A systematic review of literature on Insulin-like growth factor-2-mediated hypoglycaemia in non-islet cell tumours.

INTRODUCTION: Insulin-like growth factor-2 (IGF-2)-mediated hypoglycemia is a rare yet clinically significant entity with considerable morbidity and mortality. Existing literature is limited and fails to offer a comprehensive understanding of its clinical trajectory, management and prognostication. METHODS: Systematic review of English-language articles reporting primary patient data on IMH was searched using electronic databases (PubMed, Scopus and Embase) from any date up to 21 December 2022. Data were analysed in STATA-16. RESULTS: The systematic review contains 172 studies, including 1 Randomised controlled trial, 1 prospective observational study, 5 retrospective observational studies, 150 case reports, 11 case series and 4 conference abstracts. A total of 233 patients were analysed, averaging 60.6 ± 17.1 years in age, with comparable proportions of males and females. The commonest tumours associated with Insulin-like Growth Factor-2-mediated hypoglycaemia were fibrous tumours (N = 124, 53.2%), followed by non-fibrous tumours originating from the liver (N = 21, 9%), hemangiopericytomas (N = 20, 8.5%) and mesotheliomas (N = 11, 4.7%). Hypoglycaemia was the presenting feature of NICT in 42% of cases. Predominant clinical features included loss of consciousness (26.7%) and confusion (21%). The mean IGF-2 and IGF-1 levels were 882.3 ± 630.6 ng/dL and 41.8 ± 47.8, respectively, with no significant correlation between these levels and patient outcomes. Surgical removal was the most employed treatment modality (47.2%), followed by medication therapy. The recovery rate was 77%, with chronic liver disease (CLD) significantly associated with a poor outcome (OR: 7.23, P: 0.03). Tumours originating from fibrous tissues were significantly associated with recovery (p < .001). In the logistic regression model, CLD remained a significant predictor of poor outcomes. CONCLUSION: This systematic review highlights that most non-islet-cell tumour-hypoglycaemia (NICTH) is due to fibrous tumours. NICTs demonstrate a variable prognosis, which is fair if originating from fibrous tissue. Management such as octreotide, corticosteroids, diazoxide, embolization, radiotherapy and surgical resection have disparate success rates.

A Rare Case of Lambert-Eaton Myasthenia Syndrome Associated with Non-Hodgkin's Lymphoma: A Case Report and Review of the Literature.

Introduction: Lambert-Eaton myasthenia syndrome (LEMS) is a rare autoimmune disorder characterized by autoantibodies targeting presynaptic neuromuscular junctions. It results in muscle weakness and autonomic dysfunction. LEMS can be idiopathic or associated with neoplastic diseases, often small-cell lung cancer. This case report describes a rare instance of paraneoplastic LEMS in a man with non-Hodgkin lymphoma. Case Presentation: A 57-year-old male with non-Hodgkin lymphoma presented with progressive muscle weakness, diminished reflexes, and autonomic symptoms. Diagnosis revealed LEMS with autoantibodies against voltage-gated calcium channels. Immunosuppressive therapy and lymphoma treatment led to significant improvement in his condition. Conclusion: This case highlights the rare occurrence of paraneoplastic LEMS in a patient with non-Hodgkin lymphoma. Recognition and timely management of LEMS alongside lymphoma treatment can lead to significant clinical improvement, emphasizing the need for increased awareness of such complex associations.

The Impact of Tyrosine Kinase Inhibitors on Fatherhood in Patients With Chronic Myeloid Leukemia: A Mixed-Method Study.

INTRODUCTION: Multiple studies have demonstrated that tyrosine kinase inhibitors (TKIs) exert a significant extent of control over chronic myeloid leukemia (CML), as evidenced by studies such as the population-based Swedish CML registry, which found that patients reaching age 70 had a relative survival rate close to one when compared to the general population. Consequently, new perspectives on the safety of treatments have emerged, particularly in the context of their impact on fatherhood in men. According to the authors, this is the first study to examine the effect of TKIs on fatherhood in CML patients.  Methods: A single-center, mixed-design study (retrospective data review and phone interviews) was conducted with CML male patients in the chronic or accelerated phase, evaluating the effect of imatinib, dasatinib, and nilotinib on their fatherhood, irrespective of whether they were administered as a first, second, or third line of treatment. RESULTS: The study included interviews with 150 patients. Included were 27 patients. The average age was approximately 44.5 years. One hundred percent of the patients were in the chronic phase. The median age at first conception following TKI therapy was 36, and the median duration of TKI therapy was approximately seven years. The total number of offspring was 49; 98% were born at term and had a normal birth weight. No reports of stillbirths, fetal deaths, or congenital malformations were made. All the offspring grew and developed normally. No CML-related cancers were reported in any of the newborns. CONCLUSION: Around 98% of male CML patients receiving imatinib, dasatinib, or nilotinib did not experience a negative impact on their fatherhood or the health of their children. However, improved education for patients beginning treatment with TKIs addresses the potential psychological worry of having an unfavorable effect on their fertility or offspring, which may increase medication adherence.

Third reported case of rare necrolytic migratory erythema associated with bacteraemia due to severe zinc deficiency after revisional Roux-En-Y gastric bypass: case report and literature review.

INTRODUCTION: Obesity is a risk factor for zinc deficiency. After bariatric surgery, non-compliance to diet/vitamin supplements, surgical complications leading to vomiting/diarrhea, poor follow-up and malabsorption can precipitate or exacerbate pre-existing zinc deficiency. CASE REPORT: We report a patient with rare necrolytic migratory erythema associated with bacteraemia due to severe zinc deficiency after revisional Roux-en-Y gastric bypass (following primary laparoscopic sleeve gastrectomy). CONCLUSION: Bariatric teams should screen patients before bariatric surgery for nutritional deficiencies and continue surveillance of their nutritional status after surgery. They should maintain a high index of suspicion for zinc deficiency in patients with skin rash after bariatric surgery. LEVEL OF EVIDENCE: Level V, case report.

Pruritus as a Presenting Symptom of FIP1L1-PDGFRA-Positive Chronic Eosinophilic Leukemia.

Eosinophilia can be found in a variety of benign and malignant conditions, and a persistent eosinophilic count of more than 1500/mm3 necessitates additional investigation. Patients with FIP1L1-PDGFRA-positive chronic eosinophilic leukemia might present as asymptomatic or in a catastrophic state with multi-organ involvement. We present the case of a young male patient who was diagnosed with FIP1L1-PDGFRA chronic eosinophilic leukemia after a long history of recurrent cutaneous symptoms with no systemic signs.