Skip to main content
Top
Published in: BMC Musculoskeletal Disorders 1/2021

Open Access 01-12-2021 | Pituitary Adenoma | Case report

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

Authors: Daisuke Kamakura, Katsunori Fukutake, Kazumasa Nakamura, Shintaro Tsuge, Keiji Hasegawa, Naobumi Tochigi, Akihito Wada, Tetsuo Mikami, Hiroshi Takahashi

Published in: BMC Musculoskeletal Disorders | Issue 1/2021

Login to get access

Abstract

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.
Literature
1.
go back to reference Key C. On paraplegia depending on disease of the ligaments of the spine. Guys Hosp Rep. 1838;3:17–34. Key C. On paraplegia depending on disease of the ligaments of the spine. Guys Hosp Rep. 1838;3:17–34.
2.
go back to reference Tsukimoto H. A case report: autopsy of syndrome of compression of spinal cord owing to ossification within spinal canal of cervical spines. Nippon Geka Hokan. 1960;29:1003–7. Tsukimoto H. A case report: autopsy of syndrome of compression of spinal cord owing to ossification within spinal canal of cervical spines. Nippon Geka Hokan. 1960;29:1003–7.
3.
go back to reference Breidahl P. Ossification of the posterior longitudinal ligament in the cervical spine. “the Japanese disease” occurring in patients of British descent. Australas Radiol. 1969;13(3):311–3.CrossRef Breidahl P. Ossification of the posterior longitudinal ligament in the cervical spine. “the Japanese disease” occurring in patients of British descent. Australas Radiol. 1969;13(3):311–3.CrossRef
4.
go back to reference Yoshimura N, Nagata K, Muraki S, Oka H, Yoshida M, Enyo Y, et al. Prevalence and progression of radiographic ossification of the posterior longitudinal ligament and associated factors in the Japanese population: a 3-year follow-up of the ROAD study. Osteoporos Int. 2014;25(3):1089–98.CrossRef Yoshimura N, Nagata K, Muraki S, Oka H, Yoshida M, Enyo Y, et al. Prevalence and progression of radiographic ossification of the posterior longitudinal ligament and associated factors in the Japanese population: a 3-year follow-up of the ROAD study. Osteoporos Int. 2014;25(3):1089–98.CrossRef
5.
go back to reference Yan L, Gao R, Liu Y, He B, Lv S, Hao D. The pathogenesis of ossification of the posterior longitudinal ligament. Aging Dis. 2017;8(5):570–82.CrossRef Yan L, Gao R, Liu Y, He B, Lv S, Hao D. The pathogenesis of ossification of the posterior longitudinal ligament. Aging Dis. 2017;8(5):570–82.CrossRef
6.
go back to reference Ikegawa S, Kurokawa T, Hizuka N, Hoshino Y, Ohnishi I, Shizume K. Increase of serum growth hormone-binding protein in patients with ossification of the posterior longitudinal ligament of the spine. Spine. 1993;18(13):1757–60.CrossRef Ikegawa S, Kurokawa T, Hizuka N, Hoshino Y, Ohnishi I, Shizume K. Increase of serum growth hormone-binding protein in patients with ossification of the posterior longitudinal ligament of the spine. Spine. 1993;18(13):1757–60.CrossRef
7.
go back to reference Goto K, Yamazaki M, Tagawa M, Goto S, Kon T, Moriya H, et al. Involvement of insulin-like growth factor I in development of ossification of the posterior longitudinal ligament of the spine. Calcif Tissue Int. 1998;62(2):158–65.CrossRef Goto K, Yamazaki M, Tagawa M, Goto S, Kon T, Moriya H, et al. Involvement of insulin-like growth factor I in development of ossification of the posterior longitudinal ligament of the spine. Calcif Tissue Int. 1998;62(2):158–65.CrossRef
8.
go back to reference Lavrentaki A, Paluzzi A, Wass JA, Karavitaki N. Epidemiology of acromegaly: review of population studies. Pituitary. 2017;20(1):4–9.CrossRef Lavrentaki A, Paluzzi A, Wass JA, Karavitaki N. Epidemiology of acromegaly: review of population studies. Pituitary. 2017;20(1):4–9.CrossRef
9.
go back to reference Hoskuldsdottir GT, Fjalldal SB, Sigurjonsdottir HA. The incidence and prevalence of acromegaly, a nationwide study from 1955 through 2013. Pituitary. 2015;18(6):803–7.CrossRef Hoskuldsdottir GT, Fjalldal SB, Sigurjonsdottir HA. The incidence and prevalence of acromegaly, a nationwide study from 1955 through 2013. Pituitary. 2015;18(6):803–7.CrossRef
10.
go back to reference Stuber JL, Palacios E. Vertebral scalloping in acromegaly. Am J Roentgenol Radium Therapy, Nucl Med. 1971;112(2):397–400.CrossRef Stuber JL, Palacios E. Vertebral scalloping in acromegaly. Am J Roentgenol Radium Therapy, Nucl Med. 1971;112(2):397–400.CrossRef
11.
go back to reference Bluestone R, Bywaters EG, Hartog M, Holt PJ, Hyde S. Acromegalic arthropathy. Ann Rheum Dis. 1971;30(3):243–58.CrossRef Bluestone R, Bywaters EG, Hartog M, Holt PJ, Hyde S. Acromegalic arthropathy. Ann Rheum Dis. 1971;30(3):243–58.CrossRef
12.
go back to reference Li H, Jiang LS, Dai LY. Hormones and growth factors in the pathogenesis of spinal ligament ossification. Eur Spine J. 2007;16(8):1075–84.CrossRef Li H, Jiang LS, Dai LY. Hormones and growth factors in the pathogenesis of spinal ligament ossification. Eur Spine J. 2007;16(8):1075–84.CrossRef
13.
go back to reference Scarpa R, De Brasi D, Pivonello R, Marzullo P, Manguso F, Sodano A, et al. Acromegalic axial arthropathy: a clinical case-control study. J Clin Endocrinol Metab. 2004;89(2):598–603.CrossRef Scarpa R, De Brasi D, Pivonello R, Marzullo P, Manguso F, Sodano A, et al. Acromegalic axial arthropathy: a clinical case-control study. J Clin Endocrinol Metab. 2004;89(2):598–603.CrossRef
14.
go back to reference Nishimura S, Nagoshi N, Iwanami A, Takeuchi A, Hirai T, Yoshii T, et al. Prevalence and distribution of diffuse idiopathic skeletal hyperostosis on whole-spine computed tomography in patients with cervical ossification of the posterior longitudinal ligament: a multicenter study. Clin Spine Surg. 2018;31(9):E460–5.CrossRef Nishimura S, Nagoshi N, Iwanami A, Takeuchi A, Hirai T, Yoshii T, et al. Prevalence and distribution of diffuse idiopathic skeletal hyperostosis on whole-spine computed tomography in patients with cervical ossification of the posterior longitudinal ligament: a multicenter study. Clin Spine Surg. 2018;31(9):E460–5.CrossRef
15.
go back to reference Schmidt RF, Goldstein IM, Liu JK. Ossified ligamentum flavum causing spinal cord compression in a patient with acromegaly. J Clin Neurosci. 2013;20(11):1599–603.CrossRef Schmidt RF, Goldstein IM, Liu JK. Ossified ligamentum flavum causing spinal cord compression in a patient with acromegaly. J Clin Neurosci. 2013;20(11):1599–603.CrossRef
16.
go back to reference Berg C, Petersenn S, Lahner H, Herrmann BL, Buchfelder M, Droste M, et al. Cardiovascular risk factors in patients with uncontrolled and long-term acromegaly: comparison with matched data from the general population and the effect of disease control. J Clin Endocrinol Metab. 2010;95(8):3648–56.CrossRef Berg C, Petersenn S, Lahner H, Herrmann BL, Buchfelder M, Droste M, et al. Cardiovascular risk factors in patients with uncontrolled and long-term acromegaly: comparison with matched data from the general population and the effect of disease control. J Clin Endocrinol Metab. 2010;95(8):3648–56.CrossRef
17.
go back to reference Fatti LM, Scacchi M, Pincelli AI, Lavezzi E, Cavagnini F. Prevalence and pathogenesis of sleep apnea and lung disease in acromegaly. Pituitary. 2001;4(4):259–62.CrossRef Fatti LM, Scacchi M, Pincelli AI, Lavezzi E, Cavagnini F. Prevalence and pathogenesis of sleep apnea and lung disease in acromegaly. Pituitary. 2001;4(4):259–62.CrossRef
18.
go back to reference Dal J, Leisner MZ, Hermansen K, Farkas DK, Bengtsen M, Kistorp C, et al. Cancer incidence in patients with acromegaly: a cohort study and meta-analysis of the literature. J Clin Endocrinol Metab. 2018;103(6):2182–8.CrossRef Dal J, Leisner MZ, Hermansen K, Farkas DK, Bengtsen M, Kistorp C, et al. Cancer incidence in patients with acromegaly: a cohort study and meta-analysis of the literature. J Clin Endocrinol Metab. 2018;103(6):2182–8.CrossRef
19.
go back to reference Holdaway IM, Rajasoorya RC, Gamble GD. Factors influencing mortality in acromegaly. J Clin Endocrinol Metab. 2004;89(2):667–74.CrossRef Holdaway IM, Rajasoorya RC, Gamble GD. Factors influencing mortality in acromegaly. J Clin Endocrinol Metab. 2004;89(2):667–74.CrossRef
20.
go back to reference Holdaway IM. Excess mortality in acromegaly. Horm Res. 2007;68(Suppl 5):166–72.PubMed Holdaway IM. Excess mortality in acromegaly. Horm Res. 2007;68(Suppl 5):166–72.PubMed
21.
go back to reference Arosio M, Reimondo G, Malchiodi E, Berchialla P, Borraccino A, De Marinis L, et al. Predictors of morbidity and mortality in acromegaly: an Italian survey. Eur J Endocrinol. 2012;167(2):189–98.CrossRef Arosio M, Reimondo G, Malchiodi E, Berchialla P, Borraccino A, De Marinis L, et al. Predictors of morbidity and mortality in acromegaly: an Italian survey. Eur J Endocrinol. 2012;167(2):189–98.CrossRef
22.
go back to reference Rajasoorya C, Holdaway IM, Wrightson P, Scott DJ, Ibbertson HK. Determinants of clinical outcome and survival in acromegaly. Clin Endocrinol. 1994;41(1):95–102.CrossRef Rajasoorya C, Holdaway IM, Wrightson P, Scott DJ, Ibbertson HK. Determinants of clinical outcome and survival in acromegaly. Clin Endocrinol. 1994;41(1):95–102.CrossRef
23.
go back to reference Kasuki L, Rocha PDS, Lamback EB, Gadelha MR. Determinants of morbidities and mortality in acromegaly. Arch Endocrinol Metab. 2019;63(6):630–7.CrossRef Kasuki L, Rocha PDS, Lamback EB, Gadelha MR. Determinants of morbidities and mortality in acromegaly. Arch Endocrinol Metab. 2019;63(6):630–7.CrossRef
24.
go back to reference Nachtigall L, Delgado A, Swearingen B, Lee H, Zerikly R, Klibanski A. Changing patterns in diagnosis and therapy of acromegaly over two decades. J Clin Endocrinol Metab. 2008;93(6):2035–41.CrossRef Nachtigall L, Delgado A, Swearingen B, Lee H, Zerikly R, Klibanski A. Changing patterns in diagnosis and therapy of acromegaly over two decades. J Clin Endocrinol Metab. 2008;93(6):2035–41.CrossRef
Metadata
Title
Acromegaly presenting with myelopathy due to ossification of posterior longitudinal ligament: a case report
Authors
Daisuke Kamakura
Katsunori Fukutake
Kazumasa Nakamura
Shintaro Tsuge
Keiji Hasegawa
Naobumi Tochigi
Akihito Wada
Tetsuo Mikami
Hiroshi Takahashi
Publication date
01-12-2021
Publisher
BioMed Central
Published in
BMC Musculoskeletal Disorders / Issue 1/2021
Electronic ISSN: 1471-2474
DOI
https://doi.org/10.1186/s12891-021-04232-6

Other articles of this Issue 1/2021

BMC Musculoskeletal Disorders 1/2021 Go to the issue