Syndrome of inappropriate secretion of anti-diuretic hormone due to hypothalamic hamartoma: use of tolvaptan
Syndrome of inappropriate secretion of anti-diuretic hormone due to hypothalamic hamartoma: use of tolvaptan
Objectives: hypothalamic hamartoma (HH) typically presents with gonadotrophin-dependent precocious puberty and/or seizures. Other endocrine disturbances are rare. We describe an infant with syndrome of inappropriate secretion of anti-diuretic hormone (SIADH) and a HH.
Case presentation: a 6-week-old infant presented with seizures and life-threatening hyponatremia. A HH was identified on magnetic resonance imaging. Clinical examination and biochemistry were consistent with SIADH, and serum copeptin was high during hyponatremia, further supporting this diagnosis. Tolvaptan was effective in normalizing plasma sodium and enabling liberalization of fluids to ensure sufficient nutritional intake and weight gain and manage hunger.
Conclusions: hyponatremia due to SIADH is novel at presentation of a HH, and can be challenging to diagnose and manage. Successful management of hyponatremia in this case was achieved using tolvaptan.
hypothalamic hamartoma, syndrome of inappropriate secretion of antidiuretic hormone (SIADH), tolvaptan
895-899
Moon, Rebecca
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Soliman, Maisara
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Hoogenboom, Lieke
d2381f5d-39a6-4e27-8d83-ef49ee978e7a
Gilbert, Rodney D
c69c5b99-2a15-4502-acd8-36b254d42601
Bird-Lieberman, Georgina
2c3fc13d-ec5c-4efa-8f08-e6bcccd9e4c4
Singh, Jaspal
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Bockenhauer, Detlef
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Kumaran, Anitha
c7880b22-4f22-4d76-9272-0b21f7778192
1 September 2023
Moon, Rebecca
954fb3ed-9934-4649-886d-f65944985a6b
Soliman, Maisara
17b54a47-aad3-4b7e-bbaa-7c276ffcf038
Hoogenboom, Lieke
d2381f5d-39a6-4e27-8d83-ef49ee978e7a
Gilbert, Rodney D
c69c5b99-2a15-4502-acd8-36b254d42601
Bird-Lieberman, Georgina
2c3fc13d-ec5c-4efa-8f08-e6bcccd9e4c4
Singh, Jaspal
f6abef14-c81d-421b-8671-1c271833baa3
Bockenhauer, Detlef
81f044d1-f60b-4a20-a9a5-a91b0c1871c4
Kumaran, Anitha
c7880b22-4f22-4d76-9272-0b21f7778192
Moon, Rebecca, Soliman, Maisara, Hoogenboom, Lieke, Gilbert, Rodney D, Bird-Lieberman, Georgina, Singh, Jaspal, Bockenhauer, Detlef and Kumaran, Anitha
(2023)
Syndrome of inappropriate secretion of anti-diuretic hormone due to hypothalamic hamartoma: use of tolvaptan.
Journal of Pediatric Endocrinology and Metabolism, 36 (9), .
(doi:10.1515/jpem-2023-0136).
Abstract
Objectives: hypothalamic hamartoma (HH) typically presents with gonadotrophin-dependent precocious puberty and/or seizures. Other endocrine disturbances are rare. We describe an infant with syndrome of inappropriate secretion of anti-diuretic hormone (SIADH) and a HH.
Case presentation: a 6-week-old infant presented with seizures and life-threatening hyponatremia. A HH was identified on magnetic resonance imaging. Clinical examination and biochemistry were consistent with SIADH, and serum copeptin was high during hyponatremia, further supporting this diagnosis. Tolvaptan was effective in normalizing plasma sodium and enabling liberalization of fluids to ensure sufficient nutritional intake and weight gain and manage hunger.
Conclusions: hyponatremia due to SIADH is novel at presentation of a HH, and can be challenging to diagnose and manage. Successful management of hyponatremia in this case was achieved using tolvaptan.
Text
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Accepted/In Press date: 5 June 2023
e-pub ahead of print date: 19 June 2023
Published date: 1 September 2023
Additional Information:
Funding Information:
Research funding : No specific funding for this study received. RJM is funded by an NIHR Academic Clinical Lectureship.
Publisher Copyright:
© 2023 Walter de Gruyter GmbH, Berlin/Boston.
Keywords:
hypothalamic hamartoma, syndrome of inappropriate secretion of antidiuretic hormone (SIADH), tolvaptan
Identifiers
Local EPrints ID: 478988
URI: http://eprints.soton.ac.uk/id/eprint/478988
ISSN: 0334-018X
PURE UUID: 1d11496d-880a-4fc8-b0c3-b8b4c86e9e8f
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Date deposited: 17 Jul 2023 16:39
Last modified: 18 Apr 2024 01:54
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Contributors
Author:
Rebecca Moon
Author:
Maisara Soliman
Author:
Lieke Hoogenboom
Author:
Rodney D Gilbert
Author:
Georgina Bird-Lieberman
Author:
Jaspal Singh
Author:
Detlef Bockenhauer
Author:
Anitha Kumaran
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