Unusual Association of Epidemic Dropsy with Brachial Neuritis and Palatal Palsy
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CLINICAL BRIEF Unusual Association of Epidemic Dropsy with Brachial Neuritis and Palatal Palsy Sushama Sahoo & Arindam Bandyopadhyay & Nepal Chandra Mahapatra Received: 17 July 2011 / Accepted: 21 December 2011 / Published online: 10 January 2012 # Dr. K C Chaudhuri Foundation 2012 Abstract Epidemic dropsy (ED) results from accidental ingestion of adulterated mustard oil with argemone oil. Chief organs involved in this disease are heart, subcutane- ous tissue, eyes and kidneys. Nervous system involvement is very rare. Objective manifestation of neurological in- volvement is even rarer. The authors report two cases from the same family, who were victims of epidemic dropsy along with their parents. One of them showed objective neurologic involvement in the form of brachial neuritis and another showed palatal palsy. Keywords Epidemic dropsy . Argemone oil . Sanguinarine . Brachial neuritis . Palatal palsy Introduction Epidemic dropsy was first reported in 1877 from Calcutta by Lyon [1] and has since been reported to occur in other countries including Mauritius, Madagascar, Fiji, South Africa and Burma [2]. It is caused by contamination of cooking oil by toxic argemone oil which is completely miscible [3]. Organs involved in this disease are heart, subcutaneous tissue, eyes and kidneys. Some reports had documented few neurologic manifestations, but objective evidence of neurologic involvement is very rare. The pres- ent communication reports brachial neuritis in epidemic dropsy, an observation reported previously in one literature only [4]; and palatal palsy in epidemic dropsy, an observa- tion supposed not to have been previously reported. Case Reports Case 1 A 15-y-old boy presented with swelling of lower limbs for one mo and dyspnoea on exertion for 2 wk. One month back he suffered from low grade fever and loose motions for 3–4 d. Two days after admission, the boy started complaining of pain in both shoulders which was followed by difficulty in raising both arms. Gradually, he developed wasting of deltoids, supraspinatus and infraspinatus muscles on both sides. Neurological examination revealed a muscle power of 2/5 in supraspinatus bilaterally, 2/5 in left infra- spinatus, 3/5 in right infraspinatus and 1/5 in deltoids bilat- erally. Reflexes were sluggish in both upper limbs. There was a sensory loss on the skin over both deltoids. Case 2 A 10-y-old girl, sister of the above-mentioned boy, was also admitted at the same time with complaints of loose motions for 2–3 d initially, followed by lower limb swelling for one mo and nasal intonation of voice along with nasal regurgitation of liquids for last 5 d. Nervous system exam- ination revealed nothing except right sided palatal palsy. Their parents had got similar problem of same duration. They all consumed homemade mustard oil for 3 mo. Gen- eral survey revealed puffy faces, bilateral pitting edema with mild tenderness and erythematous overlying skin. Pulse S. Sahoo Department of Pediatric Medicine, Dr. B C Roy Postgraduate Institute of Pediatric Sciences, Kolkata, India A. Bandyopadhyay : N. C. Mahapatra Department of Pediatric Medicine, Calcutta National Medical College & Hospital, Kolkata, India S. Sahoo (*) 8/4, Santoshpur East Road, Kolkata 700 075, West Bengal, India e-mail: [email protected] Indian J Pediatr (May 2013) 80(5):428–429 DOI 10.1007/s12098-011-0676-6
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Transcript of Unusual Association of Epidemic Dropsy with Brachial Neuritis and Palatal Palsy
CLINICAL BRIEF
Unusual Association of Epidemic Dropsy with BrachialNeuritis and Palatal Palsy
Sushama Sahoo & Arindam Bandyopadhyay &
Nepal Chandra Mahapatra
Received: 17 July 2011 /Accepted: 21 December 2011 /Published online: 10 January 2012# Dr. K C Chaudhuri Foundation 2012
Abstract Epidemic dropsy (ED) results from accidentalingestion of adulterated mustard oil with argemone oil.Chief organs involved in this disease are heart, subcutane-ous tissue, eyes and kidneys. Nervous system involvementis very rare. Objective manifestation of neurological in-volvement is even rarer. The authors report two cases fromthe same family, who were victims of epidemic dropsyalong with their parents. One of them showed objectiveneurologic involvement in the form of brachial neuritisand another showed palatal palsy.
Keywords Epidemic dropsy . Argemone oil . Sanguinarine .
Brachial neuritis . Palatal palsy
Introduction
Epidemic dropsy was first reported in 1877 from Calcuttaby Lyon [1] and has since been reported to occur in othercountries including Mauritius, Madagascar, Fiji, SouthAfrica and Burma [2]. It is caused by contamination ofcooking oil by toxic argemone oil which is completely
miscible [3]. Organs involved in this disease are heart,subcutaneous tissue, eyes and kidneys. Some reports haddocumented few neurologic manifestations, but objectiveevidence of neurologic involvement is very rare. The pres-ent communication reports brachial neuritis in epidemicdropsy, an observation reported previously in one literatureonly [4]; and palatal palsy in epidemic dropsy, an observa-tion supposed not to have been previously reported.
Case Reports
Case 1 A 15-y-old boy presented with swelling of lowerlimbs for one mo and dyspnoea on exertion for 2 wk. Onemonth back he suffered from low grade fever and loosemotions for 3–4 d. Two days after admission, the boy startedcomplaining of pain in both shoulders which was followedby difficulty in raising both arms. Gradually, he developedwasting of deltoids, supraspinatus and infraspinatus muscleson both sides. Neurological examination revealed a musclepower of 2/5 in supraspinatus bilaterally, 2/5 in left infra-spinatus, 3/5 in right infraspinatus and 1/5 in deltoids bilat-erally. Reflexes were sluggish in both upper limbs. Therewas a sensory loss on the skin over both deltoids.
Case 2 A 10-y-old girl, sister of the above-mentioned boy,was also admitted at the same time with complaints of loosemotions for 2–3 d initially, followed by lower limb swellingfor one mo and nasal intonation of voice along with nasalregurgitation of liquids for last 5 d. Nervous system exam-ination revealed nothing except right sided palatal palsy.
Their parents had got similar problem of same duration.They all consumed homemade mustard oil for 3 mo. Gen-eral survey revealed puffy faces, bilateral pitting edema withmild tenderness and erythematous overlying skin. Pulse
S. SahooDepartment of Pediatric Medicine,Dr. B C Roy Postgraduate Institute of Pediatric Sciences,Kolkata, India
A. Bandyopadhyay :N. C. MahapatraDepartment of Pediatric Medicine,Calcutta National Medical College & Hospital,Kolkata, India
S. Sahoo (*)8/4, Santoshpur East Road,Kolkata 700 075, West Bengal, Indiae-mail: [email protected]
Indian J Pediatr (May 2013) 80(5):428–429DOI 10.1007/s12098-011-0676-6
rates were increased. Pulse pressures were widened in allcases. All of them had marked pallor and tachypnea. Exam-ination of CVS revealed hyperdynamic apex with cardio-megaly and ejection systolic murmur of grade II.
Investigations showed moderate anemia with reducedPCV, normal WBC and platelet counts. Serum creatinineand urea, lipid profiles and LFT were normal except lowalbumin. Echocardiography showed dilated RA and RV,dilated SVC with inspiratory collapse suggestive of system-ic venous congestion and increased PA pressure. Investiga-tion of parents revealed similar type of picture. MRI ofbrachial plexus in the boy and MRI of brain in the girl werenormal. In boy nerve conduction studies showed abnormalaxillary nerve conduction with normal distal latency andmarked reduction in compound muscle action potential(CMAP) amplitude on stimulation at Erb’s point. EMG ofboth deltoids showed a neurogenic pattern.
Strikingly all three members of the family had congestivecardiac failure (CCF). They all had consumed their ownhomemade mustard oil for last three mo. Urine samplesand mustard oil were sent for HPLC and sanguinarine, thetoxin responsible for ED was detected.Diagnosis of epidem-ic dropsy was confirmed. Following conservative treatmentwith ACE inhibitor, diuretic, vitamins A and E along withbed rest and other supportive measures both the childrenimproved symptomatically within six wk. Complete recoveryof palatal palsy in girl and brachial neuritis in boy took 3 moand 6 mo respectively.
Discussion
Epidemic dropsy results from consumption of mustard oiladulterated with argemone oil [5].Argemone seeds have gottwo main alkaloids namely sanguinarine and dihydrosan-guinarine and several other minor alkaloids[6]. Epidemicdropsy has an insidious onset. Clinical features are usuallyswelling of lower limbs, intermittent or continuous fever,anorexia, abdominal colic, vomiting and diarrhea. Bilateralsymmetrical pitting edema of lower limbs is almost aconstant finding. Erythema or rash over the edematousparts, CCF, hepatomegaly and anemia are other presentingfeatures [7]. Renal involvement and ocular manifestationsmay also occur [8].
Pathophysiology of ED is due to increased capillary per-meability caused by sanguinarine [7]. Sanguinarine enhancesglycogenolysis and increases pyruvate levels resulting inuncoupling of oxidative phosphorylation, inhibition of
Na+K+ATPase of heart leading to heart failure and increasedproduction of reactive oxygen species [4, 9]. Withdrawal ofcontaminated cooking oil is the most vital initial step intreatment. Bed rest, protein-rich diet along with supplementsof calcium, antioxidants and diuretics are commonly used[7–9].
Although controversy exists regarding neurological in-volvement in ED, paraesthesias and alteration in tendonjerks have been reported [4, 10]. In this report, case 1suggests a picture similar to brachial neuritis and case 2suggests palatal palsy in association with ED. The authorsassume that these can not be coincidental findings. Theexact mechanism of these associations however needs tobe elucidated. Early recognition of the problem and a mul-tidisciplinary approach may help in improving the prognosisof these patients.
Contributions SS,AB: Patient management, data collection andmanuscript preparation; NCM: supervised the patient managementand reviewed the manuscript.
Conflict of Interest None.
Role of Funding Source None.
References
1. Lyon IB. Textbook of medical jurisprudence for India. 1st ed.Calcutta, India: Thacker, Spink & Company; 1889. pp. 214.
2. Aronson JK. Plant poisons. In: Cook GC, Zumla AI, editors.Manson’s Tropical Diseases. 22nd ed. Philadelphia, PA: SaundersElsevier; 2009. pp. 605–7.
3. Sarkar SN, Rahman MB. A chemical method for the estimationof alkaloid present in argemone and mustard oils. Curr Sci.1945;14:196–7.
4. Prabhakar S, Khurana D, Gill KD, et al. Neurologic complicationsof dropsy: from possibility to reality. Neurol India. 2000;48:144.
5. Das M, Khanna SK. Epidemic dropsy. Natl Med J India.1998;11:207–8.
6. Mukherjee SP, Lal RB, Mathur KBL. Investigation into the epide-miology of epidemic dropsy—XII. Isolation of active substancesfrom toxic oils. Indian J Med Res. 1941;29:361–5.
7. Sharma BD, Malhotra S, Bhatia V, et al. Epidemic dropsy in India.Postgrad Med J. 1999;75:657–61.
8. Mohan M, Sood NN, Dayal N, et al. Ocular and clinico-epidemiological study of epidemic dropsy. Indian J Med Res.1984;80:449–56.
9. Upreti KK, Das M, Khanna SK. Role of antioxidants and scav-engers on argemone oil toxicity in rats. Arch Environ ContamToxicol. 1991;20:531–7.
10. Sachdev HPS, Sachdev MS, Sood LV, et al. Electrophysiologicalstudies of the eye, peripheral nerve and muscles in epidemicdropsy. J Trop Med Hyg. 1989;92:412–15.
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