OpenMed/multicare-articles huggingface dataset:
Clinical features of atypical odontalgia; three cases and literature reviews
Authors: Miho Takenoshita, Anna Miura, Yukiko Shinohara, Rou Mikuzuki, Shiori Sugawara, Trang Thi Huyen Tu, Kaoru Kawasaki, Takeru Kyuragi, Yojiro Umezaki, Akira Toyofuku
Journal: Biopsychosoc Med (2017)
Keywords: antidepressants, aripiprazole, atypical odontalgia, augmentation, persistent dento-alveolar pain disorder
MeSH terms: Case Reports

Background:
Atypical odontalgia (AO) is a disease characterized by continuous pain affecting the teeth or tooth sockets after extraction in the absence of any identifiable cause on clinical or radiographic examination. Antidepressants, such as amitriptyline, are reported to be effective in the treatment of AO; however, their efficacy varies depending on the case. In this article, we report three types of AO and discuss its heterogeneity and management.
Case presentation:
In the first case, a 58-year-old woman presented with a heavy, splitting pain in the four maxillary front post-crown teeth, as if they were being pressed from the side. Her symptoms abated with 20 mg of amitriptyline. In the second case, a 39-year-old woman presented with a feeling of heaviness pain on the right side of maxillary and mandibular molar teeth, face, whole palate, and throat. She was unable to function because of her pain. Her symptoms drastically subsided with 3 mg of aripiprazole. In the third case, a 54-year-old woman presented with a tingling sensation on the left mandibular second premolar and first molar, and an uncomfortable feeling on her provisional prosthesis that made it unbearable to keep the caps on. Her symptoms diminished with 2 mg of aripiprazole added to 30 mg of mirtazapine.
Conclusions:
AO shows various features and responses to drugs. It is considered not only a purely sensory problem, but also a considerably complex psychological problem, such as rumination about the pain. Investigating the difference in pharmacotherapeutic responses might help to advance the treatment of AO.
Intramuscular Hemangioma of Thyrohyoid Muscle
Authors: Ramesh Parajuli, Suman Thapa, Sushna Maharjan
Journal: Case Rep Otolaryngol (2016)

Intramuscular hemangiomas are rare benign vascular neoplasms. IMH accounts for less than 1% of all hemangiomas. These neoplasms commonly occur in trunk and extremities but are rare in head and neck region. The present case is a 17-year-old female patient, who presented with a painless, slowly enlarging mass in left sided upper neck for 4 years. Investigations were suggestive of vascular neoplasm. She underwent excision of the mass in toto under general anesthesia. Postoperative period was uneventful. Histopathological examination of the mass revealed it as mixed type of intramuscular hemangioma. She did not have any signs of recurrences on her last follow-up at 6 months postoperatively. This case report discusses the rare IMH arising from thyrohyoid strap muscle.
Facial Pain Associated with CPAP Use: Intra-Sinusal Third Molar
Authors: Maxime Mermod, Martin Broome, Remy Hoarau, Daniel Zweifel
Journal: Case Rep Otolaryngol (2014)

Objective. This paper describes a patient with left hemifacial pain elicited by the use of a CPAP mask. Case Report. A 74-year-old man was referred with a history of pain in the left maxillary sinus related to the use of his CPAP interface, thereby prohibiting the use of the latter. Computed tomography revealed an intra-sinusal ectopic third molar in the left maxillary sinus floor corresponding to the painful area. After removal of the ectopic tooth under local anesthesia by a Caldwell-Luc approach, the patient was relieved of his symptoms. Conclusion. Although an ectopic tooth in the maxillary sinus is rare, this case points out the importance of actively looking for a regional problem if patients cannot tolerate the CPAP interface since this can lead to issues of incompliance and medical complications due to the untreated obstructive sleep apnoea syndrome.
Immune reconstitution, glomerulonephritis, and successful treatment with rituximab
Authors: George Vasquez-Rios, John C Edwards, Saketh Tummala, Ashley Chapel, Ramez Sunna, David S Brink, Christopher Laohathai, Thanh-Mai Vo
Journal: Clin Nephrol Case Stud (2020)
Keywords: autoimmunity, glomerulonephritis, immune restoration, multiple sclerosis, reconstitution syndrome, vasculitis
MeSH terms: Case Reports

Background:
Alemtuzumab can induce secondary autoimmunity affecting multiple organs. While kidney involvement is uncommon, it can be associated with devastating forms of glomerulonephritis (GN).
Case presentation:
A 32-year-old African American woman presented with hypertension, proteinuria, and progressive renal failure. Her medical history was remarkable for secondary progressive multiple sclerosis (SPMS). She had received her first induction dose of alemtuzumab 1 year prior to presentation. Upon evaluation, she had scanning speech, multidirectional nystagmus, and mild edema. Her serum creatinine was 2 mg/dL. Urine studies revealed proteinuria and microscopic hematuria. Her serologic tests were positive for c-antineutrophil cytoplasmic antibodies (> 1 : 640). In addition, she was found to have new-onset severe thyroid dysfunction with antibodies against thyroglobulin and thyroid peroxidase. Kidney biopsy was diagnostic for pauci-immune crescentic GN. The patient was treated with methylprednisolone and rituximab with subsequent renal, thyroid, and neurological recovery.
Conclusion:
This is an atypical case of GN following therapy with alemtuzumab. We hypothesize that immune reconstitution may be a potential mechanism. Alemtuzumab is a new treatment for SPMS that can be associated with GN. Practice guidelines should address the management of its renal complications.
Sensorineural hearing loss and mild cardiac phenotype caused by an EYA4 mutation
Authors: Satoko Abe, Hidehiko Takeda, Shin-Ya Nishio, Shin-Ichi Usami
Journal: Hum Genome Var (2018)
MeSH terms: Case Reports

EYA4 is a member of the vertebrate eya gene family of transcriptional activators and plays several roles in both embryonic and inner ear development. The majority of EYA4 gene mutations are associated with autosomal dominant non-syndromic hearing loss (DFNA10). In addition, some mutations in this gene cause autosomal dominant syndromic hearing loss with dilated cardiomyopathy. EYA4 is a rare cause of sensorineural hearing loss, and only a limited number of papers regarding mutations in this gene have been published. Thus, detailed clinical features remain unclear. We conducted next-generation sequencing of a Japanese individual with progressive sensorineural hearing loss and identified an EYA4 pathogenic variant. Pure-tone audiometry revealed bilateral, nearly symmetric, moderate sensorineural hearing loss in the low and middle frequencies. Minor abnormalities were observed on the patient's electrocardiogram and echocardiography without any apparent symptoms. Next-generation sequencing is effective in elucidating the etiology of hearing loss, and the present findings suggested the possible phenotypic expansion of deafness caused by EYA4 gene mutations.
Severe fetal intracranial hemorrhage: Congenital Cytomegalovirus infection may play a role? A case report and review of literature
Authors: Letizia Capasso, Clara Coppola, Maria Vendemmia, Serena Salomè, Valentina Esposito, Chiara Colinet, Carolina Porfito, Francesco Raimondi
Journal: IDCases (2021)
Keywords: cmv infection, cytomegalovirus, intracranial fetal hemorrhage, neonate, prenatal diagnosis
MeSH terms: Case Reports

Cytomegalovirus (CMV) is the most common cause of congenital infection, with a wide spectrum of clinical manifestations and different grade of severity. We report the case of a male baby born at term with an early prenatal diagnosis of severe intracranial hemorrhage (ICH), with no other evident risk factors. Urine and blood sample were tested for CMV-DNA, and diagnosis of congenital CMV infection was established. This case describes intracranial hemorrhage as uncommon although possible sign of early fetal CMV infection. Considering that pathogenic factors cannot be defined in 25 % of term neonates with ICH, this case report highlights the importance of CMV screening in pregnant women and in term infants with prenatal ICH of unknown origin.
Case report: A rare case of cerebral herniation during glioma resection in a syphilis-positive patient
Authors: Han Wang, Qianli Lin, Fang Wang, Yong Yi, Xiaoping Xu, Jingcheng Jiang, Qingshan Deng
Journal: Front Neurol (2023)
Keywords: cerebral herniation, craniotomy, glioma, syphilis, vasculitis
MeSH terms: Case Reports

Acute intraoperative cerebral herniation is catastrophic in craniotomy and seriously affects the outcomes of surgery and the prognosis of the patient. Although the probability of its occurrence is low, it can lead to severe disability and high mortality. We describe a rare case of intraoperative cerebral herniation that occurred in a syphilis-positive patient. The patient was diagnosed with both glioma and syphilis. When the glioma was completely removed under the surgical microscope, acute cerebral herniation occurred. An urgent intervention in cerebral herniation identified a collection of colorless, transparent, and protein-rich gelatinous substances rather than a hematoma, which is a more commonly reported cause of intraoperative cerebral herniation in the literature. We have found no previous descriptions of such cerebral herniation during craniotomy in a patient with syphilis and glioma. We suspected that the occurrence of intraoperative cerebral hernia might be related to the patient's infection with syphilis. We considered the likelihood of an intraoperative cerebral herniation to be elevated when a patient had a disease similar to syphilis that could cause increased vascular permeability.
Protracted Hypocalcemia following 3.5 Parathyroidectomy in a Kidney Pancreas Recipient with a History of Robotic-Assisted Roux-en-Y Gastric Bypass
Authors: Hugo Bonatti, Naureen Iqbal, Catherine Kling, Willie Melvin, James Broome
Journal: Case Rep Transplant (2018)
MeSH terms: Case Reports

Background:
Hypocalcemia is a frequent complication of parathyroidectomy for secondary/tertiary hyperparathyroidism. In patients with a history of prior Roux-en-Y gastric bypass (RYGBP), changes in nutritional absorption make management of hypocalcemia after parathyroidectomy difficult.
Case report:
A 41-old-year morbidly obese female with c-peptide negative diabetes mellitus and renal failure had RYGBP. Following significant weight loss she underwent simultaneous pancreas-kidney transplantation. She had excellent transplant graft function but developed tertiary hyperparathyroidism with calciphylaxis. She underwent resection of 3.5 glands leaving a small, physiologic remnant remaining in situ at the left inferior position. She was discharged on postoperative day one in good condition, asymptomatic with serum calcium of 7.6 mg/dL and intact PTH of 12 pg/mL. The patient had to be readmitted on postoperative day #14 for severe hypocalcemia of 5.0 mg/dl and ionized calcium 2.4 mg/dl. She required intravenous calcium infusion to achieve calcium levels of >6.5 mg/dl. Long-term treatment includes 5 g of elemental oral calcium TID, vitamin D, and hydrochlorothiazide. She remains in the long term on high-dose medical therapy with normal serum calcium levels and PTH levels around 100 pg/mL.
Discussion:
Our patient's protracted hypocalcemia originates from a combination of 3.5 gland parathyroidectomy, altered intestinal anatomy post-RYGBP, and potentially her pancreas transplant causing additional metabolic derangement. Alternative bariatric procedures such as sleeve gastrectomy may be more suitable for patients with renal failure or organ transplants in whom adequate absorption of vitamins, minerals, and drugs such as immunosuppressants is essential.
Portal Hypertensive Biliopathy Presents with Massive Bleeding during ERCP after Balloon Sphincteroplasty in a Noncirrhotic Saudi Sickler Patient
Authors: Ahmad M Al-Akwaa, Mohammed Elsadig, Ahmed E Al-Fayaa, Mohja D Al-Shehri
Journal: Case Rep Med (2017)
MeSH terms: Case Reports

Portal hypertensive biliopathy (PHB) is described as abnormalities of the walls of the biliary tree secondary to portal hypertension. Gastrointestinal bleeding caused by PHB is rare. PHB as a cause of serious bleeding after sphincteroplasty during ERCP is extremely rare. Here, we report a case of PHB in a young Saudi male with cell sickle anemia who developed massive hemorrhage during ERCP after balloon dilation of the ampulla of Vater. We further discussed the diagnosis and management. To the best of our knowledge, no such case has been reported.
Complications using bioabsorbable cross-pin femoral fixation: a case report and review of the literature
Authors: Saqib Hasan, Samir Nayyar, Ikemefuna Onyekwelu, Kunal Kalra, Soterios Gyftopoulos, Laith M Jazrawi
Journal: Case Rep Radiol (2011)
MeSH terms: Case Reports

The use of bioabsorbable cross-pin transcondylar fixation has remained a viable option for femoral fixation in anterior cruciate ligament reconstruction. Although numerous biomechanical studies have demonstrated high fixation strength and minimal slippage with use of this method of fixation, there have been increasing reports of a variety of clinical complications associated with these implants. We reviewed the literature for all complications associated with the Bio-TransFix implant and present a case report of a patient status after ACL reconstruction using Bio-TransFix cross-pin femoral fixation with iliotibial band friction syndrome from a broken cross-pin four month post-operatively.
Segmental infantile hemangioma with milia: A case report
Authors: Elena Pastukhova, Nordau Kanigsberg
Journal: SAGE Open Med Case Rep (2023)
Keywords: milia, infantile hemangioma, milia-like cysts, segmental hemangioma
MeSH terms: Case Reports

Milia are common in children. They are small, keratinizing cysts that arise either as primary epidermoid cysts or secondary to other dermatoses, trauma or certain medications. In the paediatric population, milia are most frequently congenital and resolve spontaneously. Infantile hemangiomas are relatively common in neonates. They typically arise within the first few weeks of life, undergo a proliferative phase in the first 6 months, then begin to involute at around 12 months of age. After involution, residual skin changes may be observed, such as telangiectasia, fibrofatty tissue and redundant skin. However, there is a gap in the literature regarding concomitant milia and infantile hemangiomas. We report a case of a 5-months-old female who presented with a large, segmental infantile hemangioma of the posterior neck with milia.
Giant bilateral angiomyolipomas with spontaneous hemorrhage and inferior vena cava thrombosis in a patient with tuberous sclerosis
Authors: Meriem Menany, Safae Khnaba, Bouchaib Radouane, Mohamed Jidal, Touriya Amil, Rachida Saouab
Journal: Pan Afr Med J (2017)
Keywords: bilateral angiomyolipomas, fat thrombosis, hemorrhage
MeSH terms: Angiomyolipoma / diagnosis, Kidney Neoplasms / diagnosis, Vena Cava, Inferior / pathology, Venous Thrombosis / etiology, Case Reports, Adult, Angiomyolipoma / complications, Angiomyolipoma / diagnostic imaging, Female, Hemorrhage / etiology, Humans, Kidney Neoplasms / complications, Kidney Neoplasms / diagnostic imaging, Retroperitoneal Space / pathology, Tomography, X-Ray Computed, Tuberous Sclerosis / pathology, Vena Cava, Inferior / diagnostic imaging, Venous Thrombosis / diagnostic imaging

Renal angiomyolipomas are rare type of benign renal neoplasm. They are composed of vascular, smooth and fat elements and can be associated to phacomatosis as Tuberous Sclerosis disease. Symptomatic presentation is most frequently spontaneous retroperitoneal hemorrhage, which can be fatal. The risk of bleeding is proportional to the size of the lesion (>4 cm of diameter). Typical angiomyolipomas are benign but may have alarming properties: nuclear pleomorphism and mitotic activity, extension into the vena cava, and spread to regional lymph nodes without malignant progression. We report a Computed Tomography finding of a rare giant bilateral angiomyolipomas with spontaneous hemorrhage and inferior vena cava thrombus in a patient with tuberous sclerosis, emphasizing the importance of imagery in the positive and etiologic diagnosis.
Pelvic Inflammatory Disease: Possible Catches and Correct Management in Young Women
Authors: Chiara Di Tucci, Daniele Di Mascio, Michele Carlo Schiavi, Giorgia Perniola, Ludovico Muzii, Pierluigi Benedetti Panici
Journal: Case Rep Obstet Gynecol (2018)
MeSH terms: Case Reports

The incidence of adnexal masses increases exponentially with age and the most frequent causes in young women are physiologic cysts and pelvic abscesses with pelvic inflammatory disease (PID). Clinical examination can direct physicians to an appropriate management of adnexal mass, but the role of transvaginal ultrasound is crucial for diagnosis and treatment decision, even if it sometimes can be misleading, especially in young women. Ca 125, blood count, and CRP are useful to clarify suspected etiology of a pelvic mass, but specificity and positive predictive value are low because elevation of laboratory tests may occur in several benign conditions. In our work we present four cases of suspected pelvic masses. Despite guidelines for management of PID, the right timing to switch to surgical therapy is not clear. Therefore, the treatment decision should be based on a careful evaluation of various parameters such as signs symptoms and above all age. Moreover, we believe that, for a correct diagnosis and for the best fertility sparing treatment, it is also extremely important to refer to a gynecological oncology unit with an expert surgeon.
Achieving Lingualized Balanced Occlusion in a Fixed-Removable Rehabilitation for a Maxillary Complete and Mandibular Kennedy Class II Case
Authors: Ibrahim Tulunoglu, Samuel Cohen
Journal: Case Rep Dent (2019)
MeSH terms: Case Reports

In this case report, a method to achieve an adequate compensating curve and bilateral balanced lingualized occlusion in a case requiring maxillary complete denture and mandibular Kennedy Class II removable partial and fixed prosthodontic rehabilitation is described.
Case Report: Combined pembrolizumab, 5-fluorouracil, and cisplatin therapy were remarkably effective in p16-positive squamous cell carcinoma of unknown primary
Authors: Risako Suzuki, Kazuyuki Hamada, Ryotaro Ohkuma, Mayumi Homma, Toshiaki Tsurui, Nana Iriguchi, Tomoyuki Ishiguro, Yuya Hirasawa, Hirotsugu Ariizumi, Yutaro Kubota, Atsushi Horiike, Kiyoshi Yoshimura, Satoshi Wada, Toshiko Yamochi, Takuya Tsunoda
Journal: Front Oncol (2023)
Keywords: 5-fluorouracil, pd-1 antibody, cancer of unknown primary, cisplatin, p16, pembrolizumab, squamous cell carcinoma
MeSH terms: Case Reports

Background:
Cancer of unknown primary (CUP) is a malignant tumor without a known primary lesion with a frequency of 3-5%. It can be divided into favorable and unfavorable prognosis subsets. While recommended treatments are available for the former group, there is no established treatment for the latter. Here, we report the effective treatment of a 32-year-old woman with p16-positive squamous cell CUP with pembrolizumab plus 5-fluorouracil and cisplatin therapy.
Case presentation:
A 32-year-old woman presented with metastatic lesions in the liver, lung, bone, cervical region, abdominal region, and pelvic lymph nodes. She was diagnosed with p16-positive squamous cell carcinoma of unknown primary origin. The patient received pembrolizumab plus 5-fluorouracil and cisplatin therapy, which markedly reduced the metastasis and improved her Eastern Cooperative Oncology Group performance status after two courses.
Conclusion:
This case report highlights the potential of pembrolizumab plus 5-fluorouracil and cisplatin therapy for treating CUP with an unfavorable prognosis. p16 positivity is worth examining for squamous cell carcinoma of unknown primary origin, and if present, this therapy should be considered a promising treatment option.
Clinical and Genetic Findings of the First Report of PAPA Syndrome in Brazil
Authors: Sérgio Júlio Fernandes, Maria Isabel Valdomir Nadaf, Nauro Hudson Monteiro, Izabel Nazira Nadaf, Cleiton Ribeiro Lélis, Bianca Yumi Takano, Bárbarah Gabriella de Camargo Monteiro, Nyvea Gabriella de Camargo Monteiro, Olga Akiko Takano, Leonardo Oliveira Mendonça
Journal: Case Reports Immunol (2021)
MeSH terms: Case Reports

Background:
PAPA syndrome (MIM #604416) is a rare monogenic autoinflammatory disease genetically transmitted in an autosomal dominant trait that results from missense mutations in the proline-serine-threonine phosphatase-interactive protein 1 (PSTPIP1) gene located on chromosome 15 and is characterized by sterile pyogenic arthritis, pyoderma gangrenosum, and cystic acne. We describe the clinical and molecular findings of two related Brazilian patients with PAPA syndrome. Case Presentation. A 7-year-and-3-month-old boy with nonconsanguineous parents had had recurrent pyoarthritis since the age of 5 years and 8 months. During his last and long hospitalization, the lack of improvement with antibiotics, evidence of increased inflammatory activity, repeated arthrotomies, draining purulent fluid that had negative cultures, and the history of trauma, all on in a clinical background of pyoarthritis, led to the suspicion of an autoinflammatory syndrome. This was confirmed by the good clinical response to corticotherapy. Genetic sequencing confirmed the diagnosis of PAPA syndrome, with the pathogenic mutation c.688 G > A (p. Ala230Thr) in the PSTPIP1 gene present in the patient and in the mother.
Conclusions:
This case illustrates that in children with recurrent/recalcitrant sterile recurrent pyogenic arthritis/osteomyelitis, the possibility of an underlying immunological condition should be considered. In both, recurrent infections or recurrent inflammation, many genes involved in the inborn errors of immunity can be associated, and a correct and precocious diagnosis is necessary to avoid mobility and mortality. To the best of our knowledge, this is the first report of PAPA syndrome in Brazil.
Pernicious Anemia in an Adult with Trisomy 21
Authors: Kentaro Kamada, Osamu Kawano, Satoshi Yakuwa, Kentaro Wakasa, Kimiaki Uetake
Journal: Case Reports Immunol (2023)
MeSH terms: Case Reports

Pernicious anemia is an autoimmune disease caused by the malabsorption of vitamin B12. It usually appears in the elderly. People with trisomy 21 are susceptible to autoimmune diseases. This susceptibility is thought to be due to altered expression of the AIRE gene, which is located in the 21q22.3 region. Although pernicious anemia is not common in people with trisomy 21, AIRE is pointed out as a susceptibility gene of pernicious anemia in a genome-wide association study. Here, we report a man with trisomy 21, who suffered from the pernicious anemia. When he was in his 30 s, he visited our hospital because of diarrhea and poor oral intake. He showed thrombocytopenic purpura-like features, and was diagnosed as pernicious anemia. After supplementation of vitamin B12, he recovered from the illness. The reason for his early onset may be because of trisomy 21. Altered expression of AIRE might trigger the disease.