OpenMed/multicare-articles huggingface dataset:
New Insights about Pulmonary Epithelioid Hemangioendothelioma: Review of the Literature and Two Case Reports
Authors: Romeu Duarte Mesquita, Marta Sousa, Carmen Trinidad, Eugénia Pinto, Iosu Antón Badiola
Journal: Case Rep Radiol (2017)
MeSH terms: Case Reports

Pulmonary epithelioid hemangioendothelioma (PEH) is a rare neoplasm of vascular origin. There are three different major imaging patterns identified in thoracic manifestation of epithelioid hemangioendothelioma: (1) multiple pulmonary nodules; (2) multiple pulmonary reticulonodular opacities; and (3) diffuse infiltrative pleural thickening. Radiographically, presence of bilateral multiple nodules is the most common pattern of presentation. The diagnosis is made on the basis of histopathological findings and confirmed by positive immunohistochemistry staining. Although the prognostic factors for PEH have not yet been well established, a better prognosis is usually associated with the multinodular pattern. We report two different imagological presentations of this rare disease, based on two institutional experiences, along with a review of the relevant literature.
Case report: Primary immunodeficiency due to a novel mutation in CARMIL2 and its response to combined immunomodulatory therapy
Authors: Yu Zhu, Lili Ye, Hua Huang, Xuemei Xu, Yu Liu, Jian Wang, Yanliang Jin
Journal: Front Pediatr (2023)
Keywords: cd4+/cd8+ lymphocytes, combined immunotherapy, novel mutation, primary immunodeficiency, whole exome sequencing (wes)
MeSH terms: Case Reports

Capping protein regulator and myosin 1 linker 2 (CARMIL2) is necessary for invadopodia formation, cell polarity, lamellipodial assembly, membrane ruffling, acropinocytosis, and collective cell migration. CARMIL2 deficiency is a rare autosomal recessive disease characterized by dysfunction in naïve T-cell activation, proliferation, differentiation, and effector function and insufficient responses in T-cell memory. In this paper, we report a 9-year-old female patient with a novel pathogenic variant in CARMIL2 (c.2063C > G:p.Thr688Arg) who presented with various symptoms of primary immunodeficiencies including recurrent upper and lower respiratory infections, perioral and perineum papules, reddish impetiginized atopic dermatitis, oral ulcer, painful urination and vaginitis, otitis media, and failure to thrive. A missense mutation leading to insufficient CARMIL2 protein expression, reduced absolute T-cell and natural killer cell (NK cell) counts, and marked skewing to the naïve T-cell form was identified and indicated defective maturation of T cells and B cells. Following 1 year of multitargeted treatment with corticosteroids, hydroxychloroquine, mycophenolate mofetil, and thymosin, the patient presented with significant regression in rashes. CD4+ T-cell, CD8+ T-cell, and NK cell counts were significantly improved.
Muscle Fatigue Post-stroke Elicited From Kilohertz-Frequency Subthreshold Nerve Stimulation
Authors: Yang Zheng, Henry Shin, Xiaogang Hu
Journal: Front Neurol (2018)
Keywords: electromyography, kilohertz-frequency, muscle fatigue, stroke, transcutaneous electrical nerve stimulation
MeSH terms: Case Reports

Purpose: Rapid muscle fatigue limits clinical applications of functional electrical stimulation (FES) for individuals with motor impairments. This study aimed to characterize the sustainability of muscle force elicited with a novel transcutaneous nerve stimulation technique. Method: A hemiplegic chronic stroke survivor was recruited in this case study. Clustered subthreshold pulses of 60-μs with kilohertz (12.5 kHz) carrier frequency (high-frequency mode, HF) were delivered transcutaneously to the proximal segment of the median/ulnar nerve bundles to evaluate the finger flexor muscle fatigue on both sides of the stroke survivor. Conventional nerve stimulation technique with 600-μs pulses at 30 Hz (low-frequency mode, LF) served as the control condition. Fatigue was evoked by intermittently delivering 3-s stimulation trains with 1-s resting. For fair comparison, initial contraction forces (approximately 30% of the maximal voluntary contraction) were matched between the HF and LF modes. Muscle fatigue was evaluated through elicited finger flexion forces (amplitude and fluctuation) and muscle activation patterns quantified by high-density electromyography (EMG). Result: Compared with those from the LF stimuli, the elicited forces declined more slowly, and the force plateau was higher under the HF stimulation for both the affected and contralateral sides, resulting in a more sustainable force output at higher levels. Meanwhile, the force fluctuation under the HF stimulation increased more slowly, and, thus, was smaller after successive stimulation trains compared with the LF stimuli, indicating a less synchronized activation of muscle fibers. The efficiency of the muscle activation, measured as the force-EMG ratio, was also higher in the HF stimulation mode. Conclusion: Our results indicated that the HF nerve stimulation technique can reduce muscle fatigue in stroke survivors by maintaining a higher efficiency of muscle activations compared with the LF stimulation. The technique can help improve the performance of neurorehabilitation methods based on electrical stimulation, and facilitate the utility of FES in clinical populations.
Severe Hypoglycemia Caused by a Giant Borderline Phyllodes Tumor of the Breast: A Case Report and Literature Review
Authors: Yaoxia Liu, Min Zhang, Xudan Yang, Zhen Fan, Yi Li, Tao Wang, Ping Chen
Journal: Front Endocrinol (Lausanne) (2022)
Keywords: breast, hypoglycemia, insulin-like growth factor-2, non-islet cell tumor hypoglycemia, phyllodes tumor
MeSH terms: Breast Neoplasms / complications, Breast Neoplasms / metabolism, Breast Neoplasms / surgery, Hypoglycemia / etiology, Phyllodes Tumor / complications, Phyllodes Tumor / metabolism, Phyllodes Tumor / surgery, Case Reports, Systematic Review, Research Support, Non-U.S. Gov't, C-Peptide, Female, Humans, Insulin, Middle Aged, C-Peptide, Insulin

A case of hypoglycemic coma caused by a giant borderline phyllodes tumor of the breast has been described. The patient, a 63-year-old woman, was admitted with recurrent unconsciousness. She had a giant breast tumor with decreased blood glucose, insulin, and C-peptide. The patient's hypoglycemia resolved rapidly after resection of the breast tumor. Pathological examination indicated a borderline phyllodes tumor of the breast, and immunohistochemistry suggested high expression of insulin-like growth factor-2 (IGF-2) in the tumor tissue. A literature review is also included to summarize the clinical characteristics of such patients and to serve as a unique resource for clinical diagnosis and treatment of similar cases.
Robot-Assisted Removal of a Partially Intravesical Intrauterine Device (IUD) and Large Bladder Stone
Authors: Cassandra Heaney, Paul Lee, Andrew Winer
Journal: Case Rep Urol (2023)
MeSH terms: Case Reports

An intrauterine device (IUD) is a highly effective and widely utilized option for long-acting reversible contraception. IUDs are generally well-tolerated with a low rate of serious complications. Perforation of an IUD through the uterine wall and into the urinary bladder is a rare event that may be asymptomatic. The approach for surgical removal primarily depends on the location of the device. We present a case report of a 41-year-old woman who was found to have a partially intravesical IUD and associated 2.4 cm bladder calculus. Removal of the intravesical IUD and stone was achieved with cystoscopy, cystolitholapaxy, and robot-assisted laparoscopic cystotomy.
Transformation of Malignant Melanoma From Giant Nevus in Infantile Penis
Authors: Dong Hwan Lee, Min Ji Kim, Il Jae Lee, Dong Ha Park
Journal: Front Surg (2020)
Keywords: melanoma, pediatric surgery, pediatric tumor, penile neoplasms pathology, transformation
MeSH terms: Case Reports

Background: Malignant melanoma is the most serious type of skin cancer, and its incidence rate increases with age. Malignant melanoma in infants has been rarely reported in the literature. Herein, we report a case of malignant transformation of a nodular lesion found in the penis of a patient with a giant congenital nevus. Case presentation: A 1-month-old male patient was admitted due to the presence of a giant congenital nevus involving the lower abdomen, bilateral inguinal areas, genitals, and left thigh and knee. Six months later, nodules measuring 1 cm in diameter protruding from the genital area were noted, and a part of the nodule was removed via elliptical excision with the patient under general anesthesia. Gross examination showed an edematous lesion similar to a neurofibroma and with unclear boundaries. Biopsy revealed a malignant melanoma, with a Breslow thickness of at least 3 mm, and absence of lymphovascular invasion; the biopsy confirmed incomplete excision. The patient was scheduled for radical resection, but reconstruction was not performed following surgical resection due to the guardian's refusal. Hence, the patient only received an adjuvant medical treatment and eventually died. Conclusion: We reported a rare case of an infant with a malignant melanoma in the penis. Congenital malignant melanoma rarely occurs in infants; however, due to its fatal consequences, follow-up should be performed to assess for malignant changes.
Positive fertility outcomes in a female with classic congenital adrenal hyperplasia following bilateral adrenalectomy
Authors: Urania Dagalakis, Ashwini Mallappa, Meredith Elman, Martha Quezado, Deborah P Merke
Journal: Int J Pediatr Endocrinol (2016)
Keywords: adrenal hyperplasia, bilateral adrenalectomy, classic cah, fertility outcomes, hyperandrogenism
MeSH terms: Case Reports

Background:
Classic congenital adrenal hyperplasia (CAH) requires lifetime steroid replacement and supraphysiologic glucocorticoid dose is often required for adequate adrenal androgen suppression. Patients often suffer from long-term co-morbidities and female infertility is common.
Case presentation:
We report the use of laparoscopic bilateral adrenalectomy as a treatment for a 21 year old female with classic simple virilizing CAH and infertility. She presented as an adolescent with increasing weight gain, amenorrhea and elevated adrenal androgens despite the use of dexamethasone (250 mcg given twice daily), and fludrocortisone (150 mcg daily). An anti-androgen (flutamide 250 mg given twice daily) and a combined oral contraceptive pill were added to her regimen and prevented progressive virilization, but she eventually desired fertility. A bilateral laparoscopic adrenalectomy was performed at age 21. The right adrenal gland weighed 41.8 grams and the left gland 45.5 grams. There were no complications during the surgery. Since the surgery, she has had a total of three pregnancies, resulting in 3 healthy full-term infants. Follow-up 7 years later at age 27 revealed overall excellent health with a BMI of 25.1 kg/m(2), no evidence of adrenal rest tissue based on hormonal testing, above average quality-of-life based on 36-item short-form health survey and she has not experienced an adrenal crisis.
Conclusions:
This case highlights the use of bilateral adrenalectomy as a treatment option for female infertility in a patient with classic CAH and difficult-to-control hyperandrogenism secondary to adrenal nodular hyperplasia. Outstanding quality-of-life, disease control and fertility were achieved.
Imaging findings of ossifying fibromyxoid tumor with histopathological correlation: A case report
Authors: Soshi Ideta, Jun Nishio, Mikiko Aoki, Tetsuro Ishimatsu, Kazuki Nabeshima, Hiroshi Iwasaki, Masatoshi Naito
Journal: Oncol Lett (2013)
Keywords: bone scintigraphy, computed tomography, magnetic resonance imaging, ossifying fibromyxoid tumor

Ossifying fibromyxoid tumor (OFMT) is a soft tissue tumor of uncertain lineage that most often arises in the extremities of adults. Imaging findings of this uncommon tumor are rare. We, herein, present a case of OFMT occurring in the left thigh of a 36-year-old male. Radiological examinations revealed a well-circumscribed subcutaneous mass with an incomplete shell of peripheral ossification, suggesting a benign condition. Following complete excision, the mass was histopathologically diagnosed as an OFMT. The patient demonstrated no evidence of local recurrence within 11 months of follow-up. We describe the clinicopathological and radiological features, and review the relevant literature.
COVID-19 Infection Negative in Nasopharyngeal Swabs but Suspected in Computed Tomography and Confirmed in Bronchoalveolar Lavage Material
Authors: Robert Chrzan, Tadeusz Popiela, Maciej Małecki, Jan Skupień, Amira Bryll, Anna Grochowska
Journal: Case Rep Infect Dis (2021)
MeSH terms: Case Reports

We present a case of a patient with clinical symptoms of pneumonia, negative in several polymerase chain reaction COVID-19 tests from nasopharyngeal swabs but suspected in computed tomography and finally confirmed in bronchoalveolar lavage material.
Case report: Brachial plexopathy caused by malignant peripheral nerve sheath tumor and review of the literature
Authors: Mengjie Chen, Xiuli Li, Xinhong Feng
Journal: Front Neurol (2023)
Keywords: emg, brachial plexopathy, case report, diagnosis, malignant peripheral nerve sheath tumors
MeSH terms: Case Reports

Brachial plexopathy (BP) is easily misdiagnosed due to its complexity and varying clinical presentation. Malignant peripheral nerve sheath tumors (MPNST) can accumulate in the brachial plexus and share symptoms with BP, which may hinder the differential diagnosis between BP induced by radiation or metastases, and MPNST-derived BP, in patients with a history of breast cancer and radiation exposure. A 34-year-old Chinese female presented with MPNST. The tumor involved the brachial plexus. She had a history of breast cancer and radiotherapy. The first consideration was radiation- or breast cancer metastasis-derived BP. Clinical examination was performed. Finally, a diagnosis of MPNST of the brachial plexus was made, which guided an accurate treatment plan. This report highlights the importance of correctly diagnosing BP etiology for guiding precise treatment. BP caused by MPNST needs to be considered in clinical practice, and biopsy plays a central role in the differential diagnosis. Complete local surgical resection can prolong survival of patients with MPNST and improve treatment prognosis.
Internal Maxillary Artery-Radial Artery-Middle Cerebral Artery Bypass and STA-MCA Bypass for the Treatment of Complex Middle Cerebral Artery Bifurcation Aneurysm: A Case Report
Authors: Chaojue Huang, Shixing Qin, Guan Cao, Wei Huang, Yongjia Yu
Journal: Front Surg (2022)
Keywords: extracranial-to-intracranial bypass, high flow bypass, internal maxillary artery, middle cerebral artery bifurcation aneurysm, pediatric complex aneurysm, superficial temporal artery
MeSH terms: Case Reports

Background:
Children's complex middle cerebral artery (MCA) aneurysm is a relatively rare occurrence. When the huge aneurysm is located in the MCA bifurcation with an inconspicuous neck and involving numerous arteries, intravascular interventional surgery or aneurysm clipping are often difficult treatment options. At this point, high flow bypass revascularization is necessary as a treatment to preserve cerebral blood flow. In recent years, the internal maxillary artery (IMA) has gradually become the mainstream donor artery of thw high flow bypass. We performed internal maxillary artery -radial artery-middle cerebral artery (IMA-RA-MCA) and superficial temporal artery-middle cerebral artery (STA-MCA) bypass as the treatment of a complex MCA bifurcation aneurysm in consideration of the patient's condition and the advantage of the IMA. According to the author, this case is the youngest reported case of IMA-RA-MCA bypass at present.
Case description:
A male child, 7 years and 8 months, was admitted to the hospital due to "recurrent headache for more than 9 months," DSA indicated that there was a large wide-necked aneurysm at the bifurcation of the right MCA M1 segment, with a size of about 1.16*1.58*1.32 cm. The inflow path of the aneurysm was in front of M1 bifurcation, and one outflow path originated from the aneurysm body, and another small outflow path attached to the aneurysm body. After completing the preoperative evaluation, an extended pterional approach with zygomatic osteotomy was performed to fully expose the aneurysm and IMA, harvesting the left radial artery at the same time, then a STA-MCA bypass, IMA-RA-MCA bypass, and aneurysm trapping were performed. postoperative re-examination showed that bypass vessels and the distal middle artery vessels were patent and the aneurysm disappeared, the child has no neurological dysfunction.
Conclusions:
IMA-RA-MCA bypass is an effective high-flow cerebral blood reconstruct scheme in the treatment of complex middle cerebral artery bifurcation aneurysms. This case can provide a reference for the surgical treatment of complex middle cerebral artery bifurcation aneurysms in children.
Spontaneous Retrobulbar Haematoma
Authors: Mehmet Deveer, Nesat Cullu, Halil Beydilli, Hamdi Sozen, Onder Yeniceri, Selcuk Parlak
Journal: Case Rep Radiol (2015)

Background. Spontaneous orbital haemorrhage is a very rare condition and vision-threatening event. It may occur due to trauma, orbital surgery/injections, orbital vascular anomalies, and a variety of systemic predisposing factors. Signs of retrobulbar hemorrhage include proptosis, ophthalmoplegia, increased intraocular pressure, loss of pupillary reflexes, and optic disc or retinal pallor. Both Computed Tomography scan and Magnetic Resonance Imaging may be performed in the diagnosis. Case Report. A 31-year-old woman was referred to our hospital with a complaint of headache and blurred vision following a strong sneeze. Ophthalmological examination revealed mild Relative Afferent Pupillary Defect in left eye. Computed Tomography revealed left hyperdense retrobulbar mass and displaced optic nerve. T1 weighted hypointense, T2 weighted hyperintense and non-enhanced round shape, sharply demarcated lesion measuring 18 × 15 × 14 × mm in diameter compatible with haematoma was detected by MRI. Surgically Caldwell-Luc procedure was performed. Histological examination confirmed haematoma. Follow-up Magnetic Resonance Imaging revealed a small reduction in the size of lesion but not complete resolution. The patient's complaint was regressed. She is now free of symptoms and is still under surveillance. To our knowledge, this is the first reported case of retrobulbar haematoma caused by sneeze.
Temporary Abdominal Closure Combined With an Irrigating System Utilizing Hypochlorous Acid Solution to Decrease Abdominal Mucopurulence
Authors: Marc R Matthews, Asia N Quan, Alexandra S Weir, Kevin N Foster, Daniel M Caruso
Journal: Eplasty (2018)
Keywords: vac veraflo, abthera device, negative pressure wound therapy, temporary abdominal closure, vashe solution
MeSH terms: Case Reports

Introduction: Leaving the abdominal cavity open is a well-described and frequently utilized technique in the treatment of severe intra-abdominal sepsis. Irrigation through a negative pressure wound therapy device is a technique employed to assist in the closure of wounds as well as the reduction of bacterial contamination. Furthermore, hypochlorous acid has been found to be safe and effective in microorganismal elimination from extremity wounds. There is no literature regarding the infusion of hypochlorous solution into the abdominal cavity for intra-abdominal sepsis or mucopurulent abscesses or biofilm. Objectives: A 47-year-old man with granulomatosis polyangiitis was started on weekly rituximab. After 4 infusions, skin sloughing, ultimately diagnosed as toxic epidermal necrolysis, developed. During the hospital course, he developed sepsis and bowel perforation necessitating an exploratory laparotomy. The abdomen was left open with a temporary abdominal closure using the Abthera open abdomen negative wound therapy device; however, the abdomen remained infected with visually diffuse, thickening mucopurulence despite multiple washouts. Therefore, a VAC Vera-Flo irrigation device was combined with the Abthera open abdomen negative wound therapy device and cyclical irrigation of hypochlorous acid. After 72 hours, the purulence visually was improved and no adverse events were recorded with the placement of intra-abdominal hypochlorous acid. Conclusions: The combination of two medical devices for the intra-abdominal instillation of irrigation is considered "off-label use" from the manufacturer's recommendations. In addition, the repeated instillation of hypochlorous acid solution has not been described but was noted to have visually decreased the contaminated effluent within the intra-abdominal fluid.
Safe surgical technique: cement-augmented pedicle screw instrumentation and balloon-guided kyphoplasty for a lumbar burst fracture in a 97-year-old patient
Authors: Thomas Freude, Benjamin König, Frank Martetschläger, Sebastian Siebenlist, Markus Neumaier, Ulrich Stöckle, Stefan Döbele
Journal: Patient Saf Surg (2013)
MeSH terms: Case Reports

Background:
During the last few years, an increasing number of unstable thoracolumbar fractures, especially in elderly patients, has been treated by dorsal instrumentation combined with a balloon kyphoplasty. This combination provides additional stabilization to the anterior spinal column without any need for a second ventral approach.
Case presentation:
We report the case of a 97-year-old male patient with a lumbar burst fracture (type A3-1.1 according to the AO Classification) who presented prolonged neurological deficits of the lower limbs - grade C according to the modified Frankel/ASIA score. After a posterior realignment of the fractured vertebra with an internal screw fixation and after an augmentation with non-absorbable cement in combination with a balloon kyphoplasty, the patient regained his mobility without any neurological restrictions.
Conclusion:
Especially in older patients, the presented technique of PMMA-augmented pedicle screw instrumentation combined with balloon-assisted kyphoplasty could be an option to address unstable vertebral fractures in "a minor-invasive way". The standard procedure of a two-step dorsoventral approach could be reduced to a one-step procedure.
Cervical Spinal Osteomyelitis with Epidural Abscess following an Escherichia coli Urinary Tract Infection in an Immunocompetent Host
Authors: Abdelmoniem Moustafa, Rowida Kheireldine, Zubair Khan, Hussam Alim, Mohammad Saud Khan, Mohd Amer Alsamman, Eslam Youssef
Journal: Case Rep Infect Dis (2019)
MeSH terms: Case Reports

Spinal epidural abscess (SEA) is uncommon with an incidence reported as 0.33-1.96 abscesses per 10,000 hospital admissions per year. Two-thirds of the cases were caused by Staphylococcus aureus. Escherichia coli (E. coli) is a less common cause of SEA, and it is usually after urinary tract infection in patient with preexisting risk factor. A 69-year-old male with a past medical history significant for prostatitis was admitted with fever, altered mental status, neck pain, progressive lower extremities weakness, and frequent falls for 7 days. Both blood and urine cultures grew E. coli. Lumbar puncture showed 94 RBCs, 24 WBCs (16% neutrophils and 46% lymphocytes), and elevated protein level at 1140 mg/dl with no bacteria. C-spine MRI showed epidural abscess along the anterior and right lateral margin of the cord causing cord compression from C5 through C7, anterior perivertebral abscess from C4 through T2, marrow edema involving C6 and C7 vertebral bodies with increased signal in the intervertebral disc space at C6-C7, and consistent with osteomyelitis and discitis. Anterior cervical decompression with evacuation of anterior epidural abscess with fusion was done. The culture from the epidural abscess grew E coli. A diagnosis of SEA should be considered in patients presenting with progressive weakness and neurological deficits following UTI and is to be confirmed by MRI. E. coli could be the culprit for epidural abscess and spine osteomyelitis even in immunocompetent patients.