Acute Encephalitic Syndrome Induced by Scleromyxedema

Am J Med Sci. 2020 Aug;360(2):192-195. doi: 10.1016/j.amjms.2020.05.018. Epub 2020 May 20.

Abstract

Dermato-neuro syndrome is a potentially fatal neurological complication of scleromyxedema consisting of fever, seizures, and coma. This is an overlooked scleromyxedema case of a 62-year-old female patient from 2-years ago. She was admitted to our ICU because of high fever, colloid speech, muscle ache, and nausea. Molecular methods in the cerebrospinal fluid for neurotropic viruses ruled out acute infectious encephalitis. Her thyroid hormones were within normal values while the serum protein electrophoresis confirmed the monoclonal gammopathy of immunoglobulin G lambda (IgG(λ)), known for the last 2 years. The subsequent bone-marrow biopsy excluded the development of multiple myeloma. The patient fulfilled fundamental diagnostic criteria of scleromyxedema (monoclonal gammopathy, normal thyroid function and the appearance of marked sclerosis and induration of the skin papules on the face, neck, extremities, and skin creases) presenting as dermato-neuro syndrome, which was histologically confirmed. She demonstrated a remarkable improvement after intravenous immunoglobulin treatment during the first 24 hours. Mimics of non-infectious acute encephalitis should include the clinical diagnosis of scleromyxedema, especially when patients present in the emergency department with acute fever, coma, and skin lesions of diffuse sclerodermoid and papular type.

Keywords: Encephalitis; Fever; Immunoglobulin; Scleromyxedema.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Acute Febrile Encephalopathy / diagnosis
  • Acute Febrile Encephalopathy / etiology*
  • Acute Febrile Encephalopathy / therapy
  • Biopsy
  • Blood Protein Electrophoresis
  • Brain / diagnostic imaging
  • Coma / etiology
  • Diagnosis, Differential
  • Diagnostic Errors*
  • Female
  • Humans
  • Immunoglobulin G
  • Immunoglobulins, Intravenous / therapeutic use*
  • Immunologic Factors / therapeutic use*
  • Infectious Encephalitis / diagnosis
  • Intensive Care Units
  • Middle Aged
  • Monoclonal Gammopathy of Undetermined Significance / blood
  • Monoclonal Gammopathy of Undetermined Significance / complications*
  • Scleromyxedema / complications*
  • Scleromyxedema / diagnosis
  • Scleromyxedema / pathology
  • Scleromyxedema / therapy
  • Seizures / etiology
  • Skin / pathology
  • Thyrotropin / blood
  • Thyroxine / blood
  • Tomography, X-Ray Computed
  • Triiodothyronine / blood

Substances

  • Immunoglobulin G
  • Immunoglobulins, Intravenous
  • Immunologic Factors
  • Triiodothyronine
  • Thyrotropin
  • Thyroxine