Xanthomatous hypophysitis causing hypogonadotropic hypogonadism resulting in delayed presentation of slipped capital femoral epiphysis

Kirit Singh, Avinash Kumar Kanodia, Peter Ross, Antonia Torgersen, Jamie Maclean, Graham Leese, Kismet Hossain-Ibrahim

Research output: Contribution to journalArticle

Abstract

An 18-year-old man who underwent bilateral pinning of his hip joints after a left unstable Slipped Capital Femoral Epiphysis (right pinned prophylactically) was noted to have delayed secondary sexual characteristics and post-operative diabetes insipidus. The patient also described a history of fatigue, headache and polydipsia for the past 4 years. Endocrine investigations revealed reduced androgen levels, hypocortisolism, a borderline normal Serum ACE and secondary hypothyroidism. Magnetic Resonance Imaging of the pituitary gland identified an enhancing mass and a thickened stalk which trans-nasal endoscopic biopsy found to be necrotic with pus. Histology confirmed a diagnosis of Xanthomatous Hypophysitis, an inflammatory condition likely related to a partial rupture of a Rathke cleft cyst. The patient was subsequently commenced on Androgen, Thyroxine, Desmopressin and Hydrocortisone therapy with on-going endocrine follow-up. Although endocrine dysfunction & hypogonadism has been recognised to be a risk factor for SCFE at an atypically older age, due to reduced androgen levels leading to a weakened physeal plate, this is the first known case of a Xanthomatous Hypophysitis resulting in pituitary dysfunction and eventual SCFE. This case highlights that an increased range of pituitary disorders should be considered in late presentations of SCFE; and vice versa the risk of SCFE should be considered in patients with prolonged hypogonadotropic hypogonadism.

Original languageEnglish
Pages (from-to)1-4
Number of pages4
JournalBritish Journal of Neurosurgery
Early online date19 Nov 2018
DOIs
Publication statusE-pub ahead of print - 19 Nov 2018

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Slipped Capital Femoral Epiphyses
Hypogonadism
Androgens
Central Nervous System Cysts
Pituitary Diseases
Polydipsia
Deamino Arginine Vasopressin
Diabetes Insipidus
Suppuration
Hip Joint
Pituitary Gland
Hypothyroidism
Thyroxine
Nose
Fatigue
Headache
Hydrocortisone
Rupture
Histology
Magnetic Resonance Imaging

Keywords

  • diabetes insipidus
  • hypogonadotropic hypogonadism
  • slipped capital femoral epiphysis
  • xanthomatous hypophysitis

Cite this

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title = "Xanthomatous hypophysitis causing hypogonadotropic hypogonadism resulting in delayed presentation of slipped capital femoral epiphysis",
abstract = "An 18-year-old man who underwent bilateral pinning of his hip joints after a left unstable Slipped Capital Femoral Epiphysis (right pinned prophylactically) was noted to have delayed secondary sexual characteristics and post-operative diabetes insipidus. The patient also described a history of fatigue, headache and polydipsia for the past 4 years. Endocrine investigations revealed reduced androgen levels, hypocortisolism, a borderline normal Serum ACE and secondary hypothyroidism. Magnetic Resonance Imaging of the pituitary gland identified an enhancing mass and a thickened stalk which trans-nasal endoscopic biopsy found to be necrotic with pus. Histology confirmed a diagnosis of Xanthomatous Hypophysitis, an inflammatory condition likely related to a partial rupture of a Rathke cleft cyst. The patient was subsequently commenced on Androgen, Thyroxine, Desmopressin and Hydrocortisone therapy with on-going endocrine follow-up. Although endocrine dysfunction & hypogonadism has been recognised to be a risk factor for SCFE at an atypically older age, due to reduced androgen levels leading to a weakened physeal plate, this is the first known case of a Xanthomatous Hypophysitis resulting in pituitary dysfunction and eventual SCFE. This case highlights that an increased range of pituitary disorders should be considered in late presentations of SCFE; and vice versa the risk of SCFE should be considered in patients with prolonged hypogonadotropic hypogonadism.",
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Xanthomatous hypophysitis causing hypogonadotropic hypogonadism resulting in delayed presentation of slipped capital femoral epiphysis. / Singh, Kirit; Kanodia, Avinash Kumar; Ross, Peter; Torgersen, Antonia; Maclean, Jamie; Leese, Graham; Hossain-Ibrahim, Kismet.

In: British Journal of Neurosurgery, 19.11.2018, p. 1-4.

Research output: Contribution to journalArticle

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AU - Kanodia, Avinash Kumar

AU - Ross, Peter

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AU - Maclean, Jamie

AU - Leese, Graham

AU - Hossain-Ibrahim, Kismet

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