Indian J Pediatr DOI 10.1007/s12098-013-1300-8
SCIENTIFIC LETTER
Dengue Hemorrhagic Fever with Discolored Leg Syndrome in a Neonate Manoharan Vijoo & Ramalingam Raja & Chandrasekaran Venkatesh & Uthirakumaran Ramkumar & Palanisamy Soundararajan
Received: 1 June 2013 / Accepted: 4 November 2013 # Dr. K C Chaudhuri Foundation 2013
To the Editor: A 23-d-old male neonate was brought with history of fever of 2 days duration followed by purpuric rashes over the trunk and extremities of 1 day duration associated with echymoses and purplish discoloration over the lower extremities (Fig. 1). The neonate had received injection vitamin K at birth and there were no risk factors for developing vitamin K deficiency. Except for the bleeding manifestations and mild hepatomegaly, rest of the examination were normal. Investigations revealed thrombocytopenia with prolonged prothrombin and activated partial thromboplastin time, mildly elevated total leukocyte count and deranged liver function test. TORCH panel was negative. Late onset sepsis with disseminated intravascular coagulation (DIC) was suspected and injection cloxacillin and gentamicin were started, but stopped after a week as blood culture and CRP were negative. Dengue NS1 antigen was positive which was later confirmed by serology with IgM and IgG both being positive. The child was treated symptomatically with intravenous fluids titrated to hematocrit, whole fresh blood and fresh frozen plasma. The purplish discoloration was the first to disappear followed by echymoses and the purpura with normalization of coagulation profile and platelet count, following which the neonate was discharged home. A follow up one week later revealed a well infant with no evidence of any further bleeding. Dengue infection is uncommon in the neonatal period but can be fatal. The transmission of virus can occur during the perinatal period or the infection may be acquired in the postnatal period. The reasons for severe infection developing in the neonates is due to a process known as antibody dependent enhancement, wherein the transplacentally acquired IgG M. Vijoo : R. Raja : C. Venkatesh (*) : U. Ramkumar : P. Soundararajan Department of Pediatrics, Mahatma Gandhi Medical College & Research Institute, Puducherry 607 402, India e-mail: [email protected]
facilitates replication of virus (of a different strain) acquired postnatally [1]. In the reported case, there was no history suggestive of dengue infection in the mother either during her pregnancy or in the post partum period. This would mean that the mother might have had a sub clinical dengue infection and would have developed antibodies which would have been passively transferred to her infant. Atypical manifestations of dengue have been described in older children [2] but rarely in neonates. In our case, although the neonate had features of bleeding and vascular stasis (purplish discolouration of extremities) suggesting a picture of purpura fulminans, the peripheral pulses were well felt and Doppler study was normal. Dengue hemorrhagic fever presenting as purpura fulminans in a 4-y-old girl has been described in literature [3]. Discolored leg syndrome is a distinct entity characterized by patchy red, blue or purple discoloration of the legs and has been described in neonates 48 h following vaccination, predominantly with DPT containing vaccines [4]. The mechanism of discoloration
Fig. 1 Diffuse purpura, ecchymosis and purplish discoloration of feet in a neonate
Indian J Pediatr
is unknown and thought to occur due to vasomotor reaction. However, in the reported case, no intramuscular injections or vaccines were given during the period immediately prior to the onset of symptoms. Contributions All authors were involved in the diagnosis and management of the case, literature review and drafting of the manuscript. Professor Palanisamy Soundararajan will act as guarantor for this paper. Conflict of Interest None. Role of Funding Source None.
References 1. Kabra SK, Jain Y, Singhal T, Ratageri VH. Dengue hemorrhagic fever: Clinical manifestations and management. Indian J Pediatr. 1999;66: 93–101. 2. Gala HC, Avasthi BS, Lokeshwar MR. Dengue shock syndrome with two atypical complications. Indian J Pediatr. 2012;79:386–8. doi:10. 1007/s12098-011-0551-5. 3. Karunatilaka DH, De Silva JR, Ranatunga PK, Gunasekara TM, Faizal MA, Malavige GN. Idiopathic purpur fulminans in dengue hemorrhagic fever. Indian J Med Sci. 2007;61:471–3. 4. Kemmeren JM, Vermeer-de Bondt PE, van der Mass NA. Discoloured leg syndrome after vaccination—descriptive epidemiology. Eur J Pediatr. 2009;168:43–50.
SCIENTIFIC LETTER
Dengue Hemorrhagic Fever with Discolored Leg Syndrome in a Neonate Manoharan Vijoo & Ramalingam Raja & Chandrasekaran Venkatesh & Uthirakumaran Ramkumar & Palanisamy Soundararajan
Received: 1 June 2013 / Accepted: 4 November 2013 # Dr. K C Chaudhuri Foundation 2013
To the Editor: A 23-d-old male neonate was brought with history of fever of 2 days duration followed by purpuric rashes over the trunk and extremities of 1 day duration associated with echymoses and purplish discoloration over the lower extremities (Fig. 1). The neonate had received injection vitamin K at birth and there were no risk factors for developing vitamin K deficiency. Except for the bleeding manifestations and mild hepatomegaly, rest of the examination were normal. Investigations revealed thrombocytopenia with prolonged prothrombin and activated partial thromboplastin time, mildly elevated total leukocyte count and deranged liver function test. TORCH panel was negative. Late onset sepsis with disseminated intravascular coagulation (DIC) was suspected and injection cloxacillin and gentamicin were started, but stopped after a week as blood culture and CRP were negative. Dengue NS1 antigen was positive which was later confirmed by serology with IgM and IgG both being positive. The child was treated symptomatically with intravenous fluids titrated to hematocrit, whole fresh blood and fresh frozen plasma. The purplish discoloration was the first to disappear followed by echymoses and the purpura with normalization of coagulation profile and platelet count, following which the neonate was discharged home. A follow up one week later revealed a well infant with no evidence of any further bleeding. Dengue infection is uncommon in the neonatal period but can be fatal. The transmission of virus can occur during the perinatal period or the infection may be acquired in the postnatal period. The reasons for severe infection developing in the neonates is due to a process known as antibody dependent enhancement, wherein the transplacentally acquired IgG M. Vijoo : R. Raja : C. Venkatesh (*) : U. Ramkumar : P. Soundararajan Department of Pediatrics, Mahatma Gandhi Medical College & Research Institute, Puducherry 607 402, India e-mail: [email protected]
facilitates replication of virus (of a different strain) acquired postnatally [1]. In the reported case, there was no history suggestive of dengue infection in the mother either during her pregnancy or in the post partum period. This would mean that the mother might have had a sub clinical dengue infection and would have developed antibodies which would have been passively transferred to her infant. Atypical manifestations of dengue have been described in older children [2] but rarely in neonates. In our case, although the neonate had features of bleeding and vascular stasis (purplish discolouration of extremities) suggesting a picture of purpura fulminans, the peripheral pulses were well felt and Doppler study was normal. Dengue hemorrhagic fever presenting as purpura fulminans in a 4-y-old girl has been described in literature [3]. Discolored leg syndrome is a distinct entity characterized by patchy red, blue or purple discoloration of the legs and has been described in neonates 48 h following vaccination, predominantly with DPT containing vaccines [4]. The mechanism of discoloration
Fig. 1 Diffuse purpura, ecchymosis and purplish discoloration of feet in a neonate
Indian J Pediatr
is unknown and thought to occur due to vasomotor reaction. However, in the reported case, no intramuscular injections or vaccines were given during the period immediately prior to the onset of symptoms. Contributions All authors were involved in the diagnosis and management of the case, literature review and drafting of the manuscript. Professor Palanisamy Soundararajan will act as guarantor for this paper. Conflict of Interest None. Role of Funding Source None.
References 1. Kabra SK, Jain Y, Singhal T, Ratageri VH. Dengue hemorrhagic fever: Clinical manifestations and management. Indian J Pediatr. 1999;66: 93–101. 2. Gala HC, Avasthi BS, Lokeshwar MR. Dengue shock syndrome with two atypical complications. Indian J Pediatr. 2012;79:386–8. doi:10. 1007/s12098-011-0551-5. 3. Karunatilaka DH, De Silva JR, Ranatunga PK, Gunasekara TM, Faizal MA, Malavige GN. Idiopathic purpur fulminans in dengue hemorrhagic fever. Indian J Med Sci. 2007;61:471–3. 4. Kemmeren JM, Vermeer-de Bondt PE, van der Mass NA. Discoloured leg syndrome after vaccination—descriptive epidemiology. Eur J Pediatr. 2009;168:43–50.
