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Hypopituitarism in Men with History of Hypovolemic Shock

Batista Rafael Loch , Toscanini Andrea Cecilia, Borba Clarissa Groberio, Cunha-Neto Malebranche B
American Journal of Medical Case Reports. 2018, 6(3), 50-52. DOI: 10.12691/ajmcr-6-3-3
Published online: April 24, 2018

Abstract

Hypopituitarism is the partial or complete insufficiency of pituitary hormone secretion. It has a diverse etiology, and some causes are of vascular origin. Vascular causes of hypopituitarism are classified as hemorrhagic/ischemic or obstructive/compressive. We report the case of a patient with chronic hypopituitarism found 15 years after the occurrence of hypovolemic shock during surgery performed to remove nasal polyps. We believe that hypopituitarism in this case is very similar to that described by Sheehan, resulting from blood loss and coagulation disorders. Observation of pituitary function in patients with a history of hypovolemic shock may be necessary, as it is mandatory in patients with a history of postpartum bleeding.

1. Introduction

Hypopituitarism is a medical condition related to high morbidity and mortality if not treated properly. The diagnosis of this condition is not always clear and is sometimes characterized as idiopathic. Hypopituitarism can have many causes including compression by pituitary tumors and genetic defects. Among the causes, postpartum hemorrhage with loss of pituitary function was described by Sheehan in 1938. Among these possible vascular etiologies, include vascular ischaemia after snake bite and hypopituitarism after hemorrhagic fever and hantaviruses.

Vascular causes of hypopituitarism are classified as hemorrhagic/ischemic or obstructive/compressive. Hypopituitarism of obstructive/compressive vascular origin has been associated with carotid artery aneurysms 1, in which vascular injury leads to compression of pituitary tissue associated with hormone deficiency. Among hemorrhagic/ischemic causes, coagulative necrosis in Sheehan’s syndrome is well described, although its pathogenesis has yet to be elucidated. Infarction secondary to arrest of blood flow to the anterior lobe of the pituitary gland is the basic process. It remains unclear whether this process results from vasospasm, thrombosis, or vascular compression 2. We report the case of a patient with chronic hypopituitarism found 15 years after the occurrence of hypovolemic shock during surgery performed to remove nasal polyps. The cause of hypopituitarism in this patient appears to be ischemic due to the occurrence of pituitary deficits and partially empty sella. The patient had a favorable outcome after hormone replacement and is currently in perfect health, thus preventing histopathological investigation. However, we believe that hypopituitarism in this case is very similar to that described by Sheehan, resulting from blood loss and coagulation disorders. Observation of pituitary function in patients with a history of hypovolemic shock may be necessary, as it is mandatory in patients with a history of postpartum bleeding.

2. Case Report

An 83-year-old male patient, with complaints of daily intermittent dizziness and symptomatic postural hypotension for about 4 years. The patient has a history of sinusitis, and complaints have been associated with this diagnosis without improvement. About 15 years ago, the patient developed hypovolemic shock after severe hemorrhage following surgery to remove nasal polyps. In that episode, he had blood pressure levels of 0 x 0 mmHg, the patient developed circulatory shock, with cardio respiratory arrest and was managed with cardiovascular resuscitation was treated with volume expansion and showed a good response to treatment. After some time, the patient began to experience dizziness and malaise, being diagnosed and treated as having sinusitis. Later, during routine examination, hyponatremia was detected, with serum sodium = 120 mEq/l. Given the clinical suspicion of adrenal insufficiency, the patient underwent laboratory tests, which revealed hypothyroidism, hypogonadism, low GH and IGF-1 levels, and reduced cortisol levels (Table 1). The patient underwent cosyntropin stimulation test (250 mg IV), which revealed partial corticotropin deficiency (Table 1). Magnetic resonance imaging (MRI) of sella turcica showed partially empty sella. The patient started replacement of levothyroxine, hydrocortisone acetate, and testosterone and maintains hormone levels appropriate for age.

3. Discussion

Sheehan's syndrome occurs as a result of ischemic pituitary necrosis due to severe postpartum hemorrhage. It may be rarely seen without massive bleeding or after normal delivery. Improvement in obstetric care and availability of rapid blood transfusion coincided with a remarkable reduction in the frequency of Sheehan's syndrome particularly in western society. But it has recently been reported more often from well-developed countries. It is one of the most common causes of hypopituitarism in underdeveloped or developing countries. Enlargement of pituitary gland, small sella size, disseminated intravascular coagulation and autoimmunity have been suggested to play a role in the pathogenesis of Sheehan's syndrome in women who suffer from severe postpartum hemorrhage. The patients may seek medical advice because of various presentations ranging from non-specific symptoms to coma and the clinical manifestation may change from one patient to another. Failure of postpartum lactation and failure to resume menses after delivery are the most common presenting symptoms. Although a small percentage of patients with Sheehan's syndrome may cause abrupt onset severe hypopituitarism immediately after delivery, most patients have a mild disease and go undiagnosed and untreated for a long time. It may result in partial or panhypopituitarism and most patients has empty sella on CT or MRI 3, 4.

Sheehan observed excessive fibrin on blood vessel of patients and named it in situ thrombosis 5. Although an enlarged pituitary gland during pregnancy may become more susceptible to ischemia, primary thrombosis appears to be a possible explanation for pituitary ischemia in these patients 3. The placenta is a rich source of thromboplastin, an activator of prothrombin responsible for defibrination which may result in hypofibrinogenemia and risk of abruptio placentae. In late pregnancy, disseminated intravascular coagulation (DIC) may also be triggered by leakage of amniotic fluid or syncytiotrophoblastic microparticles containing functional tissue factor into the mother’s bloodstream 4. As a consequence, fibrin may be deposited on blood vessel walls, which explains the anatomical changes observed by Sheehan in the classical description of hypopituitarism after postpartum bleeding 5.

Snake bites may cause hypopituitarism by procoagulant enzymes in venom causing coagulative necrosis of the anterior pituitary. The first case of snake bite causing hypopituitarism was described in 1958 in Brazil 6. Later, in 1976, hypopituitarism following Russell’s viper bite 7, a common type of snake in South and Southeast Asian countries, was described. Since then, 50 cases of hypopituitarism following Russell’s viper bite (Daboia russelii and Daboia siamensis) have been reported. Russell’s viper venom is known to contain several toxins and some biologically active procoagulant enzymes activating factors V and X, resulting in fibrin formation, which may be associated with DIC 8. Russell’s viper venom also contains “hemorrhagin”, a metalloproteinase that damages vascular endothelium. Thus, venom may lead to thromboses and hemorrhages. Focal hemorrhage and microvascular fibrin deposition in the pituitary gland may be responsible for the pathological finding of infarction of the anterior pituitary 3.

Hypopituitarism associated with hemorrhagic fever with renal syndrome has been described 9. This viral infection is caused by hantaviruses, and its clinical course is characterized by fever, circulatory collapse with hypotension, hemorrhages, and renal failure 10. There is capillary leakage, suggesting that vascular endothelium is the main target of this infection. Autopsy reports show that in fatal cases there was slight enlargement of the pituitary gland, with signs of hemorrhage and necrotic foci in 75.5% of cases. Cases of survivors developing hypopituitarism revealed pituitary atrophy and empty sella. In these cases, there is shock, vascular collapse, and subsequent hypopituitarism 8.

Based on different etiologies, we have observed that pituitary necrosis, whether in patients with Sheehan’s syndrome, in patients envenomed by Russell’s viper or in patients with hemorrhagic fever with renal syndrome, presents circulatory abnormalities, often with aggravating factors such as prolonged circulatory collapse, DIC, hyperhydration, and coagulation disorders. Therefore, severe circulatory abnormalities appear to be suggestive of situations of risk for the occurrence of hypopituitarism, even in the long term. The hypopituitarism after ventricular arrhythmia has been demonstrated in a pilot study that included 44 patients which was observed GH deficiency in 27.2% of long-term patients, supporting the hypothesis that blood reduced blood flow can relate to late hypopituitarism 11.

In cases of GH deficiency suspicion, this diagnosis should be done by hypoglycemia test after insulin. However, this test is contraindicated in elderly patients and / or patients with heart disease and in those with epilepsy. The GH replacement in adults is possible, but the cost / benefit ratio is not well established. For this reason we do not do this test in our patient, despite the low level of IGF-1 12.

An empty sella is defined as a sella which, regardless of its size, is completely or partly filled with cerebrospinal fluid (CSF). Empty sella is occasionally found as a normal anatomical variation, which is referred to as primary empty sella. On the other hand, empty sella is also seen after surgery, irradiation or medical treatment of pituitary adenoma, which is called secondary empty sella. Magnetic resonance imaging (MRI) is useful in diagnosing empty sella. Primary empty sella is usually free from clinical symptoms but sometimes associated with headache, obesity, visual disturbance, non-traumatic CSF rhinorrhea and pituitary insufficiency. These associated findings constitute the empty sella syndrome. Non-symptomatic cases require no treatment but periodical follow up is necessary 13, 14.

In terms of differential diagnosis, lymphocytic hypophysitis should be kept in mind. However, this etiology predominantly affects women between the third and fourth decades of life, and often occurs after pregnancy 15. Another possibility is a ocorrence of Empty Sella Syndrome. At this condition, infarction clinically silent pituitary mass may result in the development of a partial or completely empty sella. The pituitary function often remains intact, because the surrounding tissue is fully functional. The hypopituitarism, however, may develop insidiously. Further, the increased pressure can promote intracranina herniation arachnoid membrane into the pituitary fossa 16.

Although elderly patients may have a history of falls and fractures more frequently, this was not reported by the patient or by relatives. Hypopituitarism cases after trauma are common, but relate the severity of trauma and is usually not observed empty sella in these patients 17. We believe that, in this patient, the etiology of hypopituitarism was due to circulatory shock, similar to hypopituitarism described by Sheehan. However, systematic studies are needed so that we can establish a causal relationship between hypovolemic shock and hypopituitarism. The establishment of this relationship increases the diagnostic possibilities of vascular origin of hypopituitarism.

4. Conclusion

This case shows the detection of chronic hypopituitarism with a deficiency of the thyrotropic, gonadotropic and somatotropic axes, in addition to partial deficit of the corticotropin axis in an elderly patient with a history of bleeding with progression to hypovolemic shock. We believe that in this case hypopituitarism was caused by pituitary ischemia, similar to cases of hypopituitarism syndrome after postpartum bleeding, as characterized by Sheehan. It may be important to monitor pituitary function in patients with a history or symptoms of hypovolemic shock, even after the acute phase of shock.

Declaration of Interest

The authors declare that there is no conflict of interest that could be perceived as prejudicing the imparciality of the research reported.

Funding

This research did not receive any specific grant from any funding agency in the public, comercial or not-for-profit sector.

References

[1]  Castinetti F, Reynaud R, Quentien MH, Jullien N, Marquant E, Rochette C, Herman JP, Saveanu A, Barlier A, Enjalbert A, Brue T. Combined pituitary hormone deficiency: current and future status. J Endocrinol Invest. 2014 Sep 9.
In article      PubMed
 
[2]  Wilson V, Mallipedhi A, Stephens JW, Redfern RM, Price DE. The causes of hypopituitarism in the absence of abnormal pituitary imaging. QJM Jan; 107(1): 21-4, 2014.
In article      View Article
 
[3]  Kelestimur F. Sheehan’s syndrome. Pituitary 6(4):181-8, 2003.
In article      View Article  PubMed
 
[4]  Mir SA, Masoodi SR, Wani AI, Farooqui KJ, Bashir MI. Deep vein thrombosis in a patient of Sheehan's syndrome: Autoimmunity or hypercoagulabilty. Indian J Endocrinol Metab, Oct; 17(Suppl 1):S105-6, 2013.
In article      View Article
 
[5]  Sheehan HL. Post-partum necrosis of the anterior pituitary. J Pathol Bacteriol 1937, 45:189-214, 1937.
In article      View Article
 
[6]  Wolff H. Insuficiência hipofisária anterior por picada de ofídio. Arq Bras Endocrinol Metabol 7:25-47, 1958.
In article      
 
[7]  Eapen CK, Chandy N, Kochuovarkey KL, et al. Unusual com- plication of snake bite: hypopituitarism after viper bites. In: Ohsaka A, Hayashi K, Sawai Y, eds. Animal, plant and microbial toxins, vol 2. New York, NY, Plenum Press, 467–73, 1976.
In article      View Article
 
[8]  Antonypillai CN, Wass JA, Warrell DA, Rajaratnam HN. Hypopituitarism following envenoming by Russell's vipers (Daboia siamensis and D. russelii) resembling Sheehan's syndrome: first case report from Sri Lanka, a review of the literature and recommendations for endocrine management. QJM, Feb; 104(2):97-108, 2011.
In article      View Article
 
[9]  Pekic S, Cvijovic G, Stojanovic M, Kendereski A, Micic D, Popovic V. Hypopituitarism as a late complication of hemorrhagic fever. Endocrine, Mar; 26(2):7982, 2005.
In article      View Article
 
[10]  Mäkelä S, Jaatinen P, Miettinen M, Salmi J, Ala-Houhala I, Huhtala H, Hurme M, Pörsti I, Vaheri A, Mustonen J. Hormonal deficiencies during and after Puumala hantavirus infection. Eur J Clin Microbiol Infect Dis, Jun; 29(6):705-13, 2010.
In article      View Article
 
[11]  Evaluation of long-term pituitary functions in patients with severe ventricular arrhythmia: a pilot study. J Endocrinol Invest, Nov; 37(11):1057-64, 2014.
In article      View Article
 
[12]  Jallad RS, Bronstein MD. Growth hormone deficiency in adulthood: how to diagnosis and when to treat? Arq Bras Endocrinol Metabol. Jul; 52(5):861-71, 2008.
In article      
 
[13]  Allix I, Rohmer V. What's new about hypophysitis? Rev Med Interne, Dec; 35(12):815-22, 2014.
In article      
 
[14]  Akiyama Y, Yamasaki T, Kagawa T, Moritake K. Empty sella syndrome. Nihon Rinsho 51(10):2731-6, 1993.
In article      PubMed
 
[15]  Saindane AM, Lim PP, Aiken A, Chen Z, Hudgins PA. Factors determining the clinical significance of an "empty" sella turcica. Am J Roentgenol. May; 2005: 1125-31, 2013.
In article      View Article
 
[16]  Degli Uberti EC, Teodori V, Transforini G, Tamarozzi R, Margutti A, Bianconi M, Rossi R, Ambrosio MR, Pansini R. The empty sella syndrome. Clinical, radiological and endocrinologic analysis in 20 cases. Minerva Endocrinol Jan-Mar;14(1):1-18, 1989.
In article      
 
[17]  Fernandez-Rodriguez E, Bernabel I, Castro AI, Casanueva FF. Hypopituitarism after traumatic brain injury. Endocrinol Metab Clin North Am. Mar; 44(1):151-9, 2015.
In article      View Article
 

Published with license by Science and Education Publishing, Copyright © 2018 Batista Rafael Loch, Toscanini Andrea Cecilia, Borba Clarissa Groberio and Cunha-Neto Malebranche B

Creative CommonsThis work is licensed under a Creative Commons Attribution 4.0 International License. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/

Cite this article:

Normal Style
Batista Rafael Loch, Toscanini Andrea Cecilia, Borba Clarissa Groberio, Cunha-Neto Malebranche B. Hypopituitarism in Men with History of Hypovolemic Shock. American Journal of Medical Case Reports. Vol. 6, No. 3, 2018, pp 50-52. http://pubs.sciepub.com/ajmcr/6/3/3
MLA Style
Loch, Batista Rafael, et al. "Hypopituitarism in Men with History of Hypovolemic Shock." American Journal of Medical Case Reports 6.3 (2018): 50-52.
APA Style
Loch, B. R. , Cecilia, T. A. , Groberio, B. C. , & B, C. M. (2018). Hypopituitarism in Men with History of Hypovolemic Shock. American Journal of Medical Case Reports, 6(3), 50-52.
Chicago Style
Loch, Batista Rafael, Toscanini Andrea Cecilia, Borba Clarissa Groberio, and Cunha-Neto Malebranche B. "Hypopituitarism in Men with History of Hypovolemic Shock." American Journal of Medical Case Reports 6, no. 3 (2018): 50-52.
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[1]  Castinetti F, Reynaud R, Quentien MH, Jullien N, Marquant E, Rochette C, Herman JP, Saveanu A, Barlier A, Enjalbert A, Brue T. Combined pituitary hormone deficiency: current and future status. J Endocrinol Invest. 2014 Sep 9.
In article      PubMed
 
[2]  Wilson V, Mallipedhi A, Stephens JW, Redfern RM, Price DE. The causes of hypopituitarism in the absence of abnormal pituitary imaging. QJM Jan; 107(1): 21-4, 2014.
In article      View Article
 
[3]  Kelestimur F. Sheehan’s syndrome. Pituitary 6(4):181-8, 2003.
In article      View Article  PubMed
 
[4]  Mir SA, Masoodi SR, Wani AI, Farooqui KJ, Bashir MI. Deep vein thrombosis in a patient of Sheehan's syndrome: Autoimmunity or hypercoagulabilty. Indian J Endocrinol Metab, Oct; 17(Suppl 1):S105-6, 2013.
In article      View Article
 
[5]  Sheehan HL. Post-partum necrosis of the anterior pituitary. J Pathol Bacteriol 1937, 45:189-214, 1937.
In article      View Article
 
[6]  Wolff H. Insuficiência hipofisária anterior por picada de ofídio. Arq Bras Endocrinol Metabol 7:25-47, 1958.
In article      
 
[7]  Eapen CK, Chandy N, Kochuovarkey KL, et al. Unusual com- plication of snake bite: hypopituitarism after viper bites. In: Ohsaka A, Hayashi K, Sawai Y, eds. Animal, plant and microbial toxins, vol 2. New York, NY, Plenum Press, 467–73, 1976.
In article      View Article
 
[8]  Antonypillai CN, Wass JA, Warrell DA, Rajaratnam HN. Hypopituitarism following envenoming by Russell's vipers (Daboia siamensis and D. russelii) resembling Sheehan's syndrome: first case report from Sri Lanka, a review of the literature and recommendations for endocrine management. QJM, Feb; 104(2):97-108, 2011.
In article      View Article
 
[9]  Pekic S, Cvijovic G, Stojanovic M, Kendereski A, Micic D, Popovic V. Hypopituitarism as a late complication of hemorrhagic fever. Endocrine, Mar; 26(2):7982, 2005.
In article      View Article
 
[10]  Mäkelä S, Jaatinen P, Miettinen M, Salmi J, Ala-Houhala I, Huhtala H, Hurme M, Pörsti I, Vaheri A, Mustonen J. Hormonal deficiencies during and after Puumala hantavirus infection. Eur J Clin Microbiol Infect Dis, Jun; 29(6):705-13, 2010.
In article      View Article
 
[11]  Evaluation of long-term pituitary functions in patients with severe ventricular arrhythmia: a pilot study. J Endocrinol Invest, Nov; 37(11):1057-64, 2014.
In article      View Article
 
[12]  Jallad RS, Bronstein MD. Growth hormone deficiency in adulthood: how to diagnosis and when to treat? Arq Bras Endocrinol Metabol. Jul; 52(5):861-71, 2008.
In article      
 
[13]  Allix I, Rohmer V. What's new about hypophysitis? Rev Med Interne, Dec; 35(12):815-22, 2014.
In article      
 
[14]  Akiyama Y, Yamasaki T, Kagawa T, Moritake K. Empty sella syndrome. Nihon Rinsho 51(10):2731-6, 1993.
In article      PubMed
 
[15]  Saindane AM, Lim PP, Aiken A, Chen Z, Hudgins PA. Factors determining the clinical significance of an "empty" sella turcica. Am J Roentgenol. May; 2005: 1125-31, 2013.
In article      View Article
 
[16]  Degli Uberti EC, Teodori V, Transforini G, Tamarozzi R, Margutti A, Bianconi M, Rossi R, Ambrosio MR, Pansini R. The empty sella syndrome. Clinical, radiological and endocrinologic analysis in 20 cases. Minerva Endocrinol Jan-Mar;14(1):1-18, 1989.
In article      
 
[17]  Fernandez-Rodriguez E, Bernabel I, Castro AI, Casanueva FF. Hypopituitarism after traumatic brain injury. Endocrinol Metab Clin North Am. Mar; 44(1):151-9, 2015.
In article      View Article