OpenMed/multicare-articles huggingface dataset:
Nasal Tip Reconstruction Using Integra Bilayer Wound Matrix: An Alternative to the Forehead Flap
Authors: M A Applebaum, J D Daggett, W L Carter
Journal: Eplasty (2015)
Keywords: integra, bilayer wound matrix, forehead flap, nasal reconstruction, nasal tip
MeSH terms: Case Reports

Large defects of the nasal tip with exposure of the underlying cartilage pose a significant reconstructive challenge to the plastic surgeon.
Objective:
This article presents a case of a large nasal tip defect following basal cell excision that was successfully treated with Integra bilayer wound matrix and skin grafting.
Methods:
Following tumor excision, meshed Integra bilayer wound matrix was placed over the exposed nasal tip cartilage. After 4 weeks, the silicone layer was removed and a full-thickness graft placed on the nasal tip.
Results:
This reconstruction resulted in the restoration of patient's nasal tip with good contour and color match without the need for a forehead flap.
Conclusion:
This demonstrates the reconstructive potential of this modality in patients who are not candidates for reconstruction with a forehead flap.
Clinical and Radiological Findings of Autosomal Dominant Osteopetrosis Type II: A Case Report
Authors: Priyanka Kant, Neelkamal Sharda, Rahul R Bhowate
Journal: Case Rep Dent (2013)

Osteopetrosis is a rare inherited genetic disease characterized by sclerosis of the skeleton caused by the absence or malfunction of osteoclasts. Three distinct forms of the disease have been recognized, autosomal dominant osteopetrosis being the most common. Autosomal dominant osteopetrosis exhibits a heterogeneous trait with milder symptoms, often at later childhood or adulthood. The aim of this case report is to present the clinical and radiographic features of a 35-year-old female patient with autosomal dominant osteopetrosis type II who exhibited features of chronic generalised periodontitis, and the radiographs revealed generalised osteosclerosis and hallmark radiographic features of ADO type II, that is, "bone-within-bone appearance" and "Erlenmeyer-flask deformity."
Spontaneous Recession Repair after Orthodontic Treatment: Case Report with the Use of Digital Approach for Quantification of Soft Tissue Changes
Authors: Myroslav Goncharuk-Khomyn, Oleksandr Krasnokutskyy, Mykola Boichuk, Vitaliy Rusyn, Marharyta Hliudzyk-Shemota
Journal: Case Rep Dent (2023)
MeSH terms: Case Reports

The article presents a case of spontaneous recession repair in a male patient with Class II malocclusion, division 1, after orthodontic treatment with aligners. The difference in digital recession depth was measured before and at the end of treatment by means of automatic intraoral scans superimposition within adapted software while using "Cross section" and "Measuring" instruments. Digital analysis of intraoral scans obtained before and at the end of treatment has revealed that recessions within the area of teeth 1.5, 1.4, 1.3, 1.2, 1.1, 2.1, 2.2, 2.3, 2.4, and 2.5 have improved, and recession depth reduced by 0.73 ± 0.08 mm, 1.02 ± 0.09 mm, 1.86 ± 0.13 mm, 0.72 ± 0.09 mm, 0.73 ± 0.04 mm, 0.67 ± 0.06 mm, 0.66 ± 0.07 mm, 1.50 ± 0.12 mm, 1.10 ± 0.05 mm, and 0.45 ± 0.04 mm, appropriately. The present case report emphasizes that orthodontic correction of altered tooth position (angulation, inclination, and rotation) under certain clinical conditions may be considered as an effective method for soft tissue contour optimization in cases when pre-treatment tooth position could be interpreted as a causative factor or associated with diagnosed recession. The following outcomes could be related, but not limited to creeping attachment mechanism, bone-housing centering effects, optimization of occlusal load distribution with ruling out peak zones of strain accumulation, and mucogingival stress leveling. Due to the authors' knowledge, the present case report is the first one where the signs of spontaneous recession repair after orthodontic treatment were evidenced with the intraoral scans and quantified by the specifically implemented digital analysis approach.
Report of a rare case of gorham-stout disease of both shoulders: bisphosphonate treatment and shoulder replacement
Authors: Eike Garbers, Falk Reuther, Gunther Delling
Journal: Case Rep Rheumatol (2011)
MeSH terms: Case Reports

Massive osteolysis known as Gorham-Stout disease is a rare idiopathic disorder typically affecting long bones in a unifocal pattern. Angiomatosis is strongly connected to the osteolysis. Weather angiomatosis is the cause or the result of osteolysis is subject of intense discussion (Kawasaki et al. (2003), Möller et al. (1999), Radhakrishnan and Rockson (2008)). There are about 200 cases described since 1955. Our patient is a 77-year-old female patient with osteolyses of both shoulders involving the proximal humerus, lateral clavicle, and the glenoid. Under bisphosphonate therapy, the progressive osteolysis stopped on the right side and showed progression on the left. With the patient complaining about severe rest pain and impaired function, we performed surgical reconstruction by implantation of total shoulder prosthesis three months after onset of symptoms. Our case shows a possibility of primary and early surgical reconstruction with good clinical outcome.
A Time to Pause and Reflect: When a Patient with Autoimmune Hepatitis Stops Responding to Corticosteroids
Authors: Lewis Tsang, Mitali Fadia, Shivakumar Chitturi
Journal: Case Rep Gastrointest Med (2016)
MeSH terms: Case Reports

Drug-induced liver injury (DILI) with features of autoimmunity (AI) is a challenging diagnosis to make particularly due to its apparent corticosteroid responsiveness. We present the case of a 74-year-old woman who presented with a 2-week history of jaundice and fatigue. She was initially diagnosed with autoimmune hepatitis (AIH) based on biochemical and histological characteristics and prompt response with budesonide but a biochemical relapse occurred soon after inadvertent rechallenge with irbesartan, a drug that she had discontinued prior to her presentation but was not initially considered to be a cause of her symptoms.
Atypical infections in tsunami survivors
Authors: Christian Garzoni, Stéphane Emonet, Laurence Legout, Rilliet Benedict, Pierre Hoffmeyer, Louis Bernard, Jorge Garbino
Journal: Emerg Infect Dis (2005)
MeSH terms: Disasters, Discitis / microbiology, Drug Resistance, Multiple, Bacterial, Gram-Negative Bacteria / drug effects, Gram-Negative Bacterial Infections / microbiology, Mycobacterium Infections, Nontuberculous / microbiology, Scedosporium / isolation & purification, Survivors, Thoracic Vertebrae, Case Reports, Female, Humans, Male, Middle Aged, Mycetoma / microbiology, Mycobacterium chelonae / isolation & purification

After a tsunami hit Asia in December 2004, 2 survivors had severe infections due to multidrug-resistant and atypical bacteria and rare fungi weeks afterwards. Treating these infections is challenging from a clinical and microbiologic point of view.
Case Report: Acute Intracardiac Thrombosis in Children With Coronavirus Disease 2019 (COVID-19)
Authors: Hamid Bigdelian, Mohsen Sedighi, Mohammad Reza Sabri, Bahar Dehghan, Chehreh Mahdavi, Alireza Ahmadi, Mehdi Ghaderian, Hamid Rahimi, Atefeh Sadeghizadeh, Monirsadat Emadoleslami, Seyed Nasser Mostafavi, Rana Saleh, Niloofar Javadi, Maryam Derakhshan, Zahra Pourmoghaddas, Shima Sarfarazi Moghadam
Journal: Front Pediatr (2021)
Keywords: covid-19, acute thrombosis, cardiac surgery, case series, children
MeSH terms: Case Reports

We herein describe a case series of children with SARS-CoV-2 infection (COVID-19) complicated with acute intracardiac thrombosis. The diagnosis of COVID-19 was confirmed through the reverse transcription-polymerase chain reaction (RT-PCR). Transthoracic echocardiography of patients revealed large intracardiac mobile masses resected successfully via cardiac surgery. The underlying mechanisms of this thrombus in the COVID-19 infection may be attributed to the hypercoagulation and inflammatory state of the disease incurred by the SARS-CoV-2 virus.
Case Report: Second Report of Joubert Syndrome Caused by Biallelic Variants in IFT74
Authors: Ke Zhongling, Li Guoming, Chen Yanhui, Chen Xiaoru
Journal: Front Genet (2021)
Keywords: ift74, joubert syndrome, ciliopathy, cleft lip, developmental delay, polydactyly
MeSH terms: Case Reports

Joubert syndrome (JBTS) is a rare ciliopathy characterized by developmental delay, hypotonia, and distinctive cerebellar and brain stem malformation called the molar tooth sign (MTS). We reported a 15-month-old female with dysmorphic features (flat nasal bridge, almond-shaped eye, and a minor midline notch in the upper lips), hypotonia, polydactyly, development delay, and MTS. Whole exome sequencing revealed biallelic heterozygous mutations c.535C>G(p.Q179E/c.853G>T) (p.E285*) in IFT74, which were inherited from the parents. So far, only one article reported JBTS associated with IFT74 gene mutation, and this is the second report of the fifth patient with JBTS due to variants in IFT74. All five patients had developmental delay, postaxial polydactyly, subtle cleft of the upper lip, hypotonia, and MTS, but notably without renal and retinal anomalies or significant obesity, and they shared the same mutation c.535C>G(p.Q179E) in IFT74, and c.853G>T(p.E285*) that we found was a new mutation in IFT74 that related with Joubert syndrome. Those findings highlight the need for the inclusion of IFT74 in gene panels for JBST testing.
Case report: Pembrolizumab plus Axitinib related hypothyroid myopathy in two kidney cancer patients
Authors: Andrea Boutros, Lara Vera, Federico Gatto, Giuseppe Fornarini, Elisa Zanardi
Journal: Front Oncol (2022)
Keywords: axitinib, hypothyroid myopathy, hypothyroidism, immune-relate adverse event, immunotherapy, kidney cancer, myopathy, pembrolizumab
MeSH terms: Case Reports

The first-line therapy in advanced kidney cancer has changed in recent years due to the introduction of combinations of tyrosine kinase inhibitors (TKIs) of vascular endothelial growth factor receptors (VEGFR) and immune checkpoint inhibitors (ICIs). Although immune-related adverse events are well-known, in the case of combination treatments, the determination of which drug is related to an adverse event may be challenging. We reported two cases of patients who developed muscle enzyme elevation in association with hypothyroidism during therapy with pembrolizumab plus axitinib for metastatic kidney cancer. The myopathy rapidly resolved after hormone replacement therapy with levothyroxine. Hypothyroid myopathy is a scarcely known and underreported adverse event. This adverse event may be relevant in the differential diagnosis with immune-related myositis, which has an autoimmune pathogenesis and a potentially fatal course.
Erythema Multiforme Major Associated With Community-Acquired Pneumonia: Lessons From a Case Report
Authors: Xiaomei Fan, Yong Luo, Jieluan Lu, Jinji Xu, Qing Chen, Huijuan Guo, Ping Jin
Journal: Front Pediatr (2021)
Keywords: adverse drug reactions, azithromycin, children, erythema multiforme major, human leukocyte antigen
MeSH terms: Case Reports

Background: Erythema multiforme (EM) is an acute immune-mediated inflammatory mucinous skin disorder. The etiology of pediatric EM involves infections, medications, autoimmune diseases, and genetic factors. Case Report: An 8-year-old girl with Mycoplasma pneumoniae (MP) associated community-acquired pneumonia developed erythema target-like symptoms 1 week after azithromycin administration. The erythema quickly spread throughout the body involving the oral and ocular mucous membranes, the trunk, and the extremities, and eventually developed into erythema multiform major (EMM). Through drug withdrawal and specific treatment including systemic corticosteroids and supportive care, her clinical symptoms were improved. After 31 days, most of the mucocutaneous symptoms were relieved, except pigmentation. Human leukocyte antigen (HLA) gene sequencing was performed and 20 HLA genotypes were identified. The patient follow-up lasted for 18 months. Rashes appeared on her trunk when receiving azithromycin orally after discharge and then disappeared after azithromycin withdrawal. Conclusions: Pediatric EM is a rare disease and recognition of its etiology is important for EM management. In this case, azithromycin and HLA-DQB1 * 03:01 genotype may contribute to EMM. Lesson: For drug-induced EM, rapid identification and withdrawal of the causative drugs is critical. Re-exposure to the same drug or exposure to drugs with similar chemical structures should also be avoided. Patient education and rational use of medicines are essential for pediatric patients.
Unique Phrenic Nerve-Sparing Regional Anesthetic Technique for Pain Management after Shoulder Surgery
Authors: Jason K Panchamia, David A Olsen, Adam W Amundson
Journal: Case Rep Anesthesiol (2017)
MeSH terms: Case Reports

Background:
Ipsilateral phrenic nerve blockade is a common adverse event after an interscalene brachial plexus block, which can result in respiratory deterioration in patients with preexisting pulmonary conditions. Diaphragm-sparing nerve block techniques are continuing to evolve, with the intention of providing satisfactory postoperative analgesia while minimizing hemidiaphragmatic paralysis after shoulder surgery.
Case report:
We report the successful application of a combined ultrasound-guided infraclavicular brachial plexus block and suprascapular nerve block in a patient with a complicated pulmonary history undergoing a total shoulder replacement.
Conclusion:
This case report briefly reviews the important innervations to the shoulder joint and examines the utility of the infraclavicular brachial plexus block for postoperative pain management.
Pharyngeal-cervical-brachial variant of Guillain-Barré syndrome: a case report of a rare complication following Dengue-Chikungunya co-infection
Authors: Osama Mohiuddin, Anosh Aslam Khan, Syed Hamza Bin Waqar, Ali Tariq Shaikh, Momina Mariam Marufi, Sumeen Jalees, Farah Yasmin
Journal: Pan Afr Med J (2021)
Keywords: chikungunya, dengue, guillain-barré syndrome, case report, co-infection
MeSH terms: Chikungunya Fever / complications, Dengue / complications, Guillain-Barre Syndrome / diagnosis, Case Reports, Adult, Chikungunya Fever / diagnosis, Chikungunya Fever / therapy, Coinfection, Dengue / diagnosis, Dengue / therapy, Female, Guillain-Barre Syndrome / physiopathology, Guillain-Barre Syndrome / therapy, Humans, Immunoglobulin M / immunology, Immunoglobulins, Intravenous / administration & dosage, Immunoglobulin M, Immunoglobulins, Intravenous

Pharyngeal-cervical-brachial (PCB) variant of Guillain-Barré Syndrome (GBS) is characterized by weakness in cervicobrachial and oropharyngeal region, together with areflexia of upper limbs. Being an uncommon variant, it is often misdiagnosed as other neurological conditions resembling GBS. Although most of the cases occur as a post-infectious complication, no reports describing its development following dengue-chikungunya co-infection have been documented. A young female presented with a progressive history of swallowing difficulty, bilateral arm weakness and neck weakness. Three weeks earlier, she was presented with clinical features corresponding to dengue and was symptomatically treated. Currently, hypotonia and decreased muscle strength were observed in both upper limbs and neck. Detailed investigation revealed the presence of Immunoglobulin M (IgM) antibodies against dengue antigen (NS 1) and Chikungunya virus (CHIKV), confirming the possibility of previous dengue-chikungunya co-infection. Nerve conduction studies and electromyography of upper limbs pointed towards findings consistent with the early stages of acute motor demyelinating and possible axonal neuropathy. The detection of antiganglioside antibodies (anti-GT1a antibodies), confirmed the diagnosis of the pharyngeal-cervical-brachial variant of GBS. A five days treatment of intravenous immunoglobulin (IVIG) along with physical rehabilitation was started which led to significant improvement and the patient was discharged after 15 days. PCB is an unfamiliar variant of GBS for many clinicians. Diagnosis can be made by a thorough history, clinical examination and investigations that can rule out other potential causes of cervicobrachial and oropharyngeal weakness. It also necessitates careful monitoring and followups after mono- and co-arboviral infections to prevent any debilitating neurological complications.
Case report: Rudimentary uterine horn with ovarian endometriosis manifested as pelvic ectopic kidney
Authors: Si-Fan Yin, Jia-Gui Chai, Run-Lin Feng, Zhi-Yuan Yin, Shen-Zhao Zhao, Tao Zhang, Chang-Xing Ke
Journal: Front Med (Lausanne) (2023)
Keywords: diagnosis, endometriosis, pelvic ectopic kidney, rudimentary uterine horn, treatment
MeSH terms: Case Reports

Background:
Unicornuate uterus is a congenital uterine malformation. Unicornuate uterus with rudimentary horn, ovarian endometriosis, and congenital renal agenesis are rare combinations that can be easily misdiagnosed due to the lack of typical clinical manifestations.
Case summary:
A 19-year-old woman with pelvic pain was admitted to the hospital after a month. Physical examination was unremarkable. B-ultrasound and CT scan both indicated pelvic ectopic kidney. In addition, renal scintigraphy revealed normal perfusion and function of the right kidney, but the perfusion and function of the left kidney were not visible. A left pelvic ectopic kidney was diagnosed by preoperative images. A laparoscopic left pelvic ectopic nephrectomy was performed after adequate surgical preparation. However, the postoperative pathological diagnosis revealed a rudimentary uterine horn with ovarian endometriosis and congenital renal agenesis. Fortunately, she got recovered and was discharged from the hospital after 5 days following the operation. Moreover, she received regular follow-ups at the gynecology clinic. To date, no right adnexal or uterine abnormalities have been detected on ultrasound during the follow-up visits.
Conclusion:
Rudimentary uterine horn with ovarian endometriosis and congenital renal agenesis are rare and are easily Misdiagnosed due to the lack of typical clinical manifestations. A gynecological examination is recommended for patients who may have this disease.
Case Report: Durable complete pathologic response and organ preservation following ipilimumab and nivolumab for locally advanced primary vaginal mucosal melanoma
Authors: Ahmad A Tarhini, Wissam B Hanayneh, John J Powers, Carlos M Moran Segura, Jose R Conejo-Garcia, Cesar A Lam, Ardeshir Hakam, Mitchel S Hoffman
Journal: Front Oncol (2022)
Keywords: case report, immunotherapy, ipilimumab, nivolumab, vaginal mucosal melanoma
MeSH terms: Case Reports

Optimal management of locally advanced vaginal mucosal melanoma is poorly understood because of its rarity and unique biology. Patients have a poor prognosis despite aggressive management approaches including pelvic exenteration and adjuvant radiation that carry major morbidities. We report a case of a patient in early 40's who experienced complete pathologic response and organ preservation following immunotherapy consisting of 3 cycles of ipilimumab and nivolumab. Treatment was complicated by a high-grade immune mediated hepatitis that eventually resolved with immunosuppressive therapy. Immune monitoring studies utilizing vaginal tumor biopsies showed evidence of enhanced infiltration by CD3+/CD8+ cytotoxic T-cells and increased expression of MHC-I/PD-L1 within the tumor microenvironment following immunotherapy. The patient continues to be without evidence of disease recurrence by radiologic and gynecologic examinations with more than 2 years of follow up from the time of immunotherapy initiation. To our knowledge, this is the only case report in the literature of a patient with locally advanced vaginal mucosal melanoma experiencing a durable complete pathologic response and organ preservation following immune checkpoint blockade as the only treatment approach.
An unusual cause of fatal rapid-onset ataxia plus syndrome
Authors: Ivan Kmezic, Jan Weinberg, Dan Hauzenberger, Farouk Hashim, Evangelia Kollia, Monika Klimkowska, Inger Nennesmo, Martin Paucar
Journal: Cerebellum Ataxias (2017)
Keywords: ataxia, fascia analysis, hydroxyurea, jc-virus, pml, polycythemia vera
MeSH terms: Case Reports

Background:
Progressive multifocal leukoencephalopathy (PML) is a demyelinating disorder of the central nervous system caused by reactivation of the JC-virus and is in most cases associated with underlying immunosuppression. Acquired immune deficiency syndrome (AIDS) and hematological malignancies are well-known predisposing factors for PML. However, in the past ten years, various pharmacological agents have been associated with increased risk of PML. Based on the phenomenology PML can be divided into the cerebral form and the rare cerebellar form.
Case presentation:
Here we describe a man affected by polycythemia vera (PCV) that was treated with hydroxyurea (HU) and developed PML. The initially PML presentation included ataxia as one of the main features. Brain MRI displayed widespread supratentorial and infratentorial lesions. Immunological analysis revealed absence of reactivity to a wide range of antigens. The course of disease was rapidly progressive with fatal outcome - autopsy ruled out leukemic transformation.
Conclusion:
The occurrence of PML in PCV patients is very rare and has been reported only once. Movement disorders, such as ataxia, are also less frequent. In the present case the PML was likely multifactorial.
ZD 1839 in patients with brain metastases from non-small-cell lung cancer (NSCLC): report of four cases
Authors: F Cappuzzo, C Calandri, S Bartolini, L Crinò
Journal: Br J Cancer (2003)
MeSH terms: Antineoplastic Agents / pharmacology, Brain Neoplasms / drug therapy, Brain Neoplasms / secondary, Carcinoma, Non-Small-Cell Lung / drug therapy, Carcinoma, Non-Small-Cell Lung / secondary, Lung Neoplasms / pathology, Quinazolines / pharmacology, Case Reports, Aged, Brain Neoplasms / radiotherapy, Carcinoma, Non-Small-Cell Lung / radiotherapy, Female, Gefitinib, Humans, Male, Middle Aged, Tomography, X-Ray Computed, Treatment Outcome, Antineoplastic Agents, Quinazolines, Gefitinib

The activity of ZD 1839 on brain metastases (BM) from Non-Small-Cell Lung Cancer (NSCLC) is unknown. We report four cases of BM responding to ZD 1839 theraphy.