OpenMed/multicare-articles huggingface dataset:
Facial Cellulitis Due to Type I Talon Cusp in a Pediatric Patient: A Case Report
Authors: Tulika Wakhloo
Journal: Cureus (2023)
Keywords: dental anomaly, facial cellulitis, maxillary lateral incisor, pediatric preventive dentistry, talon cusp
MeSH terms: Case Reports

Talon cusp is a rare odontogenic developmental anomaly with male predilection and multifactorial etiology. It projects as an accessory cusp-like structure from the cingulum area or the cement enamel junction (CEJ) affecting both primary and permanent dentition. It is imperative to clinically examine the developing dentition for occlusal harmony, especially in children with this anomaly. This report highlights facial cellulitis as a complication arising from a type I taloned maxillary lateral incisor and its endodontic management in a female pediatric patient. It emphasizes the clinical significance, early diagnosis, and prompt prophylactic management of the talon cusp so as to prevent the development of acute sequelae in children.
Osteolysis of the Greater Trochanter Caused by a Foreign Body Granuloma Associated with the Ethibond® Suture after Total Hip Arthroplasty
Authors: Keiji Kamo, Hiroaki Kijima, Koichiro Okuyama, Nobutoshi Seki, Shin Yamada, Naohisa Miyakoshi, Yoichi Shimada
Journal: Case Rep Orthop (2017)
MeSH terms: Case Reports

The present case shows a case of progression of osteolysis of the greater trochanter caused by a foreign body granuloma associated with the number 5 Ethibond suture in cementless THA with the direct lateral approach that was completely healed by removal of the Ethibond suture. A 55-year-old Japanese woman with secondary osteoarthritis caused by acetabular dysplasia underwent left cementless THA with the direct lateral approach. After setting of the total hip prosthesis, the gluteus medius muscle and vastus lateralis muscle were reattached to the greater trochanter through two bone tunnels using number 5 Ethibond EXCEL sutures. The left hip pain disappeared after surgery, but the bone tunnels enlarged gradually and developed osteolysis at 10 weeks. The removal of the Ethibond sutures and debridement improved the osteolysis. Histological examination showed the granuloma reaction to a foreign body with giant cell formation. The Ethibond suture has the lowest inflammatory tissue reaction and relatively high tension strength among nonabsorbable suture materials. However, number 5 Ethibond has the potential to cause osteolysis due to a foreign body granuloma, as in the present case.
Hemorrhagic Pericardial Effusion with Tamponade: A Rare Adverse Effect of Infliximab-Case Report and Literature Review
Authors: Henry D Lather, J Michelle Kahlenberg
Journal: Case Rep Rheumatol (2016)

Introduction. Antitumor necrosis factor (TNF) alpha agents are commonly used biologic therapies for a wide variety of rheumatic and inflammatory diseases. Here, we present a case of hemorrhagic pericarditis as a consequence of infliximab and review the literature on pericardial complications stemming from this drug class. Methods. For the literature review, search terms using versions of antitumor necrosis factor alpha AND pericardial effusion OR pericarditis OR pleuropericarditis OR cardiac tamponade were used. Results. Pericarditis is a rare but serious complication of anti-TNF based therapy, and hemorrhagic fluid is even more rare, with only one additional case reported. It is likely that this complication was secondary to a robust immune response to very high titer anti-infliximab antibodies. Providers should be aware that this complication can occur and that abnormal elevations in procalcitonin may accompany this unusual finding.
The Brain-Heart Link: A Case Report of a Critically Located Multiple Sclerosis Lesion in the Brainstem Leading to Recurrent Takotsubo Syndrome
Authors: Florentijn Risseeuw, Pegah Masrori, Ingrid Baar, Simon Nicolay, Constantijn Franssen, Barbara Willekens
Journal: Front Cardiovasc Med (2021)
Keywords: takotsubo syndrome, autonomic dysfunction, brainstem, case report, multiple sclerosis, stress cardiomyopathy
MeSH terms: Case Reports

Various central nervous system (CNS) diseases, including neurovascular and neuroinflammatory diseases, can lead to stress cardiomyopathy, also known as Takotsubo syndrome (TTS). We present a case of a 69-year-old woman with cardiovascular comorbidities, suffering from repeated episodes of TTS and respiratory failure due to a critical lesion in the brainstem, leading to a diagnosis of multiple sclerosis (MS). Despite aggressive treatment, intractable and recurrent symptoms in our patient occurred. Repeated bouts of autonomic dysfunction and respiratory failure ultimately led to installment of palliative care and the patient passing away. TTS should raise suspicion for underlying neurological diseases. Thorough questioning of previous neurological symptoms and extensive neurological workup is warranted. MS should be considered as a trigger of TTS also in elderly patients with cardiovascular risk factors.
STEMI after Dobutamine Stress Echocardiography in Hyperthyroid State
Authors: Mahmood Mubasher, Ashfaq Patel, Mohamed Magdi, Tahir Hamid
Journal: Case Rep Cardiol (2019)
MeSH terms: Case Reports

Uncontrolled hyperthyroidism has been associated with significant changes in cardiovascular hemodynamics. We report a case of a 39-year-old male who has been recently diagnosed with severe hyperthyroidism. He was undergoing dobutamine stress echocardiography (DSE) for evaluation of symptoms suggestive of stable angina. The exam was complicated by ST-segment elevation myocardial infarction- (STEMI-) required coronary angiography that showed mild coronary artery disease.
Intracellular and extracellular rhomboid shaped crystalline inclusions in a case of IgG lambda restricted plasma cell myeloma: a case report and review of the literature
Authors: Andres Matoso, Tina Rizack, Diana O Treaba
Journal: Diagn Pathol (2010)
MeSH terms: Bone Marrow / chemistry, Immunoglobulin G / analysis, Inclusion Bodies / chemistry, Multiple Myeloma / diagnosis, Plasma Cells / chemistry, Case Reports, Review, Aged, Bone Marrow / immunology, Bone Marrow / pathology, Bone Marrow Examination, Crystallization, Flow Cytometry, Humans, Immunohistochemistry, Immunophenotyping, Inclusion Bodies / pathology, Male, Multiple Myeloma / chemistry, Multiple Myeloma / drug therapy, Multiple Myeloma / immunology, Multiple Myeloma / pathology, Plasma Cells / immunology, Plasma Cells / pathology, Predictive Value of Tests, Immunoglobulin G

The presence of crystalline inclusions in plasma cell myeloma is a rare phenomenon and cases have been reported with rod, needle, and rectangular shaped crystals. Here, we present a case of IgG lambda restricted plasma cell myeloma with rhomboid shaped intracellular crystalline inclusions and extracellular crystal depositions in the bone marrow. Since rhomboid crystal depositions can be seen in other clinical conditions such as pseudogout, this case invites consideration of plasma cell myeloma in the differential diagnosis of patients with rhomboid crystalline deposition in the bone marrow and in sites/organs other than the bone marrow.
Hemorrhagic schwannoma of the trochlear nerve: Case report and a review of the literature
Authors: Jin Lei, Yu Li, Xueyan Wan, Junwen Wang, Chao You, Kai Zhao, Hongquan Niu
Journal: Front Oncol (2023)
Keywords: intracranial schwannoma, intratumoral hemorrhage, neurosurgical oncology, subtemporal approach, trochlear nerve
MeSH terms: Case Reports

Background:
Schwannomas of the trochlear nerve with the absence of systemic neurofibromatosis are considerably uncommon, especially complicated by intra-tumoral hemorrhage. Due to the lack of typical clinical manifestations and imaging findings, a definite diagnosis of trochlear schwannomas before surgery is particularly difficult.
Case presentation:
We report the case of a 64-year-old female patient who presented with a unilaterally intermittent headache of 2-month duration and without a remarkable neurological deficit at admission. Imaging studies revealed a well-demarcated cystic-solid lesion with mixed signals beside the brainstem and suprasellar cisterna. The patient underwent a surgical operation with total resection of the tumor by a subtemporal surgical approach. The tumor was intraoperatively found to originate from the trochlear nerve and was pathologically confirmed as a hemorrhagic schwannoma with cystic degeneration.
Conclusions:
We describe this case in detail and conduct a concomitant survey of the literature, summarizing the clinical presentations, radiological features, surgical treatment, and the possible mechanisms of hemorrhage in relevance to trochlear nerve schwannoma.
A Giant Parathyroid Adenoma Presenting as Nausea, Vomiting, and Headaches in an Adolescent Male
Authors: Jessica Muse, Rebecca Palmer, Jeanna Auriemma
Journal: Case Rep Pediatr (2023)
MeSH terms: Case Reports

Primary hyperparathyroidism is rare in the pediatric population and is typically caused by a single parathyroid adenoma. Parathyroid adenomas are almost always small and not palpable on exam but can be evaluated by neck ultrasonography or 99 m Tc-sestamibi scan. Surgical resection is the only curative treatment. In this case, a 16-year-old male presents with a 10-day history of nausea, vomiting, and headaches and is found to have a highly elevated calcium and parathyroid hormone level and a cerebral calcification in his frontal lobe noted on computed tomography. He had a palpable mass over the region of his left inferior parathyroid gland which was surgically resected with histopathology revealing a giant parathyroid adenoma. Giant parathyroid adenomas are exceptionally rare in children and adolescents and are more likely to present with severe hypercalcemic crisis than smaller adenomas. As early symptoms are often nonspecific, awareness of this clinical entity is important. There are several reports of basal ganglia calcifications in the setting of parathyroid adenoma, but, to our knowledge, this is the first report of a patient with frontal lobe calcification.
A Rare Clinical Variant of Oromandibular Limb Hypogenesis Syndrome Type I B
Authors: Ritesh Rambharos Kalaskar, Alkesh Godhane, Ashita Kalaskar, Swati Demble
Journal: Int J Clin Pediatr Dent (2016)
Keywords: aglossia, environmental factors, hypodactyli, microstomia, olhs, rudimentary ears
MeSH terms: Case Reports

Aglossia is a rare congenital malformation that often occurs as an isolated disorder or is observed in association with other congenital deformities, particularly limb defects. We present a unique case of a 7-year-old girl with aglossia, hypodactyli, rudimentary ears, retrognathic and V-shaped mandible. Her parental history revealed intrauterine exposure of medicines. The patient had problems in difficulty in eating, speech, taste sensation and hearing. The present case does not fit into Hall's classification of oromandibular limb hypogenesis syndrome (OLHS) which best describes hypoglossia and limb deformities. Therefore, the purpose of this article is to document the rare variant of OLHS which can be included in Hall's classification. How to cite this article: Kalaskar RR, Godhane A, Kalaskar A, Demble S. A Rare Clinical Variant of Oromandibular Limb Hypogenesis Syndrome Type I B. Int J Clin Pediatr Dent 2016;9(1):78-81.
Continuous Professional Oral Health Care Intervention Improves Severe Aspiration Pneumonia
Authors: Wakako Nawata, Yojiro Umezaki, Masahiro Yamaguchi, Masato Nakajima, Michiko Makino, Masahiro Yoneda, Takao Hirofuji, Takafumi Yamano, Hiroaki Ooboshi, Hiromitsu Morita
Journal: Case Rep Dent (2019)
MeSH terms: Case Reports

Professional oral health care (POHC) is known to prevent aspiration pneumonia in patients with dysphagia and/or those at the perioperative stage of surgery. However, the effect of POHC on patients suffering from aspiration pneumonia remains unknown. Here, we report a case where continual POHC intervention improved severe aspiration pneumonia. A 74-year-old male patient with a brain infarction suffered from severe aspiration pneumonia (PSI: IV, A-DROP: 3) complicated by vascular dementia and severe dysphagia. Because an antimicrobial approach following the treatment guidelines for pneumonia was not effective, we started a POHC intervention to improve his poor oral condition at the request of the attending doctor and the patient's family. The severe pneumonia markedly improved after continual POHC by the dental team. This case suggests that continual POHC intervention by a dental hygienist may improve severe aspiration pneumonia.
Management of hepatic epithelioid haemangio-endothelioma in children: what option?
Authors: K Sharif, M English, P Ramani, D Alberti, J-B Otte, P McKiernan, S Gosseye, M Jenney, J de Ville de Goyet
Journal: Br J Cancer (2004)
MeSH terms: Antineoplastic Combined Chemotherapy Protocols / therapeutic use, Hemangioendothelioma / drug therapy, Liver Neoplasms / drug therapy, Case Reports, Adolescent, Antineoplastic Agents, Alkylating / therapeutic use, Child, Child, Preschool, Female, Hemangioendothelioma / pathology, Hemangioendothelioma / surgery, Humans, Ifosfamide / therapeutic use, Liver Neoplasms / pathology, Liver Neoplasms / surgery, Liver Transplantation, Male, Neoplasm Recurrence, Local, Prognosis, Retrospective Studies, Antineoplastic Agents, Alkylating, Ifosfamide

Hepatic epithelioid haemangio-endothelioma (HEHE) is an endothelium-derived tumour of low-to-medium grade malignancy. It is predominantly seen in adults and is unresponsive to chemotherapy. Liver transplantation is an accepted indication when the tumour is unresectable. Hepatic epithelioid haemangio-endothelioma is very rare in children and results after transplantation are not reported. The aim of this study is to review the experience of three European centres in the management of HEHE in children. A retrospective review of all paediatric patients with HEHE managed in three European centres is presented. Five children were identified. Four had unresectable tumours. The first had successful resection followed by chemotherapy and is alive, without disease 3 years after diagnosis. One child died of sepsis and one of tumour recurrence in the graft and lungs 2 and 5 months, respectively, after transplant. Two children who had progressive disease with ifosfamide-based chemotherapy have had a reduction in clinical symptoms and stabilisation of disease up to 18 and 24 months after the use of platinum-based chemotherapy. HEHE seems more aggressive in children than reported in adults and the curative role of transplantation must be questioned. Ifosfamide-based chemotherapy was not effective. Further studies are necessary to confirm if HEHE progression in children may be influenced by platinum-based chemotherapy.
Case Report: A Rare Case of Autoinflammatory Phospholipase Cγ2 (PLCγ2)-Associated Antibody Deficiency and Immune Dysregulation Complicated With Gangrenous Pyoderma and Literature Review
Authors: Na Wu, Bingqing Zhang, Tao Wang, Min Shen, Xuejun Zeng
Journal: Front Immunol (2021)
Keywords: tnfα inhibitor, autoinflammatory disease, autoinflammatory phospholipase cγ2-associated antibody deficiency and immune dysregulation syndrome, gangrenous pyoderma, hyperimmunoglobulinemia e, phospholipase cγ2
MeSH terms: Immunologic Deficiency Syndromes / genetics, Phospholipase C gamma / genetics, Pyoderma Gangrenosum / genetics, Case Reports, Research Support, Non-U.S. Gov't, Review, Exons, Humans, Immunologic Deficiency Syndromes / complications, Immunologic Deficiency Syndromes / enzymology, Male, Mutation, Missense, Phenotype, Phospholipase C gamma / metabolism, Pyoderma Gangrenosum / complications, Young Adult, PLCG2 protein, human, Phospholipase C gamma

Background:
Autoinflammatory phospholipase Cγ2 (PLCγ2)-associated antibody deficiency and immune dysregulation (APLAID) is a rare autoinflammatory disease caused by gain-of-function mutations in the PLCG2 gene. Here we report a rare case of APLAID patient carrying a novel heterozygous missense PLCG2 I169V mutation with gangrenous pyoderma and concomitant high serum immunoglobulin (Ig) E level.
Methods:
The patient was diagnosed as APLAID and has been treated in our department. His phenotype and genotype were carefully documented and studied. We also conducted a comprehensive literature review on APLAID.
Results:
A 23-year-old Chinese Han man presented with recurrent fever for 18 years and vesiculopustular rashes for 9 years, along with chronic bronchitis, leukocytosis, increased C-reactive protein, immunodeficiency and high serum IgE. Skin biopsy showed chronic inflammatory cells infiltration. A paternal heterozygous missense variant in exon 6 of the PLCG2 gene p. I169V was identified. His vesiculopustular and IgE level responded to medium dose corticosteroids. After withdrawal of steroids, he developed severe arthritis and a large deteriorating ulceration resembling pyoderma gangrenosum on the left knee. Large dose corticosteroids were suboptimal. Then he received adalimumab with satisfactory response for arthritis and skin lesion. But he got an immunodeficiency-associated lymphoproliferative disorder 2 months later. Through literature review, there were a total of 10 APLAID patients reported by six English-language publications. Vesiculopustular rashes, sinopulmonary infection and immunodeficiency were the most frequent symptoms of APLAID patients. Glucocorticoids, intravenous immunoglobulin and biologics were clinically used to treat APLAID but none of these patients had a complete recovery.
Conclusions:
The rarity and diversity of APLAID make it difficult to be diagnosed. Our study reported the first case of APLAID with gangrenous pyoderma and concomitant high IgE carrying a novel PLCG2 mutation, which may expand the clinical phenotype and genotype of APLAID.
Reinduction of bevacizumab in combination with pegylated liposomal Doxorubicin in a patient with recurrent glioblastoma multiforme who progressed on bevacizumab/irinotecan
Authors: Mohammed Almubarak, Michael Newton, Ramin Altaha
Journal: J Oncol (2008)
MeSH terms: Case Reports

Glioblastoma multiforme (GBM) carries a dismal prognosis despite the current standard of multimodality treatments. Recent studies showed promising results to a regimen consisting of a VEGF inhibitor, (bevacizumab) and a topoisomerase I inhibitor (irinotecan) [BI] in recurrent GBM. However, those patients with GBM who progress on BI will succumb to their disease generally in a very short period of time. We report a case of a 56-year-old male patient with GBM who declined surgical resection and received chemoradiation with temozolomide. This treatment was withheld secondary to significant thrombocytopenia. Subsequently, he achieved stable disease for 10 months with a regimen consisting of thalidomide and tamoxifen before progressing. This was followed by bevacizumab with irinotecan [BI], for which he had a significant partial response for 8 months with subsequent progression. Reinducing the patient with bevacizumab in combination with a pegylated liposomal doxorubicin [PLD] (a topoisomerase II inhibitor) demonstrated antitumor activity with significant shrinkage of contrast enhancing mass and peritumoral edema.
Reported survival with severe mixed acidosis and hyperlactemia after toluene poisoning
Authors: Amr S Omar, Masood Ur Rahman, Said Abuhasna
Journal: Saudi J Anaesth (2011)
Keywords: lactic acidosis, hyperlactemia, toluene
MeSH terms: Case Reports

Lactic acidosis is a recognized complication of the inhalant abuse such as toluene, especially in patients with renal insufficiency. We report a case of severe metabolic acidosis and hyperlactemia due to toluene sniffing. The favorable outcome, despite extremely poor clinical symptoms, signs, laboratory and radiological findings, was unexpected. Specific aspects of the clinical course are addressed. Toluene sniffing should be considered in evaluating sever metabolic acidosis. Favorable outcome could be achieved with early diagnosis and proper interventions.
A Western Case of Intravascular Large B-Cell Lymphoma as Unusual Cause of Persistent Fever
Authors: Pier Paolo Piccaluga, Stefania Paolini, Cristina Campidelli, Nicola Vianelli, Luigi Bolondi
Journal: Case Rep Hematol (2019)
MeSH terms: Case Reports

Fever of unknown origin (FUO) is a common and challenging clinical condition that can be referred, among others, to infections, drug's effects, various inflammatory disorders, and cancers. Among the latter, lymphomas can indeed cause fever, which is therefore accounted as a lymphoma-related sign in patients' staging. Intravascular large B-cell lymphoma (IVLBCL) is a very rare tumor, characterized by lymphoma cell accumulation within sinusoids and, despite a very aggressive course, the evidence of this disease is scarce. Two variants are currently recognized, respectively, occurring in either Western (mainly characterized by neurological symptoms and skin involvement) or Eastern countries (with hemophagocytic syndrome, bone marrow, spleen, and liver involvement). We describe an atypical and unprecedented IVLBCL patient, presenting with pronounced features of Eastern cases as well as skin involvement. Due to the scant amount of neoplastic cells, the diagnosis was very challenging, with FUO being the first and for a certain time unique clinical sign. Although lymphoma was suspected, the lack of evidence for neoplastic cells delayed the final diagnosis. Eventually, only autopsy revealed the extensive involvement of different organs and tissues.
Pure sensory stroke from compression of putaminal haemorrhage: a case report
Authors: Daoming Tong
Journal: Cases J (2009)

Introduction:
The literature rarely describes putaminal haemorrhage producing pure spinothalamic sensory deficit. Here reports a case of putaminal haemorrhage in which selective impairment of the spinothalamic sensory modality was due to the compression of the hematoma.
Case presentation:
A 57 year old hypertensive man presented with a pure sensory stroke(PSS), and CT scan showed a putaminal haemorrhage. The clinical course was characterized by rapid resolution of the deficits.
Conclusion:
This case illustrates this rarely of PSS from compression of putaminal haemorrhage of good functional and vital prognosis, and stresses the value of CT scanning for diagnosis and prognosis.
Parry-Romberg Syndrome with Uhthoff's Phenomena: A Spectrum of Autoimmune Disease?
Authors: Samuel Asanad
Journal: Case Reports Immunol (2019)
MeSH terms: Case Reports

Parry-Romberg syndrome (PRS) is a rare disorder characterized by unilateral facial atrophy. Currently, the pathogenesis of PRS is poorly understood and no definitive treatment is available. This article reports the case of a 51-year-old woman with progressive hemifacial atrophy following herpes zoster infection, who presented with a concomitant chronic history of heat-induced diplopia. Magnetic resonance imaging showed unilateral cerebral white matter, periventricular, and medial longitudinal fasciculus lesions. The patient's diplopia resolved following treatment with valacyclovir. Infection has been previously considered as potential cause of PRS. However, herpes-induced PRS with ophthalmologic manifestations of Uhthoff's phenomena has not previously been reported. The present case suggests that PRS may possibly have an autoimmune etiology resembling that of multiple sclerosis.