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Fulltext Cavernous lymphangioma of the digits: a rare cause of macrodactyly

Leong, J.F., Levin, K.B., Rajkumar V., Abdullah, S., Jamari, S.

Medicine & Health, 2019;14(2):261-265.
MyJurnal

Cavernous lymphangioma is a congenital malformation of lymphatic system causing dilated lymphatic sinuses that involve the skin and subcutaneous tissues. This was an interesting case of dystrophic macrodactyly of the left ring and little finger in a 18-month-old girl who presented with swollen and sausage like fingers deformity which turned out to be an isolated cavernous lymphangioma. This tumor, although rare to occur in the extremeties, must be differentiated from other congenital vascular lesions of the hand that include arteriovenous malformations and hemangiomas. Diagnosis should be solely based on histopathological analysis of the excised tissue mass. Surgical excision is usually necessary for satisfactory functional and cosmetic outcome

Matched MeSH terms: Arteriovenous Malformations
Fulltext The Spetzler-Martin Grading System and management of patients with intracranial arteriovenous malformation in a tertiary referral hospital

Al-Edrus, S. A., Suhaimi, S. N., Noor Azman, A. R., Latif, A. Z., Sobri, M.

Malaysian Journal of Medicine and Health Sciences, 2010;6(1):91-99.
MyJurnal

Introduction: An arteriovenous malformation (AVM) is an abnormal collection of blood vessels in which arterial blood flows directly into the draining vein without the normal interposed capillaries. The Spetzler-Martin grading system has been widely accepted worldwide to estimate the pretreatment risks and predict the outcome of patients with intracranial AVM. In Malaysia, we still do not have the baseline data of this grading system. Methods: A total of 33 patients from a tertiary referral hospital diagnosed with intracranial AVM based on neuroimaging findings over a 4-year period were studied. Medical
records were traced and neuroimaging findings were analysed. The AVMs were graded according to the Spetzler-Martin grading system and Fisher’s exact test was used to assess statistical difference between the grades of the AVM and management plan for the patients. Results: Four patients were graded as Grade 1, 9 patients as Grade II, 10 patients as Grade III, 6 patients as Grade IV and 4 patients as Grade V. Ten patients were treated conservatively; six patients underwent surgery and embolisation respectively. Four patients underwent radiosurgery and a combination of embolisation while surgery and radiosurgery were given to 5 patients and 2 patients respectively. Statistically significant difference (p=0.016) was found between the Spetzler-Martin grading system and the management of intracranial AVMs. Conclusion: The management decision was not made based on the
grading of the AVMs. It is recommended that all AVM patients be routinely graded according to this system prior to treatment.

Matched MeSH terms: Intracranial Arteriovenous Malformations
Fulltext Spontaneous Thrombosis of Dural Arteriovenous Malformation: A Case Report

Sobri M., Mezlina W.Z., Subramaniam, J.H.

International Medical Journal Malaysia, 2009;8(2):39-42.
MyJurnal

Dural arteriovenous malformation (DAVM) is relatively rare and defined as abnormal connections or shunts between the arterial and the venous side of vascular tree located within the dura mater. Spontaneous closures of DAVM are rare and have been scarcely reported. This case report will describe the neuroimaging findings and classification of DAVM. A 50 year old lady presented with headache. Neuroimaging showed prominent serpinginous flow-void structures, cerebral angiogram confirmed the presence of DAVM at the occipital region. She had defaulted treatment and follow-up for 3 years. On second admission, she had a cerebral angiogram which showed normal findings with no evidence of fistulas or malformation. She was discharged well. Causes of spontaneous closure of DAVM are discussed.

Matched MeSH terms: Arteriovenous Malformations
Fulltext Hematospermia: an unusual cause of urology emergency

Goh, E.H., Tan, G.H., Rozman, Z., Nik Azuan, N.I., Fam, X.I., Mohd Hafidzul, J., et al.

Journal of Surgical Academia, 2017;7(1):72-74.
MyJurnal

Hematospermia is a distressing disorder in sexually active men resulting in great concern to the patient. We report an unusual case of hematospermia with an atypical presentation, involving a 54-year-old man presented with acute urinary retention after sexual intercourse. Although the causes are widely known, arteriovenous malformation as the cause of this disorder has not much been reported in the literature. Transcatheter embolization of internal pudendal artery is a promising option for hematospermia caused by arterial fistula or bleeding.

Matched MeSH terms: Arteriovenous Malformations
Fulltext Phenytoin-induced severe thrombocytopaenia post dexamethasone co-administration in a patient with intracerebral haemorrhage

Lau, E.F., Mazlan, M., Shanmugam, H.

JUMMEC, 2018;21(2):31-34.
MyJurnal

Phenytoin is commonly prescribed for the prophylaxis of seizures in neurosurgical patients. A phenytoininduced
serious adverse effect of thrombocytopenia has been reported in the literature. The concurrent
use of dexamethasone, another commonly prescribed drug in neurosurgical patients, has been reported to
aggravate this adverse haematological effect. We present a report of phenytoin-induced thrombocytopenia
in a patient concurrently prescribed with dexamethasone, after an intracerebral haemorrhage secondary to
a rupture of an arteriovenous malformation. The thrombocytopenia was noted after two weeks of phenytoin
medication. Phenytoin was immediately withheld, and seven units of random donor platelets were transfused.
A gradual resolution of thrombocytopenia was observed within a week.

Matched MeSH terms: Arteriovenous Malformations
Fulltext Uterine arteriovenous malformation

Hashim H, Nawawi O

Malays J Med Sci, 2013 Mar;20(2):76-80.
PMID: 23983582 MyJurnal

Uterine arteriovenous malformation (AVM) is a rare condition, with fewer than 100 cases reported in the literature. Despite it being rare, it is a potentially life-threatening condition. This case report describes a 33-year-old woman who presented with secondary post-partum hemorrhage. Transabdominal ultrasound (US) of the pelvis showed increased vascularity with multidirectional flow of the uterus and a prominent vessel, located on the left lateral wall. She also had retained product of conception, which complicated the diagnosis. A uterine artery angiogram confirmed an AVM in the fundal region with an early draining vein. Embolisation of the AVM was performed successfully.

Matched MeSH terms: Arteriovenous Malformations
Fulltext Angioarchitecture of brain arteriovenous malformations and the risk of bleeding: an analysis of patients in northeastern malaysia

Kandai S, Abdullah MS, Naing NN

Malays J Med Sci, 2010 Jan;17(1):44-8.
PMID: 22135525

Central nervous system arteriovenous malformation (AVM) is a vascular malformation of the brain and involves entanglement of veins and arteries without an intervening capillary bed. Affecting predominantly young male patients, AVM presents with different clinical manifestations namely headache, seizures, neurological deficit and intracranial haemorrhage. The patients who present acutely with intracranial bleeding have a significant morbidity and mortality. The aim is to study the angioarchitecture of brain AVM (BAVM) and determine the risk factors for intracranial bleeding. Ultimately, the goal of the study is to look for the association between volume of haematoma and architecture of BAVM.

Matched MeSH terms: Intracranial Arteriovenous Malformations
Education and Imaging. Gastrointestinal: massive intraperitoneal haemorrhage in a young woman with hereditary hemorrhagic telangiectasia

Sivarani S, Chan YK, Liam CK, Kumar G, Abdullah BJ, Mahadeva S

J Gastroenterol Hepatol, 2010 Sep;25(9):1587.
PMID: 20796159 DOI: 10.1111/j.1440-1746.2010.06469.x
Matched MeSH terms: Arteriovenous Malformations/complications*
Fulltext Development of pseudo-subarachnoid hemorrhage secondary to hypoxic-ischemic injury due to bleeding pulmonary arterio-venous malformation

Md Noh MSF, Abdul Rashid AM

BMC Neurol, 2018 Sep 28;18(1):157.
PMID: 30266082 DOI: 10.1186/s12883-018-1161-x

BACKGROUND: The computed tomography (CT) finding of a pseudo-subarachnoid hemorrhage (SAH) may lead the treating physician into a diagnostic dilemma. We present a case of a pseudo-SAH in a patient with post-resuscitative encephalopathy, secondary to a newly diagnosed bleeding pulmonary arterio-venous malformation (AVM).
CASE PRESENTATION: A 19-year-old female presented acutely with massive hemoptysis. Cardiopulmonary resuscitation (CPR) followed, and the patient was subsequently intubated for airway protection with intensive care unit (ICU) admission. Urgent CT angiography of the thorax showed a bleeding pulmonary AVM, with evidence of hemothorax. Non-contrasted cranial CT initially revealed cerebral edema. Day 3 post admission, repeat cranial CT showed worsening cerebral edema, with evidence of pseudo-SAH. Patient passed away the next day.
CONCLUSIONS: Pseudo-SAH, if present, carries a poor prognosis. It should be recognized as a potential CT finding in patients with severe cerebral edema, due to various causes. The diagnosis is vital, to avoid wrongful treatment institution, as well as determination of cause of death.

Matched MeSH terms: Arteriovenous Malformations/complications
Fulltext Aneurysms are more common than arteriovenous malformations as the cause of subarachnoid haemorrhage in the Malaysian population

Chee CP, Loh TG

Singapore Med J, 1988 Oct;29(5):427-31.
PMID: 3241969

A retrospective prospective study of 84 cases of subarachnoid haemorrhage, 24 intracranial aneurysms and 18 arteriovenous malformations (AVM's) treated in the University Hospital, Kuala Lumpur, during the post CT-scan era was carried out to verify the relative frequencies of these clinical entities in the Malaysian population. Our results show that aneurysms are commoner than arteriovenous malformations as a cause of subarachnoid haemorrhage and also the most frequent as a whole, thus refuting the previous claims that AVM's are 4 to 10 times more common than aneurysms in this part of the world. Of interest was the internal carotid artery aneurysms accounted for half of the anterior circulation aneurysms and that 2/3 of the AVM's presented with intracranial haemorrhage.

Matched MeSH terms: Intracranial Arteriovenous Malformations/complications*
Fulltext Severe left anterior descending artery stenosis with proximal arteriovenous malformation presenting as acute myocardial infarction

Oteh M, Azarisman SM, Hanim NM, Noorfaizan S

Singapore Med J, 2009 Feb;50(2):e76-8.
PMID: 19296018

Congenital coronary artery anomalies are rare, with an incidence of about 0.06-1.3 percent of all patients undergoing cardiac catheterisation. They are commonly asymptomatic, but potentially serious lesions may lead to myocardial ischaemia, infarction and/or sudden cardiac death. The occurrence of a concomitant stenotic lesion is exceedingly rare. We report an 80-year-old man who presented with acute anterior myocardial infarction. Coronary angiography revealed severe proximal left anterior descending (LAD) and arteriovenous malformation (AVM) from the first septal branch of the LAD. The LAD stenosis and the AVM were successfully treated with two Jomed covered stents.

Matched MeSH terms: Arteriovenous Malformations/diagnosis*; Arteriovenous Malformations/pathology*
Uterine arteriovenous malformation - Possible association to uterine fibroids?

Gan F, Vikneswaran V, Yu KK

Med J Malaysia, 2021 03;76(2):273-274.
PMID: 33742646

A 32-year-old, gravida 2 para 0+1, was managed in Selayang Hospital, Selangor for uterine fibroids in pregnancy and placenta previa major. The lady went into preterm labour at 33 weeks, requiring emergency Caesarean section. Intraoperatively, we found a thinned-out bulge between the intramural uterine fibroids at the posterior uterine wall, which then perforated and was repaired. Persistent bleeding post operatively led to relaparotomy and hysterectomy. Histology of the uterus reported arteriovenous malformation (AVM). We postulate the possibility of these lesions coexisting with uterine fibroids. Screening for uterine AVMs in patients with fibroids may lead to early detection with option of embolization; deferring the need for hysterectomy.

Matched MeSH terms: Arteriovenous Malformations
Fulltext Hereditary hemorrhagic telangiectasia or Osler-Weber-Rendu syndrome: management of epistaxis in 4 cases

Salina H., Lim P.S., Gendeh B.S.

International Medical Journal Malaysia, 2018;17(2):129-134.
MyJurnal

Hereditary Hemorrhagic Telangiectasia, also known as Osler-Weber-Rendu Syndrome is an autosomal
dominant disorder causing systemic abnormalities of the vascular structure. There are multiple arteriovenous malformations present in the skin and mucosal surface of the nail beds, nose, gastrointestinal tract, lungs and brain. Epistaxis is the common presentation symptom, which may require multiple hospital admissions and blood transfusions. It is extremely rare disease in our population. We report 4 cases of HHT who presented to us with moderate to severe epistaxis and how we managed these patients.

Matched MeSH terms: Arteriovenous Malformations
Fulltext Expression of osteopontin, matrix metalloproteinase-2 and -9 proteins in vascular instability in brain arteriovenous malformation

Anbarasen L, Lim J, Rajandram R, Mun KS, Sia SF

PeerJ, 2019;7:e7058.
PMID: 31275742 DOI: 10.7717/peerj.7058

Background: Matrix metalloproteinase (MMP)-2 and -9 are Osteopontin (OPN) dependent molecules implicated in the destabilization of blood vessels. OPN and MMPs have been studied in brain arteriovenous malformation (BAVM) patients' tissues and blood samples before intervention. In this study, we compared the serum level of these markers before and after treatment, as well as assessed their protein expressions in BAVM tissues to evaluate their roles in this disease.
Methodology: Serum samples from six BAVM patients and three control subjects were analyzed using enzyme-linked immunoabsorbent assay (ELISA) for OPN. A total of 10 BAVM patients and five control subjects were analyzed using Multiplex ELISA for MMPs. A total of 16 BAVM tissue samples and two normal brain tissue samples were analyzed using immunohistochemistry.
Result: MMP-2 and -9 were significantly higher in the serum of BAVM patients before and after treatment than in control patients. There were no significant differences of OPN and MMP-9 serum level in BAVM patients before and after treatment. MMP-2 showed a significant elevation after the treatment. Expression of OPN, MMP-2 and -9 proteins were seen in endothelial cells, perivascular cells and brain parenchyma of BAVM tissues.
Conclusion: Findings revealed that the level of MMP-2 and -9 in the serum correlated well with the expression in BAVM tissues in several cases. Knockdown studies will be required to determine the relationships and mechanisms of action of these markers in the near future. In addition, studies will be required to investigate the expression of these markers' potential applications as primary medical therapy targets for BAVM patients.

Matched MeSH terms: Intracranial Arteriovenous Malformations
Fulltext Neural oscillation, network, eloquent cortex and epileptogenic zone revealed by magnetoencephalography and awake craniotomy

Idris Z, Kandasamy R, Reza F, Abdullah JM

Asian J Neurosurg, 2014 Jul-Sep;9(3):144-52.
PMID: 25685205 DOI: 10.4103/1793-5482.142734

BACKGROUND: Magnetoencephalography (MEG) is a method of functional neuroimaging. The concomitant use of MEG and electrocorticography has been found to be useful in elucidating neural oscillation and network, and to localize epileptogenic zone and functional cortex. We describe our early experience using MEG in neurosurgical patients, emphasizing on its impact on patient management as well as the enrichment of our knowledge in neurosciences.
MATERIALS AND METHODS: A total of 10 subjects were included; five patients had intraaxial tumors, one with an extraaxial tumor and brain compression, two with arteriovenous malformations, one with cerebral peduncle hemorrhage and one with sensorimotor cortical dysplasia. All patients underwent evoked and spontaneous MEG recordings. MEG data was processed at band-pass filtering frequency of between 0.1 and 300 Hz with a sampling rate of 1 kHz. MEG source localization was performed using either overdetermined equivalent current dipoles or underdetermined inversed solution. Neuromag collection of events software was used to study brain network and epileptogenic zone. The studied data were analyzed for neural oscillation in three patients; brain network and clinical manifestation in five patients; and for the location of epileptogenic zone and eloquent cortex in two patients.
RESULTS: We elucidated neural oscillation in three patients. One demonstrated oscillatory phenomenon on stimulation of the motor-cortex during awake surgery, and two had improvement in neural oscillatory parameters after surgery. Brain networks corresponding to clinico-anatomical relationships were depicted in five patients, and two networks were illustrated here. Finally, we demonstrated epilepsy cases in which MEG data was found to be useful in localizing the epileptogenic zones and functional cortices.
CONCLUSION: The application of MEG while enhancing our knowledge in neurosciences also has a useful role in epilepsy and awake surgery.
KEYWORDS: Awake craniotomy; brain network; epilepsy; magnetoencephalography; neural oscillation

Matched MeSH terms: Arteriovenous Malformations
Bailouts During Neurointervention; Novel Techniques in Tackling Coil Migration and Premature Intravascular Detachment of Microcatheter Tip

Ambrosanio G, Arthimulam G, Leone G, Guarnieri G, Muto M, Muto M

World Neurosurg, 2020 10;142:167-170.
PMID: 32615295 DOI: 10.1016/j.wneu.2020.06.190

BACKGROUND: Intracranial vascular malformations are increasingly being treated via the endovascular route. Though generally safe, a multitude of intraprocedural complications that potentially lead to disastrous clinical outcomes may arise. It is crucial for the operators to be well versed with the various techniques that are available to overcome any procedure-specific complications.
METHODS: We present 2 cases in which we encountered premature intravascular detachment of the microcatheter tip and coil migration while treating a dural arteriovenous fistula and aneurysm, respectively. We used a stentriever to remove the detached microcatheter tip and suction using the reperfusion catheter to remove the migrated coil, both techniques that have not been reported in the literature thus far.
RESULTS: Detached microcatheter tip and migrated coil were successfully retrieved using a stentriever and aspiration catheter.
CONCLUSIONS: These novel techniques could potentially reduce mortality and morbidity associated with neurointervention.

Matched MeSH terms: Intracranial Arteriovenous Malformations/surgery*
Fulltext Challenges in the management of massive intraorbital and hemifacial arteriovenous malformation as causing life-threatening epistaxis

Manuel AM, Kalimuthu S, Pathmanathan SS, Narayanan P, Zainal Abidin Z, Azmi K, et al.

Asian J Surg, 2017 Apr;40(2):158-162.
PMID: 24210537 DOI: 10.1016/j.asjsur.2013.09.011

Arteriovenous malformations are congenital lesions that may evolve with time and manifest in a plethora of presentations. They can occur as torrential epistaxis when it extensively involves the facial region. Multi-imaging modalities are available to assist in characterizing the structure of the lesion as well as its location and extent. This complex disease requires a multidisciplinary team approach with preoperative embolization and surgery. We present a rare cause of life-threatening epistaxis in a gentleman with a longstanding orbital and hemifacial arteriovenous malformation and discuss the complexities involved in its management.

Matched MeSH terms: Arteriovenous Malformations/complications; Arteriovenous Malformations/therapy*
Fulltext Arteriovenous malformation in temporal lobe presenting as contralateral ocular symptoms mimicking carotid-cavernous fistula

Mohd-Tahir F, Siti-Raihan I, Wan Hazabbah WH

Case Rep Ophthalmol Med, 2013;2013:158961.
PMID: 23533876 DOI: 10.1155/2013/158961

Aim. To report a rare case of arteriovenous malformation in temporal lobe presenting as contralateral orbital symptoms mimicking carotid-cavernous fistula. Method. Interventional case report. Results. A 31-year-old Malay gentleman presented with 2-month history of painful progressive exophthalmos of his left eye associated with recurrent headache, diplopia, and reduced vision. Ocular examination revealed congestive nonpulsating 7 mm exophthalmos of the left eye with no restriction of movements in all direction. There was diplopia in left lateral gaze. Left IOP was elevated at 29 mmHg. Left eye retinal vessels were slightly dilated and tortuous. CT scan was performed and showed right temporal arteriovenous malformation with a nidus of 3.8 cm × 2.5 cm with right middle cerebral artery as feeding artery. There was dilated left superior ophthalmic vein of 0.9 mm in diameter with enlarged left cavernous sinus. MRA and carotid angiogram confirmed right temporal arteriovenous malformation with no carotid-cavernous fistula. Most of the intracranial drainage was via left cavernous sinus. His signs and symptoms dramatically improved following successful embolisation, completely resolved after one year. Conclusion. Intracranial arteriovenous malformation is rarely presented with primary ocular presentation. Early intervention would salvage the eyes and prevent patients from more disaster morbidity or fatality commonly due to intracranial haemorrhage.

Matched MeSH terms: Intracranial Arteriovenous Malformations

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