Objective:To describe the effect of hemoglobin Wayne variant on hemoglobin A1c (A1c) accuracy and to stress the importance of patient-physician communication and trust.Methods: We present the clinical history and laboratory findings of 2 patients, with a review of related literature.Results: Two older patients were diagnosed with diabetes mellitus (DM) and suffered from frequent hypoglycemia resulting from titrating their diabetes medications based on an extremely elevated A1c (>11% [97 mmol/ mol]) obtained through high-performance liquid chromatography. Discrepancies were noticed between their blood glucose logs and their A1c results. Ultimately, both were found to have heterogenous hemoglobin Wayne variant by hemoglobin electrophoresis. Repeat immunoassay found the A1c to be very low, in the 5 to 6.2% (31 to 44 mmol/ mol) range. One of the patients did not even meet diagnostic criteria for DM.Conclusion: A1c testing is susceptible to misinterpretation due to multiple interfering factors. Hemoglobin variants should be considered as a differential when there are discrepancies between A1c and blood glucose levels.Trust between the patient and the physician is essential in raising clinical suspicion and avoiding potentially lethal outcomes. (AACE Clinical Case Rep. 2019;5:e59-e61) Abbreviations: A1c = glycated hemoglobin; DM = diabetes mellitus; HPLC = high-performance liquid chromatography
Osteonecrosis is a pathological condition that could lead to a debilitating physical disease and impede daily activities. It is generally categorised into aetiology – primary (idiopathic) or secondary. When direct damage to the bone vasculature or direct injury of the bone marrow is related to an identifiable cause such as traumatic injuries, steroid or bisphosphonate use, increased alcohol intake, sickle cell disease, autoimmune diseases, chemotherapy or malignancy, it is categorised as secondary osteonecrosis. On the other hand, osteonecrosis wherein the mechanisms of development are not fully understood is categorised as primary or idiopathic osteonecrosis. This category includes inherited thrombophilia and hypofibrinolysis as potential causes. There are no clear guidelines or general agreements about anticoagulation treatment and duration in primary osteonecrosis due to thrombophilia or hypofibrinolysis. We report a case of primary osteonecrosis associated with hypofibrinolysis and successful control with lifelong direct oral anticoagulation therapy.
BACKGROUND Pseudogout, or calcium pyrophosphate dihydrate (CPPD) disease, is an inflammatory joint disease that most commonly involves the joints of the knees, ankles, and wrists. Pseudogout has also been known to involve the spine, especially the atlanto-occipital joint of the cervical spine, but there is limited documentation of its involvement in the lumbar spine. Though the atypical presentation of spinal pseudogout with findings consistent with discitis and epidural abscess has been documented, its presentation with associated chronic spinal epidural hematoma is a rare entity. OBSERVATIONS The authors present two separate cases of pseudogout involvement of the lumbar spine, one case presenting with a clinical and radiographic picture consistent with discitis and epidural abscess and the other with radiographic and operative findings consistent with a chronic epidural hematoma. LESSONS This case series demonstrates rare and atypical presentations of pseudogout within the lumbosacral spine.
Background:While it is a rare entity, spinal angiolipomas are well-defined benign tumors that have been described sporadically in the literature starting from the late 1800s. Composed of mature lipomatous and angiomatous elements, these tumors manifest neurological symptoms due to progressive spinal cord or root compression. We present a case of a thoracic spinal angiolipoma and review the relevant literature.Case Description:A 68-year-old male with ongoing bilateral lower extremity weakness was found on enhanced magnetic resonance imaging to have an extradural mass in the thoracic spine causing cord compression. A T4–T8 laminectomy and complete excision of the epidural mass resulted in reversal of the patient's neurological symptoms. Histopathology identified the mass as a thoracic spinal angiolipoma.Conclusion:Given its uncommon occurrence and excellent prognosis, our report serves as a reminder to always consider spinal angiolipoma in the differential diagnosis of epidural masses.
BACKGROUND Chordoid meningioma is a rare World Health Organization (WHO) grade 2 variant of meningioma with histological features resembling those of a chordoma. This tumor type is known for having an aggressive clinical course with a propensity for local recurrence. Most cases occur within the cranium, more specifically around the cerebral convexities. Although extracranial meningiomas of various subtypes have been documented, extracranial meningioma with a chordoid subtype is an extremely rare entity. OBSERVATIONS The authors herein report the case of a 51-year-old female who presented with a chief complaint of dysphagia and was found to have a neck mass abutting the carotid sheath. The patient underwent resection and final pathology results revealed a WHO grade 2 chordoid meningioma. LESSONS This case report demonstrates an atypical case of an extracranial chordoid meningioma adjacent to the carotid sheath. To the authors’ knowledge, this is the first reported case of a chordoid meningioma occurring within the soft tissue of the neck.
BACKGROUND Mycotic aneurysms (MAs) are rare intracranial pathologies. They are associated with spontaneous rupture, which is often the first presenting sign. Subarachnoid hemorrhage and intraparenchymal hemorrhage are the most common sequelae of ruptured MAs, with subdural hematoma being an atypical presentation. The presentation of an MA as a subdural empyema has not yet been reported in the literature. OBSERVATIONS The authors discussed a 68-year-old man who presented with subdural empyema and received surgery for evacuation. He was found to have a ruptured mycotic aneurysm intraoperatively. LESSONS This case demonstrated a rare and atypical presentation of an MA.
INTRODUCTION: Chiari malformation type I (CM-I) can cause debilitating neurologic symptoms, most notably distinctive headaches, and the only existing definitive treatment is suboccipital decompression. While CM-I is typically defined as ≥5 mm downward displacement of cerebellar tonsils, overall CM-I cerebellar morphology is highly heterogeneous, and it is uncertain if this variability represents distinct patient populations or influences clinical response to suboccipital decompression.METHODS: 186 adult CM-I patients with high-resolution T1-MRIs were retrospectively identified. Each patient's cerebellum was non-linearly and diffeomorphically transformed to template brain, with this transformation's inverse representing cerebellar deformation. A similarity matrix was generated between patients' deformations using the Pearson correlation coefficient of their inverse transformations' log Jacobian determinants within each cerebellar lobule. Spectral clustering was performed on this similarity matrix to identify patient clusters. Headache freedom after suboccipital decompression was compared between clusters by Kaplan-Meier analysis.RESULTS: Three morphological clusters of CM-I patients were identified. Cluster A is characterized by posteroinferior cerebellar fullness and platybasia; B by midline anterior-posterior compression; C solely by tonsillar descent. After suboccipital decompression, headaches were found to recur more frequently and sooner in Cluster B (p < 0.01).CONCLUSIONS: These analyses suggest that CM-I encapsulates morphologically distinct subtypes, here identified as Cluster A and Cluster B, with Cluster C representing an intermediate phenotype. These clusters vary significantly in clinical response to suboccipital decompression, which may have implications for surgical patient selection or operative technique. Future research should involve independent validation of this cluster scheme and optimization of treatment for patients in Cluster B, which is found here to have poorer surgical outcomes.
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