Cerebral Chromoblastomycosis due to Cladosporium Trichoides (Bantianum) - Part I (A Review and Case Report)
Correspondence Address: Source of Support: None, Conflict of Interest: None DOI: 10.4103/0028-3886.279712
Source of Support: None, Conflict of Interest: None
Ten cases in the last thirteen years probably represent the sujm total of world experience in cerebral fungal granulomas caused by Cladosporium trichoides. It was from a cerebral abscess in Binford's patient, a 22-year-old coloured male from Maryland, that Emmons, in 1952, cultured, identified and named the fungus for the first time and produced fatal cerebral lesions in mice and rabbits after its intravenous inoculation. In the same year Emmons also helped in identifying the organism from material sent to him by King and Collette who described the second case in a 47-year-old white male from Pennsylvania. The next two cases were reported in 1955, one from France by Segretain et al. and one from Belgium by Dereymaeker and De Somer (quoted by Symmers). In 1956, McGill and Brueck recorded this disease in a 37-year-old coloured female from Louisiana and the next case to be described was by Jaffe of Venezuela in 1958 (quoted by Duque). In 1960, Riley and Mann reported what was probably the seventh case in a 24-year-old coloured male from Louisiana. Duque in 1961, Doby-Dubois et al. in 1962 and Tommasi et al. in 1964 have recorded one each making a total of ten cases where the fungus was successfully grown; and ours is probably the eleventh.
There are at least a further 8 cases where the fungus was morphologically identified within human brain tissue, but was not grown in culture. Duque has stressed that C. trichoides andFonsecaea pedrosoi are indistinguishable when examined in their tissue phases; he mentions the case described by Lucasse et al. in 1954, where a fungus thought to be C. trichoides was isolated from the brain of a 10-year-old boy, a native of the Belgian Congo, and was later studied by Borelli who classified it as F. pedrosoi. Also included in this group are reports by Watson (1957) and Manson (1958) of South Africa (quoted by Duque); by Bobra (1958) of Canada; by Symmers (1960) of England; by Duque (1961) of Colombia; by Bagchi et al. (1962) of India; and Shimazono et al. (1963) of Japan. The particulars of some of these cases are given below.
Bobra's patient was a 57-year-old Negro suffering from malignant hypertension (blood pressure 230/150), papilledema, loss of vision, and right hemiplegia, who had an unsuspected left parieto-occipital abscess, the size of a golf ball, discovered at necropsy. Symmers' patient was a 22-year-old woman who had spent all her life on a moorland farm in England; while on a course of cortisone for the treatment of polyarteritis, she suddenly collapsed and developed a left hemiplegia. At necropsy there was a thrombosed aneurysm of the right middle cerebral artery with a zone of softening, assumed to be an infarct of the brain, sections of which showed a fungus resembling C. trichoides. Duque's patient was a 29-year-old Negro farmer who died of a granulomatous meningitis and ependymitis, and microscopic examination of the lesions and exudates revealed the fungus. Bagchi et al. in 1962 described the first Indian case in a 54-year-old male from Calcutta who had a right parietal granuloma which was examined histopathologically by Dr. F. Mariat of Institut Pasteur, Paris and revealed a fungus, morphologically resembling a Cladosporium.
If material from all granulomatous lesions of the central nervous system, with or without suppuration, is routinely inoculated upon mycological culture media it is likely that more such cases would be detected. It is unlikely though that they would all be benefited by surgery as is shown by the high mortality among the cases described including our patient who showed great promise of recovery after his first operation, but later succumbed from the after-effects of ventricular loculation due to continued activity of the disease process Successful treatment may depend upon a better understanding of the processes causing a breakdown of the immunity mechanisms in the host and the provision of more effective systemic anti-fungal remedies.
The patient, H. M., was a 24-year-old Hindu male from Panvel, a village about 55 miles from Bombay. He belonged to the community of 'Agrees' which, according to the classification of the Community Block Development Scheme, is only one step above the 'Adivasis' who are primitive tribal folk at the bottom of the social scale. The group next above the 'Agrees' are the 'Kolis' who are fisher folk for whom the 'Agrees' provide cheap labour. The 'Agrees' inhabit fishing villages 50 to 60 miles north of Bombay and also provide cheap labour for the local farmers. Our patient was economically better off than many of his community in that he owned his house and farm and was a paddy cultivator. He was twice married, his first wife having died in childbirth, and had two children by his second wife. His parents, two sisters and one brother were alive and there was no history of a similar illness in his family.
He was admitted on January 30, 1964 for severe generalised headache with neck rigidity and right hemiparesis of 48 hours' duration. We were informed that 10 days previously he had complained of sudden paraesthesiae and weakness in his right arm followed by loss of consciousness for half an hour. On his way to the hospital he vomited once and soon after admission had a right focal convulsion. General examination revealed a generalised rash, which itched, composed of scaly erythematous patches of hypopigmentation and hyperpigmentation. He was uncertain about its duration but thought that it had been there for many years. There was a warm tender swelling of his left calf indicating a deep seated abscess. Neurological examination revealed mild dysphasia, papilloedema, spastic right hemiparesis, hemihypaesthesia and impairment of position sense in the right hand and foot. Ears, nose and throat were normal. Temperature and blood pressure were normal but the pulse rate was 60 per minute. The clinical diagnosis was either subarachnoid haemorrhage from cerebral angioma or meningitis with cerebral abscess.
Investigations. Urine normal; blood sugar 108 mg. %, blood urea 12 mg. %; blood V.D.R.L. negative; haemoglobin 11.5 grammes; white cell count 10,900. Radiograph skull normal; radiograph chest showed multipie, bilateral, calcified, apical foci. EEG showed a left parietal slow wave focus. Cerebrospinal fluid was clear, colourless and contained 5 lymphocytes, proteins 20 mg.%, sugar 68 mg.%, chlorides 760 mg.%.
Having thus excluded subarachnoid haemorrhage or meningitis, a left carotid angiogram was performed and this showed a left parietal tumour without pathological vessels. Ventriculogram demonstrated downward displacement of the body and trigone of the left lateral ventricle, confirming the presence of a high left parietal tumour [Figure 1].
First operation. On February 6, 1964 a left fronto-parietal craniotomy was performed and a tense dura exposed in spite of intravenous mannitol administration. The brain was swollen and the exposed motor strip was greatly expanded. Within it were two subcortical nodules, each 2 to 3 cm. in diameter, which were enucleated. They consisted of a well-defined capsule 3 to 6 mm. in thickness with a central liquefied area containing yellowish seropurulent fluid with white and olive flakes. Immediate frozen section and bacteriological examination of the tissue revealed large colonies of pigmented mycelia leaving no doubt as to the presence of cerebral fungal invasion. During excision of a third but deep-seated nodule, the roof of the body of the lateral ventricle was opened and it was feared that ventricular contamination might occur.
Post-operative Course. The patient regained consciousness on the night of operation; he was alert but dysphasic; slight neck rigidity and dense upper motor neurone right facial and right arm paralysis were present. On the following day a plan for conducting extensive mycological investigations was outlined. It was decided to inoculate all subsequent samples of ventricular and spinal fluid, as well as collections under the craniotomy flap, on suitable culture media. Biopsies and scrapings were obtained from the skin lesions and calf abscess. Blood and twenty-four hour urine collection were taken for culture. (For reports see Part II.)
Within a few days his headache was relieved and papilloedema regressed. Dysphasia and right hemiparesis improved enough to enable him to hold brief conversations and walk unassisted. In view of the bad prognosis of systemic mycosis, it was decided to keep the patient under observation as long as possible for long-term penicillin treatment, which was found useful in the case reported by Binford et al. This was discontinued only at the end of 4 months.
Whether this was responsible or not, a week later, the patient suddenly became drowsy, incontinent and difficult to rouse. Papilledema was absent, but he had marked bradycardia and a rapidly developing left hemiparesis. EEG was repeated and showed, on the previously normal right side, slow waves with phase reversal in the temporo-parietal region. On probing the hemisphere for an abscess through the right parietal burr hole a greatly distended lateral ventricle was entered and ventricular fluid under high pressure escaped. It was slightly opalescent and contained 216 cells, mainly lymphocytes with a few polymorphs, 300 mg. per cent protein and normal sugar. The fluid was replaced by air and a ventriculogram obtained. It showed marked distension of the body, trigone, temporal and occipital horns of the right lateral ventricle but air could not be manipulated into the third ventricle or the frontal horn [Figure 2] and [Figure 3]. Air was next introduced into the left lateral ventricle and subsequent views showed filling of the entire left lateral ventricle, third ventricle, and, surprisingly, also the right frontal horn; the septum and the third ventricle were acutely angulated towards the left side [Figure 4] and [Figure 5]. In the brow-down P-A view an enormously distended right lateral ventricle was seen and beyond it a faint outline of a normal-sized right frontal horn indicating the presence of trapped air within its cavity. These films conclusively demonstrated the presence of loculation of the right lateral ventricle just behind the foramen of Monro and a freely communicating right frontal horn with the rest of the ventricular system.
Second operation. On June 20, 1964 right frontal craniotomy was performed and the loculated ventricle aspirated. The fluid was xanthochromic and contained 36 cells, mainly lymphocytes, diminished sugar and more than 300 mg. per cent protein. It was immediately inoculated on culture media and colonies of the fungus were later grown. The ventricle was opened and the choroid plexus seen ending blindly alongside the body of the caudate nucleus. The foramen of Monro and the head of the caudate nucleus were completely cut off by a septum formed by the coaptation of the medial and lateral ependymal walls. Through the septum a tunnel was created around the choroid plexus by sucking away the vascular granulation tissue surrounding it and the roof of the frontal horn entered. Owing to its inflammatory swelling the choroid plexus was almost compietely blocking the foramen. The opening was widened by cutting the anterior column of the fornix. For better communication an opening was also made through the septum pellucidum into the left lateral ventricle within which there were a few tags of granulation tissue which were biopsied. The loculaied ventricle was now in free communication with the ventricular system.
Post-operative Course. For three months after the operation the patient remained stuporose and quadruplegic. He was tube fed and a tracheostomy was performed. His muscles began to waste and he developed bed-sores; in this condition he was removed to his village on September 29, 1964 and we were informed that he died there a fortnight later.
We thank the Dean, K.E.M. Hospital, Bombay for permission to publish, our Neurologist, Dr. A. D. Desai, for helpful EEG reports and our Secretary, Mr. E. G. Warier for his valuable assistance.
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Conflicts of interest
There are no conflicts of interest.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]