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Case Reports

Indian Pediatrics 1999; 36:86-88 

Paroxysmal Torticollis of Infancy


UijaI Poddar
Surjit Singh
Lata Kumar

From the Department of Pediatrics, Postgraduate Institute of Medical Education and Research, Chandigarh 160012, India.

Repri1lt requests: Dr. Ujjal Poddar, Assista1lt Professor, Division of Pediatric Gastroenterology, Department of Gastroenterology, PGIMER,
Chadigarh 160012, India. E-mail: [email protected]

Manuscript Received: May26, 1998; Initial review completed: July 6, 1998;
Revision Accepted: August 7, 1998

Paroxysmal torticollis (PT) is a self-limiting condition which presents in infancy and generally resolves spontaneously by' two to three years of age( I). This condition was first described by Snyder in 1969(1). It is characterized, by recurrent episodes of head tilt, usually alternating from side to side, sometimes accompanied by vomiting, pallor, agitation and ataxia which subside spontaneously within a few hours or days. The etiology remains unknown. No treatment is effective. Since the first description of 12 cases, less than 40. cases have been described. We report here an infant who presented with paroxysmal torticollis.

Case Report

A 5-month-old girl was admitted for head tilting. She was youngest of the four children. She had tilting of head to the right side and irritability-for 3 days. There was no history of pallor, vomiting, seizures, trauma and drug intake. There was no past history of similar attacks. Her development was normal. On examination she weighed 5 kg; other anthropometric measurements were within normal limits. She was keeping her head tilted to right and did- not cry on manipulation. There was no other abnormalities on systemic examination. Both parents
and siblings were healthy. There was no family history of similar illness or of migraine or paroxysmal vertigo.

Over the next 3 days her tilting disappeared and she remained alright for next 3 days. But on 7th day she again developed head tilting and this time it was to the left side. Though the first episode was associated with irritability and lasted for 6 days, second episode was not associated with irritability and lasted for 2 days only. During these episodes she appeared alert and was usually not in distress. She could be fed during the episodes. After one month of discharge she was alright.

She was investigated to rule out other causes of head tilting like the. presence of intracranial space occupying lesion, atlantoaxial dislocation and. benign paroxysmal vertigo (cold caloric test).

Discussion

Although, torticollis is present in many illnesses with several different mechanisms(2-6), in paroxysmal torticollis the onset in the first months of life, the very frequent and often striking regular recurrence(5) and tendency to remit spontaneously and to disappear entirely after a few months or years make it unlike any other known form of intermittent torticollis. In most of the reported. cases the torticollis was noted upon awakening in the morning(7). Such attacks occur quite often (once or twice in a month) initially, last from hours. to days and gradually disappear or recur after long symptom free intervals, eventually to stop completely.

In our case, episodes. of torticollis started at 5 months of age, lasted for 3 to 6 days, and resolved spontaneously. The child remained completely normal in between. Associated clinical symptoms like pallor, irritability, vomiting and general malaise which were described by Snyder(1) were not seen in our patient.

There was no family history of similar episodes in our case; though this has been reported(5,8-10). Female preponderance was described in all reports(1,5,9,11). Our.patient was also a female infant. This female preponderance and occurrence of similar cases in the family indicates the possibility of some genetic causative factors.

The etiology of PT remains obscure. In his original description, Snyder(1) suggested a possible abnormality in the peripheral vestibular (labyrinthine) apparatus' similar to that which is associated with the syndrome of benign paroxysmal vertigo in children (BPV). Dunn and Snyder(2) proposed that PT was merely a precursor of BPV based upon the fact that four of his original patients developed the BPV as they became older. In our case cold caloric test was normal and thus there was no abnormality in peripheral vestibular apparatus.

Some authors(3,5,9) postulated that the abnormality was cerebellar or of vestibulocerebellar connection; they thought that it might be vascular in origin in view of paroxysmal character. They concluded that both PT and BPV could possibly be precursors of recurrent migraine(3). None of the family members of our patient had migraine. As regards other etiology, there was no history of drug intake( 6) and no cervical dislocation(5) or posterior fossa tumor(4) could be detected.

Paroxysmal torticollis of infancy is a benign disorder, does not need any medication. It is important to recognize the condition because parents can be reassured that the prognosis is good.
 

 References


1. Snyder CH. Paroxysmal torticollis in infancy: A possible form of labyrinthitis. Am J Dis Child 1969; 117: 458-460.

2. Dunn DW, Snyder CH. Benign paroxysmal vertigo of childhood. Am J Dis Child 1976;
130: 1099- 1100.

3. Eeg Olofsson 0, OdkvistL, Lindskog V, Andersson B. Benign paroxysmal vertigo in childhood. Acta Otolaryngol 1982; 93: 283- 289.

4. Fessard C. Cerebral tumors in infancy. Am J Dis Child 1968; 115: 302-308.

5. Simer G, Bergstrom B. Benign paroxysmal torticollis in infancy. Acta Pediatr Scand 1979; 68: 219-223.

6. Daele CY. Paroxysmal torticollis in infancy. Am J Dis Child 1970; 120: 88.

Hanukoghs A, Somekh E, Fried D. Benign paroxysmal torticolIis in infancy. Clin Pediatr (Phila) 1984; 23: 272-274.

8. Lipson EH, Robertson We. Paroxysmal torticollis of infancy: Familial occurrence. Am J Dis Child 1978; 132: 422-423.

9. Deonna T, Martin D. Benign paroxysmal torticollis in infancy. Arch Dis Child 1981: 56: 956-959.

10. Roulet E, Deonna T. Benign paroxysmal torticolIis in infancy. Dev Med Child Neural 1988; 30: 409-410.

11. Bratt H, Menelaus M. Benign paroxysmal torticollis of infancy. J Bone Joint Surg 1992; 74B: 449-451.

 

 

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