OpenMed/multicare-articles huggingface dataset:
High number of CD56(bright) NK-cells and persistently low CD4+ T-cells in a hemophiliac HIV/HCV co-infected patient without opportunistic infections
Authors: Giulia Fregni, Anaenza Freire Maresca, Valérie Jalbert, Anne Caignard, Daniel Scott-Algara, Elisabeth Bordé Cramer, Elisabeth Rouveix, Marie C Béné, Claude Capron
Journal: Virol J (2013)
MeSH terms: CD4-Positive T-Lymphocytes / immunology, CD56 Antigen / analysis, HIV Infections / complications, Hemophilia A / complications, Hepatitis C / complications, Killer Cells, Natural / immunology, Case Reports, Adult, CD4 Lymphocyte Count, Cell Degranulation, HIV Infections / immunology, Hemophilia A / immunology, Hepatitis C / immunology, Humans, Killer Cells, Natural / chemistry, Male, Viral Load, CD56 Antigen, NCAM1 protein, human

Background:
Both the human immunodeficiency virus (HIV) and hepatitis C virus (HCV), either alone or as coinfections, persist in their hosts by destroying and/or escaping immune defenses, with high morbidity as consequence. In some cases, however, a balance between infection and immunity is reached, leading to prolonged asymptomatic periods. We report a case of such an indolent co-infection, which could be explained by the development of a peculiar subset of Natural Killer (NK) cells.
Results:
Persistently high peripheral levels of CD56+ NK cells were observed in a peculiar hemophiliac HIV/HCV co-infected patient with low CD4 counts, almost undetectable HIV viral load and no opportunistic infections. Thorough analysis of NK-subsets allowed to identify a marked increase in the CD56bright/dim cell ratio and low numbers of CD16+/CD56- cells. These cells have high levels of natural cytotoxicity receptors but low NCR2 and CD69, and lack both CD57 and CD25 expression. The degranulation potential of NK-cells which correlates with target cytolysis was atypically mainly performed by CD56bright NK-cells, whereas no production of interferon γ (IFN-γ) was observed following NK activation by K562 cells.
Conclusions:
These data suggest that the expansion and lytic capacity of the CD56bright NK subset may be involved in the protection of this « rare » HIV/HCV co-infected hemophiliac A patient from opportunistic infections and virus-related cancers despite very low CD4+ cell counts.
Biopsychosocial Approach for Conservative Management of Abdominal Pregnancy in Previously Infertile Mother
Authors: Junita Indarti, Yudianto Budi Saroyo, Beta Andewi Resti Anggraheni, Andika Widyatama
Journal: Case Rep Med (2022)
MeSH terms: Case Reports

Background:
Abdominal pregnancy is a rare condition in which early termination is generally recommended. However, there are cases of abdominal pregnancies treated using expectant management with satisfactory outcomes. This management may be considered in special cases, such as previously infertile couples.
Case:
A case of a 27-year-old woman with infertility history complaining of vaginal bleeding was presented. Physical and ultrasound examination revealed an abdominal pregnancy with 18 weeks of gestation. Although she was ready to abort the pregnancy, she insisted to try expectant management for her pregnancy. Her baby was finally born at 25 weeks via laparotomy.
Conclusion:
Abdominal pregnancy is a rare and highly morbid form of ectopic pregnancy. It demands a skilled approach in management. A comprehensive and holistic approach is required to deliver the best outcome for the patient and her family.
Researching the delivery of health and nutrition interventions for women and children in the context of armed conflict: lessons on research challenges and strategies from BRANCH Consortium case studies of Somalia, Mali, Pakistan and Afghanistan
Authors: Michelle F Gaffey, Anushka Ataullahjan, Jai K Das, Shafiq Mirzazada, Moctar Tounkara, Abdirisak A Dalmar, Zulfiqar A Bhutta
Journal: Confl Health (2020)
Keywords: afghanistan, humanitarian health, mali, maternal and child health, mixed methods, pakistan, research challenges, somalia

Background:
The BRANCH Consortium recently conducted 10 mixed-methods case studies to investigate the provision of health and nutrition interventions for women and children in conflict-affected countries, aiming to better understand the dominant influences on humanitarian health actors' programmatic decision-making and how such actors surmount intervention delivery barriers. In this paper, the research challenges encountered and the mitigating strategies employed by the case study investigators in four of the BRANCH case study contexts are discussed: Somalia, Mali, Pakistan and Afghanistan.
Discussion:
Many of the encountered research challenges were anticipated, with investigators adopting mitigation strategies in advance or early on, but others were unexpected, with implications for how studies were ultimately conducted and how well the original study aims were met. Insecurity was a fundamental challenge in all study contexts, with restricted geographical access and concerns for personal safety affecting sampling and data collection plans, and requiring reliance on digital communications, remote study management, and off-site team meetings wherever possible. The need to navigate complex local sociopolitical contexts required maximum reliance on local partners' knowledge, expertise and networks, and this was facilitated by early engagement with a wide range of local study stakeholders. Severe lack of reliable quantitative data on intervention coverage affected the extent to which information from different sources could be triangulated or integrated to inform an understanding of the influences on humanitarian actors' decision-making.
Conclusion:
Strong local partners are essential to the success of any project, contributing not only technical and methodological capacity but also the insight needed to truly understand and interpret local dynamics for the wider study team and to navigate those dynamics to ensure study rigour and relevance. Maintaining realistic expectations of data that are typically available in conflict settings is also essential, while pushing for more resources and further methodological innovation to improve data collection in such settings. Finally, successful health research in the complex, dynamic and unpredictable contexts of conflict settings requires flexibility and adaptability of researchers, as well as sponsors and donors.
An exceptional Albanian family with seven children presenting with dysmorphic features and mental retardation: maternal phenylketonuria
Authors: Ina Knerr, Johannes Zschocke, Stefan Schellmoser, Hans G Topf, Corina Weigel, Jörg Dötsch, Wolfgang Rascher
Journal: BMC Pediatr (2005)
MeSH terms: Abnormalities, Multiple / etiology, Intellectual Disability / etiology, Microcephaly / etiology, Phenylketonuria, Maternal / diagnosis, Case Reports, Adolescent, Adult, Child, Child, Preschool, Female, Humans, Male, Phenylketonuria, Maternal / blood, Phenylketonuria, Maternal / genetics, Pregnancy

Background:
Phenylketonuria is an inborn error of amino acid metabolism which can cause severe damage to the patient or, in the case of maternal phenylketonuria, to the foetus. The maternal phenylketonuria syndrome is caused by high blood phenylalanine concentrations during pregnancy and presents with serious foetal anomalies, especially congenital heart disease, microcephaly and mental retardation.
Case presentation:
We report on an affected Albanian woman and her seven children. The mother is affected by phenylketonuria and is a compound heterozygote for two pathogenetic mutations, L48S and P281L. The diagnosis was only made in the context of her children, all of whom have at least one severe organic malformation. The first child, 17 years old, has a double-chambered right ventricle, vertebral malformations and epilepsy. She is also mentally retarded, microcephalic, exhibits facial dysmorphies and small stature. The second child, a girl 15 years of age, has severe mental retardation with microcephaly, small stature and various dysmorphic features. The next sibling, a boy, died of tetralogy of Fallot at the age of three months. He also had multiple vertebral and rib malformations. The subsequent girl, now eleven years old, has mental retardation, microcephaly and epilepsy along with facial dysmorphy, partial deafness and short stature. The eight-year-old child is slightly mentally retarded and microcephalic. A five-year-old boy was a premature, dystrophic baby and exhibits mental retardation, dysmorphic facial features, brachydactyly and clinodactyly of the fifth finger on both hands. Following a miscarriage, our index case, the youngest child at two years of age, is microcephalic and mentally retarded and shows minor facial anomalies. All children exhibit features of phenylalanine embryopathy caused by maternal phenylketonuria because the mother had not been diagnosed earlier and, therefore, never received any diet.
Conclusion:
This is the largest family suffering from maternal phenylketonuria reported in the literature. Maternal phenylketonuria remains a challenge, especially in woman from countries without a neonatal screening program. Therefore, it is mandatory to be alert for the possibility of maternal phenylketonuria syndrome in case of a child with the clinical features described here to prevent foetal damage in subsequent siblings.
Epithelioid hemangioma of lingual alveolar mucosa: an immunohistochemical case report
Authors: Rajat Nangia, Abhiney Puri, Rakhi Gupta, Sucheta Bansal, Amita Negi, Isha Chauhan
Journal: Case Rep Med (2014)

Epithelioid hemangioma is a rare benign vascular lesion that presents as a nodular lesion in the skin of head and neck region. It is a superficial vascular entity which can either be due to tumor or reactive lesion, but the exact etiology is still unknown. We hereby present a rare case which has been reported with the history of small nodular-like growth on mandibular buccal and lingual area. The excisional biopsy was performed and tissue was submitted for histopathological diagnosis. The immunohistochemistry was performed to check the expression of CD31 marker which proved that origin of epithelioid cells was vascular.
Ectopic thyroid tissue in the breast: A case report
Authors: Zheng Gong, Yan Yang, Zegang Liu
Journal: Open Life Sci (2023)
Keywords: breast, case report, ectopic thyroid tissue, embryology
MeSH terms: Case Reports

Ectopic thyroid tissue is a rare condition manifested as the appearance of thyroid tissue outside the thyroid gland. Here, we report a case of ectopic thyroid tissue in the breast. A 48-year-old Chinese woman who was diagnosed with breast cancer received modified radical mastectomy. A thyroid tissue was found on subsequent pathological examination. The ectopic thyroid tissue was confirmed by immunohistochemistry staining of thyroid biomarkers, including thyroglobulin, thyroid transcription factor-1, and thyroid peroxidase. Currently, abnormal thyroid anlage descent is the main theory to explain ectopic thyroid tissue, especially lingual thyroid. However, it is far-fetched to explain the pathogenesis of ectopic thyroid tissues existed in organs or tissues far from thyroid such as iris, cardiac, pulmonary, duodenal, adrenal, and vertebral. Here, we reviewed the previous cases of ectopic thyroid tissue in breast and proposed a "entoderm migration" theory to explain distant ectopic thyroid tissues based on embryonic development perspective.
Infection of a Ventricular Septal Defect Patch with Acremonium Species
Authors: Nosheen Nasir, Kauser Jabeen, Joveria Farooqi, Binish Arif Sultan, Afia Zafar, Aamir Hameed Khan, Muneer Amanullah, Farheen Ali
Journal: Case Rep Infect Dis (2018)
MeSH terms: Case Reports

A ventricular septal defect (VSD) patch infection with Acremonium species isolated from vegetation and blood culture is described. Antifungal treatment was discontinued after 3 months and patient developed relapse. Surgery with prolonged oral voriconazole was instituted with recovery. We emphasize importance of surgery and prolonged therapy to treat such infections.
Mixed hepatoblastoma in a young male adult: a case report and literature review
Authors: Valeria Fiaschetti, Roberto Fiori, Eleonora Gaspari, Sonia Crusco, Giovanni Simonetti
Journal: Case Rep Med (2010)
MeSH terms: Case Reports

Hepatoblastoma (HB) is a rare malignant tumour of the liver and usually occurs in the first three years of life. Most of these tumours arise in the embryo; hence it seems to be unusual that hepatoblastoma occurs in adults and is an exceptional cause of primary malignant liver tumour in adult patients. The diagnosis is often overlooked, and patients might be diagnosed at late stages of the disease at risk of increased mortality. In this paper we report a case of a 30-year-old man with mixed hepatoblastoma and abdominal pain, hepatomegaly and fever. The patient under went noninvasive diagnostic methods: duplex scanning, Computed Tomography (CT), and Magnetic Resonance imaging (MRI). In our experience, despite the important role of histological sample provided by biopsy in defining the diagnosis, very important was the role of MRI, more than ultrasonography and enhanced CT. The MRI detects tumor features as size, margins, and ratio with neighboring organs in order to get the best surgical approach.
Post traumatic type 1 diabetes mellitus (insulin-dependent): a case report
Authors: Rabie Karrouri
Journal: Pan Afr Med J (2014)
Keywords: trauma, children, diabetes, pathogenesis, stress
MeSH terms: Diabetes Mellitus, Type 1 / etiology, Life Change Events, Stress, Psychological / complications, Case Reports, Child, Diabetes Mellitus, Type 1 / psychology, Humans, Libya / ethnology, Male, Refugees / psychology, Severity of Illness Index, Tunisia

Most researchers have studied the influence of life stress as precipitating the onset of type 1 diabetes, but as the relationship between severe psychological trauma and diabetes has been a rarely studied subject in paediatric age group. Here, we report the case of a 10-year-old Libyan boy, without personal or familial diabetes mellitus history, which is presented to Moroccan medico-surgical field hospital, installed in Tunisia for refugees of the Libyan revolution, for type 1 diabetes appeared immediately after severe psychological trauma.
Abnormal Breathing Patterns Predict Extubation Failure in Neurocritically Ill Patients
Authors: Pragya Punj, Premkumar Nattanmai, Pravin George, Christopher R Newey
Journal: Case Rep Crit Care (2017)
MeSH terms: Case Reports

In neurologically injured patients, predictors for extubation success are not well defined. Abnormal breathing patterns may result from the underlying neurological injury. We present three patients with abnormal breathing patterns highlighting failure of successful extubation as a result of these neurologically driven breathing patterns. Recognizing abnormal breathing patterns may be predictive of extubation failure and thus need to be considered as part of extubation readiness.
Papillary Thyroid Carcinoma Presenting As a Cystic Neck Lesion: Case Series
Authors: Sethu T Subha, Mohd-Adzreil Bakri, Hisyam Salleh, Mohamad Doi, Abdul-Jalil Nordin
Journal: Iran J Otorhinolaryngol (2018)
Keywords: 18 f-fdg-pet ct scan, cystic neck lesion, fnac, papillary thyroid carcinoma
MeSH terms: Case Reports

Introduction:
Papillary thyroid carcinoma (PTC) constitutes 75-85% of all thyroid cancers. PTC usually presents as a subtle, commonly slow-growing, painless thyroid mass or a solitary nodule in the neck. This presentation of a cystic neck lump, without the presence of a thyroid nodule, may imitate the course of a benign disease, thus delaying diagnosis and proper treatment.
Case report:
Three cases that had been initially presented as a cystic neck lesion in which a benign etiology was considered primarily were compiled in this study. PTC was only diagnosed after surgical excision of these cystic neck lesions in the first two cases, and after performing fine needle aspiration cytology (FNAC) and an 18fluorine-fluorodeoxyglucose positron emission tomography computed tomography (18F-FDG-PET CT) scan in the latter case.
Conclusion:
PTC can sometimes present as a cystic neck mass; a presentation which is usually related to a benign lesion. This case series emphasizes that patients who appear to have a solitary cystic neck mass must be treated with a high index of clinical suspicion. Although not a first-line imaging modality, 18F-FDG-PET can be extremely useful in assessing patients with a cystic neck lesion, where diagnosis is still uncertain after standard investigations such as ultrasonography and FNAC have been performed.
Dosing of Ertapenem in an Extreme Obesity: A Case Report of 250 kg Patient
Authors: Jana Lass, Kadri Tamme, Karin Kipper, Joel Starkopf
Journal: Case Rep Crit Care (2017)
MeSH terms: Case Reports

Limited available data for dosing in obesity of the medicines used in this case are discussed, with the emphasis on ertapenem. The case illustrates the difficulties in dosing medicines to morbidly overweight patients. The number of such patients is increasing but data on adequate doses of medicines are scarce. We demonstrate that ertapenem 1,5 g i.v. once daily provided adequate drug exposure for susceptible bacteria in a 250 kg patient with normal renal function. The case suggests the usefulness of therapeutic drug monitoring of antibiotics, especially in critically ill patients.
Primary Hyperoxaluria Diagnosed Based on Bone Marrow Biopsy in Pancytopenic Adult with End Stage Renal Disease
Authors: Pardis Nematollahi, Fereshteh Mohammadizadeh
Journal: Case Rep Hematol (2015)

Inborn errors of metabolism cause increase of metabolites in serum and their deposition in various organs including bone marrow. Primary hyperoxaluria (PH) is a rare inborn error in the pathway of glyoxylate metabolism which causes excessive oxalate production. The disease is characterized by widespread deposition of calcium oxalate (oxalosis) in multiple organs. Urinary tract including renal parenchyma is the initial site of deposition followed by extrarenal organs such as bone marrow. This case report introduces a 54-year-old woman with end stage renal disease presenting with debilitating fatigue and pancytopenia. The remarkable point in her past medical history was recurrent episodes of nephrolithiasis, urolithiasis, and urinary tract infection since the age of 5 years and resultant end stage renal disease in adulthood in the absence of appropriate medical evaluation and treatment. She had an unsuccessful renal transplantation with transplant failure. The patient underwent bone marrow biopsy for evaluation of pancytopenia. Microscopic study of bone marrow biopsy led to the diagnosis of primary hyperoxaluria.
Methotrexate-induced acute leukemia: report of three cases and review of the literature
Authors: Khalid A Al-Anazi, Khalid I Eltayeb, Mohammed Bakr, Fahad I Al-Mohareb
Journal: Clin Med Case Rep (2009)
Keywords: acute lymphoblastic leukemia, acute myeloid leukemia, methotrexate, myelodysplastic syndrome, rheumatoid arthritis
MeSH terms: Case Reports

For many years, methotrexate has been used in the treatment of certain chronic medical disorders e.g. rheumatoid arthritis and psoriasis as well as a number of malignant disorders e.g. acute lymphoblastic leukemia, certain types of lymphoma and breast carcinoma. Its use has been associated with various systemic toxicities and complications. The association between methotrexate therapy and the development of lymphoma and pseudolymphoma is well established. In patients treated with methotrexate, the development of leukemia has been attributed to either the primary disorder e.g. rheumatoid arthritis or to other drugs used concomitantly e.g. cyclophosphamide. Reported here are two patients with rheumatoid arthritis and one patient with psoriasis treated with low dose methotrexate for variable periods of time. Two of these patients developed acute myeloid leukemia on myelodysplastic syndrome background, while the third patient developed pre-B acute lymphoblastic leukemia that expressed few myeloid markers and had a positive philadelphia chromosome. To our knowledge, these are the first reported cases of methotrexate-induced acute leukemia.
Heterotopic Ossification Encountered During a Complex Ventral Hernia Repair: Case Report and Literature Review
Authors: Takintope Akinbiyi, Sanjeev Kaul
Journal: Eplasty (2017)
Keywords: abdominal wall calcification, ectopic/heterotopic ossification, myositis ossificans traumatica, soft-tissue calcification, ventral hernia repair

Introduction: Heterotopic ossification involves the formation of trabecular bone outside of its usual anatomic location. While it is a well-known entity in orthopedic and spinal injury literature, it has also been observed after midline laparotomy and severe burns. Methods/Case Report: We present a case of a 69-year-old man who presented for ventral hernia repair after a prolonged postoperative course following colectomy involving an open abdomen with eventual closure with skin grafting. Results: Two large calcified objects were encountered during the excision of the skin graft from the small intestine and during the component separation. They had grown into the anterior fascia and rectus muscle and interdigitated between loops of the small bowel. After careful resection of the 2 calcified objects, a ventral hernia repair with a component separation was successfully performed. Pathology was consistent with heterotopic ossification. After 18 months, there was no clinical evidence of recurrence. Discussion: Heterotopic ossification is not frequently encountered during ventral hernia repairs, but its presence can complicate repair. Resection is the only option in the context of hernia repair. If recognized preoperatively, waiting up to a year for the bone to mature before excision has been suggested, but there is minimal data to support this. Consultation with a general surgeon is also advised in case the calcified tissue involves the underlying viscera.
Unusual combined thymic mucoepidermoid carcinoma and thymoma: a case report and review of literature
Authors: Shi-gang Wu, Yang Li, Bin Li, Xiao-ying Tian, Zhi Li
Journal: Diagn Pathol (2014)
MeSH terms: Carcinoma, Mucoepidermoid / pathology, Neoplasms, Multiple Primary / pathology, Thymoma / pathology, Thyroid Neoplasms / pathology, Case Reports, Review, Humans, Male, Middle Aged

Background:
In rare condition, combined thymic epithelial tumors showing either type A or type B thymomas areas combined with thymic carcinoma components may occur in thymus. Mucoepidermoid carcinoma (MEC) of the thymus is rare in thymic carcinoma, and so far there is no report to describe a combined epithelial tumor of thymus with MEC component. We report an unusual case of combined thymic MEC/type B2 thymoma in a middle-aged male occurring in a mass of anterior mediastinum.
Case report:
A 51-year-old Chinese male patient presented with a 6-month history of right ptosis and progressive muscle weakness. Computed tomography (CT) examination revealed a solitary, well-circumscribed mass was in the anterior mediastinum with mild heterogeneous enhancement. Histologically, the mass contained two separated components and displayed typically histological features of low-grade MEC and type B2 thymoma, respectively. There was no gradual transition of these two components observed in mass, and no enlarged lymph node was found in the surrounding tissues. A diagnosis of combined thymic MEC/type B2 thymoma was made. The patient received thymectomy to resect the mass totally. After surgery, chemotherapy with regiments of cisplatin and mitomycin, and radiotherapy of the main tumor bed were performed on the patient. There was no evidence of tumor recurrence during the period of 12 months follow-up.
Conclusion:
To our best knowledge, this is the first report of combined thymic epithelial tumor with MEC component. Although this tumor is rare, the diagnosis of a thymic MEC should be taken into consideration when a combined epithelial tumor is occasionally encountered in thymus.
Virtual slides:
The virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/9721397571157894.