OpenMed/multicare-articles huggingface dataset:
Case report: Scleromyxedema associated with a monoclonal gammapathy: Successful treatment with intravenous immunoglobulins
Authors: Shang-Shang Wang, Qin-Yi Chen, Lei-Hong Xiang
Journal: Front Immunol (2023)
Keywords: intravenous immunoglobulin therapy, monoclonal gammapathy, sclerodermoid disorders, scleromyxedema, treatment
MeSH terms: Exanthema, Paraproteinemias / complications, Paraproteinemias / diagnosis, Paraproteinemias / drug therapy, Scleromyxedema / diagnosis, Scleromyxedema / drug therapy, Case Reports, Female, Humans, Immunoglobulins, Intravenous / therapeutic use, Middle Aged, Rare Diseases, Thalidomide / therapeutic use, Immunoglobulins, Intravenous, Thalidomide

Scleromyxedema is a rare idiopathic fibromucinous disorder characterized by a generalized papular and sclerodermoid cutaneous eruption. Patients often have praraproteinemia and extracutaneous, even lethal, manifestations. Yet the prognostic and therapeutic features of scleromyxedema are poorly documented. High-dose intravenous immunoglobulin (IVIG), used either alone or in conjunction with systemic steroids and/or thalidomide, has been suggested as a first-line treatment. We report the case of a 45-year-old woman diagnosed with scleromyxedema with paraproteinemia that initially did not respond to systemic steroids, retinoids, and thalidomide but greatly improvement in terms of systemic and cutaneous symptoms after treatment with IVIG.
Case Report of Urethritis in a Male Patient Infected with Two Different Isolates of Multiple Drug-Resistant Neisseria gonorrhoeae
Authors: Lamiaa Al-Madboly, Shereen Gheida
Journal: Front Med (Lausanne) (2017)
Keywords: neisseria gonorrhoeae, arthritis, conjunctivitis, resistance, urethritis
MeSH terms: Case Reports

We report a brief description of a case suffering from bacterial urethritis, conjunctivitis, and arthritis, caused by two different isolates of multiple drug-resistant Neisseria gonorrhoeae. Initial diagnosis was dependent on the patient history, clinical findings, symptoms, and the bacteriological data. Polymerase chain reaction confirmed the identification of the pathogens. Random amplified polymorphic DNA revealed two different patterns. Susceptibility testing was performed using Kirby-Bauer disk diffusion method and the minimum inhibitory concentration was also determined. It revealed multiple drug resistance associated with β-lactamase production. Only gentamicin, rifampicin, and azithromycin were active against the test pathogens. A dual therapy was initiated using gentamicin as well as azithromycin to treat the possible co-infection with Chlamydia trachomatis. Complete recovery of the patient achieved with resolved symptoms a week later.
Prostate abscess in a paediatric patient: A rare diagnosis
Authors: Atish Vanmali, Mario Haines
Journal: SA J Radiol (2021)
Keywords: abscess, obstructive uropathy, paediatric, paediatric imaging, prostate
MeSH terms: Case Reports

Prostate abscess (PA) is an uncommon clinical manifestation that typically presents in the fifth to sixth decade of age and sporadically affects neonates. These characteristics, coupled with the atypical presentation, represent a clinical dilemma and a challenging diagnosis. A detailed history depicting the clinical course and the presence of risk factors is imperative to alert the clinician of the possibility of a PA. In this case report, we present a surgically confirmed PA, a diagnosis that is rarely encountered within the paediatric age group.
Alternative treatment for open bite Class III malocclusion in a child with Williams-Beuren syndrome
Authors: Giovanni Modesto Vieira, Eduardo Jacomino Franco, Denise Falcão Pinheiro da Rocha, Laudimar Alves de Oliveira, Rivadávio Fernandes Batista Amorim
Journal: Dental Press J Orthod (2015)
MeSH terms: Malocclusion, Angle Class III / therapy, Open Bite / therapy, Patient Care Planning, Williams Syndrome / complications, Case Reports, Anodontia / pathology, Cephalometry / methods, Child, Diastema / pathology, Diastema / therapy, Extraoral Traction Appliances, Follow-Up Studies, Humans, Macroglossia / pathology, Male, Maxilla / abnormalities, Orthodontic Appliance Design, Orthodontics, Interceptive / instrumentation, Palatal Expansion Technique / instrumentation, Rotation, Tooth Abnormalities / pathology

Williams-Beuren syndrome (WBS) is a rare genetic condition that affects approximately 1 in every 20,000 - 50,000 live births. WBS children have specific skeletal deformities, dental malformations and rare lingual muscle dysfunction. The need for orthodontic and orthognathic therapy has arisen and has been considered a real clinical challenge even for experienced professionals, once it requires a complex and individualized treatment plan. This study reports a case of orthopedic expansion of the maxilla, in which a modified facial mask was used for protraction of the maxillary complex associated with clockwise rotation of the maxilla. In addition, special considerations about treatment time and orthopedic outcomes are discussed.
Subtotal gastrectomy for gastric tube cancer using intraoperative indocyanine green fluorescence method
Authors: Ippei Yamana, Takuo Murakami, Shintaro Ryu, Jun Ichikawa, Yuki Shin, Nobuhiko Koreeda, Hiroto Sannomiya, Keisuke Sato, Tatsuya Okamoto, Yasuo Sakamoto, Yasushi Yoshida, Jun Yanagisawa, Tomoaki Noritomi, Suguru Hasegawa
Journal: Int J Surg Case Rep (2020)
Keywords: esophagectomy, gastric tube cancer, indocyanine green fluorescence
MeSH terms: Case Reports

Introduction:
Currently, the frequency of evaluating the flow of a reconstructed gastric tube using indocyanine green (ICG) fluorescence has been increasing. However, it has been difficult to decide on the operation method for patients with gastric tube cancer (GTC). We herein report a case in which ICG was effective in a patient with resection of GTC.
Presentation of case:
An 83-year-old man underwent subtotal esophagectomy with gastric tube reconstruction via the retrosternal route for esophageal cancer and right hemicolectomy for ascending colon cancer 16 years earlier. Postoperatively, the proximal part of the gastric tube had poor blood flow. Therefore, the patient underwent proximal-side resection of the gastric tube. Thereafter, free jejunal graft reconstruction was performed. The patient had not developed recurrence at that point. Recently, the patient visited the hospital complaining of nausea and chest discomfort. Upper gastrointestinal endoscopy revealed a type 0-IIa + IIc lesion located around the pylorus. A biopsy showed adenocarcinoma. Based on these findings, the patient was diagnosed with gastric tube cancer (cT1bN0M0StageI). The invasion depth of the cancer was predicted to be widespread submucosal invasion. Therefore, the patient underwent surgery. Intraoperatively, we evaluated the flow of the gastric tube after clamping the right gastroepiploic artery using ICG fluorescence. As a result, the flow of the gastric tube was deemed insufficient. Consequently, subtotal gastrectomy was performed with preservation of the right gastroepiploic artery via Roux-en-Y reconstruction.
Discussion:
ICG fluorescence is useful for evaluating the flow of the gastric tube helping to decide the operating method.
Conclusion:
We herein report a case of subtotal gastrectomy for GTC using intraoperative ICG fluorescence.
Osimertinib Resistance With a Novel EGFR L858R/A859S/Y891D Triple Mutation in a Patient With Non-Small Cell Lung Cancer: A Case Report
Authors: Yanli Yang, Xing Zhang, Ruixiao Wang, Jiayue Qin, Juan Wang, Zhimin Li, Xia Song
Journal: Front Oncol (2020)
Keywords: case report, circulating tumor dna, epidermal growth factor receptor, non-small cell lung cancer, osimertinib
MeSH terms: Case Reports

Targeted drug therapy based on the types of epidermal growth factor receptor (EGFR) gene mutations has been widely used in the diagnosis and treatment of patients with non-small cell lung cancer (NSCLC). With the development of next-generation sequencing (NGS) technology, more and more EGFR-tyrosine kinase inhibitor (TKI) resistance mutation sites have been revealed. Here, we report a novel EGFR L858R/A859S/Y891D triple mutation in plasma-derived circulating tumor DNA (ctDNA) was identified in a 53-year-old male patient with NSCLC resistant to osimertinib treatment, using an ultra-deep (20,000×) 160-gene panel through the NGS platform. Our case confirms that dynamic monitoring of liquid biopsy based on ctDNA is conducive to the selection of targeted therapy and the realization of the patient's full course management.
Single-port laparoscopic surgery for parasitic cystic teratoma of the greater omentum and bilateral ovarian teratomas: a case report
Authors: Akihiko Misawa, Eizo Kimura, Aiko Oka, Kiyono Osanai, Atsushi Suzuki
Journal: J Surg Case Rep (2023)
MeSH terms: Case Reports

Teratomas of extragonadal origin are extremely rare. The most common extragonadal site is the greater omentum. A 36-year-old woman was referred to our department for treatment of bilateral ovarian tumors. A 3- to 4-cm mass was observed in each ovary; bilateral ovarian mature cystic teratoma was diagnosed through imaging studies. The patient underwent single-port laparoscopic surgery, during which we found not only bilateral ovarian mature cystic teratomas but also an omental tumor macroscopically similar to the ovarian teratomas. The ovarian and omental teratomas were successfully removed through single-port laparoscopic surgery. The histopathological diagnosis of the omental tumor was parasitic mature cystic teratoma.
Hepatobiliary cystadenoma revealed by a jaundice: a case report
Authors: Taoufiq Harmouch, Marie-Pierre Vullierme, Alain Sauvanet, Valérie Paradis, Affaf Amarti
Journal: Case Rep Gastrointest Med (2011)
MeSH terms: Case Reports

Introduction. Hepatobiliary cystadenomas are rare benign cystic tumors and have a potential for recurrence and malignant transformation. The diagnosis may be very difficult because of absence of typical imaging feature in some cases. Case Presentation. In this paper, the authors discuss a 57-year-old woman who presented a jaundice related to hepatobiliary cystadenoma. Biological and radiological examinations have led to surgery, and the diagnosis is made after a histological examination of surgical specimens. Conclusion. This observation illustrates a hepatobiliary cystadenoma revealed by jaundice. Histology examination contributed to diagnosis. The authors discussed the mechanisms of biliary obstruction and differential diagnoses through a review of the literature.
Case Report: Successful Treatment of Steroid-Refractory Immune Checkpoint Inhibitor-Related Pure Red Cell Aplasia With Cyclosporin
Authors: Alexandre Gérard, Serena Romani, Elise Van-Obberghen, Audrey Fresse, Marine Muzzone, Nadège Parassol, Annick Boscagli, Fanny Rocher, Delphine Borchiellini, Milou-Daniel Drici
Journal: Front Oncol (2020)
Keywords: anemia, case report, cyclosporin, immune checkpoint inhibitor, nivolumab, pure red cell aplasia, steroid-refractory

Anemia associated with Immune checkpoint inhibitor (ICI) is usually hemolytic and regenerative. Cases of non-regenerative pure red cell aplasia are rare, and typically improve upon drug discontinuation and after corticotherapy. We herein report a case of nivolumab-related erythroblastopenia refractory to steroids in a melanoma patient that improved only after treatment with cyclosporin. Nivolumab had been well tolerated for 2 months after being introduced as an adjuvant treatment. Hemoglobin level then progressively decreased from 12.7 g/dl as baseline value to a nadir of 4.3 g/dL despite transfusion with a total of 29 packed red blood cells in 3 months. Extensive workup including repeated bone marrow examinations led to the diagnosis of pure red cell aplasia. Anemia persisted despite nivolumab discontinuation and over a month of corticotherapy, but improved dramatically 3 days after cyclosporin initiation and did not recur upon cyclosporin tapering. The patient remains cancer-free 9 months after nivolumab withdrawal. This case highlights the under-recognized risk of erythroblastopenia in patients treated with ICI and proves cyclosporin is a valid alternative for the treatment of steroid-refractory cases.
Dexmedetomidine-treated hyperventilation syndrome triggered by the distress related with a urinary catheter after general anesthesia: a case report
Authors: Junichi Saito, Erika Amanai, Kazuyoshi Hirota
Journal: JA Clin Rep (2017)
Keywords: dexmedetomidine, general anesthesia, hyperventilation syndrome

Background:
Hyperventilation syndrome (HVS) sometimes occurs in patients under stressful conditions and may provoke severe complications such as myocardial infarction and death. The authors report a case of HVS following general anesthesia, where a continuous intravenous infusion of dexmedetomidine was effective for HVS.
Case presentation:
A 23-year-old male patient with recurrent tongue cancer was scheduled to undergo partial glossectomy and neck dissection. Emergence from general anesthesia was prompt. Twenty-two minutes after extubation, the patient complained of unbearable distress caused by the urinary catheter. He began to cry, with an increased respiratory rate of over 40 breaths per minute. Intravenous infusion of flurbiprofen, droperidol, and morphine was not effective. Electrocardiography and laryngofiberscopy indicated the absence of acute coronary syndrome and airway obstruction, respectively. An arterial blood gas determination showed pH 7.63, PaCO2 18.2 mmHg, PaO2 143 mmHg on O2 mask 4 L/min, Ca2+ 4.29 mmol/L, and lactate 3.4 mmol/L. The patient was diagnosed with HVS. Dexmedetomidine infusion 2.0 μg/kg/h for 10 min followed by 0.7 μg/kg/h reduced respiratory rate, suppressed arousal, and disappeared the complaint of bladder distension. One hour after extubation, an arterial blood gas determination showed pH 7.33, PaCO2 51.3 mmHg, PaO2 196 mmHg on O2 mask 4 L/min, Ca2+ 4.70 mmol/L, and lactate 1.5 mmol/L. After admission to the intensive care unit, dexmedetomidine infusion was maintained at the rate of 0.2 to 0.7 μg/kg/h until the following morning, and he did not complain of distress caused by the urinary catheter.
Conclusions:
HVS can occur after emergence from general anesthesia, and dexmedetomidine infusion was effective for HVS.
A case report of wandering spleen with pedicle torsion and splenic infarction being misdiagnosed as organ inversion complicated with acute appendicitis
Authors: Shengjie Zhao, Yindi Wang, Zhiheng Wan, Hancheng Chen, Xinyu Zhao, Ruibin Li
Journal: Front Surg (2022)
Keywords: acute abdomen, diagnosis, pedicle torsion, treatment, wandering spleen
MeSH terms: Case Reports

Wandering spleen is a rare disease that is easily misdiagnosed. When combined with splenic pedicle torsion and even splenic infarction, wandering spleen is a rare and critical cause of surgical acute abdomen. We report an 18-year-old male patient with abdominal organ inversion diagnosed as acute appendicitis before operation. Laparoscopic exploration confirmed wandering spleen with splenic pedicle torsion led to splenic infarction and was complicated by appendicitis. He was treated with laparoscopic appendectomy and abdominal splenectomy. The patient recovered well after the operation and was discharged from the hospital in 7 days. During the 4-year follow-up, there was no report of complicated infections such as pneumonia or sepsis.
Late presentation of hyperandrogenism in pregnancy: clinical features and differential diagnosis
Authors: Gautam Das, Vinay S Eligar, Jyothish Govindan, D Aled Rees
Journal: Endocrinol Diabetes Metab Case Rep (2013)

Background:
Hyperandrogenic states in pregnancy are rare but arise most commonly due to new-onset ovarian pathology in pregnancy. We describe the case of a young woman who presented in the latter half of her pregnancy with features of hyperandrogenism. We review the biochemical and imaging findings and discuss the differential diagnosis.
Case presentation:
A 26-year-old woman presented in the later part of her pregnancy with widespread hirsutism. Biochemical testing confirmed hyperandrogenism (testosterone, 13.7 nmol/l and second-trimester pregnancy range, 0.9-4.9 nmol/l), although she had no history of menstrual disturbance, hirsutism or acne prior to conception. Radiological evaluation (ultrasound and magnetic resonance imaging) revealed multiple cystic lesions in both ovaries, leading to a presumptive diagnosis of hyperreactio luteinalis (HL). The implications of maternal hyperandrogenism on foetal virilisation were considered and the patient was counselled appropriately. She delivered a healthy baby boy uneventfully. Androgen levels, hirsutism and acne normalised within a few weeks of delivery.
Conclusion:
HL can occur at any stage of pregnancy and is an important differential diagnosis in pregnant patients with features of androgen excess. Most cases regress spontaneously after delivery and major interventions are usually not needed.
Learning points:
Hyperandrogenism in pregnancy is rare.Clinical features are similar to the non-pregnant state in the mother but virilisation in the foetus can have profound consequences.HL and pregnancy luteoma are the most common ovarian pathologies leading to hyperandrogenism in pregnancy.Spontaneous regression occurs in the post-partum period in the vast majority of cases and surgery is only required for local complications.
Bilateral renal mucormycosis following COVID-19 infection: A therapeutic challenge
Authors: Rajasekaran Kishor Kumar, Rajeev A Annigeri, Ram Gopalakrishnan, Sunil S Kaveripattu, Nitesh Jain
Journal: Clin Nephrol Case Stud (2022)
Keywords: covid-19, amphotericin, renal mucormycosis
MeSH terms: Case Reports

India witnessed an epidemic of mucormycosis during the second wave of the COVID-19 pandemic. Renal mucormycosis has been reported rarely, mostly from India, but only 2 cases have been reported following COVID-19 infection to date. We report a case of mucormycosis predominantly affecting kidneys in a young and previously healthy male following COVID-19 pneumonia, for which he had received corticosteroid, remdesivir, and tocilizumab. He presented with hematuria, progressive oliguria, and severe acute kidney injury (AKI) requiring dialysis. The diagnosis was made on kidney biopsy and contrast-enhanced CT (CECT) showed segmental and subsegmental renal artery pseudoaneurysms with distal occlusion of both kidneys. He underwent bilateral nephrectomy and received high-dose amphotericin (AMB) and posaconazole. He developed cardiac arrhythmia and pulmonary edema attributed to AMB-related cardiotoxicity after a cumulative ABM dose of 2,450 mg. This is the first case report describing the survival of a patient with bilateral renal mucormycosis following COVID-19 infection. Our case report highlights the importance of considering mucormycosis in a patient with post-COVID-19 AKI to make an early diagnosis and aggressive management comprising of surgical debridement and high-dose AMB to improve survival.
Anlotinib for the Treatment of Multiple Recurrent Lumbar and Sacral Cord Hemangioblastomas: A Case Report
Authors: Nan Jin, Chunxiao Sun, Yijia Hua, Xinyu Wu, Wei Li, Yongmei Yin
Journal: Front Oncol (2022)
Keywords: anlotinib, anti-angiogenesis, case report, hemangioblastoma, tyrosine kinase inhibitor (tki)
MeSH terms: Case Reports

Background:
Hemangioblastoma (HB) is a rare and highly vascularized tumor that originates from the central nervous system as well as other part of the body. They can appear sporadically or as part of von Hippel-Lindau (VHL) disease, a rare hereditary cancer syndrome. Although surgery can cure the majority of HBs, the disease shows a treatment-refractory challenge upon recurrence. HBs express a high amount of vascular endothelial growth factor (VEGF) which is responsible for angiogenesis and subsequently tumor progression. Anti-angiogenic treatment like bevacizumab has showed effect on HB, so we hypothesized that anlotinib could trigger HB regression via its inhibitory effect on VEGF.
Case presentation:
We will share our experience in treating a 62-year-old woman with multiple recurrent lumbar and sacral cord HBs. She was treated with anlotinib (8mg qd d1-14, q3w) for three months and her follow up radiological examination demonstrated marked tumor regression which was evaluated as having partial response pursuant to RECIST 1.1 system. She is currently still receiving treatment of anlotinib orally and the lesions continuously reduced.
Conclusion:
We have reported that anlotinib can cause significant radiographic response in a patient with multiple recurrent lumbar and sacral cord HBs for the first time. This might enable a novel therapeutic approach for patients with multiple recurrent HB or those with multiple lesions such as in VHL disease which are difficult to resect surgically.
Dysgerminoma with Estrogen-Producing Functioning Stroma Presenting Precocious Puberty
Authors: Shunsuke Nagase, Kanako Ogura, Karin Ashizawa, Nana Nakazawa-Tanaka, Masahiko Urao, Masaharu Fukunaga, Yuto Yamazaki, Hironobu Sasano, Toshiharu Matsumoto
Journal: Case Rep Pathol (2021)
MeSH terms: Case Reports

Dysgerminoma is a malignant ovarian germ cell tumor, and unlike sex-cord stromal tumors, endocrine manifestation is considered rare. Here, we report the first case of dysgerminoma presenting precocious puberty. The patient is a 7-year-old girl who presented with a breast development in Tanner stage 3. Serum estradiol (E2) was markedly elevated while luteinizing hormone (LH) and follicle-stimulating hormone (FSH) were suppressed below the detection limit. Microscopically, the right ovarian mass displayed nests of large polygonal cells and fibrous septa which were focally concentrated by theca-like plump spindle cells. Immunohistochemistry revealed that the spindle cells expressed various steroidogenic enzymes involved in estrogen biosynthesis including P450 aromatase. The tumor was diagnosed with pure dysgerminoma with estrogen-producing functioning stroma. After the operation, serum E2 declined below the detection limit; LH and FSH returned within the normal range. This case demonstrates that even a conventional dysgerminoma can present endocrine manifestation through functioning stroma.
Penile Urethral Hypospadias with Two Fistulae and Diverticulum in a Saanen Kid
Authors: Areeg Mohamed Almubarak, Rihab Mohamed Abdelghafar, Ahmed Abdelrahim Gameel, Nuha Muatasim Osman
Journal: Case Rep Vet Med (2016)
MeSH terms: Case Reports

Hypospadias is a rare congenital defect reported in most animal species and humans. This case study reports a hypospadiac case in a goat kid with urethral diverticulum diagnosed in Sudan for the first time. A 45-day-old male kid was presented to the Veterinary Teaching Hospital, College of Veterinary Medicine, Sudan University of Science and Technology (SUST), with a history of an increasing prescrotal swelling. At presentation in the clinic the kid was bright and alert and the vital parameters were within the normal physiological range. Ultrasonography was performed to assess the integrity of the urinary system using (3.5-5) MHz curvilinear probe and it revealed normal kidneys and a distended urinary bladder. The kid was referred to surgery and two hypospadiac urinary fistulae were found. One fistula was sutured and the other was not corrected due to catheterization failure.
Severe acidosis due to 5-oxoprolinase inhibition by flucloxacillin in a patient with shoulder prosthesis joint infection
Authors: Julia Elisabeth Lenz, Volker Alt, Thomas Dienemann
Journal: J Bone Jt Infect (2022)
MeSH terms: Case Reports

We report a case of a 64-year-old female patient with severe metabolic acidosis. Inhibition of 5-oxoprolinase by flucloxacillin was found to be the cause of the metabolic derailment.