic vascular graft infection
Authors: Lucia Blasco, Inmaculada López-Hernández, Miguel Rodríguez-Fernández, Javier Pérez-Florido, Carlos S Casimiro-Soriguer, Sarah Djebara, Maya Merabishvili, Jean-Paul Pirnay, Jesús Rodríguez-Baño, María Tomás, Luis Eduardo López Cortés
Journal: Front Med (Lausanne) (2023)
Keywords: pseudomonas aeruginosa, antibiotic resistance, bypass, phage, phage therapy, prosthetic vascular graft infection
MeSH terms: Case Reports

Clinical case of a patient with a Pseudomonas aeruginosa multidrug-resistant prosthetic vascular graft infection which was treated with a cocktail of phages (PT07, 14/01, and PNM) in combination with ceftazidime-avibactam (CZA). After the application of the phage treatment and in absence of antimicrobial therapy, a new P. aeruginosa bloodstream infection (BSI) with a septic residual limb metastasis occurred, now involving a wild-type strain being susceptible to ß-lactams and quinolones. Clinical strains were analyzed by microbiology and whole genome sequencing techniques. In relation with phage administration, the clinical isolates of P. aeruginosa before phage therapy (HE2011471) and post phage therapy (HE2105886) showed a clonal relationship but with important genomic changes which could be involved in the resistance to this therapy. Finally, phenotypic studies showed a decrease in Minimum Inhibitory Concentration (MIC) to ß-lactams and quinolones as well as an increase of the biofilm production and phage resistant mutants in the clinical isolate of P. aeruginosa post phage therapy.
Delayed Diagnosis of Acute Rheumatic Fever in a Patient with Multiple Emergency Department Visits
Authors: Inna Kaminecki, Renuka Verma, Jacqueline Brunetto, Loyda I Rivera
Journal: Case Rep Pediatr (2018)
MeSH terms: Case Reports

While the incidence of acute rheumatic fever (ARF) in the United States has declined over the past years, the disease remains one of the causes of severe cardiovascular morbidity in children. The index of suspicion for ARF in health care providers may be low due to decreasing incidence of the disease and clinical presentation that can mimic other conditions. We present the case of a 5-year-old boy with a history of intermittent fevers, fatigue, migratory joint pain, and weight loss following group A Streptococcus pharyngitis. The patient presented to the emergency department twice with the complaints described above. On his 3rd presentation, the workup for his symptoms revealed the diagnosis of acute rheumatic fever with severe mitral and aortic valve regurgitation. The patient was treated with penicillin G benzathine and was started on glucocorticoids for severe carditis. The patient was discharged with recommendations to continue secondary prophylaxis with penicillin G benzathine every 4 weeks for the next 10 years. This case illustrates importance of primary prevention of acute rheumatic fever with adequate antibiotic treatment of group A Streptococcus pharyngitis. Parents should also receive information and education that a child with a previous attack of ARF has higher risk for a recurrent attack of rheumatic fever. This can lead to development of severe rheumatic heart disease. Prevention of recurrent ARF requires continuous antimicrobial prophylaxis. Follow-up with a cardiologist every 1-2 years is essential to assess the heart for valve damage.
Recalcitrant lymphocytoma cutis successfully treated with mycophenolate mofetil
Authors: Farhan Mahmood, Iris Yh Teo, Mark G Kirchhof
Journal: SAGE Open Med Case Rep (2021)
Keywords: lymphocytoma cutis, cutaneous lymphoid hyperplasia, immunosuppressant, mycophenolate mofetil
MeSH terms: Case Reports

Lymphocytoma cutis reflects an exaggerated local immunologic reaction to a stimulus presenting on the head, neck, or upper extremities as a firm 1-3 cm erythematous and/or violaceous plaque or nodule. However, lymphocytoma cutis may be difficult to treat due to the variety of causative agents and the lack of reported successful treatments and outcomes. Here, we present a case of 68-year-old female with recalcitrant lymphocytoma cutis resistant to other first-line therapies including tacrolimus ointment and steroids. The red plaque on the patient's left cheek was eventually treated with mycophenolate mofetil. Mycophenolate mofetil was an accessible and effective therapeutic option to treat lymphocytoma cutis with minimal side effects.
Case report: Candidiasis of gastrojejunostomosis after pancreaticoduodenectomy: Preliminary experience from two cases
Authors: Thanh Khiem Nguyen, Ham Hoi Nguyen, Cong Long Nguyen, Tuan Hiep Luong, Long Doan Dinh, Van Duy Le, Kim Khue Dang, Thi Lan Tran
Journal: Front Oncol (2022)
Keywords: candidiasis, case report, gastrojejunostomosis, pancreactic cancer, pancreaticoduodenectomy
MeSH terms: Case Reports

Introduction:
Invasive Candida infection, or candidiasis, especially in gastrointestinal tract (GIT) is an infrequent but aggressive disease caused by Candida species. Candidiasis of gastrojejunostomosis after extensive gastrointestinal surgery may cause serious complications such as perforative peritonitis and anastomotic stenosis, which requires surgical interventions.
Case presentation:
Our two patients had undergone pancreaticoduodenectomy (PD), respectively, due to pancreatic ductal adenocarcinoma and intraductal papillary mucinous neoplasms of the pancreatic head. Both the patients were malnutritioned and debilitated before the surgery, and they required reoperation for postoperative Candidiasis-relevant complication.In the first case, the patient was readmitted to the hospital with symptoms of perforative peritonitis, for which he underwent surgery and had Candida found in both gastrojejunostomosis ulcer and peritoneal fluid. In our second case, the patient was admitted to the hospital twice after the first operation and diagnosed with Candida-induced gastrojejunostomosis stenosis by esophagogastroduodenoscopy (EGD) and endoscopic biopsy. Fluconazole was indicated for a 2-week regimen. Blood sample withdrawn afterward showed no evidence of fungal agents, and the anastomotic stenosis responded well to treatment. However, after 3 weeks, he came back with cachexia and symptoms of gastrojejunostomotic stenosis. EGD showed no image of fungal agents but anastomotic stenosis due to chronic inflammatory process. The patient was then reoperated to redo his gastrojejunostomosis.
Conclusion:
Candidiasis of gastrojejunostomosis after extensive gastrointestinal surgery such as PD is a very aggressive condition that may cause perforative peritonitis and anastomotic stenosis. However, there have been no publications on this disorder, and the strategic treatment remains unknown. We hereby present a report of two cases with postoperative gastrojejunostomosis candidiasis presenting with non-specific but aggressive and early clinical symptoms.
Corneal ectasia, cerulean (blue dot) cataract with acute hydrops in a child with Down's syndrome and hypothyroidism - a rare presentation
Authors: Bharat Gurnani, Kirandeep Kaur, Shivanand Narayana
Journal: GMS Ophthalmol Cases (2023)
Keywords: down’s syndrome, acute corneal hydrops, congenital cerulean cataract, keratoconus
MeSH terms: Case Reports

Down's syndrome or trisomy 21 is a genetic disorder caused by presence of all or a part of a third copy of chromosome 21. Keratoconus occurs in up to 15% of the adult population with Down's syndrome. There is a close consortium between trisomy 21 and keratoconus (a conical ectatic protrusion of the cornea), and children with Down's syndrome are also susceptible to developing thyroid disease, including hypothyroidism and thyrotoxicosis with exophthalmos. The authors describe a case report on acute corneal hydrops with congenital cerulean cataract in a patient with Down's syndrome with hypothyroidism having bilateral advanced keratoconus. As per the detailed literature review, this is the first case of Down's syndrome with hypothyroidism presenting with acute corneal hydrops.
Familial intragenic duplication of ANKRD11 underlying three patients of KBG syndrome
Authors: Milena Crippa, Daniela Rusconi, Chiara Castronovo, Ilaria Bestetti, Silvia Russo, Anna Cereda, Angelo Selicorni, Lidia Larizza, Palma Finelli
Journal: Mol Cytogenet (2015)
Keywords: ankrd11 intragenic duplication, genotype–phenotype correlations, kbg syndrome, low-level mosaicism
MeSH terms: Case Reports

Background:
KBG syndrome, a rare autosomal disorder characterised by distinctive craniofacial and skeletal features and developmental delay, is caused by haploinsufficiency of the ANKRD11 gene.
Results:
Here we describe two siblings with multiple symptoms characteristic of KBG and their mother with a milder phenotype. In the siblings, array-based comparative genomic hybridization (array CGH) identified an intragenic microduplication affecting ANKRD11 that was absent from the parents' array CGH profiles. Microsatellite analysis revealed the maternal origin of the rearrangement and interphase fluorescent in situ hybridization (i-FISH) experiments identified the rearrangement in low-level mosaicism in the mother. Molecular characterisation of the duplication allele demonstrated the presence of two mutant ANKRD11 transcripts containing a premature stop codon and predicting a truncated non-functional protein.
Conclusions:
Similarly to deletions and point mutations, this novel pathogenetic rearrangement causes haploinsufficiency of ANKRD11, resulting in KBG syndrome.
Case Report: The Use of Rituximab in Antibody-Negative Autoimmune Encephalitis
Authors: Soo-Hyun Park, Yong-Chan Kim
Journal: Front Neurol (2021)
Keywords: cd19+/cd20+, antibody-negative, autoimmune encephalitis, brain mri, rituximab, treatment
MeSH terms: Case Reports

Antibody-negative autoimmune encephalitis (AE) is challenging to diagnose because clinically suspected antibody-negative AE cases are difficult to confirm. If not treated properly, like antibody-positive AE, antibody-negative AE can cause irreparable damage to patients. Previously, immunotherapy was effective in treating patients with antibody-negative AE. We present the case of a 63-year-old man who was admitted to our hospital with altered cognition. He was diagnosed with antibody-negative AE based on CSF, brain MRI, and B-cell counts; autoimmune diseases with similar clinical symptoms were ruled out. He was treated with immunotherapy, especially rituximab, for antibody-negative AE. After 3 weeks of treatment, his mental state and brain MRI results, concomitant with a decrease in CD19+/CD20+ B-cell counts. This case report shows that patients with antibody-negative AE may respond to rituximab, similar to those with antibody-positive AE. Thus, potentially undetected antibodies could be responsible for the treatment outcome.
A male congenital pelvic arteriovenous malformation diagnosed by abdominal ultrasound: A case report and literature review
Authors: Yanhua Huang, Xiatian Liu, Hongwei Qian
Journal: Front Surg (2023)
Keywords: case report, congenital arteriovenous malformation, male, pelvis, ultrasound, urology
MeSH terms: Case Reports

Congenital pelvic arteriovenous malformation (AVM) is a rare vascular abnormality whereby arteries and veins are directly connected with malformed vascular plexus. Owing to its low incidence and nonspecific symptoms, the ultrasonographic characteristics of congenital pelvic AVM in males have been infrequently studied. A 30-year-old man visited our hospital complaining of progressive pain in the right lower abdomen and lumbar area since 2 months previously. Abdominal ultrasound (US) was performed at the initial examination and pelvic AVM was diagnosed, which was then confirmed by computed tomographic angiography. After right internal iliac artery embolization, the patient recovered uneventfully and remained asymptomatic during the 12-month follow-up period. Congenital pelvic AVM should thus be included in the differential diagnosis of pelvic cystic masses in males despite its low incidence, with US also being of great diagnostic value. We describe the ultrasonic features of AVM in detail and hope that this study may contribute to the ultrasonic diagnosis of congenital pelvic AVM in males.
Laparoscopic cholecystectomy during pregnancy: A case series
Authors: Hadi Ahmadi Amoli, Hassan Tavakoli, Ali Yaghoubi Notash, Maryam Sajad Far, Patricia Khashayar
Journal: J Minim Access Surg (2008)
Keywords: cholecystectomy, laparoscopy, pregnancy

This study was conducted to evaluate the safety of laparoscopic cholecystectomy (LC) during pregnancy. Patients who underwent LC were selected from several hospital databases, only six were performed during pregnancy. In this series, one of the two patients who had LC in the first trimester underwent elective termination of pregnancy while the other one gave birth to a term child normally. Half of the four who had the second trimester LC had normal deliveries at term whereas for the other two cesarean section was performed. None of our patients underwent LC in the third trimester. The findings of the present study suggest LC to be a safe procedure performed during the first and second trimester of pregnancy.