Lege Artis Medicinae

[A Man of Spirit – Remembering Béla Buda]

KAPÓCS Gábor

AUGUST 20, 2013

Lege Artis Medicinae - 2013;23(07-08)

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Life threatening rare lymphomas presenting as longitudinally extensive transverse myelitis: a diagnostic challenge

TOLVAJ Balázs, HAHN Katalin, NAGY Zsuzsanna, VADVÁRI Árpád, CSOMOR Judit, GELPI Ellen, ILLÉS Zsolt, GARZULY Ferenc

Background and aims – Description of two cases of rare intravascular large B-cell lymphoma and secondary T-cell lymphoma diagnosed postmortem, that manifested clinically as longitudinally extensive transverse myelitis (LETM). We discuss causes of diagnostic difficulties, deceptive radiological and histological investigations, and outline diagnostic procedures based on our and previously reported cases. Case reports – Our first case, a 48-year-old female was admitted to the neurological department due to paraparesis. MRI suggested LETM, but the treatments were ineffective. She died after four weeks because of pneumonia and untreatable polyserositis. Pathological examination revealed intravascular large B-cell lymphoma (IVL). Our second case, a 61-year-old man presented with headache and paraparesis. MRI showed small bitemporal lesions and lesions suggesting LETM. Diagnostic investigations were unsuccessful, including tests for possible lymphoma (CSF flow cytometry and muscle biopsy for suspected IVL). Chest CT showed focal inflammation in a small area of the lung, and adrenal adenoma. Brain biopsy sample from the affected temporal area suggested T-cell mediated lymphocytic (paraneoplastic or viral) meningoencephalitis and excluded diffuse large B-cell lymphoma. The symptoms worsened, and the patient died in the sixth week of disease. The pathological examination of the presumed adenoma in the adrenal gland, the pancreatic tail and the lung lesions revealed peripheral T-cell lymphoma, as did the brain and spinal cord lesions. Even at histological examination, the T-cell lymphoma had the misleading appearance of inflammatory condition as did the MRI. Conclusion – Lymphoma can manifest as LETM. In cases of etiologically unclear atypical LETM in patients older than 40 years, a random skin biopsy (with subcutaneous adipose tissue) from the thigh and from the abdomen is strongly recommended as soon as possible. This may detect IVL and provide the possibility of prompt chemotherapy. In case of suspicion of lymphoma, parallel examination of the CSF by flow cytometry is also recommended. If skin biopsy is negative but lymphoma suspicion remains high, biopsy from other sites (bone marrow, lymph nodes or adrenal gland lesion) or from a simultaneously existing cerebral lesion is suggested, to exclude or prove diffuse large B-cell lymphoma, IVL, or a rare T-cell lymphoma.

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A rare entity of acquired idiopathic generalised anhidrosis which has been successfully treated with pulse steroid therapy: Does the histopathology predict the treatment response?

ÖKTEM Özdemir Ece, ÇANKAYA Şeyda, UYKUR Burak Abdullah, ERDEM Simsek Nazan, YULUG Burak

Acquired idiopathic generalised anhidrosis is an uncommon sweating disorder characterized by loss of sweating in the absence of any neurologic, metabolic or sweat gland abnormalities. Although some possible immunological and structural mechanisms have been proposed for this rare entity, the definitive pathophysiology is still un­clear. Despite some successfully treated cases with systemic corticosteroid application, the dose and route of steroid application are controversial. Here, we present a 41-year-old man with lack of genera­lised sweating who has been successfully treated with high dose pulse intravenous prednisolone. We have discussed his clinical and histopathological findings as well as the treatment options in view of the current literature.

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[Commemorating the Lipótmező. Part 1.]

RIHMER Zoltán

[“What did Lipótmező mean to you?” My friends and acquaintances asked frequently this question in the past decades, concerning the National Institute for Psychiatry and Neurology or well known as the Lipótmező my past workplace and the role it played in my life thus far. It is difficult to give a short answer, but the three and a half decades I have spent there were certainly of decisive importance in my professional and private life as well. Since I was banned from tobacco smoking due to my disease ten years ago, I cannot keep my pipe in my mouth any more. Thus, I decided to recollect the dearest stories kept in my memory, which had the deepest impact on me during my 35 years in Lipótmező both as a doctor and as a man. ]

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To handle the HaNDL syndrome through a case: The syndrome of headache with neurologic deficits and cerebrospinal fluid lymphocytosis

ÇOBAN Eda, TEKER Ruken Serap, SERİNDAĞ Helin, SAKALLI Nazan, SOYSAL Aysun

The syndrome of headache with neurologic deficits and cerebrospinal fluid lymphocytosis (HaNDL) is a rare entity. This disease has been related to migrainous headaches. It is a benign, self-limited disorder, which is characterized by fluctuating neurological symptoms and cerebrospinal fluid lymphocytosis. We describe a case of a 47 years old man with acute onset of headache and aphasia. Cerebrospinal fluid analysis revealed a lymphocytic pleocytosis (25 cells/μl, 100% lymphocytes). Electroencephalogram showed moderate slow rhythm in the left hemisphere, with temporoparietal predominance, and without epileptiform activity. His blood tests as well as magnetic resonance imaging (MRI) results were normal. With the diagnosis of HaNDL syndrome the patient was accepted in the Department of Neurology and discharged with full recovery.

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[Thrombolysis in case of ischemic stroke caused by aortic dissection]

LANTOS Judit, NAGY Albert, HEGEDŰS Zoltán, BIHARI Katalin

[Seldom, an acute aortic dissection can be the etiology of an acute ischemic stroke. The aortic dissection typically presents with severe chest pain, but in pain-free dissection, which ranges between 5-15% of the case, the neurological symptoms can obscure the sypmtos of the dissection. By the statistical data, there are 15-20 similar cases in Hungary in a year. In this study we present the case history of an acute ischemic stroke caused by aortic dissection, which is the first hungarian publication in this topic. A 59-year-old man was addmitted with right-gaze-deviation, acute left-sided weakness, left central facial palsy and dysarthric speech. An acute right side ischemic stroke was diagnosed by physical examination without syptoms of acute aortic dissection. Because, according to the protocol it was not contraindicated, a systemic intravenous thrombolysis was performed. The neurological sypmtoms disappeared and there were no complication or hypodensity on the brain computed tomography (CT). 36 hours after the thrombolysis, the patient become restlessness and hypoxic with back pain, without neurological abnormality. A chest CT was performed because of the suspition of the aortic dissection, and a Stanford-A type dissection was verified. After the acute aortic arch reconstruction the patient died, but there was no bleeding complication at the dissection site caused by the thrombolysis. This case report draws attention to the fact that aortic dissection can cause acute ischemic stroke. Although it is difficult to prove it retrospectively, we think the aortic dissection, without causing any symptoms or complain, had already been present before the stroke. In our opinion both the history of our patient and literature reviews confirms that in acute stroke the thrombolysis had no complication effect on the aortic dissection but ceased the neurological symptoms. If the dissection had been diagnosed before the thrombolysis, the aortic arch reconstruction would have been the first step of the treatment, without thrombolysis. ]