Immune Thrombocytopenic Purpura and Paradoxical Thrombosis: A Systematic Review of Case Reports.

BACKGROUND AND AIMS:  Immune thrombocytopenic purpura (ITP) is an acquired bleeding disorder characterized by autoantibodies against platelets. The clinical presentation is variable; the main symptom is bleeding, and many patients are asymptomatic; others have nonspecific symptoms like fatigue. Uncommonly, ITP can present with paradoxical thrombosis. The risk of thrombosis in ITP may be higher than expected, which makes the management of ITP more challenging. This review aims to evaluate patients with ITP who develop thrombosis and identify potential risk factors related to thrombosis in this category of patients. MATERIALS AND METHODS:  English literature was searched using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines for adults above 18 years with primary ITP who had infarctions or thrombotic events. Patients with secondary ITP were excluded. The search included articles published up to 20th October 2021. RESULTS:  A total of 73 articles were included. Seventy-seven patients with ITP had developed infarctions and various thrombotic events. Sixty-three patients had arterial events, and 14 patients developed venous thrombotic events. CONCLUSION:  Patients with ITP have low platelets, which predispose them to bleed; despite that, serious thrombotic complications can happen in these patients and are difficult to predict. Therefore, it is critical for physicians to understand that ITP is paradoxically a prothrombotic condition and to address preventive thromboembolic measures whenever possible.

Leuprolide-Induced Hyperosmolar Hyperglycemic State in an Elderly Patient: A Case Report and Literature Review.

We present a novel case of severe hyperosmolar hyperglycemic derangement in an elderly patient - without a known history of diabetes mellitus - after the first injection of leuprolide for the treatment of metastatic prostate adenocarcinoma. Whilst the available literature provided accumulative evidence of an association between insulin resistance and the use of gonadotropin-releasing hormone (GnRH) agonists, the initial presentation of leuprolide-induced impaired glycemic tolerance with a hyperosmolar hyperglycemic state (HHS) represents a clinical rarity that was seldom reported. A literature review was conducted to explore the underlying mechanisms of leuprolide-associated glucose intolerance. Screening for diabetes is recommended for patients receiving leuprolide therapy to identify at-risk patients and close glycemic monitoring is warranted in diabetic patients to minimize serious complications from poor glycemic control induced by leuprolide.

Daptomycin-Associated Diarrhea: A Case Report and Review of the Literature.

Antibiotic-associated diarrhea (AAD) describes any unexplained diarrhea associated with the use of antibiotics. AAD develops through diverse mechanisms, ranging from pharmacologic effects on gut motility to disturbance of the function and carbohydrate metabolism of the indigenous intestinal flora and overgrowth by pathogenic micro-organisms. Clostridioides difficile-associated diarrhea (CDAD) is a subset of AAD; however, it accounts only for a small percentage of diarrhea caused by antibiotics. Diarrhea has been reported as a side effect of daptomycin use, nevertheless, it's thought to be mild and carries significantly less risk of diarrhea than other alternative treatments of S. aureus bacteremia, i.e., vancomycin or cefazolin. The authors present an interesting case of daptomycin-associated diarrhea presenting with a protracted and severe course. Patient symptoms didn't improve with empiric Clostridioides difficile therapy and CDAD testing was negative. Diarrhea promptly resolved after discontinuation of daptomycin. Furthermore, a thorough literature review was conducted and discussed in this article to raise awareness of this under-recognized complication. Clinicians should be mindful of daptomycin-associated diarrhea along with its presentation and treatment. Further studies are needed to identify the pathophysiology of daptomycin-associated diarrhea and other forms of AAD. Understanding their mechanism could help prevent, treat, and reduce the significant medical costs associated with antibiotic adverse events.

New Phenotypic Feature in a Patient With a Rare Triplication of the 22q11.2 Region Presenting With Peters Anomaly, Congenital Heart Disease, and Global Developmental Delay: A Case Report and Literature Review.

The vulnerability of chromosome 22q11.2 region to rearrangement is due to several low copy repeat (LCR) sequences. These rearrangements are involved in syndromes that share similar phenotypic features. The rearrangements of the 22q11.2 chromosomal region are common, specifically, duplications and deletions associated with congenital anomalies and developmental disabilities disorders. However, the features associated with this chromosomal rearrangement remain largely unknown. We present, to the best of our knowledge, the third patient affected by triplication of the 22q11.2 chromosome region, who presents with Peters anomaly, global developmental delay, patent ductus arteriosus, and subaortic stenosis. This case highlights a new phenotypic feature associated with triplication of this genomic region.

Thoracic Spinal Cord Compression Secondary to Metastatic Papillary Thyroid Carcinoma: An Unusual Oncological Phenomenon.

Hematogenous spread is fairly an unusual feature for papillary thyroid carcinoma (PTC) in comparison to follicular thyroid carcinoma (FTC). Thoracic spinal metastasis with complicating cord compression is an even rarer manifestation of PTC that was reported in a limited number of cases in the literature. Herein we present a 65-year-old female with a history of PTC on current radiotherapy, status post attempted surgery due to significant tumor burden and intraoperative bleeding, presented with a one-week history of rapidly progressive bilateral lower extremities weakness. Physical examination revealed paraplegia of both lower extremities with areflexia and a sensory level equivalent to the upper thoracic vertebrae. Urgent imaging depicted destructive epidural lesions at T1-T3 vertebrae with thoracic cord compression. Emergent laminectomy and debulking of these lesions were undertaken. Histopathological examination confirmed metastatic PTC. The patient proceeded to further treatment with radiotherapy following her successful neurological recovery. Thoracic vertebral metastasis is an unusual oncological phenomenon of PTC. Metastatic PTC should be considered in patients with a current or remote history of PTC who present with thoracic cord compression. Our case demonstrates that multidisciplinary management is the key to achieving a better outcome for metastatic PTC with thoracic cord compression.

Plasminogen Activator Inhibitor-1 4G/5G Polymorphism Presenting as Recurrent Ischemic Stroke: The Microthrombi Shower.

Certain clinical scenarios should alert a physician to take a deeper look into causative pathological processes. This was evident in the case of a 41-year-old man who presented for recurrent micro thromboembolic strokes, which is atypical for the patient's age. Our desire to explain the pathological process led to the rare finding of a plasminogen activator inhibitor-1 polymorphism, which has been associated with an increased risk of cerebrovascular thrombosis. A defect in this pathway leads to the inhibition of the tissue plasminogen activator protein. This genetic polymorphism has relatively been unexplored in recent medical literature, and we are hoping that our case may inspire future research that could help potential targets of risk factor stratifications as well as the development of novel pharmacological options.

Antiphospholipid Syndrome in a Patient With Autosomal Dominant Polycystic Kidney Disease: The Surface of the Moon.

Antiphospholipid syndrome (APS) is a rare coagulopathic disorder diagnosed with a combination of clinical/imaging findings with specific antibody titer elevations over a period of 12 weeks. The following case report will discuss the unusual and challenging hospital course of a patient with extensive autosomal dominant polycystic kidney disease (ADPKD) being treated for a multi-drug resistant urinary tract infection (UTI). The patient later developed multiple deep vein thrombosis (DVT) and was found to have antiphospholipid syndrome. Warfarin, the anticoagulant of choice for antiphospholipid syndrome, has a higher likelihood of intracerebral hemorrhage than direct oral anticoagulants. This is particularly challenging since patients with autosomal dominant polycystic kidney disease have a higher propensity to develop intracranial aneurysms (ICA).

Lutetium-177 Dotatate-Induced Hemolytic Anemia and Myelodysplastic Syndrome.

Lutetium-177 (177Lu) dotatate is a type of peptide receptor radioligand therapy (PRRT) using radiolabeled somatostatin for patients with progressive somatostatin receptor-positive gastrointestinal neuroendocrine tumors. While cases of therapy-related myeloid neoplasms (t-MN) have been described as a consequence of 177 Lu dotatate, there are no reports of hemolytic anemia associated with therapy. We present a case of a 68-year-old woman with metastatic low-grade neuroendocrine tumor who presented four weeks after the second dose of 177Lu dotatate with progressive fatigue and dyspnea. Laboratory workup was remarkable for hemolytic anemia. Lutetium-177 dotatate-induced hemolysis was suspected after ruling out other causes. Corticosteroid treatment was initiated with improvement in hemoglobin, and dose-reduced PRRT was planned upon discharge. Six months into the treatment course of 177Lu dotatate, macrocytic anemia was noticed on routine follow-up with normal vitamin B12 and folic acid levels. A bone marrow biopsy was done, revealing myelodysplastic syndrome (MDS) features. Given the temporal relationship between drug introduction and the objective findings, early-onset 177Lu dotatate-induced MDS was diagnosed with a plan for close hematologic follow-up. Myelodysplastic syndrome should be suspected when megaloblastic anemia develops in patients with previous 177Lu dotatate therapy. The latency period between initial treatment and MDS diagnosis reported in the literature ranges between 15 months to seven years. Apart from the unusually early onset of MDS, what is unique about our case is the development of hemolytic anemia after administration of PRRT. The clinical course and the brisk response to steroid therapy, suggest other mechanisms of PRRT toxicity besides DNA breaks, genetic mutations, and myelosuppression by an immune-mediated component that likely plays a role in 177Lu dotatate toxicity. Further investigation and monitoring are needed to identify the frequency of such adverse events and the pathophysiology of their occurrence.

Incomplete Kawasaki Disease in an Infant: A Case Report and Literature Review.

The term "incomplete Kawasaki Disease (IKD)" was first used to describe patients with coronary complications who did not fulfill the classical diagnostic criteria for Kawasaki Disease (KD). The risk of coronary artery involvement is similar if not greater in cases of IKD. However, the recognition of IKD is challenging and often delayed, especially in infants. Multiple algorithms have been formulated to identify cases of IKD utilizing supplemental clinical, echocardiographic, and laboratory features. Although fever is not required for a diagnosis of KD in the Japanese guideline, most of the current guidelines, including those of the American Heart Association (AHA), consider the presence of fever for at least seven days a requirement for the diagnosis of both KD and IKD in infants. We present a case of IKD in a four-month-old female who presented with fever for less than three days and did not follow the current AHA algorithm for IKD. An echocardiogram obtained 10 days later revealed a coronary artery aneurysm, and a retrospective diagnosis of IKD was made. A review of the literature identified similar cases with a growing consensus on the need to redefine the role of fever. Pediatricians should search for coronary artery lesions in cases of high clinical suspicion, even if the fever period is short, particularly in those less than six months. Additionally, further innovative research is directly needed to identify immunological and cellular markers that could be tested early in the course of the disease and guide the management.