Acta Pædiatrica ISSN 0803–5253
REGULAR ARTICLE
Factors associated with sclerema in infants with diarrhoeal disease: a matched case-control study Mohammod Jobayer Chisti (
[email protected])1 , Tahmeed Ahmed1 , Abu Syed Golam Faruque1 , Shuvra Saha2 , Mohammed Abdus Salam1 , Sufia Islam3 1.International Centre for Diarrhoeal Disease Research, Bangladesh (ICDDR,B), Dhaka, Bangladesh 2.Medical Research Council (UK), The Gambia 3.Department of Pharmacy, East West University, Dhaka, Bangladesh
Keywords Case-fatality, Diarrhoea, Hypothermia, Infant, Sclerema
Abstract Aim: To identify clinical and biochemical factors associated with sclerema in infants with diarrhoeal
Correspondence Dr. Mohammod Jobayer Chisti, Clinical Sciences Division, International Centre for Diarrhoeal Disease Research, Bangladesh (ICDDR,B), 68 Shaheed Tajuddin Ahmed Sarani, Mohakhali, Dhaka 1212, Bangladesh. Tel: + (88-02) 8860523-32 ext: 2334 | Fax: + (88-02) 8823116 | Email:
[email protected]
from among those admitted to the special care unit (SCU) and longer stay unit (LSU) of the Dhaka Hospital of International Centre for Diarrhoeal Disease Research, Bangladesh (ICDDR,B) for their diarrhoeal illness from May 2005 through April 2006. Sclerema as the dependant variable while
Received 23 September 2008; revised 27 November 2008; accepted 3 December 2008. DOI:10.1111/j.1651-2227.2008.01196.x
illness, and their outcome. Methods: In this case-control study, we enrolled 30 infants with clinical sepsis with sclerema (cases) and another 60, age- and sex-matched infants with clinical sepsis but without sclerema (controls)
hypoxia, hypothermia, C-reactive protein (CRP) level, serum total protein and prealbumin level were the major independent variables compared in the analysis. Differences in proportions were compared by the chi-square test and differences of mean were compared by Student’s t-test or Mann–Whitney test, as appropriate. Results: The case-fatality was significantly higher among the cases than the controls (30% vs. 2%, CI 2.9–565.5). After adjusting for confounders, infants with sclerema were more likely to be hypothermic (OR 11.6, 95% CI 1.1–126.5), and have lower serum total protein (OR 1.12, 95% CI 1.04–1.21) and prealbumin (OR 1.5, 95% CI 1.1–2.3). Conclusion: Diarrhoeal infants having clinical sepsis presenting with hypothermia, lower serum protein and prealbumin are prone to be associated with sclerema.
INTRODUCTION Sclerema is defined as a diffuse, doughy feeling of the skin and/or rapidly spreading, tallow-like hardening of the subcutaneous tissue. It is more frequently observed in neonates (a condition known as sclerema neonatorum [SN]); however, has also been reported in older infants (1–5), and the reported age of the oldest infant with sclerema in association with Pseudomonas sepsis was 106 days (2). Sclerema is characterized by deposition of crystals, primarily of triglycerides, in the subcutaneous fat that can be determined by X-ray diffraction; however, some studies reported subcutaneous fibrosis, not crystallization of fat contents, was the conspicuous feature (3–5). Most of the clinical features of sepsis are usually present in SN suggesting its clinical severity (6,7). The pathogenesis of this condition has not been fully elucidated, which is important for optimizing management to reduce deaths, although there are encouraging reports on dramatic response to exchange fresh human blood transfusion and fresh blood transfusion (8–12). However, not all infants with sepsis present with sclerema, and it is therefore likely that some as yet undermined factors might play an important role for the development of this deadly condition (13). The reported case-fatality from SN in different series ranged from 50% to 100%, and deaths usually occur within hours to days of
onset of the condition (2,14). We also observed high case fatality among children presenting to the Dhaka Hospital of International Centre for Diarrhoeal Disease Research, Bangladesh (ICDDR,B) with diarrhoea and sclerema and noted other abnormalities such as hypernatremia, hyponatremia, acidosis, hypothermia and sepsis in such populations. To our knowledge, the clinical and biochemical factors associated with sclerema in infants with diarrhoeal disease have not been described in the medical literature. We thus conducted a prospective, case-control study to identify the clinical and biochemical factors associated with this condition, in an effort to identify better preventive and therapeutic options in the management of sclerema. MATERIALS AND METHODS Patient enrollment This study was conducted at the Dhaka Hospital of ICDDR,B, Dhaka, Bangladesh, which provides care and treatment to around 110 000 diarrhoeal patients with or without associated complications and with or without other health problems each year. The vast majority of the patients come from the poor socio-economic backgrounds from urban and peri-urban Dhaka, the capital city of Bangladesh. This study was approved by Research Review Committee (RRC) and Ethical Review Committee (ERC) of ICDDR,B.
C 2008 The Author(s)/Journal Compilation C 2008 Foundation Acta Pædiatrica/Acta Pædiatrica 2009 98, pp. 873–878
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Factors associated with sclerema
Chisti et al.
A written informed consent was obtained from respective parents/guardians of the each participating infant. We enrolled most of the eligible infants from among those admitted to the special care unit (SCU), but also a few from longer stay unit (LSU) of the hospital from May 2005 through April 2006. A clinical diagnosis of sclerema was made when at least two clinicians of the hospital, including at least one of the investigators reached an agreement. Study design In this age- and sex-matched case-control study, we enrolled 30 cases and 60 controls from a total of 450 infants with clinical sepsis who were admitted to the study units during the study period. Infants presenting with sclerema, defined as diffuse, doughy feeling of the skin and/or tallow like hardening of the subcutaneous tissue in the absence of any localized skin lesion, along with features of sepsis constituted cases. We defined sepsis as presence of any two of the following: tachypnoea, tachycardia, thermo-instability (hypo or hyperthermia) and abnormal WBC count (>11 000/cc or
