Abstract
Introduction
Postpartum cerebral angiopathy (PCA) is a rare and pathophysiologically ill-characterized cerebral vasoconstriction syndrome, occurring within 30 days of a usually uncomplicated pregnancy and delivery. Its onset has been associated with the use of vasoactive medications, particularly ergot alkaloids. Other cases have occurred in the absence of these medications, prompting conjecture into possible overlap between PCA and other conditions known to cause cerebral vasoconstriction, including primary angiitis of the central nervous system and postpartum eclampsia. The vast majority of cases follow a relatively benign course; however, a fatal case has been reported. Histopathologic findings in PCA, so far limited to the fatal case and two more recent biopsies, have been nonspecific.
Objective
Here we present a second fatal case of PCA, including pre- and post-mortem histopathologic analysis. We also include a review of all PCA cases reported in the English literature.
Methods
Criteria for the clinical diagnosis of PCA are proposed and used to select case reports from the medical literature. Data pertaining to patient characteristics, clinical symptomatology, cerebral imaging findings, and clinical outcomes are compared between cases associated with the postpartum use of vasoactive medications and spontaneous cases.
Conclusions
We conclude that histopathologic findings in PCA are nonspecific and secondary to ischemic brain injury. Functional vasoconstriction is the most likely primary pathophysiologic process in PCA. The etiology in cases associated with medications may be due to idiosyncratic reactions to these agents. Significant overlap in symtomatology and clinical features exists between spontaneous cases and late postpartum eclampsia.
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References
Akins PT, Levy KJ, Cross AH, Goldberg MP, Schieber MH (1996) Postpartum cerebral vasospasm treated with hypervolemic therapy. Am J Obstet Gynecol 175:1386–1388
Al-Sous W, Bohlega S, Al-Kawi Z, McLean D, Shuiri K (1998) Post-partum cerebral angiopathy. A rare cerebrovascular complication. Eur J Neurol 5:411–416
Barinagarrementeria F, Cantu C, Balderrama J (1992) Postpartum cerebral angiopathy with cerebral infarction due to ergonovine use. Stroke 23:1364–1366
Bartynski WS, Sanghvi A (2003) Neuroimaging of delayed eclampsia. Report of 3 cases and review of the literature. J Comput Assist Tomogr 27:699–713
Bogousslavsky J, Despland PA, Regli F, Dubuis PY (1989) Postpartum cerebral angiopathy: reversible vasoconstriction assessed by transcranial Doppler ultrasounds. Eur Neurol 29:102–105
Calabrese LH, Gragg LA, Furlan AJ (1993) Benign angiopathy: a distinct subset of angiographically defined primary angiitis of the central nervous system. J Rheumatol 20:2046–2050
Calabrese LH, Furlan AJ, Gragg LA, Ropos TJ (1992) Primary angiitis of the central nervous system: diagnostic criteria and clinical approach. Cleve Clin J Med 59:293–306
Calabrese LH, Mallek JA (1988) Primary angiitis of the central nervous system. Report of 8 new cases, review of the literature, and proposal for diagnostic criteria. Medicine (Baltimore) 67:20–39
Calado S, Vale-Santos J, Lima C, Viana-Baptista M (2004) Postpartum cerebral angiopathy: vasospasm, vasculitis or both? Cerebrovasc Dis 18:340–341
Call GK, Fleming MC, Sealfon S, Levine H, Kistler JP, Fisher CM (1988) Reversible cerebral segmental vasoconstriction. Stroke 19:1159–1170
Chames MC, Livingston JC, Ivester TS, Barton JR, Sibai BM (2002) Late postpartum eclampsia: a preventable disease? Am Journal Obstet Gynecol 186:1174–1177
Chang GY (2000) Basilar artery vasospasm in postpartum cerebral angiopathy. Neurology 55:1596
Comabella M, Alvarez-Sabin J, Rovira A, Codina A (1996) Bromocriptine and postpartum cerebral angiopathy: a causal relationship? Neurology 46:1754–1756
Crippa G, Sverzellati E, Pancotti D, Carrara GC (2000) Severe postpartum hypertension and reversible cerebral angiopathy associated with ergot derivative (methergoline) administration. Ann Ital Med Int 15:303–305
de Jong-van den Berg L, Mintzes B (1995) Bromocriptine and lactation suppression: are the risks acceptable? Pharm World Sci 17:93–95
Donaldson JO (2000) Eclampsia and postpartum cerebral angiopathy. J Neurol Sci 178:1
Dziewas R, Stogbauer F, Freund M, Ludemann P, Imai T, Holzapfel C, Ringelstein PB (2002) Late onset postpartum eclampsia: a rare and difficult diagnosis. J Neurol 249:1287–1291
Ellison C, Martens R, Belkin R, Bourdette D (1988) Postpartum cerebral angiopathy: A benign variant of isolated CNS vasculitis? Neurolo 38:110–110
Farine D, Andreyko J, Lysikiewicz A, Simha S, Addison A (1984) Isolated angiitis of brain in pregnancy and puerperium. Obstet Gynecol 63:586–588
Felz MW, Barnes DB, Figueroa RE (2000) Late postpartum eclampsia 16 days after delivery: case report with clinical, radiologic, and pathophysiologic correlations. J Am Board Fam Pract 13:39–46
Garner BF, Burns P, Bunning RD, Laureno R (1990) Acute blood pressure elevation can mimic arteriographic appearance of cerebral vasculitis—(a postpartum case with relative hypertension). J Rheumatol 17:93–97
Gary Cunningham, Kenneth J. Leveno, Steven L. Bloom, John C. Hauth, Larry C. Gilstrap, Katharine D. Wenstrom. 2005. Williams Obstetrics. 1600
Geocadin RG, Razumovsky AY, Wityk RJ, Bhardwaj A, Ulatowski JA (2002) Intracerebral hemorrhage and postpartum cerebral vasculopathy. J Neurol Sci 205:29–34
Geraghty JJ, Hoch DB, Robert ME, Vinters HV (1991) Fatal puerperal cerebral vasospasm and stroke in a young woman. Neurology 41:1145–1147
Granier I, Garcia E, Geissler A, Boespflug MD, Durand-Gasselin J (1999) Postpartum cerebral angiopathy associated with the administration of sumatriptan and dihydroergotamine—a case report. Intensive Care Med 25:532–534
Henry PY, Larre P, Aupy M, Lafforgue JL, Orgogozo JM (1984) Reversible cerebral arteriopathy associated with the administration of ergot derivatives. Cephalalgia 4:171–178
Herings RM, Stricker BH (1995) Bromocriptine and suppression of postpartum lactation. The incidence of adverse cardiovascular effects in women of child-bearing age. Pharm World Sci 17:133–137
Iffy L, O’Donnell J, Correia J, Hopp L (1998) Severe cardiac dysrhythmia in patients using bromocriptine postpartum. Am J Ther 5:111–115
Iffy L, Lindenthal J, McArdle JJ, Ganesh V (1996a) Severe cerebral accidents postpartum in patients taking bromocriptine for milk suppression. Isr J Med Sci 32:309–312
Iffy L, McArdle JJ, Ganesh V (1996b) Intracerebral hemorrhage in normotensive mothers using bromocriptine postpartum. ZentralblGynakol 118:392–395
Ihara M, Yanagihara C, Nishimura Y (2000) Serial transcranial color-coded sonography in postpartum cerebral angiopathy. J Neuroimaging 10:230–233
Janssens E, Hommel M, Mounier-Vehier F, Leclerc X, Guerin du Masgenet B, Leys D (1995) Postpartum cerebral angiopathy possibly due to bromocriptine therapy. Stroke 26:128–130
Konstantinopoulos PA, Mousa S, Khairallah R, Mtanos G (2004) Postpartum cerebral angiopathy: an important diagnostic consideration in the postpartum period. Am J Obstet Gynecol 191:375–377
Kubo S, Nakata H, Tatsumi T, Yoshimine T (2002) Headache associated with postpartum cerebral angiopathy: monitoring with transcranial color-coded sonography. Headache 42:297–300
Kulig K, Moore LL, Kirk M, Smith D, Stallworth J, Rumack B (1991) Bromocriptine-associated headache: possible life-threatening sympathomimetic interaction. Obstet Gynecol 78:941–943
Langlois PF, Sharon GE, Gawryl MS (1989) Plasma concentrations of complement-activation complexes correlate with disease activity in patients diagnosed with isolated central nervous system vasculitis. J Allergy Clin Immunol 83:11–16
Lee C, Hsu TY, Ou CY, Chang SY, Soong YK (1999) Retinal detachment in postpartum preeclampsia and eclampsia: report of two cases. Changgeng Yi Xue Za Zhi 22:520–524
Leitch CR, Cameron AD, Walker JJ (1997b) The changing pattern of eclampsia over a 60-year period. Br J Obstet Gynaecol 104:917–922
Lubarsky SL, Barton JR, Friedman SA, Nasreddine S, Ramadan MK, Sibai BM (1994) Late postpartum eclampsia revisited. Obstet Gynecol 83:502–505
Makdassi R, de Cagny B, Lobjoie E, Andrejak M, Fournier A (1996) Convulsions, hypertension crisis and acute renal failure in postpartum: role of bromocriptine? Nephron 72:732–733
Martin J, Sidman R (2003) Late postpartum eclampsia: a common presentation of an uncommon diagnosis. J Emerg Med 25:387–390
Mathew R, Raj RS, Sudha P (2003) Late postpartum eclampsia without prodroma. Neurol India 51:539–540
Matthys LA, Coppage KH, Lambers DS, Barton JR, Sibai BM (2004) Delayed postpartum preeclampsia: an experience of 151 cases. Am J Obstet Gynecol 190:1464–1466
Modi M, Modi G (2000) Case reports: postpartum cerebral angiopathy in a patient with chronic migraine with aura. Headache 40:677–681
Morgans D (1995) Bromocriptine and postpartum lactation suppression. Br J Obstet Gynaecol 102:851–853
Newbould S (2002) Postpartum eclampsia. Am Fam Physician 66:1
No authors listed (1984) Postpartum hypertension, seizures, strokes reported with bromocriptine. FDA Drug Bul 14:3–4
Qureshi AI, Frankel MR, Ottenlips JR, Stern BJ (1996) Cerebral hemodynamics in preeclampsia and eclampsia. Arch Neurol 53:1226–1231
Raps EC, Galetta SL, Broderick M, Atlas SW (1993) Delayed peripartum vasculopathy: cerebral eclampsia revisited. Ann Neurol 33:222–225
Raroque HG Jr, Tesfa G, Purdy P (1993) Postpartum cerebral angiopathy. Is there a role for sympathomimetic drugs? Stroke 24:2108–2110
Ringer AJ, Qureshi AI, Kim SH, Fessler RD, Guterman LR, Hopkins LN (2001) Angioplasty for cerebral vasospasm from eclampsia. Surg Neurol 56:, 73–8; discussion 378–379
Roh JK, Park KS (1998) Postpartum cerebral angiopathy with intracerebral hemorrhage in a patient receiving lisuride. Neurology 50:1152–1154
Sekimoto E, Kunishige M, Kuriwaka R, Shinohara M, Ebisutani D, Kitamura K, Doi T, Kushiki N, Mitsui T (2002) Delayed vasospasm in late postpartum cerebral angiopathy after withdrawal of methylergometrine. Cerebrovas Dis 13:288–289
Sengar AR, Gupta RK, Dhanuka AK, Roy R, Das K (1997) MR imaging, MR angiography, and MR spectroscopy of the brain in eclampsia. AJNR Am J Neuroradiol 18:1485–1490
Singhal AB (2004a) Cerebral vasoconstriction syndromes. Top Stroke Rehabil 11:1–6
Singhal AB (2004b) Postpartum angiopathy with reversible posterior leukoencephalopathy. Arch Neurol 61:411–416
Song JK, Fisher S, Seifert TD, Cacayorin ED, Alexandrov AV, Malkoff MD, Grotta JC, Campbell MS.(2004) Postpartum cerebral angiopathy: atypical features and treatment with intracranial balloon angioplasty. Neuroradiology
Sugiyama Y, Muroi A, Ishikawa M, Tsukamoto T, Yamamoto T (1997) A benign form of isolated angiitis of the central nervous system in puerperium: an identical disorder to postpartum cerebral angiopathy? Intern Med 36:931–934
Tank PD, Chauhan AR, Bhattacharya MS, Warke HS, Raut VS (2004) Neurological complications in eclampsia: a case series. Int J Fertil Womens Med 49:61–69
Thomas SV (1998) Neurological aspects of eclampsia. J Neurol Sci 155:37–43
Trommer BL, Homer D, Mikhael MA (1988) Cerebral vasospasm and eclampsia. Stroke 19:326–329
Ursell MR, Marras CL, Farb R, Rowed DW, Black SE, Perry JR (1998) Recurrent intracranial hemorrhage due to postpartum cerebral angiopathy: implications for management. Stroke 29:1995–1998
Ursell MR, Marras CL, Farb R, Rowed DW, Black SE, Perry JR (1995) Recurrent intracranial hemorrhage due to postpartum cerebral angiopathy: implications for management. Stroke 29:1995–1998
Veltkamp R, Kupsch A, Polasek J, Yousry TA, Pfister HW (2000) Late onset postpartum eclampsia without pre-eclamptic prodromi: clinical and neuroradiological presentation in two patients. J Neurol Neurosurg Psychiatry 69:824–827
Yasuda Y, Matsuda I, Kang Y, Saiga T, Kameyama M (1993) Isolated angiitis of the central nervous system first presenting as intracranial hemorrhage during cesarean section. Intern Med 32:745–748
Zunker P, Golombeck K, Brossmann J, Georgiadis D, Deuschl G (2002) Post-partum cerebral angiopathy: repetitive TCD, MRI, MRA, and EEG examinations. Neurol Res 24:570–572
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Williams, T.L., Lukovits, T.G., Harris, B.T. et al. A fatal case of postpartum cerebral angiopathy with literature review. Arch Gynecol Obstet 275, 67–77 (2007). https://doi.org/10.1007/s00404-006-0194-3
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DOI: https://doi.org/10.1007/s00404-006-0194-3