Stubborn hiccups as a sign of massive apoplexy in a naive acromegaly patient with pituitary macroadenoma

Abstract

Pituitary apoplexy (PA) may very rarely present with hiccups. A 32-year-old man with classical acromegaloid features was admitted with headache, nausea, vomiting and stubborn hiccups. Pituitary magnetic resonance imaging (MRI) demonstrated apoplexy of a macroadenoma with suprasellar extension abutting the optic chiasm. Plasma growth hormone (GH) levels exhibited suppression (below <1 ng/mL) at all time points during GH suppression test with 75 g oral glucose. After treatment with corticosteroid agents, he underwent transsphenoidal pituitary surgery and hiccups disappeared postoperatively. The GH secretion potential of the tumor was clearly demonstrated immunohistochemically. We conclude that stubborn hiccups in a patient with a pituitary macroadenoma may be a sign of massive apoplexy that may result in hormonal remission.Learning points:Patients with pituitary apoplexy may rarely present with hiccups.Stubborn hiccupping may be a sign of generalized infarction of a large tumor irritating the midbrain.Infarction can be so massive that it may cause cessation of hormonal overproduction and result in remission.

Pituitary Apoplexy in Patients with Pituitary Neuroendocrine Tumors (PitNET)

“…In terms of clinical presentation, Cushingoid features (corticotroph PitNET) are reported in 5 studies (less than 0.005% of the patients): two males of 30, respective 33 years [ 31 , 186 ], a cases series of 4 females aged between 26 and 45 years [ 33 ], and other two women of 35, respective 47 years [ 86 , 161 ]. Acromegaly (caused by somatotroph or lactosomatotroph PitNET) was recognized in 8 patients (0.005%) aged between 26 and 50 years; a male [ 86 , 109 , 136 , 148 , 156 , 217 ] to female [ 38 , 78 ] ratio of 6 to 2. One case of gigantism (+5 SD) was reported on a 9-year-old boy with lactosomatotroph PitNET [ 32 ].…”

“…Other signs and symptoms at PA presentation include lactotroph PitNET-associated galactorrhea [ 141 , 174 , 183 ]; hyperthermia [ 180 ], pruritic skin rash associated with central adrenal insufficiency-related cortisol deficit [ 185 ], hiccups [ 109 ], epistaxis [ 91 ], hematuria [ 189 ], phonophobia [ 78 ], visual illusions [ 45 ], and symptomatic diabetic ketoacidosis [ 53 , 171 , 172 ].…”

“…In terms of the PitNET stain profile (regardless clinical expression) non-functional type was followed by somatotroph PitNETs [ 32 , 38 , 42 , 46 , 53 , 56 , 66 , 76 , 78 , 86 , 92 , 93 , 98 , 100 , 108 , 109 , 121 , 128 , 136 , 141 , 148 , 152 , 156 , 166 , 168 , 169 , 171 , 175 , 192 , 217 ], lactotroph PitNETs [ 66 , 75 , 77 , 90 , 98 , 100 , 113 , 121 , 126 , 128 , 141 , 166 , 168 , 169 , 174 , 175 , 183 , 202 , 206 ], gonadotroph PitNETs […”

“…A large number of patients required hormonal replacement due to PA-associated hypopituitarism manifested as hypocortisolism which is the most important aspect in terms of an immediate life-threatening approach [ 25 , 29 , 30 , 31 , 56 , 65 , 66 , 75 , 80 , 90 , 92 , 123 , 128 , 144 , 163 , 168 , 169 , 208 ], central hypothyroidism [ 25 , 56 , 65 , 66 , 72 , 75 , 101 , 109 , 123 , 144 , 163 , 168 , 169 , 180 , 184 , 200 ], and hypogonadotropic hypogonadism [ 25 , 56 , 65 , 66 , 72 , 109 , 128 , 168 , 169 , 180 , 198 ]. GH deficiency was also reported [ 109 , 128 , 1...…”