OpenMed/multicare-articles huggingface dataset:
Successful treatment of pediatric IgG4 related systemic disease with mycophenolate mofetil: case report and a review of the pediatric autoimmune pancreatitis literature
Authors: Melissa Mannion, Randy Q Cron
Journal: Pediatr Rheumatol Online J (2011)

Autoimmune pancreatitis is frequently associated with elevated serum and tissue IgG4 levels in the adult population, but there are few reports of pediatric autoimmune pancreatitis, and even fewer reports of IgG4 related systemic disease in a pediatric population. The standard of care treatment in adults is systemic corticosteroids with resolution of symptoms in most cases; however, multiple courses of corticosteroids are occasionally required and some patients require long term corticosteroids. In these instances, steroid sparing disease modify treatments are in demand. We describe a 13-year-old girl with IgG4 related systemic disease who presented with chronic recurrent autoimmune pancreatitis resulting in surgical intervention for obstructive hyperbilirubinemia and chronic corticosteroid treatment. In addition, she developed fibrosing medianstinitis as part of her IgG4 related systemic disease. She was eventually successfully treated with mycophenolate mofetil allowing for discontinuation of corticosteroids. This is the first reported use of mycophenolate mofetil for IgG4 related pancreatitis. Although autoimmune pancreatitis as part of IgG4 related systemic disease is rarely reported in pediatrics, autoimmune pancreatitis is also characterized as idiopathic fibrosing pancreatitis. All pediatric autoimmune pancreatitis cases reported in the world medical literature were identified via a PUBMED search and are reviewed herein. Twelve reports of pediatric autoimmune pancreatitis were identified, most of which were treated with corticosteroids or surgical approaches. Most case reports failed to report IgG4 levels, so it remains unclear how commonly IgG4 related autoimmune pancreatitis occurs during childhood. Increased evaluation of IgG4 levels in patients with autoimmune pancreatitis may shed further light on the association of IgG4 with pancreatitis and the underlying pathophysiology.
A peculiar case of syphilis infection: The great imitator is on the rise
Authors: Mjp Eijmael, Rg de Bruin, V Hira, T Koster
Journal: IDCases (2022)
Keywords: ocular syphilis, tertiary syphilis, uveitis
MeSH terms: Case Reports

The incidence of syphilis is on the rise worldwide and can pose many diagnostic and therapeutic difficulties for doctors. Given the wide variety of presenting symptoms, syphilis is also known as the great imitator, which in turn frequently leads to a pronounced diagnostic- and therapeutic delay for patients. Here we present a case report of syphilitic uveitis and papillitis accompanied by acute vision loss, a rare presentation of a tertiary syphilis infection that clinically mimicked a giant cell arteritis (GCA) or arteritic anterior ischemic optic neuropathy (AAION). The patient was treated with high-dose intravenous benzyl penicillin after which full vision was restored. By presenting this case, we hope to raise awareness for the increasing incidence of syphilis infections and stress the importance of syphilis testing in patients with otherwise unexplained uveitis.
Case Report: Acute Bulbar Palsy Plus Syndrome: A Guillain-Barré Syndrome Variant More Prone to Be a Subtype Than Overlap of Distinct Subtypes
Authors: Qian Cao, Hong Chu, Xiujuan Fu, Jiajia Yao, Zheman Xiao, Zuneng Lu
Journal: Front Neurol (2020)
Keywords: guillain-barré syndrome, miller fisher syndrome, acute bulbar palsy plus syndrome, characteristics, classification
MeSH terms: Case Reports

Objective: Acute bulbar palsy plus (ABPp) syndrome is a rare regional variant of Guillain-Barré syndrome (GBS) characterized by acute bulbar palsy combined with other cranial symptoms or ataxia without limb and neck weakness. We aim to investigate characteristics of ABPp syndrome and analyze its nosological position within the GBS spectrum. Methods: A patient with ABPp syndrome was reported, and previous case reports of patients who met the criteria for ABPp syndrome from the literature were reviewed. Results: A total of 28 patients were included in our study. Median age was 32 years. Most of the patients (78.6%) were from Asia, and 75.0% had preceding infection. The main accompanying symptoms were ophthalmoplegia (85.7%), facial palsy (60.7%), and ataxia (50.0%). There existed asymmetric weakness in the form of unilateral facial palsy (32.1%) and ptosis (3.6%). Approximately half of the patients had albuminocytological dissociation. All the tested patients were seropositive for antiganglioside antibodies, of which the two most common were immunoglobulin G (IgG) anti-GT1a (77.3%) and anti-GQ1b (59.1%) antibodies. Over one-third of the patients who underwent electrophysiological assessment showed subclinical neuropathy beyond cranial nerves. The outcome was generally favorable as 89.3% of patients made full recovery within 5 months. Conclusion: The hitherto largest case series of ABPp syndrome advances our understanding of this disease. Serologically, the presence of IgG anti-GT1a and anti-GQ1b antibodies predicts and contributes to the disease. Phenotypically, ABPp syndrome is more prone to be a separate subtype of GBS than overlap of distinct subtypes and has the potential to complement current diagnostic framework of GBS.
Management of Sjogren's Syndrome Patient: A Case Report of Prosthetic Rehabilitation with 6-Year Follow-Up
Authors: Marcos de Mendonça Invernici, Amanda Finger Stadler, Gastão Vale Nicolau, Maria Ângela Naval Machado, Antônio Adilson Soares de Lima, Marilia Compagnoni Martins
Journal: Case Rep Dent (2014)

Completely and partially edentulous patients with Sjogren's syndrome (SS) experience severe hyposalivation, xerostomia, and considerable difficulty in using tissue-supported prosthesis. This clinical paper describes the management, treatment, and 6-year follow-up of a patient diagnosed with SS type II, who uses corticosteroids and antihyperglycemic drugs. The patient received restorative, periodontal, and surgical treatments followed by implant-supported fixed prosthesis. Radiographic evaluation and probing depth showed gingival health and no bone loss after 6 years. Treatment with implant-retained dental prosthesis greatly increased comfort and function, offering an alternative to patients with SS.
Cyanoacrylate-induced pseudo-tarsorrhaphy of the eye
Authors: Upender Wali, Rana Al-Senawi, Abdullah Al-Mujaini
Journal: Oman J Ophthalmol (2009)
Keywords: abrasion, cyanoacrylate, pseudo-tarsor-rhaphy
MeSH terms: Case Reports

Cyanoacrylate, also known as superglue, is a polymer which forms a strong bond at room temperature with a variety of materials including metal, glass, plastic, rubber, skin, mucous membranes and other epithelial tissues as well. We hereby present a 22-year-old female who had an accidental instillation of the glue in her eye, with an uneventful outcome.
Giant solitary synovial osteochondromatosis of the elbow causing ulnar nerve neuropathy: a case report and review of literature
Authors: Munnan Al-Najjim, Abubakar Mustafa, Carl Fenton, Syam Morapudi, Mohammad Waseem
Journal: J Brachial Plex Peripher Nerve Inj (2013)
MeSH terms: Case Reports

Introduction:
Giant or solitary osteochondroma is part of a rare disorder known as synovial osteochondromatosis. It forms part of a spectrum of disease characterized by metaplastic changes within the joint synovium that are eventually extruded as loose bodies. It has been suggested that solitary synovial osteochondroma forms as progression of synovial osteochondromatosis through a process of either coalescence of multiple smaller bodies or the growth of a dominant synovial osteochondroma. Previous studies have shown that it occurs as a late phase of the disease. We report a rare case of giant synovial osteochondromatosis at the elbow causing ulnar nerve neuropathy and mechanical symptoms which has not been previously reported in the literature.
Case report:
We report a case of a 56 year old Western European gentleman who presented with ulnar nerve neuropathy and swelling behind the elbow. The patient underwent MR imaging and subsequent biopsy that demonstrated synovial osteochondromatosis. Initially the patient declined surgery and opted for a watch and wait approach. Five years later he returned with worsening symptoms and underwent successful surgical resection of a giant solitary synovial osteochondroma.
Conclusion:
The unique outcome in our patient despite the long interval between presentation and surgical treatment resulted in early full resolution of symptoms within a short period. It may suggest an improved prognosis as compared to multiple synovial osteochondromatosis in terms of mechanical and neurological outcomes.
Left Circumflex Artery Rupture with Left Atrial Tamponade and Functional Mitral Stenosis
Authors: Weiting Huang, Khaled Mohamed Emadeldin Moheb Hammad, Victor Tar Toong Chao, Khung Keong Yeo
Journal: Case Rep Cardiol (2018)
MeSH terms: Case Reports

The growth in percutaneous transluminal devices has enabled operators to tackle more complex, native, and post-bypass surgery anatomy. However, complications such as coronary artery dissection, coronary perforation, retrograde aortic dissection, arrhythmias, and acute coronary syndrome still occur with resulting mortality rates of up to 4.2% in complex interventions. Perforation of the circumflex artery is of particular interest in view of its position and relation to the surrounding cardiac structures. This is a site of potential fluid collection, and as the left atrium is fixed to the parietal pericardium at the entry of the pulmonary veins, fluid in the oblique sinus can accumulate enough pressure to compress the left atrium and the coronary sinus. We present a case of left circumflex artery perforation which demonstrates the physiologic complications of coronary sinus and left atrial compression and the resultant functional mitral stenosis.
When "looks" can be deceiving - Internuclear ophthalmoplegia after mild traumatic brain injury: Case report and literature review
Authors: Shaikh Hai, Adel Elkbuli, Kyle Kinslow, Mark McKenney, Dessy Boneva
Journal: Int J Surg Case Rep (2019)
Keywords: blunt trauma, exotropia, ino, wemino

Introduction:
Walled-eyed monocular internuclear ophthalmoplegia (WEMINO) syndrome is a sub-variant of internuclear ophthalmoplegia (INO) and involves the same clinical findings with the addition of exotropia of the ipsilateral eye. Causes typically include multiple sclerosis (MS) and ischemia (hemorrhagic or embolic) but can be secondary to blunt trauma as seen in our presented case.
Presentation of case:
A 27-year-old man presented with new-onset visual changes, diplopia, and strabismus following a motor vehicle collision. Physical exam showed left ocular exotropia and slight hypertropia on forward gaze with deficiency of left convergence and disconjugate eye movements on horizontal gaze with right nystagmus on rightward gaze. Imaging showed hyperintensities in the right middle cerebellar peduncle and left temporal-occipital white matter likely consistent with diffuse axonal injury but otherwise nonspecific. The patient was treated conservatively with left eyepatch and exhibited improvement of exotropia and diplopia at 1 week follow up.
Discussion:
Common causes of WEMINO syndrome include MS and ischemia with no prior reports, to our knowledge, being secondary to the blunt trauma seen in our case. Patients with WEMINO present with the typical signs of failure of ipsilateral adductive movement during lateral along with ipsilateral exotropia. Management involves treating the underlying disorder, if possible, with conservative measures with traumatic origins.
Conclusion:
Until now, WEMINO syndrome secondary to trauma has not been previously documented. Our patient was effectively treated with conservative measures alone.
Good Outcome for an Individual with Severe Facial Anomalies and Hypogonadotropic Hypogonadism: A Consequence of His Cognitive Function, Pragmatic Approach, and Temperament
Authors: Timothy L Groh, William Zarella, Peter A Lee
Journal: Case Rep Pediatr (2021)
MeSH terms: Case Reports

The multiple factors that determine outcomes for individuals with visible developmental errors and/or atypical development of the reproductive system are not fully understood. This case report of an individual with Bosma arhinia microphthalmia syndrome causing severe facial anomalies and hypogonadotropic hypogonadism is used to highlight factors that impacted his adjustment from childhood through adulthood. Key factors include his temperament, intact cognitive ability, and pragmatic approach for controlling his physical and social environment. His successful adjustment even in the face of significant early life challenges demonstrates that positive outcomes are attainable for individuals with significant developmental errors. His story and experiences with the health-care system offer insight into some factors that may be pertinent to resilience and lifelong adjustment for patients with similar conditions and the importance of continually seeking the patient's perspective to tailor treatment across the lifespan.
Single-incision laparoscopic splenectomy with innovative gastric traction suture
Authors: G Srikanth, M D Wasim, A Sajjad, Neel Shetty
Journal: J Minim Access Surg (2011)
Keywords: gastric traction suture, itp, single-incision laparoscopic splenectomy
MeSH terms: Case Reports

Laparoscopic splenectomy is now the gold standard for patients with idiopathic thrombocytopenic purpura (ITP) undergoing splenectomy. There are a few reports in literature on single-incision laparoscopic (SIL) splenectomy. Herein, we describe a patient undergoing SIL splenectomy for ITP without the use of a disposable port device. We report a 20-year-old female patient with steroid-refractory ITP having a platelet count of 14,000/cmm who underwent a SIL splenectomy. Dissection was facilitated by the use of a single articulating grasper and a gastric traction suture and splenic vessels were secured at the hilum with an endo-GIA stapler. She made an uneventful postoperative recovery and was discharged on the second postoperative day. She is doing well with no visible scar at 8-month follow-up.
Resection of ruptured hepatic teratoma in an adult
Authors: Jonathan Ramkumar, Andrew Best, Ananta Gurung, Anne-Marie Dufresne, George Melich, Elena Vikis, Shawn MacKenzie
Journal: Int J Surg Case Rep (2018)
Keywords: case report, general surgery, hepatic teratoma, hepatobiliary, surgery, teratoma

Introduction:
Extragonadal locations of teratomas are uncommonly reported in the literature. Teratomas are neoplasms usually found in the gonadal organs: ovaries and testis. The majority of teratomas are found in the pediatric age group. Furthermore, teratomas originating in the liver are exceedingly rare with only 11 case reports in adult populations.
Presentation of case:
We present a case of a 65 year-old female who presented to hospital with sudden onset abdominal pain from a centrally located ruptured hepatic teratoma on CT scan. The patient underwent urgent surgery. The diagnosis of cystic mature teratoma was confirmed on histopathology. Patient was discharged on post-operative day 5. At 12 week follow-up, no post-operative complications were identified.
Discussion:
Hepatic teratomas are a rarely encountered neoplasm, especially in the adult population. Our case report is unique, as it represents the only clinical presentation of mass rupture in an adult liver teratoma. CT scan identified a well circumscribed mass containing adipose tissue, fluid, and calcifications characteristic of teratoma. Complete surgical resection is mainstay treatment. A definitive diagnosis of a mature teratoma is confirmed by histopathological findings.
Conclusion:
Presented is a rare case of ruptured hepatic teratoma in an adult who underwent surgical resection.
Diabetic neuropathic cachexia: a case report
Authors: Deeb D Naccache, William B Nseir, Moshe Z Herskovitz, Mogher H Khamaisi
Journal: J Med Case Rep (2014)

Introduction:
We report a case of the rare entity of diabetic neuropathic cachexia, in order to remind clinicians that these cases still exist.
Case presentation:
A 71-year-old Moslem Arab man with type 2 diabetes along with diabetic neuropathic cachexia complicated by a hyperfunctioning autonomous thyroid nodule, and undiagnosed acromegaly came under our care. We report the unique challenges as to what are the priorities to consider in the course of investigation and treatment. This case emphasizes the fast recovery from this remediable disorder, with antineuropathic medication and exogenous insulin to serve as an anabolic hormone on top of its hypoglycemic effect. Shared pathophysiologic aspects of diabetic neuropathic cachexia, cancerous etiologies and acute phase response are discussed.
Conclusions:
Diabetic neuropathic cachexia is an integral differential diagnosis, whenever an intense neuropathic pain dominates patient complaints, accompanied with anorexia, weight loss as well as mood and sleep disturbances. This is an original case report of interest to internists, endocrinologists, diabetologists and pain clinic practitioners. Raising the suspicion of diabetic neuropathic cachexia early and concomitant to weight loss investigation, might curtail suffering and prompt early recovery from a severe illness that has a good prognosis.
Multiple Cranial Nerve Palsy Concomitant with Leptomeningeal Involvement in Multiple Myeloma: A Case Report and Review of Literature
Authors: Neda Pak, Fatemeh Shakki Katouli, Amir Reza Radmard, Mohammad Hassan Shaki Katuli, Mohamad Mehdi Rezwanifar, Negar Sepehri Boroujeni
Journal: Int J Hematol Oncol Stem Cell Res (2018)
Keywords: diagnosis, multiple myeloma, neuroimaging
MeSH terms: Case Reports

Neurologic symptoms are quite common in multiple myeloma, but direct invasion of central nervous system is extremely rare. Leptomeningeal multiple myeloma, as a rare neurological manifestation of multiple myeloma, presents with impairment of consciousness, cranial nerve palsies and convulsions. Here, we describe a 52-year- old male patient, known case of multiple myeloma, who presented with bilateral visual loss and difficulty in swallowing.
Myxedema Coma Complicated by Pancytopenia
Authors: Charles T Mupamombe, Felix M Reyes, Derek B Laskar, Joseph Gorga
Journal: Case Rep Med (2019)
MeSH terms: Case Reports

Hypothyroidism is common, with an extreme manifestation of myxedema coma if untreated. Hematologic consequences of myxedema coma include mild leukopenia and anemia, rarely pancytopenia. We present a patient with typical symptoms of myxedema coma, but found to be pancytopenic, with sustained response to levothyroxine and blood transfusion for anemia.
Multiorgan WU Polyomavirus Infection in Bone Marrow Transplant Recipient
Authors: Erica A Siebrasse, Nang L Nguyen, Melisa J Willby, Dean D Erdman, Marilyn A Menegus, David Wang
Journal: Emerg Infect Dis (2016)
Keywords: wu polyomavirus, epithelial cells, immunosuppression, transplant, tropism, viruses
MeSH terms: Lung / virology, Polyomavirus / isolation & purification, Polyomavirus Infections / diagnosis, Respiratory Tract Infections / virology, Case Reports, Research Support, N.I.H., Extramural, Research Support, Non-U.S. Gov't, Research Support, U.S. Gov't, Non-P.H.S., Bone Marrow Transplantation, Child, Preschool, Female, Humans, Transplant Recipients

WU polyomavirus (WUPyV) was detected in a bone marrow transplant recipient with severe acute respiratory distress syndrome who died in 2001. Crystalline lattices of polyomavirus-like particles were observed in the patient's lung by electron microscopy. WUPyV was detected in the lung and other tissues by real-time quantitative PCR and identified in the lung and trachea by immunohistochemistry. A subset of WUPyV-positive cells in the lung had morphologic features of macrophages. Although the role of WUPyV as a human pathogen remains unclear, these results clearly demonstrate evidence for infection of respiratory tract tissues in this patient.
Cannabis Vaping-Induced Acute Pulmonary Toxicity: Case Series and Review of Literature
Authors: Sreedhar Adapa, Vijay Gayam, Venu Madhav Konala, Srinadh Annangi, Mina P Raju, Vishnu Bezwada, Christine McMillan, Hussain Dalal, Amrendra Mandal, Srikanth Naramala
Journal: J Investig Med High Impact Case Rep (2020)
Keywords: acute respiratory distress syndrome, acute respiratory failure, cannabidiol, cannabinoid hyperemesis syndrome, cannabis, terpenes, tetrahydrocannabinol
MeSH terms: Acute Lung Injury / etiology, Cannabis / adverse effects, Plant Oils / adverse effects, Vaping / adverse effects, Case Reports, Review, Adult, Electronic Nicotine Delivery Systems, Female, Humans, Inhalation, Male, Tomography, X-Ray Computed, Young Adult, Plant Oils

The use of cannabis for recreational as well as medicinal use is on the rise recently with more states legalizing it. We conducted a review analysis of the literature published on acute respiratory failure from vaping cannabis oil. We have also summarized the clinical details (age, length of stay, mode of ventilation, common clinical findings, and steroid use) along with common laboratory abnormalities. This article aims to educate health care providers on the clinical manifestations and management strategies for vaping-induced acute respiratory failure. We also discussed the different available formulations of cannabis oil and key ingredients responsible for the vaping-associated lung injury.
Case Report: Localized bullous pemphigoid induced by local triggers: a case series and a proposal for diagnostic criteria based on a literature review
Authors: Lluís Corbella-Bagot, Javier Gil-Lianes, Javier Fernández-Vela, Ignasi Martí-Martí, Marta Alegre-Fernández, Irene Fuertes, Patricia Garbayo-Salmons, Xavier Bosch-Amate, Antonio Guilabert, José M Mascaró Jr
Journal: Front Immunol (2023)
Keywords: bullous pemphigoid, case report, criteria, localized bullous pemphigoid, precipitating factors, radiotherapy, rosacea, triggers
MeSH terms: Pemphigoid, Bullous / diagnosis, Pemphigoid, Bullous / etiology, Skin Diseases, Vesiculobullous, Review, Case Reports, Antibodies, Female, Humans, Multicenter Studies as Topic, Precipitating Factors, Research, Antibodies

Introduction:
Localized bullous pemphigoid (LBP) is an infrequent bullous pemphigoid (BP) variant restricted to a body region. According to the most compelling evidence, LBP occurs in patients with pre-existent serum antibodies against the basement membrane zone, which occasionally acquire the capacity to induce disease after the influence of different local factors acting as triggers.
Methods:
We hereby present a multicenter cohort of 7 patients with LBP developed after local triggers: radiotherapy, thermal burns, surgery, rosacea, edema and a paretic leg. In addition, we conducted a review of the literature, and we propose a set of diagnostic criteria for LBP, also based on our case series and the 2022 BP guidelines from the European Academy of Dermatology and Venereology.
Results:
During follow-up, three of the patients from our series evolved to a generalized BP, with only one requiring hospitalization. Our literature search retrieved 47 articles including a total of 108 patients with LBP, with a 63% with a potential local precipitating factor previous to their diagnosis. LBP mostly affected older females, and a subsequent generalized progression occurred in 16.7% of the cases. The most frequently involved areas were the lower limbs. Radiation therapy and surgery were responsible for the inducement of nearly 2 in 3 cases of LBP. We observed a significantly higher risk of generalization in cases where the trigger led to the developing of LBP earlier (p=0.016). Our statistical analysis did not detect any other prognosis factor for generalization when assessing direct immunofluorescence, histological and serological results, or other patient related factors.
Conclusion:
LBP should be suspected in patients with recurrent localized bullous eruptions. The presence of a trauma history in the same anatomic area is reported in most cases.