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Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature.

ABSTRACT: Introduction: Anti-PD1 agents are widely used in the treatment of solid tumors. This has prompted the recognition of a class of immune-related adverse events (irAEs), due to the activation of autoimmune T-cells. Pembrolizumab is an anti-PD1 agent, which has been related to an increased risk of various neurological irAE (n-irAEs). Here, we present a rare case of pembrolizumab-induced neuropathy of cranial nerves. Case Report: A 72-year-old patient was diagnosed with a lung adenocarcinoma in February 2018 (EGFR-, ALK-, and PDL1 90%). According to the molecular profile, pembrolizumab was started. After three administrations, the patient developed facial paresis, ptosis, ophthalmoplegia, and dysphonia. As brain metastases and paraneoplastic markers were excluded, a drug-related disorder was suspected and pembrolizumab was discontinued. A nerve conduction study and electromyography excluded signs of neuropathy and myopathy at four limbs, and repetitive nerve stimulation was negative. However, altered blink reflex and nerve facial conduction were consistent with an acute neuropathy of the cranial district. Thus, the patient was treated with two cycles of intravenous immunoglobulins (IVIg), which rapidly allowed improvement of both symptoms and neurophysiological parameters. However, the patient died in October 2018 for a progression of lung tumor. Discussion: Only 16 cases of pembrolizumab-related neuropathies have been described so far. Our case is of particular interest for the isolated involvement of cranial nerves and the prompt response to IVIg. Conclusion: N-irAEs are insidious conditions that require solid knowledge of onco-immunotherapy complications: it is mandatory not to delay any treatment that would potentially modify the course of a neurological complication.

SUBMITTER: Bruno F

PROVIDER: S-EPMC8144636 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Project description:BackgroundIntestinal Ganglioneuromatosis (IG) is a rare disorder of the enteric nervous system. In pediatric age it is often associated with genetic syndromes such as Neurofibromatosis 1 (NF1), multiple endocrine neoplasia type 2B (MEN2B) and Cowden syndrome (PTEN mutation), and ganglioneuromas (GNs) may be sometimes the first sign of the disease. Isolated GNs are rare and sporadic. Clinical symptom vary and depend on the size and on the location of the GNs. This disorder affects intestinal motility and it, consequently, causes changes in bowel habits, abdominal pain, occlusive symptoms and rarely lower gastrointestinal bleeding secondary to ulceration of the intestinal mucosa. On the other hand, patients can remain asymptomatic for many years.Case presentationWe describe a 9-year-old boy referred to our emergency department for right lower quadrant abdominal pain. No familial history for gastrointestinal disorders. No history of fever or weight loss. At physical examination, he had diffused abdominal pain. Abdominal ultrasonography showed a hypoechoic formation measuring 41.8 mm by 35 mm in the right lower quadrant of the abdomen. Routine blood tests were normal, but fecal occult blood test was positive. Abdominal TC confirmed the hypodense formation, of about 5 cm in transverse diameter, in the right hypochondrium that apparently invaginated in the caecum-last ileal loop. Colonoscopy showed in the cecum an invaginated polypoid lesion of the terminal ileal loop. Laparoscopic resection of the polypoid lesion was performed. Histological diagnosis of the large neoplasm observed in the terminal ileum was diffuse ganglioneuromatosis. NF1, RET and PTEN gene tests resulted negative for specific mutations. At the 1 year follow-up, the patient presented good general condition and blood tests, fecal occult blood test, esophagogastroduodenoscopy, colonoscopy and MR-enterography were negative.ConclusionsOnly few cases are reported in literature of IG in pediatric age. Although rare, the present case suggests that this disorder must be taken in consideration in every patient with GI symptoms such as abdominal pain, constipation, lower intestinal bleeding, in order to avoid a delayed diagnosis.

Project description:Introduction and importance Subarachnoid hemorrhage (SAH) is mostly associated with head trauma. Non-traumatic subarachnoid hemorrhage is mostly due to vascular abnormalities: either hemorrhage from ruptured aneurysm or bleeding from arteriovenous malformation. Aneurysmal hemorrhage is the biggest cause in non-traumatic cases. Warfarin is associated with cerebral intraparenchymal hemorrhage, but it is rarely associated with SAH. Case presentation Here, we report the case of a 45-year-old male patient who was admitted to the neurology ward of our hospital due to acute ischemic stroke. The patient was treated with a vitamin K antagonist (warfarin). However, on the third day, his condition deteriorated (his GCS regressed from 11/15 to 5/15). His pupils were anisocoric. Brain CT showed extensive subarachnoid hemorrhage without intraparenchymal involvement. Cerebral magnetic resonance angiography ruled out aneurysmal rupture. The patient was intubated and transferred to the intensive care unit. Due to his poor condition, neurosurgical intervention could not be done. The patient was managed conservatively, but the patient passed away 4 days later in the intensive care unit. Clinical discussion Non-traumatic SAH is mostly caused by aneurysmal rupture. Warfarin increases the risk of intracranial hemorrhage and mostly causes intraparenchymal hemorrhage. Isolated warfarin-related SAH without parenchymal involvement is a rare event. Here we present a young male patient with an isolated warfarin-induced SAH. Conclusion Warfarin is rarely associated with isolated subarachnoid hemorrhage. This case highlights a young male patient with spontaneous SAH after warfarin therapy for acute ischemic stroke. Aneurysmal rupture and trauma should be excluded before a diagnosis of warfarin-induced SAH is made. Highlights • Non-traumatic subarachnoid hemorrhage (SAH) is mostly caused by aneurysmal rupture.• Warfarin increases the risk of intracranial hemorrhage and mostly causes intraparenchymal hemorrhage. Isolated warfarin-related SAH without parenchymal involvement is a rare event.• Here we present a young male patient with isolated SAH due to warfarin treatment for acute ischemic stroke.

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Pembrolizumab-Induced Isolated Cranial Neuropathy: A Rare Case Report and Review of Literature.

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