Literature DB >> 20157722

Flexion contractures possibly reflect the existence of hypocortisolism: two case reports.

Viorica Harbuz1, Hélène Bihan, Jeffrey Salama, Gérard Reach, Régis Cohen.   

Abstract

The diagnosis of adrenal insufficiency is discussed in case of low blood pressure and digestive symptoms. Rare inaugural abdomino-crural muscle contracture can be a misleading symptom. Here we report two new cases. A 50-year-old woman presenting a leaning forward walking attitude and negligence for the past 2 months was referred to the neurologic unit. Abdomino-crural contracture, clinical hypogonadism, and hyponatremia directed towards a panhypopituitarism, which was confirmed by subsequent investigations. Pituitary MRI found an empty sella turcica. The outcome was dramatic after hormone replacement therapy, with drawing up of the trunk and re-establishment of walking after a few days. The second case is a 58-year-old man, hospitalized with altered general condition, with a weight loss of 22 kg, and anorexia in the aftermath of a comminuted fracture of the right lower limb, complicated by pseudoarthrosis. There was amyotrophy on the extremities with intense cruralgia. The patient had an antalgic attitude in the flexion affecting his rehabilitation. During 1 year, the symptoms were mimicking psychiatric disorders, malabsorption, or cancer before the final diagnosis of central hypocorticism with normal MRI was established. The evolution was remarkable after a few days of therapy with hydrocortisone, where the myalgia disappeared, the patient quickly gained weight, and the disappearance of the retractions allowed rehabilitation. These two observations emphasize the delayed diagnosis of adrenal insufficiency in the case of abdomino-crural-related symptoms and the presence of misleading neurological symptoms. The mechanisms of this syndrome remain unknown.

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Year:  2010        PMID: 20157722     DOI: 10.1007/s00415-010-5477-8

Source DB:  PubMed          Journal:  J Neurol        ISSN: 0340-5354            Impact factor:   4.849


  12 in total

Review 1.  Flexion contractures possibly reflect the existence of hypocortisolism.

Authors:  Tetsuo Nishikawa
Journal:  Intern Med       Date:  2003-08       Impact factor: 1.271

2.  Flexion contractures in Addison's disease.

Authors:  P E WISENBAUGH; H M HELLER
Journal:  J Clin Endocrinol Metab       Date:  1960-05       Impact factor: 5.958

3.  Hyperkalemic neuromyopathy in Addison's disease.

Authors:  R H POLLEN; R H WILLIAMS
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4.  Quadriceps muscle function and fatigue in women with Addison's disease.

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Journal:  Muscle Nerve       Date:  2001-08       Impact factor: 3.217

5.  Isolated adrenocorticotropin deficiency and flexion contractures syndrome.

Authors:  Vassiliki Syriou; Anestis Moisidis; Nikolaos Tamouridis; Krystallenia I Alexandraki; Margarita Anapliotou
Journal:  Hormones (Athens)       Date:  2008 Oct-Dec       Impact factor: 2.885

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Journal:  Ann Endocrinol (Paris)       Date:  1951       Impact factor: 2.478

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Authors:  R L Blandford; A K Samanta; A C Burden; F D Rosenthal
Journal:  Br Med J (Clin Res Ed)       Date:  1985-07-13

8.  Resolution of stiff-man syndrome with cortisol replacement in a patient with deficiencies of ACTH, growth hormone, and prolactin.

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Journal:  N Engl J Med       Date:  1984-06-07       Impact factor: 91.245

9.  Myopathy in Addison's disease.

Authors:  F Mor; P Green; A J Wysenbeek
Journal:  Ann Rheum Dis       Date:  1987-01       Impact factor: 19.103

10.  'Dementia with contractures' as presenting signs of secondary adrenocortical insufficiency.

Authors:  J J van der Sande; A P van Seters; A R Wintzen
Journal:  Clin Neurol Neurosurg       Date:  1986       Impact factor: 1.876

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  3 in total

1.  Hypopituitarism due to primary empty sella and uncommon muscular symptoms.

Authors:  Enrique Anton
Journal:  Rheumatol Int       Date:  2011-01-22       Impact factor: 2.631

2.  Treatable Bedridden Elderly -Recovery from Flexion Contracture after Cortisol Replacement in a Patient with Isolated Adrenocorticotropic Hormone Deficiency.

Authors:  Takamasa Tanaka; Norihiko Terada; Yoshiki Fujikawa; Takushi Fujimoto
Journal:  Intern Med       Date:  2016-10-15       Impact factor: 1.271

3.  Stiff Person-Like Syndrome: An Unusual Presentation of Pituitary Macroadenoma with Panhypopituitarism.

Authors:  Kian Guan Goh; Abdul Hanif Khan Yusof Khan; Azraai Nasruddin
Journal:  Case Rep Neurol       Date:  2022-03-18
  3 in total

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