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Case report: quantification of methadone-induced respiratory depression using toxicokinetic/toxicodynamic relationships
Authors: Bruno Mégarbane, Xavier Declèves, Vanessa Bloch, Christophe Bardin, François Chast, Frédéric J Baud
Journal: Crit Care (2007)
MeSH terms: Methadone / blood, Naloxone / administration & dosage, Narcotic Antagonists / administration & dosage, Narcotics / blood, Respiratory Insufficiency / physiopathology, Case Reports, Adult, Chromatography, High Pressure Liquid, Drug Monitoring, Humans, Infusions, Intravenous, Isomerism, Male, Methadone / poisoning, Narcotics / poisoning, Poisoning / blood, Poisoning / drug therapy, Poisoning / physiopathology, Respiration, Respiratory Insufficiency / blood, Respiratory Insufficiency / chemically induced, Narcotic Antagonists, Narcotics, Naloxone, Methadone

Introduction:
Methadone, the most widely delivered maintenance therapy for heroin addicts, may be responsible for life-threatening poisonings with respiratory depression. The toxicokinetics and the toxicokinetic/toxicodynamic (TK/TD) relationships of methadone enantiomers have been poorly investigated in acute poisonings. The aim of this study was to describe the relationships between methadone-related respiratory effects and their corresponding concentrations.
Methods:
We report a 44-year-old methadone-maintained patient who ingested a 240-mg dose of methadone. He was found comatose with pinpoint pupils and respiratory depression. He was successfully treated with intravenous naloxone infusion over the course of 31 hours at a rate adapted to maintain normal consciousness and respiratory rate. We performed a TK/TD analysis of the naloxone infusion rate needed to maintain his respiratory rate at more than 12 breaths per minute (as toxicodynamics parameter) versus plasma R,S- and R-methadone concentrations (as toxicokinetics parameter), determined using an enantioselective high-performance liquid chromatography assay.
Results:
Initial plasma R,S-methadone concentration was 1,204 ng/ml. Decrease in plasma R- and S-methadone concentrations was linear and demonstrated a first-order pharmacokinetics (maximal observed concentrations 566 and 637 ng/ml, half-lives 16.1 and 13.2 hours, respectively). TK/TD correlation between naloxone infusion rate and R,S- and R-methadone concentrations fitted well a sigmoidal Emax model (concentration associated with a half-maximum effect [EC50] 334 and 173 ng/ml, Hill coefficient 10.0 and 7.8, respectively). In our chronically treated patient, EC50 values were in the range of previously reported values regarding methadone analgesic effects, suggesting that plasma methadone concentrations to prevent withdrawal are lower than those associated with methadone analgesic effects.
Conclusion:
After the ingestion of a toxic dose of a racemic mixture, plasma R- and S-enantiomer concentrations decreased in parallel. Despite large inter-individual variability in methadone toxicokinetics and toxicodynamics, TK/TD relationships would be helpful for providing quantitative data regarding the respiratory response to methadone in poisonings. However, further confirmatory TK/TD data are needed.
Successful extracorporeal membrane oxygenation resuscitation of patient with cardiogenic shock induced by phaeochromocytoma crisis mimicking hyperthyroidism: A case report
Authors: Tao Wang, Qiancheng Xu, Xiaogan Jiang
Journal: Open Life Sci (2021)
Keywords: ecmo, cardiocirculatory system collapse, hyperthyroidism, intensive care unit, phaeochromocytoma crisis
MeSH terms: Case Reports

A 29-year-old woman presented to the emergency department with the acute onset of palpitations, shortness of breath, and haemoptysis. She reported having an abortion (56 days of pregnancy) 1 week before admission because of hyperthyroidism diagnosis during pregnancy. The first diagnoses considered were cardiomyopathy associated with hyperthyroidism, acute left ventricular failure, and hyperthyroidism crisis. The young woman's cardiocirculatory system collapsed within several hours. Hence, venoarterial extracorporeal membrane oxygenation (VA ECMO) was performed for this patient. Over the next 3 days after ECMO was established, repeat transthoracic echocardiography showed gradual improvements in biventricular function, and later the patient recovered almost completely. The patient's blood pressure increased to 230/130 mm Hg when the ECMO catheter was removed, and then the diagnosis of phaeochromocytoma was suspected. Computed tomography showed a left suprarenal tumour. The tumour size was 5.8 cm × 5.7 cm with central necrosis. The vanillylmandelic acid concentration was 63.15 mg/24 h. Post-operation, pathology confirmed phaeochromocytoma. To our knowledge, this is the first case report of a patient with cardiogenic shock induced by phaeochromocytoma crisis mimicking hyperthyroidism which was successfully resuscitated by VA ECMO.
Unusual Presentation of Type 1 Idiopathic Macular Telangiectasia
Authors: Zaïnab Bentaleb Machkour, Philippe Denis, Laurent Kodjikian
Journal: Case Rep Ophthalmol Med (2017)
MeSH terms: Case Reports

Purpose. To report unusual presentation of type 1A idiopathic macular telangiectasia (IMT). Methods. Two middle-aged women with bilateral IMT were examined. Results. Both patients presented with a gradual vision loss in both eyes. Fundus examination was unremarkable in one case and showed small macular telangiectasia in both eyes in the other case. Fluorescein angiography (FA) revealed early bilateral macular punctuated hyperfluorescence corresponding to the dilated capillaries in both cases. FA and fundus examination confirmed also the absence of vascular abnormalities in the middle or anterior fundus periphery in one case. Spectral-domain optical coherence tomography (SD-OCT) showed cystoid macular edema in both cases. No signs of retinal vein occlusions were detected in both cases and other differential diagnoses were excluded. Based on these findings, the patients were diagnosed with bilateral type 1A IMT according to Gass and Blodi classification and were treated with intravitreal antivascular endothelial growth factor (anti-VEGF) injections and focal laser photocoagulation. Twelve months later, SD-OCT revealed partial regression of the exudative signs and significant VA improvement. Conclusion. We described two patients with an unusual presentation of type 1A IMT with bilateral presentation, affecting two middle-aged women, with occult and without peripheral involvement in one case. The description of more cases of bilateral type 1 IMT should be helpful to more precisely define the pathophysiologic mechanism that could be different from a localized Coats' disease of the macula area.
Anaphylactic shock during cement implantation of a total hip arthroplasty in a patient with underlying mastocytosis: case report of a rare intraoperative complication
Authors: Anita Ten Hagen, Pieter Doldersum, Tom van Raaij
Journal: Patient Saf Surg (2016)
Keywords: anaphylaxis, bone cement, case report, hip surgery, implantation syndrome, mastocytosis
MeSH terms: Case Reports

Background:
Cemented total hip arthroplasty (THA) is a safe and common procedure. In rare cases life threatening bone cement implantation syndrome (BCIS) may occur, which is commonly caused by pulmonary embolism (PE).
Case presentation:
We describe the rare case of a 70-year old patient who underwent an elective total hip replacement. Before surgery he was diagnosed with underlying systemic indolent mastocytosis, a rare pathological disorder that may result in anaphylaxis after massive systemic mast cell activation. Triggers may be IgE-mediated, direct mast cell activation, or unclear. Some patients may be at risk for severe non IgE-mediated reactions, such as those experienced with nonsteroidal anti-inflammatory drugs, or with perioperative muscle relaxants. During cementing of the acetabular component, our patient developed acute hypotension (blood pressure dropped from 90/50 to 60/40 mmHg, and saturation dropped from 95 to 80 %). The differential diagnosis of acute PE was excluded (no signs of breathing abnormalities during physical examination, normal arterial blood sample, and no electrocardiography or cardiac ultrasound abnormalities). The patient was diagnosed with acute anaphylactic shock, which was successfully managed by 100 % oxygen administration, rapid fluid induction, and vasoconstrictive drug therapy. He recovered hemodynamically within 15 min, did not lose consciousness, and did not develop angioedema or an urticarial rash. Forty-five minutes after onset of the symptoms, the surgical procedure was completed after inserting a press fitted uncemented femoral stem component. The patient was transported to the Intensive Care Unit (ICU) for optimal monitoring. Our patient had an uneventful recovery. Within six hours after surgery he started to ambulate following our standard fast-track rehabilitation regime. Post-operative day one he was discharged to the specialized Orthopedic Department, and after five hospital days discharged to his home. Twelve months after THA surgery our patient was satisfied with an optimal functional status of his hip joint replacement.
Conclusion:
The differential diagnosis of anaphylactic shock must be taken into consideration in patients with acute hypotension during cementing of total hip arthroplasty components. Patients with underlying mastocytosis are at particular risk of this potential life-threatening intra-operative complication. This rare entity should be taken into consideration during the pre-operative risk stratification and shared decision-making process for elective cemented joint replacement.
Techniques for Management of Portal Vein Thrombosis during Liver Transplantation
Authors: Navdeep Singh, Kenneth Washburn, Sylvester Black, Austin Schenk
Journal: Case Rep Transplant (2020)
MeSH terms: Case Reports

Portal vein thrombosis (PVT) poses a unique challenge in liver transplant. The management of PVT differs according to the extent of thrombosis. Anastomosis of a donor portal vein to a varix is a viable option when an adequate size varix is identified on preoperative imaging or intraoperatively. Here, we describe our experience in two liver transplant cases with cavernous transformation of the portal vein where the donor portal vein was anastomosed to a varix using a donor iliac vein interposition graft.
Transvaginal appendectomy in morbidly obese patient
Authors: Mehmet Ali Yagci, Cuneyt Kayaalp, Mustafa Ates
Journal: Case Rep Surg (2014)

Introduction. Laparoscopic appendectomy has significant benefits in obese patients. However, morbid obesity can be accepted as an exclusion criterion for natural orifice transluminal endoscopic surgery (NOTES). Here, we present a transvaginal appendectomy in a 66-year-old morbidly obese (BMI 36 kg/m(2), ASA III) patient. Case and Technique. Acute appendicitis was suspected based on history, physical examination, laboratory tests, and ultrasound findings. During laparoscopic surgery, a 5 mm trocar was inserted through the umbilicus and a 5 mm telescope was placed. A 12 mm trocar and a 5 mm grasper were inserted separately through the posterior fornix of the vagina under laparoscopic guidance. The appendix was divided with an endoscopic stapler through the transvaginal 12 mm trocar and removed from the same trocar. The operating time was 75 minutes with minimal blood loss (<10 mL). The patient was discharged 16 hours after surgery uneventfully and she did not require any analgesic administration. Conclusion. To the best of our knowledge, this is the first clinical case that focuses on the transvaginal appendectomy at morbid obesity. We can say that morbid obesity does not constitute an obstacle for treatment of acute appendicitis by transvaginal endoscopic surgery.
Case Report: Pulmonary Vein Isolation as a Tailored Treatment for Recurrent Ventricular Tachycardia During Hemodialysis in a Patient With Right Coronary Artery Chronic Total Occlusion
Authors: Cosmin Cojocaru, Adelina Pupăză, Corneliu Iorgulescu, Sebastian Onciul, Lucian Câlmâc, Radu Vătăşescu
Journal: Front Cardiovasc Med (2022)
Keywords: catheter ablation, electrical storm, ischaemic cardiomyopathy, paroxysmal atrial fibrillation, trigger
MeSH terms: Case Reports

Background:
Catheter ablation of the ventricular substrate can reduce ventricular tachycardia (VT) recurrence and mortality in an electrical storm (ES). However, identification and specific treatment of plausible triggers is mandatory and may lead to the resolution of ES.
Objective:
This case presentation seeks to exemplify how pulmonary vein isolation (PVI) may represent a tailored treatment of ES in cases of ventricular substrate, which only becomes arrhythmogenic during high-rate episodes of paroxysmal atrial fibrillation (PAF).
Results:
A 54-year-old male with a history of inferior myocardial infarction (MI) and long-term hemodialysis was referred for repetitive implantable cardioverter-defibrillator (ICD) shocks for apparently scar-related monomorphic VT episodes preceded by PAF initiation strictly during hemodialysis. He had recently undergone ICD implantation for similar episodes of ES preceded by the rapid-ventricular response (RVR) PAF during hemodialysis. The patient had no other history of VTs. Electrocardiogram (EKG) changes occurred exclusively during PAF and suggested functional myocardial ischemia. Coronary angiography demonstrated isolated right coronary artery (RCA) chronic total occlusion (CTO). Cardiac magnetic resonance demonstrated RCA-territory residual myocardial viability and mild LV systolic dysfunction. Surgical revascularization was not feasible due to a history of bilateral above-the-knee post-traumatic amputation and severe calcification of internal mammary (IMA) and radial arteries. Subsequent CTO-percutaneous coronary intervention attempt was unsuccessful. The difficulty of assessing LV-substrate ablation end-points due to the "functional" character of the substrate, which only became arrhythmogenic during hemodialysis-related PAF, was considered. Consequently, PVI was performed rather than VT/VF substrate ablation. Twelve months after PVI, the patient remains free of PAF and VT/VF despite chronic hemodialysis sessions.
Conclusion:
The ES episodes can be triggered by situational factors, such as RVR-PAF and functional ischemia, during hemodialysis in patients with CTO with otherwise no episodes of VT. Tailored treatment of such factors may lead to long-term VT freedom.
Case Report: A Novel Gross Deletion in PAX3 (10.26 kb) Identified in a Chinese Family With Waardenburg Syndrome by Third-Generation Sequencing
Authors: Jie-Yuan Jin, Lei Zeng, Bing-Bing Guo, Yi Dong, Ju-Yu Tang, Rong Xiang
Journal: Front Genet (2021)
Keywords: pax3, waardenburg syndrome, deletion, structural variants, third generation sequencing
MeSH terms: Case Reports

Waardenburg syndrome (WS) is a group of autosomal-dominant hereditary conditions with a global incidence of 1/42,000. WS can be categorized into at least four types: WS1-4, and these are characterized by heterochromia iridis, white forelock, prominent nasal root, dystopia canthorum, hypertrichosis of the medial part of the eyebrows, and deaf-mutism. WS3 is extremely rare, with a unique phenotype (upper limb abnormality). Heterozygous mutations of PAX3 are commonly associated with WS1, whereas partial or total deletions of PAX3 are often observed in WS3 cases. Deletions, together with insertions, translocations, inversions, mobile elements, tandem duplications, and complexes, constitute structural variants (SVs), which can be fully and accurately detected by third-generation sequencing (TGS), a new generation of high-throughput DNA sequencing technology. In this study, after failing to identify the causative gene by Sanger sequencing, SNP-array, and whole-exome sequencing (WES), we finally detected a heterozygous gross deletion of PAX3 (10.26kb, chr2: 223153899-223164405) in a WS family by TGS. Our description would enrich the genetic map of WS and help us to further understand this disease. Our findings also demonstrated the value of TGS in clinical genetics researches.
Acute post-traumatic encephalocele in a child: CT and MRI features
Authors: Abhinav Aggarwal, Ashish Kumar Gupta, Aakriti Kapoor Aggarwal
Journal: BJR Case Rep (2016)
MeSH terms: Case Reports

Orbital trauma is a commonly encountered entity in clinical practice, especially in cases of head trauma. Although fractures of the orbit are rare, they can present in an emergency setting owing to associated complications such as orbital encephaloceles. We present a case of a paediatric male patient who presented with post-traumatic proptosis and diminution of vision and was diagnosed with orbital encephalocele. The child's vision recovered completely and proptosis of the eye decreased, compared with the time of presentation. Despite persistent counselling, the patient and his parents did not give consent for surgical correction and he was discharged as leaving against medical advice. Post-traumatic orbital encephaloceles are caused as a result of blunt cranial trauma. Early treatment and diagnosis is extremely important in the optimum management and good long-term prognosis of the patient.
Toxoplasmosis in a patient who was immunocompetent: a case report
Authors: Aneta K Taila, Ameet S Hingwe, Laura E Johnson
Journal: J Med Case Rep (2011)

Introduction:
Toxoplasma gondii is an obligate intracellular protozoan that infects up to one-third of the world's population. Although this case is not the first of its kind, it is clinically important since it will help doctors keep a broad differential diagnosis in mind when attending to similar patients.
Case presentation:
We present the case of a 20-year-old man of Middle Eastern heritage presenting with only generalized lymphadenopathy who was diagnosed with acute toxoplasmosis.
Conclusion:
This case illustrates the important fact that toxoplasmosis can present with just simple lymphadenopathy, and thus can be confused with other infections such as Epstein-Barr virus and other mononucleosis-like illnesses such as cytomegalovirus, HIV with acute retroviral syndrome, cat scratch disease, leishmaniasis and syphilis. This case underlines why appropriate testing should be performed in confusing cases, and helps increase the knowledge about the diagnosis of this disease.
Polyarticular Septic Arthritis Due to Non-Typeable Haemophilus influenzae With Concomitant New-Onset Acute Gouty Arthritis
Authors: Brian Sanders, Mohammed Abdulfatah, Mossab Aljuaid, Ibrahim Tawhari
Journal: J Investig Med High Impact Case Rep (2019)
Keywords: arthritis, gout
MeSH terms: Arthritis, Gouty / complications, Arthritis, Infectious / microbiology, Haemophilus Infections / complications, Haemophilus influenzae, Case Reports, Anti-Bacterial Agents / therapeutic use, Arthritis, Gouty / pathology, Arthritis, Infectious / complications, Arthritis, Infectious / drug therapy, Arthritis, Infectious / pathology, Haemophilus Infections / drug therapy, Haemophilus Infections / microbiology, Haemophilus Infections / pathology, Humans, Male, Middle Aged, Anti-Bacterial Agents

Haemophilus influenzae is serologically classified into two main categories based on the presence or absence of the polysaccharide capsule. Strains that possess polysaccharide capsules are identified as typeable Haemophilus influenzae, whereas strains that do not have capsules are identified as non-typeable Haemophilus influenza. Only on very rare occasions, Haemophilus influenzae affects adult joints, and almost 95% of cases have been identified as type b serotypes. Coexistence of gouty and septic arthritis is rare but has been reported. We herein report a case of polyarticular septic arthritis caused by non-typeable Haemophilus influenzae in an adult with concomitant new-onset gouty arthritis. The case was successfully treated with surgical debridement and a 4-week course of ceftriaxone.
A common anesthesiology procedure for a patient with an uncommon combination of diseases: a case report
Authors: Aliki Tympa, Dimitrios Hassiakos, Nikolaos Salakos, Aikaterini Melemeni
Journal: Case Rep Anesthesiol (2012)

Administering neuraxial anesthesia to a patient with an underlying neurological disease and a combination of four other pathological disorders can be challenging. We report in this paper the case of a 45-year-old woman with neurological deficit due to ischemic brain infarct, multiple sclerosis, antiphospholipid syndrome, and β-heterozygous thalassemia that was subjected to abdominal hysterectomy and bilateral salpingoophorectomy under epidural anesthesia for ovarian cancer.
Suspected Acute Respiratory Distress Syndrome Associated With the Use of Intravenous Lipid Emulsion Therapy in a Dog: A Case Report
Authors: Heike Botha, Samuel Hollis Jennings, Saya A Press, Stephanie A Istvan
Journal: Front Vet Sci (2019)
Keywords: ards, acute respiratory distress syndrome, adverse drug reaction, canine, intravenous lipid emulsion toxicity, tetrahydrocannabinol, toxicosis
MeSH terms: Case Reports

A 12-year-old male neutered Bichon Frise presented to the Emergency Department for stupor and bradycardia after ingestion of chocolate covered 450 mg (90 mg/kg) tetrahydrocannabinol. The patient was hospitalized for supportive care, IV fluid therapy and monitoring in the intensive care unit. During hospitalization the patient became comatose and bradypneic. Treatment with intravenous lipid emulsion (ILE) therapy was instituted to accelerate toxin elimination, reduce the risk of complications related to progressive obtundation and shorten hospitalization time. Five hours after infusion, the patient developed severe respiratory distress and was ultimately euthanized. Post-mortem histologic evaluation of lung revealed severe pulmonary edema consistent with acute respiratory distress syndrome. There are infrequent reports of adverse effects associated with ILE therapy for toxicosis in veterinary medicine despite reports of complications such as acute respiratory distress syndrome in human literature. The purpose of this report is to describe the potential for a severe adverse event after treatment of a toxicosis with ILE therapy.
A 41-Year-Old Patient with a Rare Cause of Severe Abdominal Sepsis Misdiagnosed as PID
Authors: Anastasia Mikuscheva, David Becker, Mark Thompson-Fawcett
Journal: Case Rep Surg (2018)
MeSH terms: Case Reports

Infectious pelvic inflammatory disease is a common condition and a frequent cause of abdominal pain in a young female patient. In a patient who has not completed family planning, the diagnosis is often made with a low threshold and treatment started on a low suspicion of diagnosis to avoid a negative impact on fertility. Here, we present a case of a 41-year-old woman who was misdiagnosed with infectious pelvic inflammatory disease and treated ineffectively with antibiotics when the underlying condition of her persistent abdominal pain was a midgut neuroendocrine tumor that had caused bowel perforation and formation of an abscess in the pouch of Douglas.