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Fatal inflammatory hypophysitis.

Elizabeth A McIntyre1, Petros Perros

  • 1Endocrine Unit, Newcastle Hospitals NHS Trust, Freeman Hospital, Newcastle upon Tyne NE7 7DN, UK.

Pituitary
|February 24, 2007
PubMed
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This case study details a rare presentation of Wegener's granulomatosis (WG) initially mimicking other conditions due to acute pituitary failure. Early diagnosis and treatment are crucial for managing this severe autoimmune disease.

Area of Science:

  • Endocrinology
  • Rheumatology
  • Neurology

Background:

  • Wegener's granulomatosis (WG) is a rare autoimmune vasculitis typically affecting the respiratory tract and kidneys.
  • Pituitary involvement in WG is uncommon, and its presentation with acute anterior pituitary failure is exceptionally rare.

Observation:

  • A young female presented with acute hypoglycaemia, later diagnosed with panhypopituitarism and an inflammatory pituitary mass.
  • Initial investigations, including cANCA, were negative, complicating the diagnosis.
  • The patient developed progressive vision loss, diarrhoea, rash, scleritis, and proteinuria, with subsequent positive cANCA serology.

Findings:

  • The pituitary mass showed lymphocytic infiltration, consistent with an autoimmune process.
  • The patient responded to immunosuppressive therapy (steroids and cyclophosphamide).

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  • This case represents the first documented instance of WG presenting primarily with acute anterior pituitary failure without initial signs of other organ involvement.

    • Implications:

    • Highlights the importance of considering WG in patients with unexplained pituitary dysfunction, even with initially negative serology.
    • Suggests that early recognition and aggressive immunosuppression can alter the disease course.
    • Underscores the diverse and potentially misleading initial presentations of WG, necessitating a broad differential diagnosis.