Brain metastases from urachal carcinoma
response to GnRH (done only the first patient) was adequate, corroborating pituitary integrity. Basal assessment in our patients was done under neuroleptic and anti-epileptic therapy. Dynamic testing, however, was done withholding treatment. Clozapine has been shown to lower IGF-19 and sodium valproate has been shown to lower GH in normal subjects10 without, however, compromising the response of GH in dynamic testing11;12 and particularly in the ITT.13 IGF-1 levels were low in the second patient and low-normal in the first patient, implying in the latter satisfactory 24-h (time integrated) overall secretion of GH. Although our patients’ MRI studies were normal, a pathological process, attributable to NA can be suggested, which disrupted hypothalamic function, while sparing, up to the moment of the study, the pituitary per se, thus enabling the various degrees of pituitary response to dynamic testing. Deficiencies in the hypothalamic–pituitary–adrenal axis have been described in NA,5 as well as low levels of HbA1c, possibly resulting from the shortened half-life of acanthocytes.14
REFERENCES 1. Hardie RJ, Pullon HWH, Harding AE et al. Neuroacanthocytosis. A clinical, haematological and pathological study of 19 cases. Brain 1991; 114: 13–49. 2. Rubio JP, Danek A, Stone C et al. Chorea-acanthocytosis: genetic linkage to chromosome 9q21. Am J Hum Genet 1997; 61: 899–908. 3. Schwartz MS, Monro PS, Leigh PN. Epilepsy as the presenting feature of neuroacanthocytosis in siblings. J Neurol 1992; 239: 261–262. 4. Sakai T, Mawatari S, Iwashita H, Goto I, Kuroiwa Y. Choreoacanthocytosis. Clues to clinical diagnosis. Arch Neurol 1981; 38: 335–338. 5. Terao S, Sobue G, Takahashi M et al. Disturbance of hypothalamic–pituitary hormone secretion in familial chorea-acanthocytosis. No To Shinkei 1995; 47: 57–61. 6. Rose SR. Disorders of thyrotropin synthesis, secretion, and function. Curr Opin Pediatr 2000; 12: 375–381. 7. Feldman A, Bloomgarden ZT. Hypophysiotropic hormone testing in a patient with hypothalamic hypopituitarism. Am J Med 1986; 80: 1006–1010. 8. Jambart S, Turpin G, de Gennes JL. Panhypopituitarism secondary to head trauma: evidence for a hypothalamic origin of the deficit. Acta Endocrinol (Copenh) 1980; 93: 264–270. 9. Melkersson KI, Hulting AL, Brismar KE. Different influences of classical antipsychotics and clozapine on glucose-insulin homeostasis in patients with schizophrenia or related psychoses. J Clin Psychiatry 1999; 60: 783–791. 10. Shiah IS, Yatham LN, Lam RW, Zis AP. Divalproex sodium attenuates growth hormone response to baclofen in healthy human males. Neuropsychopharmacology 1998; 18: 370–376. 11. Invitti C, Danesi L, Dubini A, Cavagnini F. Neuroendocrine effects of chronic administration of sodium valproate in epileptic patients. Acta Endocrinol (Copenh) 1988; 118: 381–388. 12. Franceschi M, Perego L, Cavagnini F et al. Effects of long-term antiepileptic therapy on the hypothalamic–pituitary axis in man. Epilepsia 1984; 25: 46–52. 13. Abraham RR, Dornhorst A, Wynn V et al. Corticotrophin, cortisol, prolactin and growth hormone responses to insulin-induced hypoglycaemia in normal subjects given sodium valproate. Clin Endocrinol (Oxf) 1985; 22: 639–644. 14. Ogawa T, Seki H, Okita N, Nomura H, Takase S. A case of choreaacanthocytosis associated with low glycohemoglobin A1c. Rinsho Shinkeigaku 1993; 33: 344–346.
Brain metastases from urachal carcinoma Takanobu Kaido1 MD, Hirotsugu Uemura2 MD, Yoshihiko Hirao2 MD, Ryunosuke Uranishi1 MD, Noriyuki Nishi1 MD, Toshisuke Sakaki1 MD ª 2003 Elsevier Ltd. All rights reserved.
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Department of Neurosurgery, Nara Medical University, Nara, Japan, Department of Urology, Nara Medical University, Nara, Japan
Summary We present a case of brain metastases of the urachal carcinoma, which is extremely rare and malignant. Contrastenhanced MRI was employed to detect them. A large mass was removed surgically and 4 other small metastases were treated by gamma knife radiosurgery. Six weeks after radiosurgery, the 4 lesions had disappeared on MRI. We emphasise the importance of early diagnosis using MRI and treatment by radiosurgery for this rare condition. ª 2003 Elsevier Ltd. All rights reserved. Journal of Clinical Neuroscience (2003) 10(6), 703–705 0967-5868/$ - see front matter ª 2003 Elsevier Ltd. All rights reserved. doi:10.1016/S0967-5868(03)00016-X
Keywords: urachal carcinoma, brain metastasis, gamma knife radiosurgery Received 12 August 2002 Accepted 30 September 2002 Correspondence to: Takanobu Kaido MD, Department of Neurosurgery, Nishinara National Hospital Shichijo 2-789, Nara-city, Nara, 630-8053, Japan. Tel.: +81-742-45-4591; Fax: +81-742-48-3512; E-mail:
[email protected]
INTRODUCTION Brain metastasis of the urachal carcinoma is extremely rare. This is the first case of this disease detected using contrast-enhanced MRI and treated with gamma knife radiosurgery (GKS), and the third case of brain metastasis, to our knowledge. CASE REPORT A 62 year old man had a seizure 5 years after partial cystectomy and chemotherapy for urachal carcinoma (adenocarcinoma) of the urinary bladder. He also had a history of operations for local recurrence and lung metastases 2 years after the first surgery. A contrast-enhanced MRI revealed an well-enhanced mass, 3 cm in diameter, in the right frontal region (Fig. 1A). Moreover, 4 small masses in the right cerebellar and left occipital regions were also detected (Fig. 1B). The right frontal tumour was totally removed. The histological diagnosis of the tumour was metastatic adenocarcinoma from urachal carcinoma (Fig. 2). The postoperative course was uneventful. Four other small metastases were treated by 201-source cobalt-60 gamma knife. Six weeks after GKS, these 4 lesions had disappeared on contrast-enhanced MRI (Fig. 3A). Three months after removal of the tumour, contrast-enhanced MRI revealed recurrence of the frontal tumour at the operation site and 10 small metastases at a different site from the previous small lesions. The recurrent right frontal tumour was removed again. The 10 new metastases were radiated with GKS. Six months after the first operation, the general condition of the patient suddenly deteriorated. Brain MRI revealed invasion of tumour in the right parietal lobe where the tumour had been removed and new, small spotty lesions (Fig. 3B). Soon therefore, this patient died. DISCUSSION This is the first reported case of detection of small metastatic lesions using MRI, the first report of gamma knife radiosurgery Journal of Clinical Neuroscience (2003) 10(6)
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Fig. 1 (A, B) Contrast-enhanced T1-weighted axial MR images before gamma knife radiosurgery (GKS) showing the right frontal well-enhanced mass (A) and the right cerebellar enhanced small mass (B, arrow).
Fig. 2 Photomicrograph of a tumour specimen in the right frontal lobe showing metastatic adenocarcinoma (H & E stain, original magnification 100).
(GKS) for brain metastases of urachal carcinoma, and the third report of brain metastasis of this cancer to our knowledge. The urachus arises embryologically as a vestigial remnant of the allantois.1;2 The urachal carcinoma is an uncommon malignancy. The annual incidence is 0.01% of all cancer cases in adults,1 Early and correct diagnosis is necessary because of poor prognosis due to the aggressive nature. Distant metastases are common in the lung, omentum, liver, bone, and iliac and inguinal groups of lymph nodes.1 However, Journal of Clinical Neuroscience (2003) 10(6)
Fig. 3 (A, B) Contrast-enhanced T1-weighted axial MR images. MR image at six weeks after GKS (A) showing disappearance of lesion. MR image at six months after operation (B) showing tumor invasion at the operated region and new spotty lesion in the left frontal lobe.
brain metastases are extremely rare and have been previously reported in only 2 cases.3;4 Radiosurgery, or stereotactically focused irradiation, has been widely used for central nervous system diseases such as brain tumour(s). It is considered to be a relatively safe procedure with small risks, such as radiation necrosis, and rarely, induction of brain tumour.5 A previous study reported that most cases of urachal carcinoma have appeared to be resistant to radiotherapy.1 However, in the present case, GKS was effective in treating small metastases from urachal carcinoma. The present report also showed that contrast-enhanced MRI was very useful in the detection of small metastases from urachal carcinoma. We predict that cases of this rare but important metastatic brain tumour will increase in number hereafter with long-term and careful follow up using contrast-enhanced MRI, and GKS will be available to treat it.
REFERENCES 1. Sheldon CA, Clayman RV, Gonzalez R, Williams RD, Fraley EE. Malignant urachal lesions. J Urol 1984; 131: 1–8. 2. Rubell D, Porges RF. Carcinoma of urachus: a case report and review. Obstet Gynecol 1972; 39: 753. 3. Fujiwara S, Takaki T, Hikita T, Kanzaki H, Kuroiwa S. Brain metastasis from urachal carcinoma. Surg Neurol 1988; 29: 475–476. 4. Tewari MK, Khosla VK, Sharma BS, Vashistha RK, Khandelwal NK, Kak VK. Brain metastasis from urachal carcinoma: case report. Surg Neurol 1994; 42: 340–342.
ª 2003 Elsevier Ltd. All rights reserved.
‘ Ecstasy’-induced subarachnoid haemorrhage
5. Kaido T, Hoshida T, Uranishi R, Akita N, Kotani A, Nishi N, Sakaki T. Radiosurgery-induced brain tumor. J Neurosurg 2001; 95: 710–713.
‘Ecstasy’-induced subarachnoid haemorrhage: an under-reported neurological complication? Gabriel Yin Foo Lee MBBS (HONS) MS, Grace Wooi Kee Gong MBBS, Nikitas Vrodos FRACS, Brian Patrick Brophy FRACS Department of Neurosurgery, Royal Adelaide Hospital, Adelaide, Australia
Summary In the face of escalating recreational use of ‘Ecstasy’ (3,4-methylenedioxymethamphetamine, MDMA), physicians need to be aware of its possible adverse effects. We report two young patients who suffered subarachnoid haemorrhage following ingestion of ‘Ecstasy’ tablets. Angiographic studies demonstrated features consistent with vasculitis in both cases. Recognition of this association is important and highlights the significance of eliciting a careful drug history, particularly in cases of ‘angiogram negative’ subarachnoid haemorrhage. ª 2003 Elsevier Ltd. All rights reserved. Journal of Clinical Neuroscience (2003) 10(6), 705–707 0967-5868/$ - see front matter ª 2003 Elsevier Ltd. All rights reserved. doi:10.1016/S0967-5868(03)00151-6
Keywords: ecstasy, MDMA, subarachnoid haemorrhage, vasculitis Received 13 November 2002 Accepted 6 December 2002 Correspondence to: Dr Gabriel Lee, Department of Neurosurgery, Sir Charles Gairdner Hospital, Hospital Avenue, Nedland, Western Australia. Tel.: +61-8-93463333; Fax: +61-8-93463824; E-mail:
[email protected]
INTRODUCTION It is increasingly well recognised that recreational use of Ecstasy (MDMA) can lead to life threatening consequences. While neurological complications are uncommon, this may represent an under-reported phenomenon. We present two young patients in whom Ecstasy use was associated with non-aneurysmal subarachnoid haemorrhage. CASE 1 A 29-year-old woman experienced a sudden onset of severe occipital headache which was associated with nausea and vomiting 5 days prior to presentation to the Royal Adelaide Hospital. The patient complained of severe photophobia. On clinical examination, the patient was alert and orientated. There was marked nuchal rigidity. Neurological examination did not reveal any focal deficit. CT head scan was normal. Subsequent lumbar puncture revealed blood stained CSF with photospectrometric evidence of xanthochromia. The patient underwent conventional four vesselcerebral angiography which demonstrated evidence of ‘beading’ ª 2003 Elsevier Ltd. All rights reserved.
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of vessels within the posterior circulation. The angiographic appearances were most marked on the (L) superior cerebellar artery (Fig. 1). Further investigation with MRI/MRA revealed no significant abnormality. Serum testing revealed an erythrocyte sedimentation rate (ESR) of 11 and C-reactive protein (CRP) of 4. Antinuclear antigen (ANA) was negative. Following the patient’s admission, her friends subsequently volunteered information of illicit drug use by the patient. On repeated questioning, the patient admitted that she had used ‘Ecstasy’ on the day of ictus. She denied having previously used ‘Ecstasy’ or any other recreational drugs. The patient was observed without neurological deterioration. She was discharged at Day 13 of her admission. The delay was due to persistent headaches. Follow-up cerebral angiography 2 months later showed that the abnormalities had completely resolved. CASE 2 A 24-year-old man smoked marijuana and subsequently consumed one and a half ‘Ecstasy’ tablets while at a party. Several hours later, he experienced a sudden severe retro-orbital headache and felt unwell. Soon after arriving home, he suffered a witnessed episode of generalised tonic-clonic seizure lasting approximately 1 min. This was followed by a brief period of post-ictal confusion. On arrival at the emergency department, he was alert and orientated. Neurological examination was otherwise unremarkable. He had a further generalised seizure in the emergency department which was aborted by intravenous midazolam. The patient underwent a CT head scan. This revealed the presence of subarachnoid haemorrhage which was mainly localised to several parasagittal sulci towards the vertex (Fig. 2). Cerebral angiography demonstrated evidence of focal ‘beading’ of peripheral branches to the (R) anterior cerebral cortex consistent with angiographic arteritis. MRI/MRA head scan confirmed the presence of subarachnoid haemorrhage but no other abnormalities. The patient remained well subsequently with resolution of his headaches and no further seizures. He was discharged at Day 4 following his admission. DISCUSSION MDMA, also known as ‘Ecstasy’, is a three ring-substituted, methoxylated analogue of methamphetamine. It was first synthesized in Germany in 1914 and used initially as an appetite suppressant.1;2 During the late 1970s and 1980s, it found a secondary use in the United States as an adjunct to psychotherapy.1;2 The current widespread recreational use of MDMA by teenagers can primarily be traced back to the British dance hall phenomenon called ‘raves’.3 The use of MDMA was reported by 8% of 15- and 16-year-old British students surveyed in 1994.4 A more recent survey at three dance events in Edinburgh, Scotland demonstrated that over 80% of participants had previously used Ecstasy and amphetamines. Furthermore, 35% stated that they used Ecstasy on a weekly basis.5 Its use has since spread across to other continents.3;6 A random survey of illicit drug use by American undergraduate students revealed that 24% of those surveyed reported use of MDMA, exceeding use of lysergic acid diethylamide (LSD) and cocaine.3 Furthermore, MDMA has also become a popular choice for University students and attendees at rave parties in major coastal American cities.3 Studies from Australia 7 and Germany 6 have similarly demonstrated a high rate of MDMA usage. A 1999 survey of drug use in Norwegian adolescents further showed that MDMA was used by adolescents who also used other legal and illegal substances in a polydrug-use pattern.8 Journal of Clinical Neuroscience (2003) 10(6)