OpenMed/multicare-articles huggingface dataset:
Case Report: BCG-Triggered Hemophagocytic Lymphohistiocytosis in an Infant With X-Linked Recessive Mendelian Susceptibility to Mycobacterial Disease Due to a Variant of Chronic Granulomatous Disease
Authors: Suleiman Al-Hammadi, Amal M Yahya, Abdulla Al-Amri, Amar Shibli, Ghazala B Balhaj, Mohamed I Tawil, Ranjit Vijayan, Abdul-Kader Souid
Journal: Front Pediatr (2021)
Keywords: bcg disease, bacillus calmette-guérin, united arab emirates, immunodeficiency, inborn error of immunity, newborns, tuberculosis, vaccines
MeSH terms: Case Reports

In the United Arab Emirates, BCG (Bacillus Calmette-Guérin) is administered to all newborns. We present here a young infant with an inborn error of immunity (IEI) who developed fatal adverse events to this live-attenuated vaccine. This male infant received BCG (Serum Institute of India Pvt., Ltd., India) on Day 11 of life. On Day 25, he developed fever, followed by cervical lymphadenitis and bilateral otitis media with fluid drainage. On Day 118, he was admitted with severe hemophagocytic lymphohistiocytosis (HLH), and passed away on Day 145. The diagnostic exome sequencing test identified a hemizygous nonsense variant, NM_000397.3(CYBB):c.676C>T, p.Arg226* (rs137854592). Pathogenic variants of CYBB [cytochrome b(-245), beta subunit; Mendelian Inheritance in Man [MIM] accession code, 300481] are known to cause "immunodeficiency 34, mycobacteriosis, X-linked" (IMD34, MIM#300645) and "chronic granulomatous disease, X-linked" (CGDX, MIM#306400). The natural history of his illness is consistent with "X-linked recessive Mendelian susceptibility to mycobacterial disease (MSMD)." This entity is responsible for his BCG disease and is a likely trigger of his HLH. This disastrous event underlines the importance of developing worldwide policies that target BCG disease prevention, especially in communities with high prevalence of IEI. Moreover, screening for genetic causes of MSMD in the community could pave the way, at least partially, for scale-up of tuberculosis (TB) prevention.
Radial, renal and craniofacial anomalies: Baller-Gerold syndrome
Authors: Jyotsna Murthy, Ramesh Babu, Padmasani Venkat Ramanan
Journal: Indian J Plast Surg (2008)
Keywords: baller-gerold syndrome, craniosynostosis, crossed ectopia, ectopic kidneys, microcephaly, radial agenesis, radial club hand, reflux, renal agenesis, vesico ureteric reflux
MeSH terms: Case Reports

The Baller-Gerold syndrome is a rare syndrome with very few cases published in literature. Craniosynostosis and radial aplasia are striking features, easy to diagnose. However, there are many differential diagnoses. Often, the question raised is whether the Baller-Gerald syndrome is a distinct entity. We report a patient with findings of craniosynostosis and radial aplasia consistent with the diagnosis of the Baller-Gerold syndrome. Genotypic heterogeneity could possibly underlie the phenotypic variability exhibited by these cases.
Benzocaine and lidocaine induced methemoglobinemia after bronchoscopy: a case report
Authors: Sophie Kwok, Jacqueline L Fischer, John D Rogers
Journal: J Med Case Rep (2008)

Introduction:
Methemoglobinemia is a rare cause of hypoxemia, characterized by abnormal levels of oxidized hemoglobin that cannot bind to and transport oxygen.
Case presentation:
A 62-year-old male underwent bronchoscopy where lidocaine oral solution and Hurricaine spray (20% benzocaine) were used. He developed central cyanosis and his oxygen saturation was 85% via pulse oximetry. An arterial blood gas revealed pH 7.45, PCO2 42, PO2 282, oxygen saturation 85%. Co-oximetry performed revealed a methemoglobin level of 17.5% (normal 0.6-2.5%). The patient was continued on 15 L/minute nonrebreathing face mask and subsequent oxygen saturation improved to 92% within two hours. With hemodynamic stability and improved SpO2, treatment with methylene blue was withheld.
Conclusion:
Methemoglobinemia is a potentially lethal condition after exposure to routinely used drugs. Physicians should be aware of this complication for early diagnosis and treatment.
Injectable Chemotherapy Downstaged Oral Squamous Cell Carcinoma from Nonresectable to Resectable in a Rescue Dog: Diagnosis, Treatment, and Outcome
Authors: Shuang Cai, Ti Zhang, Chad Groer, Melanie Forrest, Daniel Aires, Vern Otte, Sally Barchman, Abby Faerber, Marcus Laird Forrest
Journal: Case Rep Vet Med (2018)
MeSH terms: Case Reports

This case report documents the diagnosis, treatment, and outcome of a nonresectable oral squamous cell carcinoma in a dog with initial poor prognosis. An approximately 4-year-old female Staffordshire Bull Terrier presented with a large mass on the front of lower jaw which was diagnosed as oral papillary squamous cell carcinoma by histopathology. CT scans revealed invasion of the cancer to the frenulum of the tongue. The mass was inoperable due to location, expansiveness, and metastatic lymph nodes. The dog received 4 treatments of intralesional hyaluronan-platinum conjugates (HylaPlat™, HylaPharm LLC, Lawrence, Kansas) at 3-week intervals. Clinical chemistry and complete blood count were performed one week after each treatment and results were within normal limits. Complications included bleeding due to tumor tissue sloughing, as well as a single seizure due to unknown causes. Upon completion of chemotherapy, CT showed that the mass had regressed and was no longer invading the lingual frenulum, and multiple lymph nodes were free of metastasis. The mass thus became resectable and the dog successfully underwent rostral bilateral mandibulectomy. Over one year after chemotherapy and surgery, the cancer remains in complete remission.
Seizure and hepatosplenomegaly-rare manifestation of parvovirus B-19: a case report and review of the literature
Authors: Yadav Kamlesh, Gupta Pallav, Murari Manjula, Malik Rohan
Journal: J Trop Med (2011)
MeSH terms: Case Reports

Parvovirus B19 is the etiologic agent of erythema infectiosum (fifth disease), a fever-rash illness occurring in childhood. We present a 10 month old child with high grade fever for 10 days, generalized tonic-clonic seizure, bilateral cervical lymphadenopathy, generalized maculopapular rash, hematemesis and malena. Bone marrow aspiration and liver biopsy were done. EBV serology and parvovirus PCR were also performed. Bone marrow aspiration and biopsy showed giant pro-erythroblast consistent with parvovirus infection. PCR showed amplification of parvovirus genomic sequences. Present case highlights an atypical presentation of Parvovirus B19 infection as fever, rash and hepatosplenomegaly.
A rare case report of amlodipine-induced gingival enlargement and review of its pathogenesis
Authors: Sanjeev Joshi, Sucheta Bansal
Journal: Case Rep Dent (2013)

Gingival enlargement is a common clinical feature of gingival and periodontal diseases. It is an unwanted side effect of certain systemic drugs given for nondental treatment. It is being reported with three main groups of drugs like calcium channel blockers (CCBs), immunosuppressants, and anticonvulsants. Among calcium channel blockers, nifedipine causes gingival hyperplasia in about 10% of patients, whereas the incidence of amlodipine-, a third generation calcium channel blocker, induced gingival hyperplasia is very limited. There are very few reports of amlodipine-induced gingival enlargement at a dose of 5 mg. We report a case of amlodipine-induced gingival enlargement in a 45-year-old hypertensive patient taking amlodipine at a dose of 5 mg.
Canagliflozin-associated diabetic ketoacidosis: a case report
Authors: Peter R Chai, Caitlin Bonney, Eike Blohm, Edward W Boyer, Kavita M Babu
Journal: Toxicol Commun (2017)
Keywords: canagliflozin, sglt inhibitors, adverse events, diabetes, diabetic ketoacidosis

Canagliflozin is a novel sodium-glucose cotransporter-2 (SGLT-2) inhibitor approved for the management of diabetes. We report the presentation and management of two cases of canagliflozin associated diabetic ketoacidosis (DKA) and discuss the mechanism of canagliflozin associated DKA. Patient 1, a 55 year old woman maintained on canagliflozin for diabetes mellitus II presented to the emergency department (ED) with 24 hours of nausea and vomiting. She was diagnosed with DKA featuring hypotension, hyperglycemia, ketosis and acidosis. A second 54 year old man also maintained on canagliflozin for diabetes mellitus I presented to the ED with 24 hours of nausea and vomiting. He was diagnosed with DKA with similar manifestations as patient 1. Both patients underwent massive volume resuscitation and intravenous insulin therapy with resolution of ketosis and acidosis. By inhibiting SGLT-2, canagliflozin promotes glucosuria, which in turn can produce up to a 10% decrease in total plasma volume rendering patients maintained on canagliflozin susceptible to dehydration. Inhibition of SGLT-2 also leads to glucagon secretion, which in the volume deplete individual, can exacerbate DKA. Physicians should be aware of the rapid onset of DKA in patients maintained on canagliflozin after just minor additional fluid losses.
Case Report: Prolapsed Ureterocele-A Differential Diagnosis of Urethral Cists
Authors: Francisco Renan Doth Sales, Georgia Alexsandra Colantonio Dourado, Ana Carolina Montes Ribeiro, Humberto de Holanda Madeira Barros, David Sucupira Cristino, Antonio Augusto Guterres Castro, Francisco José Cabral Mesquita
Journal: Case Rep Urol (2020)
MeSH terms: Case Reports

Ureterocele is a cystic dilatation of submucosal distal ureter. It presents a higher incidence in infants and young children but is rare in adults. The urethral prolapse of ureterocele is extremely rare, and its clinical presentation includes vulvar mass, hematuria, and urinary tract dysfunction. We present a case of ureterocele prolapse in a 45-year-old woman who has a 3-day-evolution vulvar mass and intense urethral bleeding. The patient underwent armed cystoscopy and ureteroscopy, ureterocele resection, and biopsy. She evolved with good postoperative condition and was then discharged.
Retroperitoneal angiomyolipoma: a case report and review of the literature
Authors: Andrew Strahan, Jonathan King, Scott McClintock
Journal: Case Rep Radiol (2013)

Retroperitoneal angiomyolipoma is a rare tumour that is difficult to diagnose preoperatively. We present a case of retroperitoneal angiomyolipoma that highlights its diagnostic dilemma. We also performed a literature review and present a review of retroperitoneal angiomyolipoma.
Surgical Repositioning of a Dilacerated Impacted Incisor
Authors: Vivek Kumar Adlakha, Preetika Chandna, Sumir Gandhi, Saroj Chopra, Namita Singh, Shamsher Singh
Journal: Int J Clin Pediatr Dent (2011)
Keywords: dental trauma, dilaceration, impacted
MeSH terms: Case Reports

Dilaceration is one of the most common complications of trauma to deciduous dentition. The possible causes of dilaceration are trauma and developmental disturbances, while some authors suggest an association with some developmental syndromes. Dilaceration can be seen in both the permanent and deciduous dentition. The present case report describes surgical repositioning of a dilacerated impacted maxillary central incisor in a 9-year-old boy.
Case Report: A Case of Perifoveal Exudative Vascular Anomalous Complex With a Good Prognosis
Authors: Min Fu, Pan Hu, Gang Zhang, Ludonghan Huang, Huan Xu, Ju Huang, Meihui Wu, Yanli Chen
Journal: Front Med (Lausanne) (2021)
Keywords: age-related macular degeneration (amd), case report, good prognosis, micropulse laser (mpl), perifoveal exudative vascular anomalous complex (pevac)
MeSH terms: Case Reports

Significance: Perifoveal exudative vascular anomalous complex (PEVAC) is a unique clinical lesion. It manifests as isolated lesions and is easily misdiagnosed. Thus far, few PEVAC case reports have been published. PEVAC is typically inconsistent with other reported macular lesions. Purpose: To report our 24-month follow-up experience on the treatment of PEVAC with a micropulse laser (MPL). Case Report: A 56-year-old Chinese woman with no history of other diseases complained of decreased vision in her left eye that had persisted for more than 1 year. Comprehensive ophthalmic examinations were performed, including a vision test, slit lamp fundus exam, optical coherence tomography (OCT), optical coherence tomography angiography (OCT-A), fluorescein fundus angiography (FFA) and indocyanine green angiography (ICGA). Intravitreal injection of ranibizumab was ineffective, and bleeding, exudation and visual acuity were not improved. After two rounds of micropulse laser (MPL) treatment, the patient was followed up, and the prognosis was good. Conclusion: PEVAC is very rare, and early diagnosis is important, as the lesions readily cause irreversible damage. Our results indicate that an MPL can be used as an alternative treatment for PEVAC patients.
Case Report: A Case of Trimethoprim/Sulfamethoxazole-Triggered Hypotensive Shock: Cytokine Release Syndrome Related to Immune Checkpoint Inhibitors and Drug-Induced Hypersensitivity Syndrome
Authors: Tetsuya Urasaki, Makiko Ono, Toshiaki Mochizuki, Koichi Takeda, Aya Nishizawa, Eri Fukagawa, Motohiro Fujiwara, Yoshinobu Komai, Shigehisa Kitano, Takeshi Yuasa, Junji Yonese, Shunji Takahashi
Journal: Front Oncol (2021)
Keywords: cytokine release syndrome, drug-induced hypersensitivity syndrome, immune checkpoint inhibitor, immune-related adverse event, trimethoprim/sulfamethoxazole
MeSH terms: Case Reports

Currently, only a few reports exist on the cytokine release syndrome (CRS) as one of the severe immune-related adverse events (irAEs) induced by immune checkpoint inhibitors (ICIs). Notably, it is very rare that grade 4 CRS related to ICI therapy overlaps with the drug-induced hypersensitivity syndrome (DiHS). A 46-year old woman with metastatic kidney cancer had grade 3 interstitial pneumonitis induced by four cycles of combination therapy of anti-programmed death-1 and anti-cytotoxic T lymphocyte-4 antibodies after right cytoreductive nephrectomy. Prophylactic administration of trimethoprim/sulfamethoxazole (TMP/SMX) was started concomitantly with prednisolone therapy to treat the interstitial pneumonitis. She developed hypotensive shock when reducing the dosage of prednisolone, and required intubation and ventilation using vasopressors at the intensive care unit. She subsequently exhibited prominent leukocytosis and an increased level of C-reactive protein, suggesting markedly increased cytokine levels. Interestingly, facial edema and erythema increased in association with pyrexia, leukocytosis, liver dysfunction, and renal failure, suggesting that she developed DiHS. She received hemodialysis three times, a plasma exchange, and anti-interleukin-6 therapy to treat severe renal dysfunction, a thrombotic thrombocytopenic purpura-suspected condition, and possible grade 4 CRS, respectively. Although these therapies did not elicit sufficient effects, high-dose administration of intravenous immunoglobulin was successful. With steroid mini-pulse therapy and the subsequent administration of prednisolone, she recovered successfully. To the best of our knowledge, this is the first report that ICIs and TMP/SMX can induce hypotensive shock accompanied with CRS and DiHS during immunosuppressive therapy for an irAE. Importantly, the prophylactic administration of TMP/SMX should be performed cautiously to avoid severe drug reactions such as CRS or DiHS.
A rare cause of postpartum low back pain: pregnancy- and lactation-associated osteoporosis
Authors: Rabia Terzi, Hasan Terzi, Tülay Özer, Ahmet Kale
Journal: Biomed Res Int (2014)
MeSH terms: Lactation Disorders / physiopathology, Low Back Pain / physiopathology, Osteoporosis / physiopathology, Pregnancy Complications / physiopathology, Case Reports, Adult, Breast Feeding, Female, Humans, Lactation, Lactation Disorders / etiology, Low Back Pain / etiology, Lumbar Vertebrae / physiopathology, Osteoporosis / etiology, Postpartum Period, Pregnancy

Pregnancy- and lactation-associated osteoporosis (PLO) is a rare form of osteoporosis. It results in severe low back pain in the last trimester of pregnancy and in the postpartum period, decreases in height, and fragility fractures, particularly in the vertebra. The current case report presents a 32-year-old patient who presented with back and low back pain that began in the last trimester of the pregnancy and worsened at two months postpartum and who was diagnosed with pregnancy- and lactation-associated osteoporosis after exclusion of other causes; the findings are discussed in view of the current literature. PLO is a rare clinical condition causing significant disability. PLO must be kept in mind in the differential diagnosis in patients presenting with low back pain during or after pregnancy. The patients must be evaluated for the risk factors of PLO, and an appropriate therapy must be initiated.
Case report: Multiple disconnection patterns revealed by a multi-modal analysis explained behavior after a focal frontal damage
Authors: Elena Monai, Erica Silvestri, Marta Bisio, Annachiara Cagnin, Marco Aiello, Diego Cecchin, Alessandra Bertoldo, Maurizio Corbetta
Journal: Front Neurol (2023)
Keywords: behavior, brain lesion, disconnection, multi-modal mapping, networks
MeSH terms: Case Reports

Introduction:
There is overwhelming evidence that focal lesions cause structural, metabolic, functional, and electrical disconnection of regions directly and indirectly connected with the site of injury. Unfortunately, methods to study disconnection (positron emission tomography, structural and functional magnetic resonance imaging, electroencephalography) have been applied primarily in isolation without capturing their interaction. Moreover, multi-modal imaging studies applied to focal lesions are rare.
Case report:
We analyzed with a multi-modal approach the case of a patient presenting with borderline cognitive deficits across multiple domains and recurrent delirium. A post-surgical focal frontal lesion was evident based on the brain anatomical MRI. However, we were able to acquire also simultaneous MRI (structural and functional) and [18F]FDG using a hybrid PET/MRI scan along with EEG recordings. Despite the focality of the primary anatomical lesion, structural disconnection in the white matter bundles extended far beyond the lesion and showed a topographical match with the cortical glucose hypometabolism seen both locally and remotely, in posterior cortices. Similarly, a right frontal delta activity near/at the region of structural damage was associated with alterations of distant occipital alpha power. Moreover, functional MRI revealed even more widespread local and distant synchronization, involving also regions not affected by the structural/metabolic/electrical impairment.
Conclusion:
Overall, this exemplary multi-modal case study illustrates how a focal brain lesion causes a multiplicity of disconnection and functional impairments that extend beyond the borders of the anatomical irrecoverable damage. These effects were relevant to explain patient's behavior and may be potential targets of neuro-modulation strategies.
Primary Angioplasty for Cardiac Allograft Vasculopathy Presenting as ST-Elevation Acute Myocardial Infarction during Endomyocardial Biopsy
Authors: Bruno Ramos Nascimento, Thalles Oliveira Gomes, Júlio César Borges, Guilherme Rafael Sant'anna Athayde, Sílvio Amadeu de Andrade, Maria da Consolação Vieira Moreira
Journal: Case Rep Transplant (2013)

Cardiac allograft vasculopathy is still a major issue, with significative mortality in heart transplant patients, and the best therapeutic options are not yet established. The progressively higher survival rates after transplantation have made it a major concern. This is a case report about a patient who underwent cardiac transplantation due to chagasic cardiomiopathy. During an endomyocardial biopsy more than 2 years after the transplant, the patient arrested in ventricular fibrillation, with ST-elevation in anterior leads after defibrillation. The angiography showed total occlusion of proximal left anterior descending artery, promptly treated with primary angioplasty, with excellent angiographic and clinical results.
Zancolli procedure and nerve repair with sural graft as a treatment for patient with claw hand due to complete rupture of ulnar and median nerve: A case report
Authors: Wahyu Widodo, Agus Waryudi, Zecky Eko Triwahyudi
Journal: Int J Surg Case Rep (2018)
Keywords: claw hand, median and ulnar nerve, sural graft, zancolli procedure

Introduction:
Combined injuries of peripheral nerve of upper extremity are usually the result of severe trauma to the extremity, and are often associated with substantial soft tissue, vascular, and bony injuries. The most common form of such combined injury is a low median-ulnar palsy, usually due to laceration of the volar wrist. It is a devastating injury, and with delayed presentation the injury is going to be more difficult to reconstruct.
Presentation of case:
Ten-year-old girl was admitted to hospital with numbness of her left palm and fingers, 5 months before admission. She was hit by a car while riding a bicycle from opposite direction from she was heading and cut her left forearm by licensed plate. Initially treated at nearby clinic but later couldn't extend her fingers after the pain subsided and left untreated for 5 months.
Discussion:
Traditionally, number of static procedures have been described to correct the claw hand deformity by preventing MCP joint hyperextension. Nevertheless, these procedures are reserved for those having ability to extend IP joints while MCP joint hyperextension were prevented. Conversely, static procedure like Zancolli capsulodesis is simple and do not sacrifice any motors in an already compromised extremity. Combined with sural graft, the outcome for the patient is hopefully better than Zancolli procedure alone.
Conclusion:
Zancolli procedure along with nerve repair with sural graft as a treatment of claw hand due to complete rupture of ulnar and median nerve is one of feasible treatment options to correct claw hand deformity with such combined palsy.
Case report: Bilateral central serous chorioretinopathy-like abnormalities in a man with pulmonary arterial hypertension
Authors: Xinrong Zhou, Jingxiang Zhang, Limin Gu, Hao Zhou, Jingfa Zhang
Journal: Front Med (Lausanne) (2022)
Keywords: case report, central serous chorioretinopathy, fundus abnormalities, hypertension - complications, macular edema, pulmonary arterial hypertension, vascular disease
MeSH terms: Case Reports

Background:
Pulmonary arterial hypertension (PAH) leads to progressive increases in pulmonary vascular resistance, right heart failure, and death if left untreated. Ocular complications secondary to PAH were less reported. In this study, we reported a case of bilateral visual loss and metamorphopsia in a patient with PAH, who developed central serous chorioretinopathy (CSCR)-like abnormalities and optic disc atrophy.
Case summary:
A 45-year-old man presented with decreasing central vision and metamorphopsia in both eyes. He had a history of PAH and 6-year history of low-dose oral sildenafil treatment. Slit-lamp examination revealed prominent dilated and tortuous episcleral and conjunctival vessels. Ultrawide-field color picture showed retinal pigment epithelial mottling and atrophy in ring-like configurations. Ultrawide-field autofluorescence showed multiple irregular hyper-autofluorescence with a constellation-like pattern surrounding the optic nerve head and macular region. Optical coherence tomography angiography (OCTA) b-scan demonstrated CSCR-like changes. Swept-source optical coherence tomography (SS-OCT) analysis showed optic nerve atrophy with enlarged cup/disc ratio in right eye, which was confirmed with perimetry. Fluorescein angiography (FA) showed marked leakage of macula and optic nerve head with time, cystoid macular edema, early blocking with late staining of the flecks as shown in the backgrounds of infrared and autofluorescence, and mild leakage in peripheral retina. Indocyanine green angiography (ICGA) showed dilation, tortuosity and congestion of all vortex veins without obvious leakage.
Conclusion:
Undertreated PAH may cause the congestion of the choroid and induce CSCR-like abnormalities.