ABSTRACT:
Hydatid cysts are due to parasitic infection caused by the cestode tapeworm Echinoccocus. In humans the hydatid disease commonly involves the liver (75%) & the lungs (15%). The remaining (10-15%) cases involve the other organs of the body-kidney, spleen, heart, brain, bones, pancreas, breast, ovaries, scrotum, thyroid gland, inguinal canal & soft tissues.The disseminated intra-peritoneal hydatid disease is a very rare finding1. We report such a case wherein the abdominal cavity is seen to contain multiple hydatid cysts .Hydatid cysts were also found in the left scrotum, lungs, liver & spleen.

KEY WORDS:
Hydatid cyst, Echinococcus, Peritoneal hydatidoses, Scrotum

CASE REPORT:
A 42 years Hindu male presented with complaints of left scrotal swelling for 6 months, progressive distention of abdomen for 1 year & generalized weakness for 1 ½ years. He had past history of hydatid disease 13 years back, for which cystectomy was done in 1994. He was not taking any drugs. There is no history suggestive of diabetes mellitus, hypertension, tuberculosis, coronary artery disease or history of Filariasis.

On examination, patient was of thin built with blood pressure 110/70 mmHg (both upper limb), pulse rate 84/min (Regular), Respiratory rate of 24/min and temperature of 37oC. He had diminished vesicular breath sounds in both lower lung fields. His abdomen was distended. On inspection, dilated veins were present over the abdomen with flow below upwards. There was a mid line supraumblical scar mark. Flanks were full and contour was irregular. Umblicus was central and everted (Figure 4). On palpation, there was no local rise of temperature, no tenderness, no fluid thrill and shifting dullness. There were multiple ill defined cystic masses all over the abdomen of varying size. There was bilateral scrotal mass which was soft and non-tender with increased scrotal volume. The liver was not enlarged and spleen was not palpable. On auscultation, bowel sound was normal, no bruit heard. Cardiovascular system and central nervous system examinations were within normal limit.

ROUTINE INVESTIGATIONS:
Hb%-10.4gm%, TLC-12,000/mm3, N50E28L22, ESR-48mm(1st hours), FBS-74mg/dl, Blood urea-31mg/dl, Sr creatinine-1.58mg/dl, Sr Na+-143meq/lt, Sr K+-4.9meq/lt, Urine analysis-normal study,ECG-normal.

LIVER FUNCTION TESTS:
Sr Billirubin (total)-0.89mg/dl, Direct-0.22mg/dl, Indirect-0.67mg/dl, SGOT-26.39IU/lt, SGPT-18.40IU/lt, Sr Alkaline phosphatase-149.38IU/lt, HBsAg-negative, Anti HCV Ab-negative.

ULTRASOUND (ABDOMEN & PELVIS):
Multiple hydatid cysts of liver, spleen, left scrotum & through out abdomen, varing from size 1 mm to 6 mm, B/L moderate Hydrocele (Figure 3).

CHEST X-RAY (PA-VIEW):
Circular opacity of size 8x2cm in right basal zone (? hydatid cyst).

C.T.SCAN (ABDOMEN):
Multiple hydatid cyst of liver, spleen, B/L lower lobes of lungs, involving mesentery & retoperitoneal region. Nonvisualization of RT kidney
(? replaced by hydatid cysts) (Figure 1, 2).

C.T.SCAN OF BRAIN: Normal

IMMUNOLOGICAL TEST:
Echinococcus antibody titer - 1:64 (Normal 1:32)

TREATMENT GIVEN:
Surgical resection of the cysts was not possible. Albendazole-400 mg PO BD for life long (with monthly monitoring of Liver function Tests) given. On first two follow ups there was decrease in the abdominal distension & the liver function test was normal.

DISCUSSION:
Hydatid disease is a parasitic infection caused by the Echinococcus granulosus. The dogs are the definite host and the adult worms are found in their small intestine. Human get infected either by contact with the definitive host or by consuming vegetables and water, contaminated with the hydatid ova2. Hydatid disease is endemic in the cattle grazing areas like India, Pakistan, Middle-East, Africa, South America, and New-zealand1. The close association of people with sheep and dogs and the unavailability of clean potable water supplies in India make it a region endemic to disease. Majority of the cases of Hydatid disease seen come from rural areas or people who have settled in urban centers after spending life in villages. Most of the people acquire the disease during their childhood, but do not present with the clinical signs and symptoms until late adult hood. The natural progression of an untreated cyst may include calcification and death of the cyst; however, more frequently the cyst gradually enlarges3.

The liver (75%) and the Lungs (15%) is the commonest site of involvement, although no site in the body may be completely immune from it4. This atypical and rare presentation of the disease may be seen in kidneys (3%), usually the upper and the lower pole of the kidney may be involved. The spleen may be involved in about 4% of the cases and is associated with splenomegaly, fever and abdominal pain4. Cerebral Hydatid cysts occur in only 2% of all the cases reported. The region of the middle cerebral artery distribution specially the parietal lobe is most frequently involved. Cardiac Hydatid cyst is very rare (0.02-2%) and most commonly affects the left ventricular chamber specially the left ventricle in 50%-60% of cases4. The other sites that have been reported to be involved are bones, pancreas, breast, ovaries, scrotum, thyroid glands, inguinal canal and soft tissues.

Hydatid cysts can also be found rarely in the peritoneum1. Most of these cases are the result of traumatic or surgical rupture of a hepatic, splenic or mesenteric cyst. The prevalence of peritoneal hydatid cysts in cases of abdominal Hydatid disease is approximately 13%. In our case the patient had already undergone cystectomy and it is likely that these findings may be associated to previous surgical rupture, although spontaneous rupture of micro cysts into the peritoneum may also occur in about 12% of the cases1. The hydatid cysts may resemble a multiloculated mass filling the entire peritoneal cavity. Such a condition is referred to as peritoneal hydatidoses .

Hydatid cyst may be solitary or multiple. The type of the imaging modality used depends on the site and the size of the hydatid cyst. Ultrasonogarphy (USG) is the first line of screening for abdominal hydatidosis and it is especially useful for detection of cystic membrane, septa, and hydatid sand. CT scan best demonstrates cyst wall calcification and cyst infection. CT scan imaging is also the modality of choice in peritoneal seedling1. The CT scan shows well defined solitary or multiple cysts that may be thin walled or thick walled. A hydatid cyst typically demonstrates a high attenuation value at unenhanced CT even without calcification. Multivesicular cysts can depict a typical honeycomb pattern. The septa represent the walls of the daughter cysts housed within the mother cyst. A “wheel spoke” pattern can be observed when the daughter cysts are separated by hydatid matrix1.

There are different types of serological tests which can be carried out for the diagnosis, screening and follow up of patients with hydatid disease. These include the immunoelectrophoresis, enzyme-linked immunosorbent assay (ELISA), latex agglutination and indirect haemagglutination (IHA) test 4. The diagnosis of HD can thus be established with the help of radiologic and serologic finding4. The diagnosis is also easier when the lesion has multiple locations involving different organs or when daughter cysts, germinal membrane detachment and calcification are present 2.

Surgery is the mainstay of treatment for hydatid cysts of the Liver. Laparotomy is the most common surgical approach6. Liver resection and pericystectomy are procedures that resect the closed cysts with a wide safety margin; however they are considered too radical procedures for hydatid cyst removal. Conservative procedures such as cystectomy and omentoplasty for hydatid disease should be the standard surgical procedure because of their safety, simplicity, and effectiveness in fulfilling the surgical treatment criteria of hydatid disease6. The peritoneal hydatidosis has also been successfully surgically removed with similar conservative procedures5, 7.

CONCLUSION:
Symptomatic or large cysts should be surgically treated. In cases suspected of having peritoneal spillage, antihelminthic drugs should be administered8. In addition, small asymptomatic cysts, some daughter cysts, and peritoneal secondary cysts and splenic cysts may also be effectively treated with Albendazole8.

REFERENCES:

1. Pedrosa I, Saiz A, Arrazola J, Ferreiros J, Pedrosa CS. Hydatid disease: radiologic and pathologic features and complications. Radiographics 2000; 3:795-817.
2. Polat P, Kantarci M, Alper F, Suma S, Koruyucu MB, Okur A.: Hydatid Disease from Head to Toe. Radiographics 2003; 23:475-94.
3. Ammann RW, Eckert J. Cestodes. Echinococcus. Gastroenterol Clin North Am 1996; 3:655-89.
4. Husen YA, Nadeem N, Aslam F, Bhaila I. Primary splenic hydatid cyst: a case report with characteristic imaging appearance. J Pak Med Assoc 2005; 5:219-21
5. Karavias DD, Vagianos CE, Kakkos SK, Panagopoulos CM, Androulakis JA. : Peritoneal echinococcosis. World J Surg 1996; 20:337-40.
6. Buttenschoen K, Carli Buttenschoen D. Echinococcus granulosus infection: the challenge of surgical treatment. Langenbecks Arch Surg 2003; 4:218-30.
7. Hamamci EO, Besim H, Korkmaz A. Unusual locations of hydatid disease and surgical approach. ANZ J Surg 2004; 5:356-60
Balik AA, Celebi F, Basglu M, Oren D, Yildirgan I, Atamanalp SS. Intra-abdominal extrahepatic

ABSTRACT:
Organophosphorus induced delayed neuropathy (OPIDN) is a delayed sequel of organophosphorus poisoning, occurs rarely1,2,6 after an acute cholinergic crisis and is confused with Guillain Barre’ Syndrome (GBS) or other causes of polyneuropathy. We report one case of OPIDN which we managed conservatively with steroids & we found improvement of CMAP (Compound Muscle Action Potential) within one month.

KEYWORDS:
Organophosphorus (OP), OPIDN, Axonal degeneration, neuropathic target esterase (NTE), Pralidoxim (PAM).

INTRODUCTION:
OPIDN refers to delayed effects of organophosphorus poisoning (OP poisoning), occurs due to binding of esterase enzyme called neuropathic target esterase (NTE)2,3 and cause predominant axonal degeneration (dying back axonopathy5) and minor demylenation7. Typically patient presents 3-5 weeks after OP poisoning with lower extremity weakness, bilateral foot drop, glove and stocking type of sensory loss5. Present case study documents one case of OPIDN due to tri Ortho cresyl phosphate poisoning after one month of cholinergic crisis and prevention of OPIDN is by early treatment of cholinergic crisis with PAM (before organophosphorus compound ages), steroids and other supportive measures (e.g splintage, physiotherapy and carbamazepine for neuropathic pain).

CASE REPORT:
A 19 year male presented to the hospital with complains of tingling sensation and weakness of lower limbs and hands 15 days back. He was apparently alright 15 days back, had 4 episodes of loose motion which subsided with medication. Then he had tingling sensation over lower limbs and hands and weakness of lower limbs followed by weakness and clumsiness of both hands. Weakness was progressive and in one week the patient was not able to walk. No history of fever, dog bite, vaccination, previous similar episode, contact, diabetes, leprosy was elicited.

Allegedly he took insecticides and suffered from acute cholincrgic crisis one and half month ago for which he was hospitalized for 11 days and recovered. On general examination the patient was conscious, afebrile with pulse rate 80/min, BP 126/80mm Hg without any other positive findings. CNS examination revealed intact cranial nerves, wasting of hypothenar, thenar (Fig. 1), calf and peroneal muscles of limbs (Fig. 2). Hypotonia involving flexor and extensor of ankle, weakness of abductor pollicis berevis, adductor pollicis and oppones pollicis seen. Long flexors and extensors of digits were normal. Lower limbs evertor, invertor, flexor and extensor of ankle were 0/5 power on both sides. Extensor and flexor of knee and hip were normal. Supinator jerk was diminished, ankle jerk was absent on both sides. Rest deep tendon jerks were normal. Plantar was nonresponsive both sides. There was in-coordinated movement, Rhomberg's test was positive and gait was high stepping. Sensory examination revealed all modalities diminished below ankle and wrist bilaterally. Peripheral nerves were normal.

Laboratory investigation revealed:
o Hb 12.7 gm%, TLC 10,700/mm3, DC N-69 L-30 E-l, ESR 8mm/lst hr, FBS 85 mg% .
o Serum Na'-132meq/lt, Serum K-4.8meq/lt, Serum urea -28mg%, Serum creatinine -1.18 mg%.
o HIV ELISA - Negative, CSF study showed TLC 3/mm3 (all lymphocytes) RBC absent, Glucose 61mg%, Protein 27 mg%, TB ELlSA- Negative.

NERVE CONDUCTION STUDY:
Initial Nerve Conduction Study:
Motor - Compound muscle action potential (CMAP) was absent in bilateral Median, Common peroneal nerve (CPN) , bilateral posterior tibial nerve (PTN).
CMAP in bilateral ULNAR - Amplitude less than normal, velocity in right ulnar less than normal , Left ulnar velocity within the normal limits.
Sensory - In bilateral Median Velocity was reduced, Right ulnar velocity was reduced no potential in Right sural, left Sural velocity and amplitude within normal limit.

Repeat Nerve conduction Study: (After one month)
CMAP- left median no potential, Right median small amplitude potential
Velocity in the above is reduced
Conduction in bilateral ulnar as before
No CMAP in bilateral CPN and bilateral PTN
Sensory as before

DISCUSSION:
Chronic complication of OP poisoning are neurobehavioral and poly-neuropathy1. Sensory motor polyneuropathy or OPIDN typically ocours after 3 to 5 weeks of exposure where motor deficit dominates the clinical picture5. Patient initially complains of tingling sensation over extremities, symmetric lower extremity weakness, and leg cramping and calf pain. Atrophy of calf muscles, ataxia and bilateral foot drop gradually ensues. It may progress to cause truncal weakness. Cranial nerves and autonomic system are not involved3. Sensory symptoms resolve over months. In mild cases motor symptoms may resolve over 15 months or may not resolve at all3. Severe cases may develop spasticity due to spinal cord involvement in the form of diffused spinal cord atrophy predominantly of thoracic spine2,5. Neuropathology of OPIDN demonstrates that large distal neurons tend to be affected first. There is slowing and stoppage of axonal flow, blockage transport of protein and other substances as well as demyelination7. Axonal degeneration precedes demyelination and there is "dying back" axonopathy5. Site of OP attack is a membrane bound target esterase called neuropathic target esterase (NTE)2,3. Function of NTE is not known. It is present in brain, spinal cord and peripheral nerve6, lymphocytes4 where specific OP binds and ages1 by losing 1 carbon and forming P-O carbon linkage. Critical mass of 70% of NTE must be inhibited to cause clinical symptom6. The delay in symptoms is due to the fact that not all axons will degenerate at the same time and nerve may continue to function until significant number of axons is degenerated.

OPIDN diagnosis is made by clinical and neurophysiologic examination and ruling out other possibilities. Currently platelet, RBC and lymphocyte NTE inhibition for confirming neuropathic potential of organophosphorus is under study . Use of hen test as screening test of neurotoxicity is flawed1.

Treatment of OPIDN is conservative and can be prevented by early and prolonged use of PAM. Tingling sensation over palm and sole responds to carbamazepine5.

REFRENCE:
1. Angelo Moretto et al: Poisoning by organophosphorus insecticides and sensory neuropathy; J. Neurol. Neurosurg. Psychiatry: 1998;64;463-468.
2. Cnia-Chang Chuang et al: Delayed Neuropathy and Myelopathy after Organophosphate Intoxication: NEJM, Volume 347:1119-1121 October 3, 2002 Number 14.
3. J. Mirandal et al : Muscular Strength and vibration thresholds during two years after acute poisoning with organophosphate insecticides. Occupational and Environmental Medicine BMJ 2004; 61: e4.
4. M. Lotti et al: Inhibition of lymphocyte neuropathy target esterase predicts the development organophosphate induced delayed polyneuropathy:J. Arch of Toxicology, Vol 59/3 Oct 1986, Pg 176-179.
5. R. Hierons and M.K. Johnson : Clinical and Toxicological investigations of a case of delayed neuropathy in man after acute poisoning by an organophosphorus pesticide: J Arch of Toxicology, Issue Volume 40, Number 4/Deceber , 1978.
6. Singh H, Sharma N: Neurological Syndromes Following organo-phosphate poisoning, Year 2000 , Neurology India, Volume 48, issue 4, Page 308-13.
7. Versik P et al: Chronic Toxic Neurophaty in Qualis: British Lek Letsy 106 (10), 293 – 296, 2005.

ABSTRACT:
Delayed cerebellar ataxia is a rare acute self limiting complication of falciparum malaria [2]. This complication is predominantly found in Sri Lanka though few cases have been reported from India and Africa [2,4]. We have recently encountered such a patient in our hospital. The case is described below with pertinent literatures reviewed.

KEY WORDS:
Delayed cerebellar ataxia, Plasmodium falciparum

INTRODUCTION:
Delayed cerebellar ataxia found in falciparum malaria is usually an acute self limiting and isolated ataxia without cerebral involvement [4]. The patient usually shows features of midline cerebellar involvement and is characterized by cerebellar gait and truncal ataxia [1,4]. Most of the patients are afebrile before the onset of symptoms [1,4].

CASE REPORT:
A 45 year old engineer was admitted to the hospital in September '07 with complaints of difficulty in maintaining balance with a tendency to fall in all directions for the past four days. He developed high grade fever, intermittent in nature associated with chills and rigor and subsided with profuse sweating. He was admitted in the local hospital and was detected having falciparum malaria by MP slide and was treated with anti-malarials and antibiotics. The patient became afebrile after five days and was discharged. Four days later He developed instability of gait such that he lurched and fell in all directions – this left him unable to walk. He was then referred to our hospital for further investigations and management.
There was no history of diabetes mellitus, hypertension, tuberculosis or sickle cell disease.

Family history is also not suggestive of diabetes mellitus, hypertension, tuberculosis or sickle cell disease.

On admission the patient was found to be conscious afebrile. There was mild pallor, no cyanosis, icterus, clubbing, edema, Lymph node or thyroid enlargement, JVP was not raised, Pulse was 80/ min regular, B.P. 150/ 84 mm of Hg, Respiratory rate of 18/ min and temperature was 36.4 ° C.

Neurological examination revealed normal higher mental function. All cranial nerves were found normal. Motor system examination revealed normal bulk, tone and power. The gait was broad based with irregular stepping and a tendency to fall in all direction. No sensory impairment or autonomic involvement was found. On finger nose testing revealed past pointing; the knee heel shin test was found to be clumsy bilaterally. Disdiadochokinesia was present bilaterally. The deep tendon reflexes were normal, plantar was bilaterally flexor. The cranium and spine was normal and there were no signs of meningeal irritation.

The respiratory, cardiovascular, and gastrointestinal system was found to be normal.

INVESTIGATION:
On investigation Hb% was 11.2 gm/dl, DC N72 E3 L25; Total leukocyte count was 7100/cmm and ESR was 6 mm in 1st hr. Routine blood biochemistry  Fasting blood glucose 110 mg/dl, urea 28 mg/dl, creatinine 1.4 mg/dl, serum Na+ 147mEq/l and K+ 4.0 mEq/l. Malaria parasite by ICT was found to be positive. CT scan of brain was found to be normal and no abnormality was detected by pure tone audiometry bilaterally. CSF study was found to be normal.
The patient was diagnosed as having delayed cerebellar ataxia following falciparum malaria. He was treated with a full course of inj. Artesunate for 5 days. The patient rapidly improved and by the 5th day he was able to walk on his own. The cerebellar signs disappeared on the 6th day and he was discharged with advice after 7 days of admission.

DISCUSSION:
Neurologic features found in malaria are usually not consistent; the commonest being altered sensorium followed by seizures [1]. But cerebellar involvement is the most consistent neurological manifestation of complicated as well as uncomplicated malaria as Purkinje cells are susceptible to damage due to hyperpyrexia [4].

RBCs with parasitized P. falciparum adhere to the endothelial wall of capillaries in a certain phase of its cycle. Parasites derive some nutrition from endothelium. This phenomenon occurs maximally in the capillaries of brain [1]. Focal hemorrhage or non hemorrhagic infarcts in cortex, basal ganglia, thalamus, pons and cerebellum have all been described in cerebral malaria [1]. Immunologic mechanism, as a cause for delayed cerebellar ataxia has been postulated; cerebellar demyelination following falciparum malaria has been reported in several patients from Sri Lanka and India occurring several days after recovery [1,4]. The CT scan of brain is normal [1,2]. The disease has an excellent prognosis with complete recovery in 3 months [2,4]. The patients are treated symptomatically though some physicians prefer to use steroids.

CONCLUSION:
Cerebellar involvement in falciparum malaria may be due to selective clogging of cerebellar micro vasculatures with parasitized RBCs, perivascular hemorrhage, microscopic infarcts, shrinkage of purkinje cells and perivascular clusters of microglia, carrying a high mortality rate [1,3] or, may be due to immunologic mechanism as in the case of delayed cerebellar ataxia [1,4].

Delayed cerebellar ataxia is a rare complication of cerebellar involvement following falciparum malaria and may be considered in the differential diagnosis in patients showing cerebellar signs especially if the patient gives a past history of fever. This is the first reported case of delayed cerebellar ataxia from our institution.

REFERENCES:
1. Adult cerebral malaria: prognostic importance of Imaging findings and correlation with postmortem findings; Tufail F. Patankar, MD, Dilip R. Karnad, MD, Prashant G. Shetty, MD, Anand P. Desai, MD, Srinivasa R. Prasad, MD;Radiology 2002;224:811-816
2. Delayed cerebellar ataxia following Falciparum malaria: Hussam Alsoub, MD : Annals of Saudi Medicine,1999;Vol 19, no. 2: 128-129
3. MR of cerebral malaria; Yves Sébastien Cordoliani, Jean-Luc Sarrazin, Dominique Felten, Eric Caumes, Cristophe Lévêque, Alain Fisch; AJNR Am Neuroradiol 19:871-874, May 1998