pregnant women might be in risk for central sinus vein thrombosis ( csvt ) and vertebral artery dissection ( vad ) . opalski syndrome , a rare variant of lateral medullary syndrome ( lms ) with concomitant ipsilateral hemiparesis , results from the ipsilateral extension of the medullary infarct to the superior cervical cord caudal to the pyramidal decussation . its etiology might be iatrogenic following induced delivery and coughing episodes . a concomitant venous - arterial disease ( opalski syndrome and incidental csvt ) following induced delivery intriguing neurological findings followed by neuroimaging confirmed the diagnosis , leading to successful conservative treatment . a 32-year - old healthy woman with 2 previous normal deliveries and a recent induced vaginal delivery was referred from another hospital diagnosed with csvt . family and personal history was noncontributory , except for migraine , no smoking history and psychotherapy for anxiety . the latest pregnancy was normal up to the 28th week during which she complained of dyspnea and coughing ( on supine position ) that spontaneously resolved . no further complaints appeared until the 30th week when she suspected reduced fetal movement which was diagnosed as a nonviable fetus , leading to induced vaginal delivery on the following day . one week later , headaches and neck tenderness ( right side ) appeared , for which conservative management was recommended . her unremarkable neurological examination in the emergency room ( er ) led the physician to discharge her for ambulatory follow - up , diagnosed as two days later , now complaining of blurred vision , vomiting and urinary retention , the patient was referred to the er again and was admitted to the gynecology ward . her gynecological and ophthalmological evaluations were normal ; 500 cm of urine was found in a post - voiding ultrasound , and an unexplained hypertension was treated . one day after admission , a new right hemiparesis ( arm > leg ) appeared . a ct venography ( ctv ) demonstrated csvt of the left transverse and sigmoid sinuses ( fig . heparin was initiated , and the patient was referred to our er for  evaluating the option of an endovascular treatment. we found discrepancies between the neurological signs and the isolated diagnosis of csvt , and suspected brain stem involvement . a reevaluation of the ctv ( that unintentionally included an arterial phase ) revealed right vad ( v3 - 4 segments ; fig . 1b ) , further confirmed by mri showing an ischemic infarct damage of the brain stem ( lateral and anterior medullary infarcts ) and cerebellum , explaining the patient 's symptoms ( fig . 2a aneurysms or stenosis were excluded . on physical examination , tachycardia ( heart rate 93 ) and hypertension ( 166/113 mm hg ) were noted . the patient was alert , suffering from headache and presenting the following neurological findings suggestive of opalski syndrome : ipsilateral hemiparesis ; vestibular signs ( vertigo , diplopia , nystagmus and vomiting ) ; cerebellar signs ( side ataxia , dysdiadochokinesia , nystagmus and dysarthria ) ; lateral spinothalamic tract signs ( contralateral deficits in pain and temperature sensation , with preserved proprioception ) ; nucleus ambiguus signs ( dysphagia , hoarseness , palate and tongue deviation to the right ) ; spinal trigeminal nucleus signs ( ipsilateral loss of pain and temperature sensation from v2 - 3 distribution , with normal v1 and reduced sensation of the palate ) ; bilateral positive babinski signs ; descending sympathetic fiber signs ( partial ipsilateral horner 's syndrome : myosis and ptosis ) , and diplopia due to limited upward and lateral gaze of the right eye . heparin treatment maintained a ptt range of 60 - 70 s , followed by unfractionated heparin treatment ( 80 mg twice a day ) with rigorous blood pressure ( bp ) control ( systolic 110140 mm hg ) . gradual improvement could be noted , and the patient started to walk by day 4 and swallow by day 6 . a thorough workup included an echocardiography ( mild tricuspid regurgitation ) , bilateral lower limb doppler examination , hematological workup ( crp , folic acid , vitamin b12 , homocysteine , factor v leiden , cytoplasmic and perinuclear anca ( antineutrophil cytoplasmic antibodies ) , rheumatoid factor , prothrombin , complement factors , anti - thrombin 3 , -2 glycoprotein , cardiolipin igm and igg , proteins s and c , and lupus anticoagulant ) . the patient was discharged after 9 days , further improving , being fully conscious and less hoarse , eating by herself , having less diplopia , nystagmus and hypoesthesia , and walking independently . after 3 months of rehabilitation , she resumed a normal lifestyle with further improvement on a recent mri . opalski   first described this phenomenon in 1946 as a variant of lms with concomitant ipsilateral hemiparesis explained by the ipsilateral extension of the medullary infarct to the superior cervical cord caudal to the pyramidal decussation . others have explained these combined findings as a result of vertebral artery ( va ) occlusion / stenosis or vad [ 3 , 4 ] , compromising the medullary penetrating arteries that arise from distal vas or anterior spinal arteries . opalski syndrome might be due to either ischemic disturbances ( such as distal vad ) and/or due to hemodynamically vulnerable areas such as the borderline zone between the anterior and posterior spinal arteries and between the vertebral and spinal arteries . concomitant lms with ipsilateral hemiparesis , supported by neuroimaging , led us to this rare diagnosis . either the induced delivery and/or the unexplained coughs could have caused the vad leading to brain stem lesions . cases of dissections with or without concomitant csvt following deliveries have been reported [ 6 , 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 ] ; however , our case differs from those found in the literature in that : ( 1 ) the gynecological course was more complicated [ intrauterine fetal death ( iufd ) and induced delivery ] ; ( 2 ) our patient experienced unexplained dyspnea and coughing 2 weeks before iufd diagnosis ; ( 3 ) our patient suffered from concomitant vad , opalski syndrome and csvt , and ( 4 ) there were suspected findings of bilateral damages in the brain stem - spinal cord junction . women of child - bearing age have a higher incidence of stroke compared to age - matched men [ 6 , 10 , 11 , 12 ] , with only mild differences between pregnant and nonpregnant patients . nonpregnant women have a lower incidence of hemorrhagic than ischemic strokes ; however , these frequencies become similar during pregnancy , suggesting pregnancy as a risk factor for hemorrhagic strokes [ 11 , 12 ] . strokes usually occur during the third trimester and the puerperium , up to 1 month postpartum . eclampsia / preeclampsia remains the main cause for both types of strokes ( up to 47% ) . hemorrhagic strokes carry a poor prognosis [ 20 , 21 ] . during the peripartum period , the overall risk for strokes ( 13.110.3/100,000 ) and csvt ( 11.6/100,000 ) is similar . among nonpregnant aged - matched females , csvt occur less frequently [ 6 , 18 ] , suggesting that in the peripartum period , women are prone to develop csvt ( approx . 5 - 20% of all csvt occur during this period ) , mainly during the first 14 days postpartum . notably , csvt by itself is considered as a cause for stroke in up to 38% of cases during the postpartum period [ 6 , 9 , 14 , 15 , 16 ] . extracranial dissections commonly present as neck pain and headache ( occipital and posterior cervical regions ) , and might be proceeded by ischemic symptoms within the first weeks of the dissection . unlike carotid dissections , vad more frequently extend intracranially and involve the subarachnoid space causing meningism . less than 30% of the cases have simultaneous dissections of two or more vessels , for which an underlying connective tissue disorder should be suspected [ 17 , 18 , 31 ] . cervicocephalic arterial dissection ( ccad ) is a rare postpartum complication , with only 16 reported cases to date [ 7 , 8 , 9 , 10 , 11 , 13 , 14 , 15 , 16 , 17 ] . the rate of spontaneous ccad in the general population is 2.6/100,000 , of which 2.4% can occur during the postpartum period , meaning that there are 6.2/10,000,000 annually [ 9 , 23 , 24 ] , representing 6% of spontaneous ccad in women under 50 years of age . extracranial dissections seem to occur more frequently among pregnant women . up to 90% of vad reversible cerebral vasoconstriction syndrome was associated with stroke during the postpartum period among 4/6 patients compared with only 3/96 aged - matched controls . contrary to reversible cerebral vasoconstriction syndrome which has a favorable outcome , intracranial dissections in postpartum patients are characterized by a worse outcome [ 7 , 30 ] . the different occurrence between intra- and extradural dissections could possibly be explained by histological changes that appear once arteries ( vas and internal carotid arteries ) become  intradural :  the change is characterized first by a diminution in thickness of the adventitial and medial coats , and second , by a very gross reduction or loss of elastic fibers in the media and external elastic lamina . our patient needed a tradeoff between bp reduction ( due to the possible hemorrhagic transformation in the stroke area ) and the general permissive hypertension ( for dissections ) . the second issue concerns the usage of antiplatelet / anticoagulation drugs , to which caution was given due to the possible hemorrhagic transformation of a stroke . once anticoagulation treatment is initiated , it will be generally replaced by antiplatelet therapy for approximately 6 months , due to the fact that most ccad resolve after 3 - 6 months . neither therapy ( antiplatelet vs. anticoagulant ) was found superior for treating acute dissection [ 26 , 27 ] . once the possibility of a connective tissue disorder is excluded , the risk of recurrence is 1% within 1.4 - 8.6 years and up to 2% within the first months following the dissection [ 28 , 32 ] . our patient presented vascular pathologies of two different origin : one of venous origin ( csvt ) and one of arterial origin ( vad ) . both could have been complications of the peripartum period and of the induced delivery . hypercoagulability should also be considered since it can either cause or be a consequence of iufd . since no postmortem examination of the fetus was done , these issues as well as the reason for the iufd remained unsolved . despite two previous normal pregnancies / deliveries and noncontributory personal / familial coagulopathies , transient hemodynamic as well as hematologic changes during pregnancy and puerperium might be considered as additional causes . pregnancy might alter all three factors of virchow 's triad ( blood flow , blood vessel wall and constitution of blood ) , and patients are therefore prone to develop vascular diseases , as seen in our patient . opalski syndrome is caused by compromised blood supply and/or hemodynamic vulnerability , both likely occurring during puerperium and/or induced delivery . it is well accepted that nearly all posterior inferior cerebellar artery ( pica ) occlusions and more than 50% of va occlusions ( mainly if a pica origin is included ) might manifest as lms . importantly , 75% of all lms cases are related to va occlusion and 12% to pure pica occlusions . the intradural v4 segment ascends to the lower pontine border and forms the basilar artery , giving rise to the pica en route ( 10 mm distal to the dura perforation ) . our patient 's right v3 - 4 dissection included the pica , presenting as right medullary and cerebellar infarcts . this dissection might explain both constituents of opalski syndrome : ( 1 ) lms signs might be caused by the pica - va - related brain stem strokes , while ( 2 ) concomitant ipsilateral hemiparesis might be caused by the involvement of the anterior spinal artery arising from v4 that normally supplies the ventral two thirds of the spinal cord , including the pyramidal tract . opalski   first described this phenomenon in 1946 as a variant of lms with concomitant ipsilateral hemiparesis explained by the ipsilateral extension of the medullary infarct to the superior cervical cord caudal to the pyramidal decussation . others have explained these combined findings as a result of vertebral artery ( va ) occlusion / stenosis or vad [ 3 , 4 ] , compromising the medullary penetrating arteries that arise from distal vas or anterior spinal arteries . opalski syndrome might be due to either ischemic disturbances ( such as distal vad ) and/or due to hemodynamically vulnerable areas such as the borderline zone between the anterior and posterior spinal arteries and between the vertebral and spinal arteries . concomitant lms with ipsilateral hemiparesis , supported by neuroimaging , led us to this rare diagnosis . either the induced delivery and/or the unexplained coughs could have caused the vad leading to brain stem lesions . cases of dissections with or without concomitant csvt following deliveries have been reported [ 6 , 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 ] ; however , our case differs from those found in the literature in that : ( 1 ) the gynecological course was more complicated [ intrauterine fetal death ( iufd ) and induced delivery ] ; ( 2 ) our patient experienced unexplained dyspnea and coughing 2 weeks before iufd diagnosis ; ( 3 ) our patient suffered from concomitant vad , opalski syndrome and csvt , and ( 4 ) there were suspected findings of bilateral damages in the brain stem - spinal cord junction . women of child - bearing age have a higher incidence of stroke compared to age - matched men [ 6 , 10 , 11 , 12 ] , with only mild differences between pregnant and nonpregnant patients . nonpregnant women have a lower incidence of hemorrhagic than ischemic strokes ; however , these frequencies become similar during pregnancy , suggesting pregnancy as a risk factor for hemorrhagic strokes [ 11 , 12 ] . strokes usually occur during the third trimester and the puerperium , up to 1 month postpartum . eclampsia / preeclampsia remains the main cause for both types of strokes ( up to 47% ) . hemorrhagic strokes carry a poor prognosis [ 20 , 21 ] . during the peripartum period , the overall risk for strokes ( 13.110.3/100,000 ) and csvt ( 11.6/100,000 ) is similar . among nonpregnant aged - matched females , csvt occur less frequently [ 6 , 18 ] , suggesting that in the peripartum period , women are prone to develop csvt ( approx . 5 - 20% of all csvt occur during this period ) , mainly during the first 14 days postpartum . notably , csvt by itself is considered as a cause for stroke in up to 38% of cases during the postpartum period [ 6 , 9 , 14 , 15 , 16 ] . extracranial dissections commonly present as neck pain and headache ( occipital and posterior cervical regions ) , and might be proceeded by ischemic symptoms within the first weeks of the dissection . unlike carotid dissections , vad more frequently extend intracranially and involve the subarachnoid space causing meningism . less than 30% of the cases have simultaneous dissections of two or more vessels , for which an underlying connective tissue disorder should be suspected [ 17 , 18 , 31 ] . cervicocephalic arterial dissection ( ccad ) is a rare postpartum complication , with only 16 reported cases to date [ 7 , 8 , 9 , 10 , 11 , 13 , 14 , 15 , 16 , 17 ] . the rate of spontaneous ccad in the general population is 2.6/100,000 , of which 2.4% can occur during the postpartum period , meaning that there are 6.2/10,000,000 annually [ 9 , 23 , 24 ] , representing 6% of spontaneous ccad in women under 50 years of age . extracranial dissections seem to occur more frequently among pregnant women . up to 90% of vad reversible cerebral vasoconstriction syndrome was associated with stroke during the postpartum period among 4/6 patients compared with only 3/96 aged - matched controls . contrary to reversible cerebral vasoconstriction syndrome which has a favorable outcome , intracranial dissections in postpartum patients are characterized by a worse outcome [ 7 , 30 ] . the different occurrence between intra- and extradural dissections could possibly be explained by histological changes that appear once arteries ( vas and internal carotid arteries ) become  intradural :  the change is characterized first by a diminution in thickness of the adventitial and medial coats , and second , by a very gross reduction or loss of elastic fibers in the media and external elastic lamina . our patient needed a tradeoff between bp reduction ( due to the possible hemorrhagic transformation in the stroke area ) and the general permissive hypertension ( for dissections ) . the second issue concerns the usage of antiplatelet / anticoagulation drugs , to which caution was given due to the possible hemorrhagic transformation of a stroke . once anticoagulation treatment is initiated , it will be generally replaced by antiplatelet therapy for approximately 6 months , due to the fact that most ccad resolve after 3 - 6 months . neither therapy ( antiplatelet vs. anticoagulant ) was found superior for treating acute dissection [ 26 , 27 ] . once the possibility of a connective tissue disorder is excluded , the risk of recurrence is 1% within 1.4 - 8.6 years and up to 2% within the first months following the dissection [ 28 , 32 ] . our patient presented vascular pathologies of two different origin : one of venous origin ( csvt ) and one of arterial origin ( vad ) . both could have been complications of the peripartum period and of the induced delivery . hypercoagulability should also be considered since it can either cause or be a consequence of iufd . since no postmortem examination of the fetus was done , these issues as well as the reason for the iufd remained unsolved . despite two previous normal pregnancies / deliveries and noncontributory personal / familial coagulopathies , transient hemodynamic as well as hematologic changes during pregnancy and puerperium might be considered as additional causes . pregnancy might alter all three factors of virchow 's triad ( blood flow , blood vessel wall and constitution of blood ) , and patients are therefore prone to develop vascular diseases , as seen in our patient . opalski syndrome is caused by compromised blood supply and/or hemodynamic vulnerability , both likely occurring during puerperium and/or induced delivery . it is well accepted that nearly all posterior inferior cerebellar artery ( pica ) occlusions and more than 50% of va occlusions ( mainly if a pica origin is included ) might manifest as lms . importantly , 75% of all lms cases are related to va occlusion and 12% to pure pica occlusions . the intradural v4 segment ascends to the lower pontine border and forms the basilar artery , giving rise to the pica en route ( 10 mm distal to the dura perforation ) . our patient 's right v3 - 4 dissection included the pica , presenting as right medullary and cerebellar infarcts . this dissection might explain both constituents of opalski syndrome : ( 1 ) lms signs might be caused by the pica - va - related brain stem strokes , while ( 2 ) concomitant ipsilateral hemiparesis might be caused by the involvement of the anterior spinal artery arising from v4 that normally supplies the ventral two thirds of the spinal cord , including the pyramidal tract . combined venous - arterial disease should be considered among patients at high - risk for both pathologies , such as women during puerperium , even when their medical history is unremarkable . discrepancies between neurological findings and incidental neuroimaging findings can mislead . a clear - cut mechanism for the combined pathology could not be found ; however , the combination of induced delivery combined with hypercoagulability and/or hemodynamic vulnerability during puerperium seems a reasonable option . it seems plausible that either the coughs and/or the induced delivery have caused the vad that , in turn , led to opalski syndrome . conservative treatment combining bp control and anticoagulation seems an appropriate first step for these kinds of combined pathologies .
background and purposewe present the first case of combined arterial ( vertebral artery dissection ) and venous [ central sinus vein thrombosis ( csvt ) ] diseases presenting as opalski syndrome in a female patient following induced delivery.case descriptiona 32-year - old woman was admitted to our institute two weeks after induced delivery with intriguing neurological findings that were finally diagnosed as a combined venous - arterial disease . 
 although she was referred diagnosed with csvt , her neurological findings indicated wallenberg  plus syndrome with ipsilateral hemiparesis ( opalski syndrome ) , further confirmed by neuroimaging revealing arterial disease ( vertebral artery dissection ) combined with incidental acute csvt . 
 coagulation , gynecological and cardiac problems were ruled out . 
 treatment consisted of continuous heparin with rigorous control of her blood pressure . 
 nine days later , the patient was discharged with prominent improvements . 
 most of the symptoms resolved following 3 months of rehabilitation.conclusionsatypical strokes ( such as opalski syndrome ) might present in postpartum patients . 
 this rare diagnosis should be suspected in patients with wallenberg 
 plus syndrome , and neuroimaging studies for determining the presence of arterial disease and brain stem lesions should be performed . 
 concomitant csvt is rare and might mislead . 
 fine diagnosis followed by immediate conservative treatment can be of great benefit .