scholarly journals Rosai-Dorfman-Destombes (RDD) disease presenting as palindromic rheumatism

2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Amin Haghighat Jahromi ◽  
Aaron M. Goodman ◽  
Carl K. Hoh
Keyword(s):  
Lower Extremity ◽  
Nasal Congestion ◽  
Young Patients ◽  
Inflammatory Disorder ◽  
Palindromic Rheumatism ◽  
Case Presentation ◽  
First Case ◽  
Articular Pain ◽  
Work Up ◽  
Rare Diagnosis

Abstract Background Rosai-Dorfman-Destombes (RDD) disease, is a rare proliferative and inflammatory disorder of non-Langerhans cell histiocytes. Case presentation We report a 35-year-old woman, who originally presented with recurrent episodes of lower extremity joint/bone pain and chronic nasal stuffiness and congestion. Her worsening nasal congestion was due to an obstructing nasal cavity lesion which was subsequently biopsied. Pathology was consistent with RDD. 18F-FDG PET images demonstrated intense uptake in the paranasal sinuses and a large pelvic lymph node. Focal osseous lesions with intense 18F-FDG uptake were also observed in the lower extremity, corresponding to areas of peri-articular pain. Rheumatologic work-up was consistent with palindromic rheumatism. She was diagnosed with immune-related disseminated RDD, presenting as palindromic rheumatism. Conclusions This is the first case of RDD presenting as palindromic rheumatism. RDD should be considered as a possible but rare diagnosis in young patients with sinus-related symptoms and lymphadenopathy. The disease can on rare occasions be disseminated and can also present as immune-related RDD, such as in this patient.

scholarly journals The differential for chest pain: when the most common cause is not the answer-a case of de novo esophageal bezoar

2021 ◽  
Vol 7 (1) ◽  
Author(s):  
Kevin Climaco ◽  
Daniel Roubik ◽  
Robert Gorrell
Keyword(s):  
Chest Pain ◽  
Pulmonary Embolus ◽  
Clinical Case ◽  
De Novo ◽  
Case Presentation ◽  
Knowing How ◽  
Initial Resuscitation ◽  
Nasogastric Tubes ◽  
Work Up ◽  
Rare Diagnosis

Abstract Background Having a broad differential and knowing how to manage the different possibilities in a patient with chest pain is important. Esophageal bezoars are rare entities and are even less common in patients without any recent hospitalizations, known achalasia, or nasogastric tubes. Despite their rarity, having it in one’s differential, and knowing how to manage it is important. Case presentation This case presents a patient with mega-esophagus secondary to an esophageal bezoar; and runs through the gamut of morbid chest pathophysiology, its differential, work-up, and management. The case is interesting in that the patient’s initial presentation brings to mind a bevy of feared chest issues to include myocardial infarction, dissection, pulmonary embolus, achalasia, and perforation. Conclusion This clinical case highlights more than just the rare diagnosis of esophageal bezoar. It also goes through initial resuscitation, key concerns, “can’t miss diagnoses”, and finally discusses the feared end state of an esophageal perforation.

Platelet distribution width in young patients with chronic venous insufficiency

Vascular ◽  
2021 ◽  
pp. 170853812110296
Author(s):  
Mustafa Aldemir ◽  
Ahmet Yuksel ◽  
Mehmet Inanir ◽  
Salih Metin ◽  
Yusuf Velioglu ◽  
...  
Keyword(s):  
Lower Extremity ◽  
Clinical Features ◽  
Chronic Venous Insufficiency ◽  
Venous Insufficiency ◽  
Young Patients ◽  
Control Group ◽  
Platelet Distribution Width ◽  
Distribution Width ◽  
Research Hospital ◽  
Lymphocyte Ratio

Objective Platelet distribution width (PDW) has been reported in a wide range of pathological settings. In this study, we aimed to investigate the relationship between PDW and lower extremity chronic venous insufficiency (CVI) by comparing the levels of PDW and other parameters derived from complete blood count (CBC) tests in young individuals with or without lower extremity CVI. Methods This prospective clinical study was conducted between January 2020 and December 2020. A total of 108 patients, 72 patients with lower extremity CVI (study group) and 36 healthy volunteers (control group) were enrolled from the Bursa Yuksek Ihtısas Educatıon Research Hospıtal and the Bolu Abant Izzet Baysal University Training and Research Hospital. The age range of the participants was between 18 and 50 years old. Participants’ baseline clinical features and CBC parameters including PDW, white blood cell, hemoglobin, hematocrit, mean corpuscular volume, red cell distribution width, neutrophil, lymphocyte, platelet count, mean platelet volume, plateletcrit, neutrophil-to-lymphocyte ratio, and platelet-to-lymphocyte ratio were compared between the two groups. Results The groups were statistically similar in terms of baseline clinical features. The median PDW value was significantly higher for the CVI patients relative to the control group (17.6 vs 16.8; p < 0.001). In terms of other CBC parameters, there were no significant differences between the groups. According to ROC analysis, area under the curve of PDW was 0.749 (95% confidence interval: 0.653–0.846 and p < 0.001). If the value of PDW was accepted as 17, it could predict CVI with 76% sensitivity and 59% specificity, whereas a PDW value of 17.5 could predict CVI with 51% sensitivity and 81% specificity. Conclusion Platelet distribution width might be a useful marker to determine an increased inflammatory response and thrombotic status in young patients with CVI.

scholarly journals Immediate post partum macular subretinal bleeding in a highly myopic patient: a case report

2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Karen Bitton ◽  
J.-L. Bacquet ◽  
F. Amoroso ◽  
S. Mrejen ◽  
M. Paques ◽  
...  
Keyword(s):  
Valsalva Maneuver ◽  
Post Partum ◽  
Pathologic Myopia ◽  
Pathological Myopia ◽  
Case Presentation ◽  
Sudden Loss ◽  
Corrected Visual Acuity ◽  
First Case ◽  
History Of ◽  
Macular Hemorrhage

Abstract Background Pathologic myopia is a major cause of visual impairment and blindness. Case presentation We report a case of an immediate post partum macular subretinal bleeding observed in a highly myopic patient. A 30-years-old woman presented two days after childbirth for sudden loss of vision in her right eye. Multimodal imaging showed macular hemorrhage masking a subtle yellowish linear lesion corresponding to lacker crack. Due to the lack of evidence for choroidal neovascularization, a simple clinical and imaging monitoring was recommended. Six weeks later, we noted an improvement in her best-corrected visual acuity and a decreased in size of the macular hemorrhage. Conclusions This is the first case reporting a macular subretinal bleeding on macular lacquer cracks in a highly myopic patient in immediate post partum. Valsalva maneuver associated with vaginal delivery could explain the occurrence of the hemorrhage associated with lacquer crack. However, natural history of pathological myopia could not be excluded.

scholarly journals Emphysematous pyelonephritis with air bubble in the inferior vena cava

2020 ◽  
Vol 26 (1) ◽  
Author(s):  
Tiffany A. Perkins ◽  
Alberic Rogman ◽  
Murali K. Ankem
Keyword(s):  
Septic Shock ◽  
Inferior Vena Cava ◽  
Inferior Vena ◽  
Vena Cava ◽  
Rare Presentation ◽  
Emphysematous Pyelonephritis ◽  
Case Presentation ◽  
Air Bubble ◽  
First Case ◽  
The Right

Abstract Background Emphysematous pyelonephritis (EPN) with gas in the inferior vena cava (IVC) is a rare presentation and to our knowledge, this is the first case report in the urologic literature. Case presentation A 35-Year-old obese diabetic Hispanic female presented to the emergency room with a clinical picture of septic shock. Prompt computerized tomography scan revealed EPN with gas throughout the right renal parenchyma and extending to the right renal vein, IVC, and pulmonary artery. She died before surgical intervention Conclusion This case demonstrates that patients presenting with severe EPN have a high mortality risk and providers should acknowledge that septic shock, endogenous air emboli, or a combination of both could result in cardiovascular collapse and sudden death.

Primary stromal sarcoma of breast: A case report and literature review

2021 ◽  
pp. 1-7
Author(s):  
Mohammad Al-Wiswasy ◽  
Mahmoud Al-Balas ◽  
Raith Al-Saffar ◽  
Hamzeh Al-Balas
Keyword(s):  
Malignant Tumors ◽  
Histopathological Examination ◽  
Prognostic Significance ◽  
Total Duration ◽  
Young Patients ◽  
Soft Tissue Sarcomas ◽  
Breast Mass ◽  
Resection Margins ◽  
Stromal Sarcoma ◽  
First Case

INTRODUCTION: Primary breast sarcoma (PBS) was first described in 1887 as a rare heterogeneous neoplasm arising from the mesenchymal tissue of the mammary gland accounting for less than 5% of all body soft-tissue sarcomas and less than 0.1% of all malignant tumors of the breast. CASE PRESENTATION: A 31-year old lady presented with left breast mass which she felt four years before during which the mass increased in size from 1 to 6 cm in the largest diameter, diagnosed clinically as a benign fibroadenoma without any further cytological or histopathological confirmation. Histopathological examination of the excised breast mass reveals undifferentiated, primary stromal sarcoma of the breast (PSSB), which was followed by mastectomy three weeks later with reconstructive breast surgery with a total duration of follow-up of 3 years thereafter. This is the first case of PSSB reported in Jordan. DISCUSSION/CONCLUSION: PSSB is the generic term given to malignant breast tumors thought to arise from the specialized mesenchymal stroma of the breast but lacking an epithelial component with a phylloides pattern. PSSB is difficult to diagnose preoperatively due to its rarity and inadequate imaging methods to establish an exact diagnosis. The histology of the patient mass may be the leading factor for the management of these tumors. Even in very young patients, a progressively growing breast mass should alert the clinician to investigate for malignancy and verify the results by biopsy. Surgery with adequate resection margins represents the only potentially curative modality with prognostic significance. Adjuvant chemotherapy and radiotherapy are not very beneficial. The prognosis is dismal for patients with lymph node involvement and the size of the tumor has a lesser bearing on the outcome.

scholarly journals Robotic-assisted proximal gastrectomy using the double-flap technique for early gastric cancer with situs inversus totalis: a case report

2021 ◽  
Vol 7 (1) ◽  
Author(s):  
Atsushi Takeno ◽  
Toru Masuzawa ◽  
Shinsuke Katsuyama ◽  
Kohei Murakami ◽  
Kenji Kawai ◽  
...  
Keyword(s):  
Gastric Cancer ◽  
Situs Inversus ◽  
Autosomal Recessive ◽  
Intraoperative Complications ◽  
Proximal Gastrectomy ◽  
Situs Inversus Totalis ◽  
Upper Body ◽  
Robot Assisted ◽  
Case Presentation ◽  
First Case

Abstract Background The robotic system has been applied in the treatment of gastric cancer (GC), and the procedure has been found to be safe and feasible. Situs inversus totalis (SIT) is a relatively rare autosomal recessive congenital anomaly. We successfully performed robot-assisted proximal gastrectomy (RAPG) and handsewn double-flap esophagogastrostomy for GC in a patient with SIT. Case presentation A 71-year-old woman was referred to us with an asymptomatic ulcerative lesion in the upper body of the stomach. Computed tomography revealed that she had SIT. She was diagnosed with cT1bN0M0, cStageIA gastric cancer. RAPG with lymph node dissection and handsewn double-flap esophagogastrostomy was performed. Robotic surgery enabled the surgeon to perform the surgery without changing his position and experiencing any confusion resulting from the patient’s reversed anatomy. It took 448 min, and no intraoperative complications occurred. Her postoperative course was uneventful; she was discharged on postoperative day 10. The final pathologic report showed pT1b1N0M0, pStage IA. Conclusions This is the first case describing RAPG with handsewn double-flap esophagogastrostomy for a SIT patient with early GC.

Fetal pulmonary choristoma: report of first case and literature review

2020 ◽  
Vol 9 (1) ◽  
Author(s):  
Anupam Nanda ◽  
Rajinder Nanda ◽  
Seema Thakur ◽  
Tej Prakash Gupta ◽  
Sudhir Jain ◽  
...  
Keyword(s):  
Literature Review ◽  
Exome Sequencing ◽  
Posterior Fossa ◽  
Spinal Column ◽  
Fetal Autopsy ◽  
Case Presentation ◽  
Antenatal Ultrasound ◽  
Clinical Exome Sequencing ◽  
First Case ◽  
Pathologic Findings

AbstractObjectivesLung tissue choristoma is a very rare disorder where mature lung tissues develop in the site not normal to the lung.Case presentationWe hereby report a first case of fetal pulmonary choristoma in a 23–24 weeks fetus where antenatal ultrasound showed a mass in posterior fossa with severe ventriculomegaly. The mass extended inferiorly in cervical spinal column and thereafter extended in the skin over the back of fetus. Fetal autopsy confirmed these findings. Pathologic findings showed mature lung tissues with bronchi, bronchioles, and alveoli. Clinical exome sequencing showed normal results.ConclusionsWe describe the antenatal ultrasound, fetal autopsy and pathologic findings of an intracranial and cutaneous pulmonary choristoma.

scholarly journals First reported case of penile prosthesis infection from brucellosis: case report

2020 ◽  
Vol 26 (1) ◽  
Author(s):  
Nabil Nabil Moohialdin ◽  
Ahmad Shamsodini ◽  
Steven K. Wilson ◽  
Osama Abdeljaleel ◽  
Ibrahim Alnadhari ◽  
...  
Keyword(s):  
Case Report ◽  
Broad Spectrum ◽  
Surgical Intervention ◽  
Penile Prosthesis ◽  
Raw Milk ◽  
Prosthesis Infection ◽  
Case Presentation ◽  
First Case ◽  
Milk Ingestion

Abstract Background Infection after the penile prosthesis can be devastating to both the patient and surgeon with various complications and consequences. After introduction of antibiotic-coated implants, the rate of infection has dramatically decreased, but still we see uncommon organisms causing infection. We present a first case report of penile prosthesis infection by brucellosis due to raw milk ingestion. To our knowledge, this is the first reported case of brucellosis penile prosthesis infection. Case presentation We present a first case report of penile prosthesis infection by brucellosis due to raw milk ingestion. A 75-year-old, diabetic male patient presented with penile prosthesis infection 5 months post-penile exchange surgery due to mechanical malfunctioning of 2-piece penile prosthesis which was inserted 11 years ago. The initial treatment with broad spectrum antibiotics did not subside the infection. After diagnosis of brucellosis, the antibiotic was changed to anti-brucellosis (Rifampicin + Tetracycline). The patient improved dramatically and was discharged home with smooth follow-up course. Conclusion Brucellosis can cause infection of penile prosthesis and can be treated with anti-brucellosis antibiotics without necessitating surgical intervention and removal of prosthesis.

scholarly journals Neonate born with ischemic limb to a COVID-19 positive mother: management and review of literature

2020 ◽  
Vol 10 (1) ◽  
Author(s):  
Shahana Perveen ◽  
Karmaine A. Millington ◽  
Suchitra Acharya ◽  
Amit Grag ◽  
Vita Boyar
Keyword(s):  
Differential Diagnosis ◽  
Compartment Syndrome ◽  
Upper Extremity ◽  
Preterm Neonate ◽  
Review Of Literature ◽  
Case Presentation ◽  
Ischemic Limb ◽  
History Of ◽  
Hand Amputation ◽  
Work Up

AbstractObjectivesTo describe challenges in diagnosis and treatment of congenital neonatal gangrene lesions associated with history of maternal coronavirus disease 2019 (COVID-19) infection.Case presentationA preterm neonate was born with upper extremity necrotic lesions and a history of active maternal COVID-19 infection. The etiology of his injury was challenging to deduce, despite extensive hypercoagulability work-up and biopsy of the lesion. Management, including partial forearm salvage and hand amputation is described.ConclusionsNeonatal gangrene has various etiologies, including compartment syndrome and intrauterine thromboembolic phenomena. Maternal COVID-19 can cause intrauterine thrombotic events and need to be considered in a differential diagnosis.

scholarly journals A Case of COVID-19-Related Thrombocytopenia and Leukopenia in an Adolescent with Mild Symptoms

Children ◽  
2021 ◽  
Vol 8 (6) ◽  
pp. 509
Author(s):  
Lydia Kossiva ◽  
Athanasios Thirios ◽  
Eleni Panagouli ◽  
Alexandros Panos ◽  
Stavroula Lampidi ◽  
...  
Keyword(s):  
Bone Marrow ◽  
Viral Infection ◽  
Nasal Congestion ◽  
Pediatric Population ◽  
Clinical Manifestations ◽  
Bone Marrow Aspiration ◽  
White Blood Count ◽  
Chronic Obstructive ◽  
First Case ◽  
The Impact

Since the beginning of the COVID-19 pandemic, there have been numerous reports and reviews on the complications caused by the disease, analyzing the acute and chronic consequences. The main symptoms of SARS-CoV-2 are dry cough, fever, and fatigue. COVID-19 appears to affect all systems, including renal, cardiovascular, circulatory, and respiratory systems, causing chronic obstructive pulmonary disease. We report on a 14-year-old male adolescent, who presented with thrombocytopenia (platelet count 92 × 109 /L) and leukopenia (white blood count 4.2 × 103 /μL) that was observed two months ago. Ten days before the first blood test, a viral infection with nasal congestion and runny nose was reported, without other accompanying symptoms. Viral antibodies screening revealed positivity for all the three specific COVID-19 antibodies. Further haematological evaluation with bone marrow aspiration revealed non-specific dysplastic features of the red cell and megakaryocyte progenitors. Although haematological alterations due to COVID-19 infection are available from adult patients’ reports, the effect of COVID-19 infection in the pediatric population is underestimated and this is the first case with such haematological involvement. Noteworthy, in the current case, the impact of the COVID-19 infection was not related to the severity of the disease, as the symptoms were mild. In similar cases, bone marrow aspiration would not be performed as a part of routine work-up. Thus, it is important when evaluating pediatric patients with COVID-19 infection to search and report those alterations in order to better understand the impact and the spectrum of clinical manifestations of the specific viral infection in children and adolescents.

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