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RECURRENT OPISTHOTONUS IN CATATONIA: AN ATYPICAL PRESENTATION

NARAYANA MANJUNATHA, URVAKHSH MEHERWAN MEHTA, JOHN P.

ABSTRACT

Opisthotonus is known to occur in , rabies, cerebral , neurosyphilis, acute cerebral injury and other medical conditions. Opisthotonus, so far, has not been reported in any major psychiatric disorder. Authors report a case of recurrent opisthotonus presenting concurrently with other catatonic signs which showed dramatic response to combination of lorazepam and electroconvulsive therapy (ECT). Clinicians should consider the possibility of catatonia in the differential diagnosis of opisthotonus since catatonia can be treated easily with benzodiazepines and ECT.

Key words: Catatonia, opisthotonus, posturing

DOI: 10.4103/0019-5359.58881 PMID: ****

INTRODUCTION and behavioral signs.[7] Catatonic signs consist of motor abnormalities in the form Opisthotonus is a state of extreme of posturing, gegenhalten, waxy flexibility, hyperextension of the head, neck and spine echolalia/ echopraxia, rigidity, stereotypies that presents as severe, prolonged and psychological pillow.[8] The PUBMED characterized by a strongly arched and rigid index search using combination of terms back, hyperextended neck and arms, with like “catatonia” and “opisthotonus/ catatonia [1] the heels bent back. Opisthotonus is known and opisthotonos” [both opisthotonus and to occur in tetanus, rabies, cerebral malaria, opisthotonos are accepted in the literature, neurosyphilis, , encephalitis, cerebral but opisthotonus (rather than -tonos) is more palsy, partial , poisoning frequently cited and better accepted] yielded and adverse effects of medications.[2-6] 3 case reports,[9-11] but no authors considered opisthotonus as a catatonic sign. Authors report Catatonia is a neuropsychiatric condition a case of catatonia in which opisthotonus (arc with unique combinations of mental, motor de cercle) presented as a form of posturing (which itself is a catatonic sign). Department of Psychiatry, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India CASE REPORT Correspondence: Dr. N. Manjunatha Department of Psychiatry, A 26-year-old formally educated married man National Institute of Mental Health and Neurosciences hailing from a rural background, working as a (NIMHANS), Bangalore - 560 029, India E-mail: [email protected] migrant construction worker with negative past,

Indian J Med Sci, Vol. 63, No. 11, November 2009 OPISTHOTONUS IS ATYPICAL CATATONIC SIGN 513 family and personal history, presented to our signs — stupor, mutism, withdrawal, posturing emergency unit on the 8th day of illness (for the and autonomic instability. The baseline Bush- fi rst time for medical attention) of abrupt onset, Francis catatonia rating scale (BFCRS)[12] score characterized by a polymorphic clinical picture was 25. A detailed neurological and systemic without precipitating factors. At the onset examination ruled out localizing neurological of illness, i.e., 8 days prior to presentation, signs, hepatosplenomegaly and other clinical the patient abruptly became anxious and features that could suggest the presence irritable for a day. Thereafter, he absconded of primary medical/ neurological conditions from home and wandered aimlessly in public such as cerebral malaria, neurosyphilis, etc. places, exhibiting disinhibited behavior over The patient was diagnosed provisionally as the next 2 days. When he was brought back having brief psychotic disorder (DSM–IV home by police on the 4th day, he was noted to TR) with prominent catatonic signs, and be irritable and started claiming that all family was initiated on treatment with intravenous members were in danger and that the police lorazepam 2 mg thrice daily, after an initial were on their trail. Soon after being brought statum intravenous dose of 4 mg. While in back home, the patient became extremely the hospital, the patient developed facial agitated, screaming loudly, running out onto the grimacing [due to facial muscle spasm, giving streets, hitting walls and doors with closed fi sts an appearance of a sneering grin like that of and being sleepless throughout the night. This risus sardonicus], motor stereotypies of lower extreme psychomotor excitement continued for jaw, verbal stereotypies, mitgehen, posturing the next 36 hours and then calmed down. and psychological pillow. The above catatonic features were accompanied by episodic and During the next 2 days, the clinical picture recurrent opisthotonus, or arc de cercle, changed to mutism, posturing, withdrawal, each episode lasting for 10 to 20 minutes, negativism and passing urine in clothes. During with multiple such episodes occurring over this period, the patient was noted to hold his the course of 24 hours with a gap of a few hands in a folded position continuously (posture minutes between episodes. These episodes of namasthe, the traditional way of greeting of opisthotonus co-occurred with fl uctuating each other in Indian culture) for about 30 to catatonic signs and features of autonomic 45 minutes at a time. There was no history of instability (tachycardia, fluctuating blood substance abuse, consumption/ administration pressure and sweating). The patient was noted of any medications (including neuroleptics) to have low-grade intermittent fever (maximum in the recent past; fever; motor weakness; of 99°F), which responded to antipyretics. This loss of consciousness; seizures; recent open clinical picture resembled earlier descriptions wound; head injury; or dog bite. There were no of malignant/ lethal catatonia.[13,14] Authors symptoms of mania, depression, schizophrenia also ruled out psychogenic opisthotonus and or suicidal attempts/ ideation. myoclonic seizures in this patient by history and observation in hospital. On examination in the emergency care unit on the 8th day of illness, the patient had catatonic Baseline routine biochemical parameters

Indian J Med Sci, Vol. 63, No. 11, November 2009 514 INDIAN JOURNAL OF MEDICAL SCIENCES did not reveal any abnormality. Repeat associated with other catatonic signs. Catatonia laboratory parameters after occurrence of can be treated easily with lorazepam and ECT, opisthotonus, such as blood glucose, creatinine- thus avoiding extensive investigations to rule phosphokinase, liver and renal parameters, out other causes of opisthotonus. electrolytes, hemogram, urine microscopy and culture, revealed no abnormality. Authors consider opisthotonus in this patient Computed tomography scan of head, as well as as a variant of stereotypic posturing, which cerebrospinal fl uid analysis (routine, acid-fast is a catatonic sign. Posturing is considered and India ink staining with Herpes Simplex Virus as one of the more distinctive phenomena in antibody titers), did not reveal any abnormality. catatonia.[15] It is described as a specific, Electroencephalography showed well-defi ned uncomfortable and often bizarre positioning alpha rhythm with lorazepam-induced fast beta of body parts against gravity with complete activity and no epileptiform discharges. akinesia maintained for hours, days and sometimes weeks. Different forms of posturing The patient was maintained on parenteral involving limbs (classic posturing), head nutrition and supportive medical care in the (psychic pillow) and eyes (staring) have been intensive care unit. Dramatic improvement was described, which can broadly be classifi ed into observed with intravenous lorazepam 2 mg two types — manneristic and stereotypic.[16] thrice daily and one session of modifi ed bilateral Manneristic posture is an odd stilted posture electroconvulsive therapy (ECT). Over the that is an exaggeration of a normal posture next 3 days, the patient recovered completely and not rigidly preserved, while a stereotyped from catatonia, with BFCRS score of zero. posture is an abnormal and nonadaptive Lorazepam was subsequently tapered off and posture that is rigidly maintained (e.g., stopped over the next 4 days. No antipsychotics psychological pillow). were used till the patient recovered from catatonia. Authors consider catatonic signs in this patient as part of acute psychosis (DSM- Following complete recovery from catatonia IV). There were no clinical or laboratory on the 13th day of illness, the patient revealed findings suggestive of a primary underlying that he wandered away from home during organic cause that could explain the above the initial period of excitement in response to clinical picture. Neuroleptic/ drug-induced command auditory . The patient extrapyramidal side effect as cause of was discharged on risperidone 4 mg/day with a opisthotonus was also ruled out, as the patient fi nal diagnosis of brief psychotic disorder (DSM– had not received any neuroleptics prior to IV). The patient has not subsequently attended the appearance of opisthotonus (as he was the follow-up. brought to medical attention for the fi rst time). DISCUSSION However, it is our contention that irrespective of whether an underlying organic cause is Understanding opisthotonus as a catatonia sign established, opisthotonus in this patient could has treatment implications, especially when it is still be considered as a catatonic sign since

Indian J Med Sci, Vol. 63, No. 11, November 2009 OPISTHOTONUS IS ATYPICAL CATATONIC SIGN 515 it appeared along with other catatonic signs 2. Ries CR, Scoates PJ, Puil E. Opisthotonos following the initial period of excitement and following propofol: A nonepileptic perspective and resolved with symptomatic treatment along treatment strategy. Can J Anaesth 1994;41:414-9. with the other catatonic signs. Therefore, the 3. Wong KT, Lum LCS, Lam SK, Huang CC, authors concluded that recurrent opisthotonus Liu CC, Chang YC. Enterovirus 71 infection and neurologic complications. N Engl J Med in this patient was a catatonic sign, i.e., a form 2000;342:356. of catatonic posturing. 4. Hendrickson RG, Morocco AP, Greenberg MI. Acute dystonic reactions to “Street Xanax”. N The authors propose, therefore, that the Engl J Med 2002;346:1753. neurobiological substrate of opisthotonus is 5. Idro R, Otieno G, White S, Kahindi A, Fegan similar to that of catatonic posturing, involving G, Ogutu B, et al. Decorticate, decerebrate a deficit in ‘termination’ of movements, and opisthotonic posturing and seizures in mediated by the posterior parietal cortex.[17] Kenyan children with cerebral malaria. Malar J Compromised GABAergic (neuron producing 2005;7:4:57. gamma-aminobutyric acid) inhibition in the 6. Chauhan VV, Pickens AT. Opisthotonus in spinal cord has also been suggested as a neurosyphilis: A case report. J Emerg Med 2008 possible etiological factor in opisthotonic in press. posturing.[2] The dramatic improvement in 7. Braunig P, Kruger S. History. In: Caroff SN, Mann SC, Francis A, Fricchione GL, editors. Catatonia opisthotonic posturing following intravenous – from psychopathology to neurobiology. lorazepam in this patient provides strong Washington DC: American Psychiatric Publishing support to the above theory. Inc; 2004. p. 1-14. 8. Taylor MA. Clinical examination. In: Caroff CONCLUSION SN, Mann SC, Francis A, Fricchione GL, editors. Catatonia – from psychopathology Opisthotonus may be a catatonic sign in to neurobiology. Washington DC: American medical emergency. Clinicians should Psychiatric Publishing Inc; 2004. pp. 45-64. consider the possibility of idiopathic catatonia 9. Dekleva KB, Husain MM. Sporadic encephalitis in their differential diagnosis of opisthotonus. lethargica: A case treated successfully with ECT. Opisthotonus co-occurring with other catatonic J Neuropsychiatry Clin Neurosci 1995;7:237-9. signs in the absence of other features 10. Diesing TS, Wijdicks EF. Arc de cercle and dysautonomia from anoxic injury. Mov Disord suggesting an underlying organic cause should 2006;21:868-9. be treated primarily with benzodiazepines and 11. Engquist A, Jørgensen BC, Andersen HB. ECT. Catatonia after epidural morphine. Acta Anaesthesiol Scand 1981;25:445-6. REFERENCES 12. Bush G, Fink M, Petrides G, Dowling F, Francis A. Catatonia I. Rating scale and standard 1. Spillane J. Involuantary movements. In: examination. Acta Psychiatr Scand 1996;93:129- Bickerstaff’s neurological examination in clinical 36. practice. 6th ed. Oxford: Blackwell Science Ltd; 13. Castillo E, Rubin RT, Holsboer-Trachsler E. 1996. p. 160-75. Clinical differentiation between lethal catatonia

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Indian J Med Sci, Vol. 63, No. 11, November 2009