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LETTER TO THE EDITOR
Year : 2016  |  Volume : 7  |  Issue : 1  |  Page : 58-59  

Alopecia universalis in a patient with Sheehan's syndrome


1 Department of Dermatology, Maulana Azad Medical College and Associated Lok Nayak Hospital, New Delhi, India
2 Department of Medicine, Maulana Azad Medical College and Associated Lok Nayak Hospital, New Delhi, India
3 Department of Radiodiagnosis, Maulana Azad Medical College and Associated Lok Nayak Hospital, New Delhi, India

Date of Web Publication18-Jan-2016

Correspondence Address:
Dr. Naresh Kumar
16/554, Joshi Road, Karol Bagh, New Delhi - 110 005
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2229-5178.174322

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How to cite this article:
Bansal S, Kumar N, Kochhar AM, Ghai R. Alopecia universalis in a patient with Sheehan's syndrome. Indian Dermatol Online J 2016;7:58-9

How to cite this URL:
Bansal S, Kumar N, Kochhar AM, Ghai R. Alopecia universalis in a patient with Sheehan's syndrome. Indian Dermatol Online J [serial online] 2016 [cited 2020 Jul 14];7:58-9. Available from: http://www.idoj.in/text.asp?2016/7/1/58/174322

Sir,

Sheehan's syndrome results from ischemic necrosis of the pituitary gland due to severe postpartum hemorrhage. This condition is still being seen frequently in developing countries such as India. Alopecia in Sheehan's syndrome usually consists of loss of axillary and pubic hair and results due to gonadotropin deficiency.[1]

A 55-year-old nondiabetic woman presented with fever of 5 days and altered sensorium of 2 days duration. She had no history of headache, vomiting, seizure, trauma, or radiation exposure. Since the last 10 years, she had been having gradually progressive fatigue and generalized hair loss. She gave a history of postpartum hemorrhage during her last delivery 16 years ago, which was followed by lactational failure and amenorrhea. She was also a known case of coronary artery disease with severe systolic dysfunction since 10 years. On examination, jugular venous pressure was raised; she had pedal edema, and generalized loss of hair including scalp, eyebrows, axillary, and pubic hair [Figure 1]. There was no evidence of any scarring or atrophy. Her ankle jerk reflex was delayed. and rest of the systemic examination was normal. She had normocytic normochromic anemia with hemoglobin of 105 g/L. Her renal and liver functions were mildly deranged. Her electrolyte, arterial blood gas analysis, and ultrasound abdomen were normal. Routine urine examination showed 30–40 pus cells/hpf. Her hormonal profile was suggestive of panhypopituitarism with low levels of serum cortisol, ACTH, fT3, fT4, TSH, GH, FSH, LH and prolactin [4.74 µg/dL (3.9–22.4), 10.12 pg/mL (0–46), 1.36 pg/dL (1.8–4.6), 0.631 ng/dL (0.93–1.71), 0.11 mIU/mL (0.27–4.2), 0.09 ng/mL (0.06–5.0), 0.497 mIU/mL (25.8–134.8), 0.10 mIU/mL (7.7–58.5), and 3.2 ng/mL (3.4–24.1), respectively]. Magnetic resonance imaging (MRI) of pituitary gland showed empty sella [Figure 2]. Pituitary antibodies could not be done due to financial constraints. In addition to continuing treatment of her coronary ailment and urinary tract infection, she was started on hormone replacement therapy. However, patient succumbed to sepsis and related complications.
Figure 1: Photograph of patient showing alopecia

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Figure 2: Sagittal section of brain showing empty sella on magnetic resonance imaging

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The pituitary gland undergoes major physiological, anatomical, and immunological changes during pregnancy, which predisposes to various disorders such as Sheehan's syndrome, lymphocytic hypophysitis, apoplexy of pituitary adenoma, and others. Sheehan's description of postpartum hypopituitarism promoted the belief that PPH leads to necrosis of the enlarged pituitary gland of pregnancy and hypopituitarism develops. The fact that only one third of women with severe postpartum bleeding develop hypopituitarism and Sheehan's syndrome has been reported to develop even after uncomplicated pregnancy and delivery suggest factors other than ischemia in the pathogenesis of Sheehan's syndrome.[1],[2] Development of alopecia universalis six years after her last delivery reflects slow clinical progression and suggests factors other than ischemia in its pathogenesis. Tissue necrosis could release sequestered antigens, triggering autoimmunity of the pituitary, and delayed hypopituitarism in Sheehan's syndrome.[3] The presence of antipituitary antibodies (APAs) and antihypothalamus antibodies (AHAs) have been demonstrated in patients with SS, suggesting that autoimmunity may play a role in the pathogenesis of this syndrome.[3],[4]

Alopecia is generally reported as loss of pubic hair and axillary hair in Sheehan's syndrome due to loss of gonadotrophic function. However, our patient had alopecia universalis. Alopecia universalis is characterized by loss of hair of entire scalp including eyebrows and body hairs. It is a severe form of alopecia areata, which is thought to be an autoimmune condition. It occurs in <1% of total cases of alopecia areata. Patients with alopecia universalis usually have a poorer prognosis; treatment failure is seen in most of these patients. Chance of spontaneous remission is low, and that of recurrence higher. Our patient had alopecia universalis since 10 years without any remission. Detection of hypopituitarism and Sheehan's syndrome was delayed as the patient mistakenly attributed her symptoms to her cardiac ailment.

Alopecia areata has been described in association with a number of autoimmune diseases including vitiligo, Hashimoto's thyroiditis, diabetes mellitus, systemic lupus erythematosus, and so on.[5] Alopecia areata has rarely been reported with hypopituitarism related to idiopathic primary hypophysitis and Sheehan's syndrome.[6],[7] Alopecia universalis has been reported with dysfunction of hypothalamic–pituitary–adrenal axis in triple H syndrome in few cases and the mechanism involved is thought to be autoimmune.[8] The association of panhypopituitarism due to Sheehan's syndrome and alopecia universalis in our patient can be explained by autoimmunity, which could be an etiological factor for both. To the best of our knowledge, this is the first report of alopecia universalis associated with Sheehan's syndrome–related panhypopituitarism.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Ozkan Y, Colak R. Sheehan syndrome: Clinical and laboratory evaluation of 20 cases. Neuro Endocrinol Lett 2005;26:257-60.  Back to cited text no. 1
    
2.
Bala M, Brünnler T, Guralnik V, Schölmerich J, Schäffler A. A 36-year old female patient presenting with hypoglycemic coma. Internist (Berl) 2009;50:606-11.  Back to cited text no. 2
    
3.
Goswami R, Kochupillai N, Crock PA, Jaleel A, Gupta N. Pituitary autoimmunity in Patients with Sheehan's syndrome. J Clin Endocrinol Metab 2002;87:4137-41.  Back to cited text no. 3
    
4.
De Bellis A, Kelestimur F, Sinisi AA, Ruocco G, Tirelli G, Battaglia M, et al. Anti-hypothalamus and anti-pituitary antibodies may contribute to perpetuate the hypopituitarism in patients with Sheehan's syndrome. Eur J Endocrinol 2008;158:147-52.  Back to cited text no. 4
    
5.
Lee NR, Kim BK, Yoon NY, Lee SY, Ahn SY, Lee WS. Differences in comorbidity profiles between early-onset and late-onset alopecia areata patients: A retrospective study of 871 Korean patients. Ann Dermatol 2014;26:722-6.  Back to cited text no. 5
    
6.
Ajith C, Gupta S, Bhansali A, Radotra BD, Kanwar AJ, Kumar B. Alopecia areata associated with idiopathic primary hypophysitis. Clin Exp Dermatol 2005;30:250-2.  Back to cited text no. 6
    
7.
Sheriff FG, Howlett WP, Kilonzo KG. Post-partum pituitary insufficiency and livido erticularis presenting a diagnostic challenge in a resource limited setting in Tanzania: A case report, clinical discussion and brief review of existing literature. BMC Endocr Disord 2012;12:4.  Back to cited text no. 7
    
8.
Ichiki K, Nakamura T, Fujita N, Honda K, Hiraga T, Ishibashi S, et al. An endocrinopathy characterized by dysfunction of the pituitary-adrenal axis and alopecia universalis: Supporting the entity of a triple H syndrome. Eur J Endocrinol 2002;147:357-61.  Back to cited text no. 8
    


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