BMC Endocrine Disorders | |
Post-partum pituitary insufficiency and livedo reticularis presenting a diagnostic challenge in a resource limited setting in Tanzania: a case report, clinical discussion and brief review of existing literature | |
Kajiru G Kilonzo1  William P Howlett1  Faheem G Sheriff1  | |
[1] Kilimanjaro Christian Medical Centre, Moshi, Tanzania | |
关键词: Africa; Livedo reticularis; Sheehan’s syndrome; Lymphocytic hypophysitis; Post partum panhypopituitarism; | |
Others : 1086446 DOI : 10.1186/1472-6823-12-4 |
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received in 2011-10-01, accepted in 2012-05-14, 发布年份 2012 | |
【 摘 要 】
Background
Pituitary disorders following pregnancy are an important yet under reported clinical entity in the developing world. Conversely, post partum panhypopituitarism has a more devastating impact on women in such settings due to high fertility rates, poor obstetric care and scarcity of diagnostic and therapeutic resources available.
Case presentation
A 37 year old African female presented ten years post partum with features of multiple endocrine deficiencies including hypothyroidism, hypoadrenalism, lactation failure and secondary amenorrhea. In addition she had clinical features of an underlying autoimmune condition. These included a history of post-partum thyroiditis, alopecia areata, livedo reticularis and deranged coagulation indices. A remarkable clinical response followed appropriate hormone replacement therapy including steroids. This constellation has never been reported before; we therefore present an interesting clinical discussion including a brief review of existing literature.
Conclusion
Post partum pituitary insufficiency is an under-reported condition of immense clinical importance especially in the developing world. A high clinical index of suspicion is vital to ensure an early and correct diagnosis which will have a direct bearing on management and patient outcome.
【 授权许可】
2012 Sheriff et al.; licensee BioMed Central Ltd.
【 预 览 】
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Figure 1. | 41KB | Image | download |
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【 参考文献 】
- [1]Karaca Z, Tanriverdi F, Unluhizarci K, Kelestimur F: Pregnancy and pituitary disorders. Eur J Endocrinol 2010, 162(Suppl 3):453-475.
- [2]Gutenberg A, Hans V, Maximilian J: Primary hypophysitis: clinical-pathological correlations. Eur J Endocrinol 2006, 155(Suppl 1):101-107.
- [3]Zargar AH, Singh B, Laway BA, Masoodi SR, Wani AI, Bashir MI: Epidemiologic aspects of postpartum pituitary hypofunction (Sheehan's syndrome). Fertil Steril 2005, 84(Suppl 2):523-528.
- [4]Cénac A, Soumana I, Develoux M, Touta A, Bianchi G: Sheehan's syndrome in Sudano-Sahelian Africa 40 observations. Bull Soc Pathol Exot 1991, 84(Suppl 5):686-692.
- [5]Sidibe EH: Sheehan’s syndrome: experience in Africa. Ann Med Interne (Paris) 2000, 151(Suppl 5):345-351.
- [6]Caturegli P, Newschaffer C, Olivi A, Pomper M, et al.: Autoimmune hypophysitis. Endocr Rev 2005, 26:599-614.
- [7]Ouma JR, Farrell VJ: Lymphocytic infundibulo- neurohypophysitis with hypothalamic and optic pathway involvement: report of a case and review of the literature. Surg Neurol 2002, 57(Suppl 1):49-53.
- [8]Castle D, De Villiers JC, Melvill R: Lymphocytic adenohypophysitis. Report of a case with demonstration of spontaneous tumour regression and a review of the literature. Br J Neurosurg 1988, 2(Suppl 3):401-405.
- [9]Ozawa Y, Shishiba Y: Recovery from lymphocytic hypophysitis associated with painless thyroiditis: clinical implications of circulating antipituitary antibodies. Acta Endocrinol 1993, 128:493-498.
- [10]Diederich S, Franzen N, Bahr V, Oelkers W: Severe hyponatremia due to hypopituitarism with adrenal insufficiency: report on 28 cases. Eur J Endocrinol 2003, 148:609-617.
- [11]Raff H: Glucocorticoid inhibition of neurohypophysial vasopressin secretion. Am J Physiol 1987, 252:635-644.
- [12]Erkut ZA, Pool C, Swaab DF: Glucocorticoids suppress corticotropin-releasing hormone and vasopressin expression in human hypothalamic neurons. J Clin Endocrinol Metab 1998, 83:2066-2073.
- [13]Ajith C, Gupta S, Bhansali A, Radotra B, Kanwar A, Kumar B: Alopecia areata associated with idiopathic primary hypophysitis. Clin Exp Dermatol 2005, 30:250-252.
- [14]Honegger J, Fahlbusch , Bornemann : Lymphocytic and granulomatous hypophysitis: experience with nine cases. Neurosurgery 1997, 40(Suppl 4):713-722.
- [15]Goswami R, Kochupillai N, Crock PA, et al.: Pituitary autoimmunity in patients with Sheehan's syndrome. J Clin Endocrinol Metab 2002, 87(Suppl 9):4137-4141.
- [16]Errarhay S, Kamaoui I, Bouchikhi C, et al.: Sheehan’s Syndrome: A case report and literature review. [http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3066722/pdf/LJM-4-081.pdf] webcite
- [17]Kristof RA, Van Roost D, Klingmüller D, et al.: Lymphocytic hypophysitis: non-invasive diagnosis and treatment by high dose methylprednisolone pulse therapy? Neurol Neurosurg Psychiatry 1999, 67:398-402.
- [18]Lee HC, Lee EJ, Lee KW, Ahn KJ, Jung TS, Kim DI, Huh KB: Computed tomographic correlation with pituitary function in Sheehan's syndrome. Korean J Intern Med 1992, 7(Suppl 1):48-53.
- [19]Bakiri F, Bendib SE, Maoui R, Bendib A, Benmiloud M: The sella turcica in Sheehan's syndrome: computerized tomographic study in 54 patients. J Endocrinol Invest 1991, 14(Suppl 3):193-196.
- [20]Molitch ME, Gillam MP: Lymphocytic Hypophysitis. Horm Res 2007, 68(Suppl 5):145-150.
- [21]Ji JD, Lee SY, Choi SJ, et al.: Lymphocytic hypophysitis in a patient with systemic lupus erythematosus. Clin Exp Rheumatol 2000, 18(Suppl 1):78-80.
- [22]Golden R: Livedo reticularis in systemic lupus erythematosus. Arch Dermatol 1963, 87:299-301.
- [23]Tan EM, Cohen AS, Fries JF, Masi AT, McShane DJ, Rothfield NF, et al.: The 1982 revised criteria for the classification of systemic lupus erythematosus. Arthritis Rheum 1982, 25:1271-1277.
- [24]Leung GK, Lopes MB, Thorner MO, Vance ML, Laws ER: Primary hypophysitis: a single-center experience in 16 cases. J Neurosurg 2004, 101:262-271.
- [25]Tubridy N, Saunders D, Thom M, Asa SL, Powell M, Plant GT, Howard R: Infundibulohypophysitis in a man presenting with diabetes insipidus and cavernous sinus involvement. J Neurol Neurosurg Psychiatry 2001, 71:798-801.
- [26]Lecube A, Francisco G, Rodriguez D, Ortega A, Codina A, Hernandez C, Simo R: Lymphocytic hypophysitis successfully treated with azathioprine: first case report. J Neurol Neurosurg Psychiatry 2003, 74:1581-1583.