Anti-NXP2-Positive Paraneoplastic Dermatomyositis With Histopathologic Changes Confined to the Acrosyringia

Am J Dermatopathol. 2017 Jan;39(1):e3-e7. doi: 10.1097/DAD.0000000000000694.

Abstract

Background: Paraneoplastic syndromes consist of a group of disorders that are not related to the extension of the primary tumor or its metastases and that might be the first manifestation of a hidden neoplasm. It is a well-known association between dermatomyositis (DM) and cancer, especially gynecological tumors in women and lung cancer in men.

Methods: We describe the case of a 67-year-old male who developed muscular weakness and pruritic skin lesions. Skin biopsies were performed and histologic findings were consistent with DM.

Results: Skin biopsy showed interface dermatitis with vacuolar degeneration of the basal layer, dermal mucin deposits, and necrotic keratinocytes in the acrosyringia, a finding that has been previously reported in lupus erythematous but not in DM. Autoimmunity tests showed positivity for antinuclear antibodies and anti-NXP2, a recently described antibody associated with juvenile DM and, more rarely, with paraneoplastic DM.

Conclusion: We present the first case in the literature with histopathologic changes of DM affecting the acrosyringia. Besides, our patient autoimmunity results support the utility of the new myositis-specific autoantibodies and its relation with a clinical phenotype.

Publication types

  • Case Reports

MeSH terms

  • Adenosine Triphosphatases / immunology*
  • Adrenal Cortex Hormones / therapeutic use
  • Aged
  • Autoantibodies / analysis*
  • Biomarkers / analysis
  • Biopsy
  • Carcinoma, Non-Small-Cell Lung / complications*
  • Carcinoma, Non-Small-Cell Lung / pathology
  • Carcinoma, Non-Small-Cell Lung / therapy
  • DNA-Binding Proteins / immunology*
  • Dermatomyositis / drug therapy
  • Dermatomyositis / etiology
  • Dermatomyositis / immunology*
  • Dermatomyositis / pathology
  • Humans
  • Immunoassay
  • Lung Neoplasms / complications*
  • Lung Neoplasms / pathology
  • Lung Neoplasms / therapy
  • Male
  • Paraneoplastic Syndromes / drug therapy
  • Paraneoplastic Syndromes / etiology
  • Paraneoplastic Syndromes / immunology*
  • Paraneoplastic Syndromes / pathology
  • Skin / drug effects
  • Skin / immunology*
  • Skin / pathology
  • Tomography, X-Ray Computed
  • Treatment Outcome

Substances

  • Adrenal Cortex Hormones
  • Autoantibodies
  • Biomarkers
  • DNA-Binding Proteins
  • Adenosine Triphosphatases
  • MORC3 protein, human