OpenMed/multicare-articles huggingface dataset:
A rare case of sinonasal glomangiopericytoma post operative accidental diagnosis and managment-A case report
Authors: Nitin Sharma, Dushyant Mandlik, Purvi Patel, Parin Patel, Aditya Joshipura, Mitesh Patel, Srinal Mankiwala, Ashutosh Vatsyayan, Tulika Dubey, Kintan Sanghvi, Diva Shah, Shubhada Kanhere, Shailesh Talati, Kaustubh Patel
Journal: Int J Surg Case Rep (2019)
Keywords: endoscopic sinus surgery, glomangiopericytoma, haemangiopericytoma, nasal, tumor

Introduction:
Glomangiopericytoma is a rare neoplasm of low malignant potential. It is a rare type of haemangiopericytoma located in nasal cavity. This neoplasm has good prognosis and complete surgical excision is treatment of choice. This case report is representing one such neoplasm. This reporting is done in line with the SCARE criteria (Agha et al., 2018 [1]).
Case presentation:
We presenting a case of 54 year old male patient from upper socioeconomic status who presented at our institution with history of surgery (endoscopic sinus surgery with Septoplasty) 15 days back. A Final histopathology report suggested glomangiopericytoma. Since primary surgery was not done as per oncologic principals, patient was advised for adjuvant radiation. At our institute patient was evaluated again. Revision surgery was done. Patient was discharged next day.
Discussion:
Glomangiopericytoma is a rare neoplasm with incidence of less than 0.5% of all neoplasms of sinonasal cavity. Prognosis is very good after complete surgical excision. It often confuses clinicians with nasal polyps. Here also patient was operated initially considering as benign polyposis outside. Patient was re-operated again to ensure the complete clearance.
Conclusion:
This is the typical case of converting dual modality treatment to single modality with the help of knowledge, communication, transparent team work. This also a rare type of neoplasm and by reporting this rare case we are contributing to data pool of nasal tumors where lack of reporting is major obstacle in the formation of uniform treatment guidelines.
Fifth annual workshop of cytoreductive surgery for advanced ovarian cancer and peritoneal surface malignancies
Authors: Krishnansu S Tewari
Journal: Gynecol Oncol Res Pract (2016)
Keywords: cytoreduction, ovarian cancer, workshop
MeSH terms: Case Reports

The Fifth Annual Advanced Course in Cytoreductive Surgery for Ovarian Cancer and Peritoneal Surface Malignancies was held at and sponsored by the Division of Gynecologic Oncology at the the University of California, Irvine on Friday and Saturday, October 9-10, 2015. The workshop was comprised of didactic modules, historical treatise, an impassioned tribute, a cadaver laboratory, and heated intraperitoneal chemotherapy demonstration. This was a not-for-profit workshop, and registration fees were used to support course faculty travel to U.C. Irvine and to pay for the cadavers. The original 56 available spots were filled within three weeks of the initial announcement, prompting procurement of two additional cadavers to satisfy registration overflow and accommodate the six U.C. Irvine fellows-in-training. While international participation in the Workshops continues to rise, we have also noted more U.S.-trained Gynecologic Oncologists among the registrants.
Case Report: The First Reported Case of Bullous Lichen Planus-Systemic Lupus Erythematosus Overlap Syndrome
Authors: Yi Liu, Xuelei Liang, Haixuan Wu, Fenglin Zhuo
Journal: Front Med (Lausanne) (2021)
Keywords: alopecia, bullous lichen planus, case report, overlap syndrome, systemic lupus erythematosus
MeSH terms: Case Reports

Introduction: Lichen planus/lupus erythematosus overlap syndrome is rarely seen in the clinic and has the characteristic clinical manifestations, histopathology, and immunopathology of lichen planus (LP) and lupus erythematosus (LE). This is the first reported case of bullous lichen planus (BLP)/systemic lupus erythematosus (SLE) overlap syndrome with hair loss as the first symptom. Case Presentation: A 48-year-old female presented with alopecia for half a year, and skin lesions accompanied by itching on her face, trunk, and limbs for 3 months. She had a history suggestive of photosensitivity. Laboratory tests and histopathology were performed for diagnosis. Histopathologic features of the upper arm and back of the hand were consistent with BLP, whereas the scalp lesion indicated LE. Laboratory examination indicated positive for antinuclear antibody (ANA) (1:160), leukopenia, increased urinary protein, decreased C3/C4, and normal BP180. The patient was given glucocorticoid combined with acitretin and immunosuppressive therapy after a definite diagnosis of BLP/SLE overlap syndrome. The lesions of the patient disappeared and some hair had regrown during the two years of follow-up. Conclusion: This is the first reported case of BLP/SLE overlap syndrome which responded well to glucocorticoids, retinoids, and immunosuppressive drugs. Multiple biopsies from characteristic lesions will guide doctors to avoid misdiagnoses and delayed treatment.
Lymphoepithelioma-like carcinoma of the bladder: a case report and review of the literature
Authors: Kenichi Mori, Tadasuke Ando, Takeo Nomura, Fuminori Sato, Hiromitsu Mimata
Journal: Case Rep Urol (2013)

Lymphoepithelioma-like carcinoma (LELC) in the bladder is uncommon with a reported incidence of 0.4%-1.3% of all bladder carcinomas. In Japan, some occurrences of LELC have been reported in the renal pelvis and ureter but only two in the bladder. A bladder tumor was identified in a 70-year-old man suffering from macroscopic hematuria for 2 months. Sections of the transurethral tumor resection showed invasive high-grade urothelial carcinoma. The patient was diagnosed with local invasive bladder tumor, and cystectomy with ileal conduit formation was performed. The final pathological evaluation was predominant LELC with urothelial carcinoma. We present a new case of LELC in the bladder and performed a review of all published cases of LELC in the urinary tract to obtain its characteristics and prognostic guide.
Diffuse Neurofibroma in a Patient with Unknown History of NF1
Authors: Mahmoud A K Ebrahim, Mishal M AlMutairi, Khaled M Hindi, Jassem M Bastaki
Journal: Case Rep Otolaryngol (2018)
MeSH terms: Case Reports

Malignant peripheral nerve sheath tumor (MPNST) is a rare disease in the parotid gland with a poor prognosis in most cases. The disease most often develops in the setting of neurofibromatosis type 1 (NF1) but can also occur sporadically. Herein, we report a rare case of MPNST in the parotid gland, in a patient with no previous history of NF1. Initial investigations of the patient, which consisted of laboratory investigations, ultrasound imaging of the swelling, fine-needle aspiration (FNA), computed tomography (CT) scan, and magnetic resonance imaging (MRI) of the neck and swelling, were compatible with a benign pleomorphic adenoma of the parotid gland. However, intraoperatively, the dissection was challenging as the tumor was adherent to the neighboring tissue. A diagnosis of MPNST arising from a diffuse neurofibroma was established based on clinicopathologic features of the disease. The patient, who exhibited clinical features compatible with (NF1), proceeded for radiotherapy following surgery to continue his treatment.
Long-term complete response with third-line PARP inhibitor after immunotherapy in a patient with triple-negative breast cancer: a case report
Authors: Roberta Caputo, Martina Pagliuca, Matilde Pensabene, Sara Parola, Michelino De Laurentiis
Journal: Front Oncol (2023)
Keywords: brca mutation, parp inhibitor, advanced breast cancer, immunotherapy, triple negative breast cancer
MeSH terms: Case Reports

While standard treatment has shown efficacy in patients with breast cancer gene (BRCA) mutations, recurrence rates are high and additional effective therapies are needed. Olaparib, a poly adenosine diphosphate-ribose polymerase (PARP) inhibitor, approved for the treatment of metastatic germline BRCA1/BRCA2 breast cancer (BC), has demonstrated evidence of a progression-free survival (PFS) benefit, good safety profile, and improved quality of life compared with standard chemotherapy. We here describe the case of a patient with BRCA1 mutated advanced BC and a long history of response to chemotherapy and immunotherapy who received systemic treatment with olaparib. First diagnosed in March 2011 at the age of 38 years with early-stage BC of the right breast, she underwent quadrantectomy plus ipsilateral axillary lymphadenectomy and adjuvant treatments with chemotherapy regimen containing 5-fluorouracil, epirubicin, and cyclophosphamide (FEC) followed by radiotherapy. Five years later, following a contralateral nodule detection leading to left breast quadrantectomy, she received adjuvant systemic treatment with docetaxel plus cyclophosphamide and radiotherapy. Gene testing showed a germline BRCA1 deleterious variant, and she underwent bilateral prophylactic mastectomy and oophorectomy. One year later, skin metastasis and bone infiltrations were detected, and she was started on first-line systemic treatment. The patient was enrolled in the IMpassion131 trial (investigating atezolizumab addition to paclitaxel) but unblinding showed that she was randomized in the placebo arm. She received second-line systemic therapy with LAG525 plus carboplatin (CLAG525B2101 trial) resulting in a PFS of 14 months. At disease progression, she was eligible for systemic third-line therapy with olaparib (300 mg twice daily) and had a complete response after 6 months of therapy and a PFS of 40 months at the time of writing. To the best of our knowledge, this is the first report of a complete response following treatment with third-line systemic olaparib in a long-responding patient and relatively good tolerability and quality of life, pre-treated with both chemotherapy and immunotherapy.
Atypical Clinical and Diagnostic Features in Ménétrier's Disease in a Child
Authors: Michael Chung, Jaime Pittenger, Deborah Flomenhoft, Jeffrey Bennett, Eun-Young Lee, Harohalli Shashidhar
Journal: Case Rep Gastrointest Med (2011)
MeSH terms: Case Reports

Ménétrier's disease is one of the rarest protein-losing gastropathies in childhood. It is characterized clinically by non-specific gastrointestinal symptoms and edema, biochemically by hypoalbuminemia, and pathologically by enlarged gastric folds. In adults, this disease can be devastating with significant morbidity and mortality. In childhood, it is a self-limiting, transient and benign illness. Its treatment is largely supportive with total parenteral nutrition (TPN) while oral intake is encouraged. Acute onset of vomiting in healthy school age children can be initially explained by acute viral gastroenteritis. However, persistent vomiting associated with hematemesis and severe abdominal pain should warrant further work-up. This case report illustrates a self-limiting and rare cause of protein-losing enteropathy called Ménétrier's disease that presented with several variant clinical features not typically described in association with this entity.
Primary hyperoxaluria detected by bone marrow biopsy: case report
Authors: F Nachite, M Dref, A Fakhri, H Rais
Journal: BMC Clin Pathol (2017)
Keywords: bone marrow biopsy, oxalate crystals, primary hyperoxaluria
MeSH terms: Case Reports

Background:
Primary hyperoxaluria is a rare disease with an estimated prevalence of 1 to 3 cases per million. It is due to a hepatic enzyme deficiency responsible for an endogenous overproduction of oxalate. Oxalate crystals commonly deposit in the kidney and more rarely in bone marrow. The literature has reported, to the best of our knowledge, only two cases of hyperoxaluria diagnosed by bone marrow biopsy and our case is the only one that does not show radiological bone lesions.
Case presentation:
A young 22 year old chronic hemodialysis patient with nephrocalcinosis. The patient had a personal and family history of recurrent kidney stones. He presented bone pain with worsening of his general state. On physical examination, no organomegaly was detected. Biological check-up showed only a normochromic and normocytic regenerative anemia resistant to treatment and a bone marrow biopsy was performed. It showed deposits of crystals of oxalate in the bone marrow surrounded by inflammatory reaction against foreign bodies. Given our context, no liver biopsy or genetic studies, which are gold standard of diagnosis testing, were done. The diagnosis of primary hyperoxaluria was made based on morphological characteristics of crystals, his medical and family history, and the absence of any secondary cause of the condition. Since curative treatment is not available in our country, the patient only receives a palliative treatment.
Conclusion:
Primary hyperoxaluria is rarely evoked by the histological study of a bone marrow biopsy. The lack of the possibility of the only effective treatment in our context and the diagnosis, usually late, of this pathology are at the origin of the fatal evolution of the disease in almost all the cases.
Case report: High-frequency repetitive transcranial magnetic stimulation for treatment of hereditary spastic paraplegia type 11
Authors: Songmei Chen, Zhiqing Zhou, Meng Ren, Xixi Chen, Xiaolong Shi, Sicong Zhang, Shutian Xu, Xiaolin Zhang, Xingyuan Zhang, Wanlong Lin, Chunlei Shan
Journal: Front Neurol (2023)
Keywords: corticospinal tract, diffusion tensor imaging, hereditary spastic paraplegia, lower extremity spasticity, repetitive transcranial magnetic stimulation, walking ability
MeSH terms: Case Reports

Hereditary spastic paraplegia (HSP) is a heterogeneous group of inherited neurodegenerative disorders that currently have no cure. HSP type 11 (SPG11-HSP) is a complex form carrying mutations in the SPG11 gene. Neuropathological studies demonstrate that motor deficits in these patients are mainly attributed to axonal degeneration of the corticospinal tract (CST). Repetitive transcranial magnetic stimulation (rTMS) is a non-invasive technique that can induce central nervous system plasticity and promote neurological recovery by modulating the excitability of cortical neuronal cells. Although rTMS is expected to be a therapeutic tool for neurodegenerative diseases, no previous studies have applied rTMS to treat motor symptoms in SPG11-HSP. Here, we report a case of SPG11-HSP with lower extremity spasticity and gait instability, which were improved by applying high-frequency rTMS (HF-rTMS) at the primary motor cortex (M1). Clinical and physiological features were measured throughout the treatment, including the Modified Ashworth Scale (MAS), Berg Balance Scale (BBS), the timed up and go (TUG) test and the 10-meter walk test time (10 MWT). The structure and excitability of the CST were assessed by diffusion tensor imaging (DTI) and transcranial magnetic stimulation (TMS), respectively. After treatment, the patient gained 17 points of BBS, along with a gradual decrease in MAS scores of the bilateral lower extremity. In addition, the TUG test and 10 MWT improved to varying degrees. TMS assessment showed increased motor evoked potential (MEP) amplitude, decreased resting motor threshold (RMT), decreased central motor conduction time (CMCT), and decreased difference in the cortical silent period (CSP) between bilateral hemispheres. Using the DTI technique, we observed increased fractional anisotropy (FA) values and decreased mean diffusivity (MD) and radial diffusivity (RD) values in the CST. It suggests that applying HF-rTMS over the bilateral leg area of M1 (M1-LEG) is beneficial for SPG11-HSP. In this study, we demonstrate the potential of rTMS to promote neurological recovery from both functional and structural perspectives. It may provide a clinical rationale for using rTMS in the rehabilitation of HSP patients.
Oral High-Dose Thiamine Improves the Symptoms of Chronic Cluster Headache
Authors: Costantini Antonio, Tiberi Massimo, Zarletti Gianpaolo, Pala Maria Immacolata, Trevi Erika
Journal: Case Rep Neurol Med (2018)
MeSH terms: Case Reports

Cluster headache is a rare painful primary disorder occurring in either episodic or chronic patterns. Several authors found that the hypothalamus, the brain region regulating endocrine function and autonomic system, is involved in the pathophysiology of cluster headache. Some authors have found in patients affected by this disease abnormality in glucose metabolism. Considering the role of thiamine in brain function, in energetic metabolism, and in pain modulation, we treated a patient affected by cluster headache with oral high-dose thiamine. We report a 41-year-old man suffering from primary chronic cluster headache since the age of 15 years. The patient began oral therapy with high-dose thiamine in December 2016. Oral thiamine supplementation led to a dramatic improvement of the symptoms. The therapy was effective in reversing all the symptoms of the disease. Our observation suggests that a thiamine deficiency due to enzymatic abnormalities or to dysfunction of the circulation of thiamine in the intracellular space could cause a neuronal selective impairment in the centers that are involved in this disease and could have an important role in the pathogenesis of the symptoms of cluster headache.
Perineural Injection for Treatment of Root-Signature Signs Associated with Lateralized Disk Material in Five Dogs (2009-2013)
Authors: Sarah Giambuzzi, Theresa Pancotto, Jeffrey Ruth
Journal: Front Vet Sci (2016)
Keywords: fluoroscopy, intervertebral disk disease, lateralized disk, perineural, root-signature
MeSH terms: Case Reports

Intervertebral disk disease (IVDD) is common in dogs; cervical IVDD accounts for 13-25% of all cases. Ventral slot decompression provides access to ventral and centrally extruded or protruded disk material. However, procedures to remove dorsally or laterally displaced material are more difficult. This case series describes the use of perineural injection as a potential treatment option for dogs experiencing root-signature signs associated with lateralized disk material in the cervical spine. Five dogs underwent fluoroscopically guided perineural injection of methylprednisolone ± bupivacaine. Most patients experienced improvement in root-signature signs and remained pain free without the assistance of oral pain medication. These findings suggest the perineural injection of methylprednisolone ± bupivacaine represents a viable option for dogs with cervical lateralized disk material causing root-signature signs.
Oral and Lower Extremity Ulcers as the Initial Presentation of Granulomatosis with Polyangiitis
Authors: Mohammed Omar Al Salihi, Bianca Dominguez, Viresh Mohanlal, S J Carlan
Journal: Case Rep Med (2022)
MeSH terms: Case Reports

Background:
Granulomatosis with polyangiitis (GPA) is a small vessel vasculitis characterized by lung and kidney involvement. It is typically a disease of white females and has a poor prognosis with the average life expectancy of 5 months for a patient without treatment. Oral and skin ulcers are considered to be rare presentations.
Case:
A 39-year-old black male presented to the hospital with oral and skin ulcers and was diagnosed with GPA based on the biopsies of both cutaneous lesions and kidney. He was started on rituximab with minimal improvement. Later he was admitted to the ICU and had plasmapheresis, and he gradually improved and was discharged home 8 days after admission.
Conclusion:
GPA is an aggressive vascular disorder resulting in possible organ system damage and failure. The role of the sickle cell trait in this patient is undefined, but this combination of gender, race, and presenting symptoms in GPA is extremely unusual.
Acute Hemorrhagic Edema of Infancy after Coronavirus Infection with Recurrent Rash
Authors: Hannah Chesser, Jeffrey M Chambliss, Eric Zwemer
Journal: Case Rep Pediatr (2017)
MeSH terms: Case Reports

Purpura, particularly when accompanied by fever, is a worrisome finding in children. Acute hemorrhagic edema of infancy (AHEI) is a benign type of small-vessel leukocytoclastic vasculitis that presents with progressive purpura and has an excellent prognosis. Patients with AHEI present with large, target-like purpuric plaques affecting the face, ear lobes, and extremities. While the rapid onset of these skin findings can be dramatic, the child with AHEI is usually well appearing with reassuring laboratory testing. We describe a case of a previously healthy 8-month-old female who presented with progressive purpura in a nondependent distribution, low-grade fevers, and extremity swelling. An extensive workup was performed prior to making the diagnosis of AHEI. Coronavirus was implicated as the likely triggering pathogen, and the patient suffered a recurrence of purpuric rash and swelling several weeks after her initial presentation.
Interferon gamma as an immune modulating adjunct therapy for invasive mucormycosis after severe burn - A case report
Authors: Dina M Tawfik, Caroline Dereux, Jan-Alexis Tremblay, Andre Boibieux, Fabienne Braye, Jean-Baptiste Cazauran, Meja Rabodonirina, Elisabeth Cerrato, Audrey Guichard, Fabienne Venet, Guillaume Monneret, Didier Payen, Anne-Claire Lukaszewicz, Julien Textoris
Journal: Front Immunol (2022)
Keywords: mucormycosis, biomarkers, immunotherapy, interferon gamma adjunct therapy, severe burn injuries
MeSH terms: Burns / complications, Mucormycosis / diagnosis, Mucormycosis / drug therapy, Mucormycosis / etiology, Case Reports, Research Support, Non-U.S. Gov't, Adult, Combined Modality Therapy, Female, HLA-DR Antigens, Humans, Interferon-gamma / therapeutic use, Recombinant Proteins, HLA-DR Antigens, Recombinant Proteins, Interferon-gamma

Background:
Mucormycosis is a deadly fungal infection that mainly affects severely immunocompromised patients. We report herein the case of a previously immunocompetent adult woman who developed invasive cutaneous mucormycosis after severe burn injuries. Interferon-gamma (IFN-γ) treatment was added after failure of conventional treatment and confirmation of a sustained profound immunodepression. The diagnosis was based on a reduced expression of HLA-DR on monocytes (mHLA-DR), NK lymphopenia and a high proportion of immature neutrophils. The immune-related alterations were longitudinally monitored using panels of immune-related biomarkers.
Results:
Initiation of IFN-γ was associated with a rapid clinical improvement and a subsequent healing of mucormycosis infection, with no residual fungi at the surgical wound repair. The serial immunological assessment showed sharp improvements of immune parameters: a rapid recovery of mHLA-DR and of transcriptomic markers for T-cell proliferation. The patient survived and was later discharged from the ICU.
Conclusion:
The treatment with recombinant IFN-γ participated to the resolution of a progressively invasive mucormycosis infection, with rapid improvement in immune parameters. In the era of precision medicine in the ICU, availability of comprehensive immune monitoring tools could help guiding management of refractory infections and provide rationale for immune stimulation strategies in these high risk patients.
A rare case of chronic Gianotti-Crosti syndrome: A case report
Authors: Laura D Chin, Carmen Liy-Wong
Journal: SAGE Open Med Case Rep (2023)
Keywords: gianotti–crosti syndrome, acrodermatitis, acrodermatitis papulosa infantum, infantile papular acrodermatitis, papular acrodermatitis of childhood
MeSH terms: Case Reports

Gianotti-Crosti syndrome, also known as papular acrodermatitis of childhood, is a common, self-limiting dermatosis often seen in children with triggers including viral and bacterial infections along with immunizations. Lesions are generally described as asymptomatic, skin colored to erythematous papules and papulovesicles that often spontaneously resolve within weeks. Here, we will discuss Gianotti-Crosti syndrome and present a rare case of chronic Gianotti-Crosti syndrome in an otherwise healthy 3-year-old male persisting for over 20 months. From this report, we aim to better educate the dermatologic community on the extremes of the Gianotti-Crosti syndrome disease course to improve diagnosis and treatment of symptomatic patients.