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Acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report

BACKGROUND: Acromegaly is a rare disease caused by high serum levels of growth hormone (GH) and insulin-like growth factor 1 (IGF-1), often originating from a pituitary adenoma. Spinal and peripheral joint abnormalities are caused by these hormonal hypersecretions. In particular, the response to GH...

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Autores principales: Kamakura, Daisuke, Fukutake, Katsunori, Nakamura, Kazumasa, Tsuge, Shintaro, Hasegawa, Keiji, Tochigi, Naobumi, Wada, Akihito, Mikami, Tetsuo, Takahashi, Hiroshi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8045308/
https://www.ncbi.nlm.nih.gov/pubmed/33853563
http://dx.doi.org/10.1186/s12891-021-04232-6
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author Kamakura, Daisuke
Fukutake, Katsunori
Nakamura, Kazumasa
Tsuge, Shintaro
Hasegawa, Keiji
Tochigi, Naobumi
Wada, Akihito
Mikami, Tetsuo
Takahashi, Hiroshi
author_facet Kamakura, Daisuke
Fukutake, Katsunori
Nakamura, Kazumasa
Tsuge, Shintaro
Hasegawa, Keiji
Tochigi, Naobumi
Wada, Akihito
Mikami, Tetsuo
Takahashi, Hiroshi
author_sort Kamakura, Daisuke
collection PubMed
description BACKGROUND: Acromegaly is a rare disease caused by high serum levels of growth hormone (GH) and insulin-like growth factor 1 (IGF-1), often originating from a pituitary adenoma. Spinal and peripheral joint abnormalities are caused by these hormonal hypersecretions. In particular, the response to GH is involved in the onset of ossification of the spinal ligament in vitro, especially ossification of the posterior longitudinal ligament (OPLL). However, because acromegaly and OPLL are rare diseases, we seldom encounter them in combination. To the best of our knowledge in the English-language literature, this is the first reported case of acromegaly presenting with thoracic myelopathy due to OPLL. CASE PRESENTATION: A 47-year-old woman presented with lower extremity weakness and paresthesia, gait disorder, and bladder disorder without any trauma. The patient’s most remarkable symptom was paraplegia, and we diagnosed myelopathy due to cervical and thoracic OPLL. Furthermore, we suspected acromegaly because of the characteristic facial features, and we found a pituitary adenoma by contrast-enhanced MRI. Cervical and thoracic decompression, posterior fixation, and pituitary adenoma resection were performed. CONCLUSION: We report a case of acromegaly that was detected after the diagnosis of OPLL. The main challenge in acromegaly is delayed in diagnosis. Even in this case, the facial features characteristic of acromegaly had appeared at least 9 years ago. Early diagnosis and treatment of acromegaly improve prognosis and reduce exposure to GH and IGF-1 through early intervention and seem to suppress the progression of ligament ossification. Orthopedic surgeons and neurosurgeons need to keep in mind that acromegaly is associated with bone/joint lesions and ossification of the spinal ligament and should aim to diagnose acromegaly early.
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spelling pubmed-80453082021-04-14 Acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report Kamakura, Daisuke Fukutake, Katsunori Nakamura, Kazumasa Tsuge, Shintaro Hasegawa, Keiji Tochigi, Naobumi Wada, Akihito Mikami, Tetsuo Takahashi, Hiroshi BMC Musculoskelet Disord Case Report BACKGROUND: Acromegaly is a rare disease caused by high serum levels of growth hormone (GH) and insulin-like growth factor 1 (IGF-1), often originating from a pituitary adenoma. Spinal and peripheral joint abnormalities are caused by these hormonal hypersecretions. In particular, the response to GH is involved in the onset of ossification of the spinal ligament in vitro, especially ossification of the posterior longitudinal ligament (OPLL). However, because acromegaly and OPLL are rare diseases, we seldom encounter them in combination. To the best of our knowledge in the English-language literature, this is the first reported case of acromegaly presenting with thoracic myelopathy due to OPLL. CASE PRESENTATION: A 47-year-old woman presented with lower extremity weakness and paresthesia, gait disorder, and bladder disorder without any trauma. The patient’s most remarkable symptom was paraplegia, and we diagnosed myelopathy due to cervical and thoracic OPLL. Furthermore, we suspected acromegaly because of the characteristic facial features, and we found a pituitary adenoma by contrast-enhanced MRI. Cervical and thoracic decompression, posterior fixation, and pituitary adenoma resection were performed. CONCLUSION: We report a case of acromegaly that was detected after the diagnosis of OPLL. The main challenge in acromegaly is delayed in diagnosis. Even in this case, the facial features characteristic of acromegaly had appeared at least 9 years ago. Early diagnosis and treatment of acromegaly improve prognosis and reduce exposure to GH and IGF-1 through early intervention and seem to suppress the progression of ligament ossification. Orthopedic surgeons and neurosurgeons need to keep in mind that acromegaly is associated with bone/joint lesions and ossification of the spinal ligament and should aim to diagnose acromegaly early. BioMed Central 2021-04-14 /pmc/articles/PMC8045308/ /pubmed/33853563 http://dx.doi.org/10.1186/s12891-021-04232-6 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Kamakura, Daisuke
Fukutake, Katsunori
Nakamura, Kazumasa
Tsuge, Shintaro
Hasegawa, Keiji
Tochigi, Naobumi
Wada, Akihito
Mikami, Tetsuo
Takahashi, Hiroshi
Acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report
title Acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report
title_full Acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report
title_fullStr Acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report
title_full_unstemmed Acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report
title_short Acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report
title_sort acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8045308/
https://www.ncbi.nlm.nih.gov/pubmed/33853563
http://dx.doi.org/10.1186/s12891-021-04232-6
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