Table 2.
Clinical characteristic of relapsing of Immunoglobulin G4 (IgG4)-related hypophysitis during steroid therapy
| Author, year | Sex, age, race | Manifestations before steroid treatment | Steroid type and dosage | Symptoms and signs after steroid treatment | Treatment after relapse and result |
|---|---|---|---|---|---|
| Taniguchi, 2006 [11] | M 75 y, Japanese | Autoimmune pancreatitis, uveitis, organizing pneumonia, panhypopituitarism with mass (Lymphocytic hypophysitis)b | Pred 50 mg/day for 1 week then taper to 10 mg/day within 7 months | Recurrent pituitary mass and organizing pneumonia | Pred 50 mg/day then 20 mg/day, contracted pituitary mass |
| Haragushi, 2010 [18] | M 68 y, Japanese | Diabetes insipidus and gradual loss of anterior pituitary function with mass (Lymphocitic hypophysitis)b, retroperitoneal fibrosis | Hydrocortisone replacement for 4 years | Headache, pituitary swelling | Pred 30 mg/day for 2 weeks then 10 mg/day, decrease pituitary swelling |
| Leporati, 2011 [2] | M 75 y, Caucasian | Panhypopituitarism with mass, sphenoid mass | Pred 40 mg/day taper to 10 mg/day over 4 weeks | Recurrent headache | Pred 15 mg/day, then taper/reescalation and suspend until 1.3 years, improved headache but hypopituitarism |
| Caputo, 2014 [27] | M 40 y, Vietnamese | Lacrimal gland mass, diabetes insipidus, panhypopituitarism with mass (Lymphocytic hypophysitis)a, enlarged infraorbital nerve | Pred 30 mg/day for 3 months | Enlarging pituitary mass with new optic nerve compression | Azathioprine 75 mg twice daily whilst weaning Pred for 10 months, recovery from adrenal insufficiency and growth hormone deficiency, ongoing bilaterally enlarged infraorbital nerves but normal pituitary size |
| Ohkubo, 2013 [6] | M 70 y, Japanese | Hashimoto’s thyroiditis, pancreatic and retroperitoneal mass, salivary gland enlargement, pituitary mass, bitemporal hemianopsia (Lymphocytic hypophysitis)a | Pred 40 mg/day for 2 weeks then taper to 5 mg/day | Reduction in pituitary lesion, improved vision but new DI and panhypopituitarism | Pred 30 mg/day then taper to 5 mg/day for 2 months, no further pituitary mass reduction and did not restore the pituitary function |
| Hydrocortisone 100 mg on day of surgery + Pred 5 mg/day for 1 month | |||||
| Ngaosuwan, 2015 (the presented case) | M 43 y, Thai | Frontal lobe seizure, multiple pituitary hormone deficiency with pituitary mass (Lymphocytic hypophysitis)a | Pred 15 mg/day for 6 weeks, 10 mg/day for 3 months, and 7.5 mg/day for 3 months | Bitemporal hemianopsia, Headache, Inflammation of optic chiasm | Pred 60 mg/day for 2 weeks, gradually decrease to 30 mg/day for 4 weeks, 20 mg/day for 4 weeks, and 10 mg/day for maintenance, complete recovery of vision, contracted pituitary mass, decreased optic chiasmatic swelling, but did not restore pituitary function |
Pred Prednisolone, mg milligrams
aInitial diagnosis based on initial histopathology
bInitial diagnosis based on clinical manifestation and imaging