Indmedica Home | About Indmedica | Medical Jobs | Advertise On Indmedica
Search Indmedica Web
Indmedica - India's premier medical portal

Current Pediatric Research

Infantile Tremor Syndrome - New observations

Author(s): Vinod H Ratageri, T.A.Shepur, Santosh Byakod

Vol. 10, No. 1 (2006-10 - 2006-12)

Curr Pediatr Res 2006; 10 (1 & 2): 9-11

Vinod H Ratageri, T.A.Shepur, Santosh Byakod

Department of Pediatrics, Karnataka Institute of Medical Science, Hubli-580021, Karnataka.

Key words: ITS, clinical features, vitamin C, Folic acid

Accepted February 07, 2006

Abstract

Infantile Tremor syndrome is a peculiar condition most commonly seen in Indian subcontinent. Although the number of cases with this syndrome reduced in recent years, finding of additional features made us to present this series. Total eleven cases were seen during Nov 2003 to Oct 2005 (3 year). Mean age of presentation was 11.2 months ( range 6 to 18 months). The male to female ratio was 5: 6. There was no seasonal variation. Tremors, pigmentation, delayed milestones, pallor and hepatomegaly was present in all cases. Splenomegaly was present in 06 (54%) cases. LRTI was associated presenting feature in 09 (82% )cases and also CCF in 03 ( 27 %) cases. The striking feature noticed was presence of scurvy in all cases on x-ray imaging studies. All children received vitamin C, folic acid and vitamin B12 and antibiotics.

The mean duration of tremor phase was 23.2days with range 5 to 44 days. There was no mortality. Vitamin C and folic acid plays significant therapeutic role in ITS management.

Introduction

Infantile Tremor Syndrome (ITS) is a well known clinical entity. Although reported from outside India [1-3], seen more commonly in Indian subcontinent [4]. The clinical features of this syndrome are tremors, skin pigmentation, developmental and mental retardation and anaemia. But in recent years, in addition to the above characteristic features we noticed other findings [5]. Hence, we are presenting our observations on ITS in recent years.

Subjects and Methods

All children presenting with typical features of ITS, admitted in between November 2003 to October 2005 (3 years) in pediatric medical ward at Karnataka Institute of Medical Sciences, Hubli, Karnataka were analyzed.

A detailed history and physical examination was carried out. Investigations including complete haemogram, blood culture and sensitivity, urine microscopy stool microscopy,

CSF examination, chest x-ray, knee x-ray, Mantoux test and gastric lavage for AFB, HIV test were done for all children.

Results

The total number of cases seen during this period was 11. All children belong to age group between 6 months to 18 months with mean age 11.2 months and all of them belong to lower socioeconomic group. The male to female ratio was 5:6. All children were exclusively breastfed till the time of admission except two in which in addition to breast feedings, small quantities of biscuits and rice were given. Out of 11 children, four each admitted during winter and rainy season and three in summer. Seventy three percent of (73%) them belong to severe malnutrition as per IA P classification.

Table I shows clinical features at presentation. In addition to characteristic features hepatomegaly was present in all cases and splenomegaly in 06 (54%) cases.

Three cases (27 ) had hemoglobin (HB) less than 5gm/ dl and in remaining cases Hb varied between 5 to 10gm. Dimorphic anaemia was predominant picture (63 %) on peripheral smear. Macrocytic anaemia was present in 4 (37 cases. There was no evidence of tuberculosis and HIV in any of our children. Urine and stool examination was normal and blood culture was sterile in all cases. CSF examination was within normal limits except in two case where CSF protein was raised just above normal X-ray imaging studies reveled presence of scurvy changes in all children and pneumonic patches in seven cases (77%). C.T. scan head done in two cases was normal.

Table I Clinical features at presentation (n=11)

Sl. No. Clinical features No. of cases (%)
1. Tremors
Localized 09 (82)
Generalized 02 (18)
2. Pigmentation
Localized 07 (63)
Generalized 04 (37)
3. Pallor 11 (100)
4. Delayed milestones/
Mental retardation
11 (100)
5. Hepatomegaly 11 (100)
6. Splenomegaly 06 (54)
7. LRTI 09 (82)
8. C C F 03 (27)

All children received vitcofol (contains folic acid and Vitamin B12), and B12, Vitamin C and intravenous antibiotics. Three of these children required blood transfusion in addition to above medication. For control of tremors propranalol carbamazepine and phenobarbitone it one was used in 04, 01 and 01 cases respectively carbamazepine The total duration required for control of tremors in our cases ranged from 5 to 44 days with mean duration of 23.2 days. There was no mortality.

Discussion

We observed that girls outnumbered boys. In many series its been noticed that boys affected more commonly [6]. However, chaparwal et al in their study noticed equal sex distribution [7]. We also observed that there was no specific seasonal predominance in our cases where as earlier studies noticed seasonal variation [8-10] indicating probably other than viral etiology playing the role in causation of ITS. Apart from typical clinical features, we also noticed presence of splenomegaly in more than in 50% of cases. Bajapai et al noticed splenomegaly only in 2 cases (out of 11). The probable reason for this high incidence in our cases could be due to simultaneous occurrence of LRTI.

The striking feature noticed in our cases was presence of scurvy changes on imaging studies. No other features of scurvy was present in any of our cases. The proposed mechanisim explained elsewhere [5]. The mean duration taken for tremor control in our cases was 23.2 days with range from 5 to 44 days. Tandon et al reported the mean duration of tremors as 43.4 days with a range from 3to 400 [12]. In other studies reported that mean being 50.5 days with range from 3-225 days [13]. Duration of tremor was reduced in our cases probably due to addition of vitamin C.

Majority of our children had LRTI at the time of presentation. Although it has been observed in earlier studies the presence of LRTI [14-15].The exact reason for this high incidence of LRTI is not known but may be due to presence of less antioxidants due to deficiency of Vitamin C. In view of presence of scurvy and decreased duration of tremor phase with addition of vitamin c, we feel that folic acid and vitamin C plays a significant role in management of ITS.

In summary compared to the earlier reports, we in our children observed occurrence of ITS more in girls, no seasonal variation, Scurvy changes on imaging studies and decreased duration of tremor phase, in addition to this also seen higher incidence of LRTI and occurrence of splenomegaly.

References

  1. Arya LS, Singh M, Aram GN. Farahmand S. Infantile tremor syndrome. Indian J Pediatrics 1988;55: 913-918.
  2. Gupte S. Infantile tremor syndrome . In the short text book of Peadiatrics 8th Edition, New Delhi: Jay pee Brothers 1988; 558-561.
  3. Amin MN, Zeki JM. Infantile Tremor Syndrome in Iraqi Kurdistan. Indian J Peadiatrics 2005; 72: 839-842.
  4. Sharada B, Bhandari B. Infantile tremor syndrome. Indian Pediatrics 1987; 24: 415-421.
  5. Ratageri VH, Shepur TA, Patil MM, Hakeem MA. Scurry in infantile tremor syndrome. Indian J Pediatrics 2005; 72: 883-884.
  6. Gupta BD, Maheshwari RK, Miglani N. Infantile tremor syndrome Indian J Peadiatrics 1978; 45:221-228.
  7. Chaparrwal B L, Singh S D, Mehta S, Pohowalla JN. Magnesium level in Serum and CSF in meminago-encehalitic syndrome. Indian J.Paediatrics 1971; 38: 331-333.
  8. Bajpai PC, Misra PK, Tandon PN. Further observation on infantile tremor syndrome. Indian Peadiatrics 1968; 5: 297-307.
  9. Garg BK, Srivatsava JR. Infantile tremor syndrome. Indian J Peadiatrics 1969; 36: 215-218.
  10. Dikshit AK. Nutritional Dystrophy and anaemia. Indian J Child Health 1957; 6: 132-136.
  11. Bajapai PC, Misra PK, Tandon PN, Nigam GJ, Vora VC et al. Serum Vitamin B12 in infantile tremor syndrome. Indian J Medical Research 1968; 56: 1398-1405.
  12. Tandon PN, Bajapai PC. The infantile tremor syndrome. In Tropical Neurology Ed. Spillac JD, London, Oxford University Press 1973; P 37.
  13. Sachdeva KK, Manchanda SS, Lal H. The syndrome of tremors mental regrenion and anaemia in infants and young children –A study of 102 cases. Indian Peadiatrics 1965 ; 2: 239-251.
  14. Jadhav M, Webb JJK, Vaishnava S. Vitamin B12 deficiency in Indian infants-A Clinical syndrome Lancet 1962; 2: 903-906.
  15. Kaul K K, Prasanna N, Chowdhary RM. Some clinical observation and impression on a syndrome of tremors in infants from India. J Peadiatrics 1963; 63: 1158-1166.

Correspondence:
Vinod H. Ratageri
Department of Paediatrics
Karnataka Institute of Medical Science
Hubli.580021
India
E-mail: ratageri(at) rediffmail.com

Access free medical resources from Wiley-Blackwell now!

About Indmedica - Conditions of Usage - Advertise On Indmedica - Contact Us

Copyright © 2005 Indmedica