OpenMed/multicare-articles huggingface dataset:
Prolonged Dyspnea after Interscalene Block: Attributed to Undiagnosed Addison's Disease and Myasthenia Gravis
Authors: John G Skedros, Casey J Kiser, Shaun D Mendenhall
Journal: Case Rep Med (2011)
MeSH terms: Case Reports

This report describes a patient who had a series of daily interscalene nerve blocks to treat pain following a shoulder manipulation for postsurgical stiffness. She experienced acute respiratory compromise that persisted for many weeks. All typical and unusual causes of these symptoms were ruled out. Her treating pulmonologist theorized that the ipsilateral carotid body had been injured. However, it was subsequently determined that the constellation of symptoms and their prolonged duration were best explained by a poor stress response from Addison's disease coupled with exacerbation of early onset myasthenia gravis. This patient's case is not a typical reaction to interscalene nerve blocks, and thus preoperative testing would not be recommended for myasthenia gravis or Addison's disease without underlying suspicion. We describe this report to inform physicians to consider a workup for these diagnoses if a typical workup rules out all usual causes of complications from an interscalene block.
Appearances Are Deceptive: Two Case Reports in Teenagers on the Conservative Laparoscopic Surgery for Adnexal Torsion with Apparent Infarction
Authors: A Jayawardane, M Patabendige, C Gunathilaka
Journal: Case Rep Obstet Gynecol (2022)
MeSH terms: Case Reports

Introduction. Torsion of the ovary, tube, or both is estimated to be responsible for 2-7% of all gynaecological emergencies. Oophorectomy is commonly performed for adnexal torsion with a possible negative impact on fertility in women of reproductive age. Case Presentations. We report two cases of teenage girls presented with adnexal torsion describing their laparoscopy features. Detorsion without additional surgical intervention could save their ovaries. Discussion. Detorsion is a more conservative surgical approach that should be considered in all younger women with ovarian torsion.
Cheilitis glandularis: Case report with respect to immunohistochemical findings
Authors: Reinhard E Friedrich, Thomas Löning
Journal: GMS Interdiscip Plast Reconstr Surg DGPW (2016)
Keywords: igg4-related disease, cheilitis glandularis, lip surgery, minor salivary gland
MeSH terms: Case Reports

Cheilitis glandularis (CG) is a rare benign affection of the lip mucosa. The etiology and pathogenesis of CG are unknown. Surgical measures are the leading therapeutic options to treat CG. This case report on a 55-year-old female illustrates the recurrent affection of the lips over several years and local therapy. Furthermore, the study of the resected glands intends to differentiate the lesions with respect to the recently introduced concept of immunoglobulin G4-related diseases.
Case Report: Successful Management of a Refractory Plasmablastic Lymphoma Patient With Tislelizumab and Lenalidomide
Authors: Lili Cheng, Qi Song, Mengke Liu, Yan Wang, Hongmei Yi, Ying Qian, Pengpeng Xu, Shu Cheng, Chaofu Wang, Li Wang, Weili Zhao
Journal: Front Immunol (2021)
Keywords: epstein-barr virus, immune checkpoint inhibitor, lenalidomide, plasmablastic lymphoma, tislelizumab
MeSH terms: Antibodies, Monoclonal, Humanized / administration & dosage, Antineoplastic Combined Chemotherapy Protocols / therapeutic use, Lenalidomide / administration & dosage, Plasmablastic Lymphoma / drug therapy, Case Reports, Research Support, Non-U.S. Gov't, Aged, Female, Humans, Antibodies, Monoclonal, Humanized, tislelizumab, Lenalidomide

Plasmablastic lymphoma (PBL) is a rare and aggressive hematological malignancy. PBL commonly occurs in immune incompetent patients, such as those with human immunodeficiency virus (HIV), post-transplant status, or immunosenescence. Given its rarity, there is no specific standard treatment for PBL. However, small case series have shown that intensive chemotherapies combined with anti-myeloma agents such as bortezomib and lenalidomide were effective in treating PBL. Unfortunately, some fragile patients could not tolerate intensive chemotherapeutic regimens, especially the elderly patients. Here we presented a 76-year-old female PBL patient refractory to miniCHOP regimen combined with bortezomib but achieved complete remission when treated with tislelizumab combined with lenalidomide, indicating that immune therapy may be a potential treatment for PBL. To our knowledge, this is the first chemoresistant PBL patient that has been successfully treated with checkpoint inhibitor plus lenalidomide, thus providing new insight towards PBL management.
Coronavirus Disease 2019 (COVID-19) Associated Hemolytic Uremic Syndrome in a Toddler
Authors: Kristie Searcy, Apoorva Jagadish, Patricia Pichilingue-Reto, Radhakrishna Baliga
Journal: Case Rep Pediatr (2022)
MeSH terms: Case Reports

Coronavirus disease 2019 (COVID-19) is a heterogenous, predominantly pulmonary disease caused by severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) that has resulted in catastrophic illness around the world. Thrombotic microangiopathy (TMA) is a triad of hemolytic anemia, thrombocytopenia, and end organ damage. This is present in severe cases of COVID-19 and in hemolytic uremic syndrome (HUS) commonly caused by Escherichia coli (E.coli) 0157:H7. We report a novel case of a toddler who presented with classic features suggestive of HUS characterized by bloody diarrhea followed by thrombocytopenia, hemolytic anemia, and acute kidney injury, in whom a polymerase chain reaction (PCR) test for SARS-CoV-2 was positive.
Treatment of Light Chain Deposition Disease Using Bortezomib-Based Regimen Followed by Thalidomide-Based Regimen in a Saudi Male
Authors: Bappa Adamu, Mushabab Al-Ghamdi, Mustafa Ahmad, Khaled O Alsaad
Journal: Case Rep Nephrol (2016)
MeSH terms: Case Reports

Light chain deposition disease (LCDD) is a rare illness with, as yet, no clear evidence-based guidelines for its treatment. To the best of our knowledge, LCDD has not been previously reported from Saudi Arabia. We present in this report, a 38-year-old Saudi male who presented with clinical features suggestive of hypertensive nephropathy but kidney biopsy later revealed the diagnosis of LCDD. His serum creatinine at presentation was 297 μmol/L which came down to 194 μmol/L on treatment with Bortezomib, Cyclophosphamide and Dexamethasone. His 24-hour protein excretion at presentation was 6 g/L which also came down to less than 1 g/day. He was later placed on Cyclophosphamide, Thalidomide, and Dexamethasone regimen because of persistent high titres of serum free light chains. He went into remission with undetectable serum free light chains and remained so for three years at the time of writing this report. We conclude that LCDD, though rare, does occur in Saudi population. The treatment of LCDD is challenging but the use of Bortezomib, a proteosome inhibitor, is promising. However, suboptimal response may require further treatment with other therapeutic options such as chemotherapy with alkylating agents or high-dose Melphalan with autologous stem cell transplant.
Air embolism after endoscopic retrograde cholangiopancreatography in a patient with budd Chiari syndrome
Authors: Beatriz Wills-Sanin, Yenny R Cárdenas, Lucas Polanco, Oscar Rivero, Sebastian Suarez, Andrés F Buitrago
Journal: Case Rep Crit Care (2014)

Endoscopic retrograde cholangiopancreatography is a procedure commonly used for the diagnosis and treatment of various pancreatic and biliary diseases. Air embolism is a rare complication, which may be associated with this procedure. This condition can be manifested as cardiopulmonary instability and/or neurological symptoms. Known risk factors include: sphincterotomy; application of air with high intramural pressure; anatomic abnormalities; and chronic hepatobiliary inflammation. It is important for the health-care staff, including anesthesiologists, interventional gastroenterologists, and critical care specialists, amongst others, to promptly recognize air embolism and to initiate therapy in a timely fashion, thus preventing potentially fatal outcomes. We submit a brief review of the literature and a case report of air embolism which occurred in the immediate postoperative stage of an endoscopic retrograde cholangiopancreatography, performed in a woman with a history of liver transplantation due to Budd Chiari syndrome and biliary stricture.
Enthesitis as a component of dactylitis in psoriatic juvenile idiopathic arthritis: histology of an established clinical entity
Authors: Katherine S L Tuttle, Sara O Vargas, Michael J Callahan, Donald S Bae, Peter A Nigrovic
Journal: Pediatr Rheumatol Online J (2015)
Keywords: dactylitis, enthesitis, enthesitis related arthritis, juvenile idiopathic arthritis, psoriatic arthritis, psoriatic juvenile idiopathic arthritis
MeSH terms: Arthritis, Juvenile / pathology, Arthritis, Juvenile / physiopathology, Rheumatic Diseases / pathology, Rheumatic Diseases / physiopathology, Case Reports, Research Support, Non-U.S. Gov't, Adolescent, Antirheumatic Agents / therapeutic use, Arthritis, Juvenile / drug therapy, Cartilage Diseases / pathology, Etanercept, Extracellular Matrix / pathology, Female, Finger Joint / pathology, Finger Joint / physiopathology, Humans, Immunoglobulin G / therapeutic use, Methotrexate / therapeutic use, Receptors, Tumor Necrosis Factor / therapeutic use, Rheumatic Diseases / drug therapy, T-Lymphocytes / pathology, Toe Joint / pathology, Toe Joint / physiopathology, Treatment Outcome, Antirheumatic Agents, Immunoglobulin G, Receptors, Tumor Necrosis Factor, Etanercept, Methotrexate

Context:
Imaging of dactylitis in adult psoriatic arthritis suggests a pathophysiological role for enthesitis. However, histological definition of the dactylitic digit is unavailable.
Objective:
We evaluated the role of enthesitis in dactylitis associated with psoriatic juvenile idiopathic arthritis (psJIA) in a child who underwent detailed imaging and histologic evaluation.
Design:
Radiographs, ultrasound and high-resolution magnetic resonance imaging were employed to define the locus of disease in two dactylitic digits from a 14-year-old girl with psJIA. Biopsies were stained with hematoxylin and eosin, CD3, CD20, CD4, CD8 and CD117 and examined via light microscopy.
Results:
Radiologic features of dactylitis included enhanced signal at digital entheses without accompanying synovitis or tenosynovitis. Histologically, finger and toe tissue exhibited hypervascular tenosynovium with a fibromyxoid expansion of fibrous tissue. This was accompanied by sparse to moderate perivascular lymphocytic inflammation consisting predominantly of T cells, with occasional admixed B cells and mast cells. Neutrophils and plasma cells were absent. Fibrocartilage exhibited reactive features including increased extracellular myxoid matrix, binucleation, and focal necrosis, without cellular inflammation. Fibrinous synovitis and mild reactive synoviocyte hyperplasia were seen.
Conclusions:
Dactylitis in psJIA bears a radiographic resemblance to the condition in adult psoriatic arthritis. Histologic hallmarks include an expanded mast cell-populated extracellular myxoid matrix, cartilage degeneration, and a T cell-rich perivascular inflammatory infiltrate. These findings help to define enthesitis as a clinicopathologic entity.
Asymptomatic nonsyndromic multiple supernumerary premolars
Authors: Seema Gupta, Mridula Goswami
Journal: Int J Clin Pediatr Dent (2012)
Keywords: impacted, nonsyndromic, supernumerary premolars
MeSH terms: Case Reports

The apparently morphologically normal finding of multiple supernumerary teeth in absence of an associated systemic condition or syndrome is an uncommon phenomenon. Surgical removal of supernumerary teeth is indicated if eruption of the adjacent teeth has been delayed; altered eruption, displacement of adjacent teeth is evident or pathologies such as cystic lesion and resorption of adjacent tooth have occurred. If the risks of surgery outweigh the benefits of removal, the teeth may be left in situ and a regular radiographic monitoring should be made. How to cite this article: Gupta S, Goswami M. Asymptomatic Nonsyndromic Multiple Supernumerary Premolars. Int J Clin Pediatr Dent 2012;5(1):84-86.
Motor cortex neuroplasticity following brachial plexus transfer
Authors: Stefan Dimou, Michael Biggs, Michael Tonkin, Ian B Hickie, Jim Lagopoulos
Journal: Front Hum Neurosci (2013)
Keywords: mri, brachial plexus transfer, motor cortex, neuroimaging, phantom limb

In the past decade, research has demonstrated that cortical plasticity, once thought only to exist in the early stages of life, does indeed continue on into adulthood. Brain plasticity is now acknowledged as a core principle of brain function and describes the ability of the central nervous system to adapt and modify its structural organization and function as an adaptive response to functional demand. In this clinical case study we describe how we used neuroimaging techniques to observe the functional topographical expansion of a patch of cortex along the sensorimotor cortex of a 27-year-old woman following brachial plexus transfer surgery to re-innervate her left arm. We found bilateral activations present in the thalamus, caudate, insula as well as across the sensorimotor cortex during an elbow flex motor task. In contrast we found less activity in the sensorimotor cortex for a finger tap motor task in addition to activations lateralized to the left inferior frontal gyrus and thalamus and bilaterally for the insula. From a pain perspective the patient who had experienced extensive phantom limb pain (PLP) before surgery found these sensations were markedly reduced following transfer of the right brachial plexus to the intact left arm. Within the context of this clinical case the results suggest that functional improvements in limb mobility are associated with increased activation in the sensorimotor cortex as well as reduced PLP.
Endoscopic pyloroplasty for severe gastric outlet obstruction due to alkali ingestion in a child
Authors: Seyed Mohsen Dehghani, Mitra Aldaghi, Hazhir Javaherizadeh
Journal: Gastroenterol Hepatol Bed Bench (2016)
Keywords: endoscopic pyloroplasty, gastric injury, obstruction
MeSH terms: Case Reports

A common belief is that alkali ingestion causes severe esophageal damage and limited gastric injury due to the buffering action of acid. Gastric injury has been observed in patients who ingested alkali. Gastric outlet obstruction (GOO) secondary to caustic ingestion occurs due to fibrosis after resolution of the acute injury and inflammation, most commonly 6 to 12 weeks after initial ingestion. The traditional treatment for GOO related to ingestion of corrosive agents is surgery. Experience with endoscopic balloon dilation of corrosive-induced GOO is limited in children. This is the first report of endoscopic pyloroplasty in a child with GOO due to caustic alkalis ingestion that was treated with balloon dilation (using TTS balloon ranging from 6-15 mm) in Iran. Four dilation sessions were required for symptomatic relief of dysphagia. After one year of follow up, weight gain was normal.
Case report: Young-onset large vessel ischemic stroke due to hyperhomocysteinemia associated with the C677T polymorphism on 5,10-methylenetetrahydrofolate reductase and multi-vitamin deficiency
Authors: Jiro Fukae, Hiroto Eguchi, Yoichi Wada, Atsuhito Fuse, Rika Chishima, Mitsuyoshi Nakatani, Asuka Nakajima, Nobutaka Hattori, Yasushi Shimo
Journal: Front Neurol (2023)
Keywords: 10-methylenetetrahydrofolate reductase, 5, antiepileptic drug, cerebral infarction, epilepsy, folic acid, hyperhomocysteinemia, vitamin b12
MeSH terms: Case Reports

Hyperhomocysteinemia is an important risk factor for cerebral infarction. Herein, we report on a 30-year-old man previously diagnosed with epilepsy who presented with right hemiplegia and total aphasia. Magnetic resonance imaging showed a fronto-temporal ischemic lesion due to occlusion of the left middle cerebral artery. Clinical testing and imaging demonstrated that he had hyperhomocysteinemia induced by multiple factors including the C677T polymorphism on 5.10-methylenetetrahydrofolate reductase (MTHFR), and multiple vitamin deficiencies. The C677T polymorphism on MTHFR is closely related to hyperhomocysteinemia and folate deficiency in epileptic patients who are taking multiple anti-convulsants. Given hyperhomocysteinemia can independently cause stroke at a young age, physicians should periodically examine plasma homocysteine and serum folic acid levels in epileptic patients who are on long-term regimens of multiple anti-epileptic drugs.
Hypertrophic scar of the conjunctiva presenting as an eyelid mass: an unusual complication after surgical treatment of a chalazion
Authors: Jun Hyuk Son, Joon Hyuk Choi, Su-Ho Lim
Journal: Springerplus (2016)
Keywords: chalazion, eyelid mass, hypertrophic scar

Purpose:
To present a rare case of a conjunctival hypertrophic scar presenting as eyelid mass after surgical treatment of a chalazion.
Case presentation:
A 74-year-old man visited our clinic with severe ocular discomfort and excessive lacrimation since several months in his left eye accompanied by itching of the upper eyelid. Examination of the anterior segment revealed a 6 mm (horizontal) × 8 mm (vertical) round, immobile, hard, mushroom-shaped protruding mass on the tarsal conjunctiva of his left eye. There was no associated pigmentation, ulceration, or tenderness. Excisional biopsy of the benign conjunctival tumor was performed using radiofrequency electrosurgical systems. The region of the excised conjunctiva was well-healed on postoperative day 14, and there was no recurrence until 1 year post-surgery. Histopathological examination demonstrated thick interlacing collagenous fibrous bundles oriented in random directions and fibroblastic proliferation. Immunohistochemical staining revealed that spindle-shaped fibroblasts were positive for CD34 and negative for smooth muscle actin. The excessive collagenous tissue was stained blue by Masson trichrome stain. These findings were consistent with a hypertrophic scar of the conjunctiva.
Conclusion:
This short report demonstrates that a hypertrophic scar of the conjunctiva can develop after surgery of a chalazion and cause severe ocular discomfort and excessive lacrimation. These lesions can be easily removed using simple excision.
Spontaneous tumor lysis syndrome in the setting of B-cell lymphoma
Authors: Mateusz Opyrchal, Travis Figanbaum, Amit Ghosh, Vincent Rajkumar, Sean Caples
Journal: Case Rep Med (2010)
MeSH terms: Case Reports

Tumor lysis syndrome (TLS) presenting in absence of chemotherapy is a rare occurrence. One of the true oncological emergencies, it can lead to significant morbidity and mortality. TLS is a phenomena usually associated with tumor cell death after treatment. The etiology of the spontaneous TLS is not well understood, which complicates the diagnosis. TLS is well known to oncologists but physicians outside of this specialty have little or no experience with this condition. Early recognition and treatment are the keys to limiting the sequela of the condition. Spontaneous tumor lysis syndrome is rare but presents added risks to the patient because of the potential for delayed diagnosis and no benefit of pretreatment. Diagnosis may be further delayed because this may be the first symptom of underlying malignancy. Therefore, it is imperative that all clinicians are familiar with the syndrome to assure timely recognition.
Clinical and molecular characterization of a de novo 19p13.3 microdeletion
Authors: Pietro Palumbo, Orazio Palumbo, Maria Pia Leone, Raffaella Stallone, Teresa Palladino, Leopoldo Zelante, Massimo Carella
Journal: Mol Cytogenet (2016)
Keywords: 19p133 microdeletion, intellectual disabilities, snp-array analysis
MeSH terms: Case Reports

Background:
Structural rearrangements of chromosome 19p13.3 are a rare condition, and their phenotypic consequences remain not well defined, because of the variability of clinical manifestations. Increasing knowledge of new 19p13.3 microdeletion is useful to clarify the phenotypic variability observed in some patients. In a small number of recent papers, patients with intellectual disabilities, multiple congenital anomalies and microdeletion of the chromosome band 19p13.3 have been described. However, little is known about genes responsible for clinical features in patients carriers of 19p13.3 microdeletion; thus, increasing number of reported cases will be helpful to investigate the contribution of candidate genes, providing bases for future investigations.
Case presentation:
Here, we report on a 10-years-old girl referred to our genetics clinic due to intellectual disability, attention deficit, behavioral and speech delay, hypotonia, facial dysmorphisms, eye anomalies and congenital malformations. Using an high resolution SNP array, we identified a de novo microdeletion of chromosome 19p13.3, resulting in the heterozygous loss of 27 RefSeq genes and a miRNA, partially overlapping with three others deletions already reported in literature, but extending downstream (centromeric) for additional 386 Kb. This chromosomal region includes 13 genes amongst of which we suggest for the first time the APC2, PLK5 and MBD3 genes as potential functional candidates for neurodevelopmental and behavioral phenotypes observed.
Conclusions:
Here we describe a patient with a 19p13.3 microdeletion that spans to the downstream chromosomal region with respect to the overlapping deletions previously reported in several other cases. The neurobehavioral features observed in our case has extended the phenotypic spectrum associated with the 19p13.3 microdeletion. New candidate genes are proposed for the neurobehavioral phenotype observed in our case.
Encysted hydrocele of the canal of Nuck in an 11-month-old child with a past history of duodenal atresia and Arnold-Chiari malformation: A case report
Authors: Zlatan Zvizdic, Emir Milisic, Adisa Chikha, Irmina Sefic, Amra Dzananovic, Semir Vranic
Journal: Medicine (Baltimore) (2019)
MeSH terms: Arnold-Chiari Malformation / complications, Cysts / congenital, Duodenal Obstruction / complications, Genital Diseases, Female / congenital, Intestinal Atresia / complications, Peritoneal Diseases / congenital, Case Reports, Cysts / pathology, Female, Genital Diseases, Female / pathology, Humans, Infant, Inguinal Canal, Peritoneal Diseases / pathology, Familial duodenal atresia

Rationale:
Hydrocele of the canal of Nuck is a rare developmental disorder and represents of a homolog of hydrocele of spermatic cord in males. Hydrocele of the canal of Nuck is a very rare cause of inguinal swelling in female infants and children. It results from the failure of obliteration of the distal portion of evaginated parietal peritoneum within the inguinal canal, which forms a sac containing fluid.
Patient concerns:
We describe a case of hydrocele of the canal of Nuck in an 11-month-old girl with a past medical history of duodenal atresia and Arnold-Chiari malformation.
Diagnosis:
Physical examination and ultrasound revealed a soft, cystic, noncompressible, and non-fluctuant labial mass measuring approximately 5 cm.
Interventions:
The patient underwent surgical exploration through a right skin crease incision. The cystic lesion was histologically confirmed to be a non-communicated hydrocele of canal of Nuck.
Outcomes:
The child is doing well at 1-year follow-up with no swelling or recurrence on the operated side.
Lessons:
Hydrocele of the canal of Nuck is a rare developmental disorder but should be considered in a differential diagnosis in young girls with an inguino-labial swelling.