We re-evaluated the patient several months after discharge and after intensive physiotherapy. persisted and, in addition, he developed dysarthria and dysphagia. Neurological evaluation exposed a mild remaining VII cranial nerve deficit, dysarthria with intelligible conversation, remaining lower limb hyposthenia (MRC 4/5), bilateral dysmetria and dyssynergia with remaining prevalence at top and lower limbs, severe ataxia with multidirectional oscillations and widened foundation in orthostatic position, requiring bilateral support. Radiological examinations were free from alterations. Diagnostic hypotheses included acute polyradiculoneuritis with common involvement of the cranial nerves (Miller-Fisher syndrome) and rhombencephalitis. Despite bad results from mind MRI, electroneurography, and blink reflex test, a diagnostic lumbar puncture exposed mild protein elevation (51 mg/dL) and 28 leukocytes/mm3(mainly lymphocytes) with no evidence of viral or bacterial infection. Viral and bacterial serological checks showed only positivity for anti-HBcAg antibodies. After ruling out infectious causes, we treated the patient with intravenous immunoglobulins (7) for five days, resulting in a regression of the remaining VII cranial nerve deficit. Subsequent checks, including chest-abdomen CT and thyroid and testicles ultrasound, as well as 5-Iodo-A-85380 2HCl neoplastic serological markers, were negative. When the oligoclonal bands were finally available (>6 specifically liquor bands, type 2 interpretative criterion), high-dose steroid therapy (1g methylprednisolone iv for five days) was initiated, resulting in an improved trunk control and ataxia. The rheumatological panel showed positivity for anti-ENA antibodies (SSA, Pm/Scl-100), p-ANCA, granular ANA+ with 1:320 dilution, anti-cardiolipin IgG, and minor positivity for anti-recoverin antibodies. Because of this dysimmune process, a post-hospital salivary gland biopsy was planned, also considering the maternal familiarity with Sjogren’s disease. An ophthalmologist exam did not reveal any ocular involvement. We re-evaluated the patient several months after discharge and after rigorous physiotherapy. The dizziness and the feeling of light-headedness experienced completely disappeared, while ataxia was still detectable but 5-Iodo-A-85380 2HCl improved in the last 20-30 days after i.v. immunoglobulins. == Conversation == The absence of classical associated conditions such as retinopathy or malignancy made analysis and treatment more challenging. Moreover, while the patient experienced multiple comorbidities contributing to his overall medical complexity, we have ruled out some other possible condition that could have caused such symptoms. A critical limitation of this case statement is the lack of definitive evidence linking anti-recoverin antibodies to cerebellitis. While the presence of anti-recoverin antibodies in the patient’s serum and the medical improvement with immunomodulatory therapy support this hypothesis, we have not found these antibodies in CSF. However, the medical improvement after immunomodulatory therapy shows the importance of considering autoimmune etiologies in individuals with cerebellar syndromes. == Summary == In conclusion, we present a case of anti-recoverin positive cerebellitis without retinopathy or neoplasia. While the underlying mechanism is still unclear, we believe that anti-recoverin antibodies may have affected the patient’s symptoms. Further study is 5-Iodo-A-85380 2HCl needed to elucidate the pathophysiology of this condition and set up definitive diagnostic and restorative recommendations. == Funding: == This study received no specific grant from funding agencies in the public, commercial, or not-for-profit industries. == Ethic Committee: == The patient gave educated consent in the use Mouse monoclonal antibody to LRRFIP1 of his personal data for study and teaching purposes. == Discord of Interest: == Each author declares that he or she has no commercial associations (e.g. consultancies, stock ownership, equity interest, patent/licensing set up etc.) that might pose a discord of interest in connection with the submitted article. == Authors Contribution: == Collecting data, writing and initial draft preparation MM; Review and editing GB, LZ. == Recommendations ==.