NEONATAL BONE INFARCTION FOLLOWIN-G CORD SEPSIS: CASE REPORT J.U.V. Monu, MD, and A.A. Okolo, MD Benin-City, Nigeria

An unusual case of infarction of the tibia in a neonate who presented with sepsis following poor cord management at birth is reported. He was managed as for necrotizing enterocolitis and had limited debridement of infarcted limb because the parents refused amputation of the limb. This patient was a diagnostic problem. Necrotizing fasciitis and in retrospect purpura fulminans are considered as possible diagnoses and are discussed briefly. (J Nati Med Assoc. 1992;84:81-84.) Key words *neonatal bone infarction * cord sepsis Proper cord care is an issue that cannot be overemphasized in the third world, especially in tropical Africa where more than 70% of all births take place outside a hospital environment and under the supervision of traditional birth attendants. Poor cord hygiene has been associated with high mortality rates following grave conditions such as neonatal tetanus, necrotizing enterocolitis (NEC), necrotizing fasciitis (NF), and ascending omphalitis in the tropics.1'3 To this growing list of potentially fatal neonatal conditions, following indifferent cord management, we add the unusual case of neonatal tibial infarction. Diagnoses of osteomyelitis, NF, and retrospectively purpura fulminans (PF) were considered and are discussed briefly.

From the Departments of Radiology and Child Health, University of Benin, Benin-City, Nigeria. Requests for reprints should be addressed to Dr J.U.V. Monu, Department of Radiology, Bowman Gray School of Medicine, 300 S Hawthorne Rd, Winston-Salem, NC 27103. JOURNAL OF THE NATIONAL MEDICAL ASSOCIATION, VOL. 84, NO. 1

CASE REPORT A 2.6-kg male infant was born at home to a 28-year-old mother. Birth was assisted by a traditional birth attendant who dressed the cord stump with a medicinal concoction of herbs. He presented at the age of 10 days to the special care baby unit at the University of Benin Teaching Hospital on referral from a peripheral clinic with a 7-day history of fever, a 3-day history of generalized skin blisters, aind a 1-day history of swelling and discoloration of the right lower limb. On presentation, the baby appeared ill, irritable, and deeply jaundiced. He had a temperature of 39.60 C, tachypnea, and tachycardia (160/minute). There were bullous eruptions in various stages of evolution on the trunk and limbs. The umbilicus was erythematous and surrounded by prominent veins covered by pustulous scaly material. The abdomen was distended, and there was evidence of hepatosplenomegaly. The liver was firm, nontender, and palpable up to 4 cm below the costal margin. The distal third of the right leg and dorsum of the foot were discolored bluish black. The proximal leg was erythematous, edematous, and very tender. The foot was cold to touch, lacked pain sensation and the dorsalis pedis pulse was absent. There was no pedal crepitus noted. The infant was assessed to have generalized sepsis with omphalitis and gangrenous right leg and foot. He was admitted to the unit, worked up for sepsis, and started on a combined drug regimen of parenteral gentamicin 5 mg/kg/day, cloxacillin 100 mg/kg/day, and metronidazole 7.5 mg/kg/day in divided doses. On admission, his packed cell volume was 45% and micro ESR was 20 mm in the first hour, and the chest radiograph was normal. Abdominal radiographs showed changes of pneumatosis intestinalis in the right iliac area. The right lower limb was significantly larger than the left, 81

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Figure 1. Note the right lower limb soft tissues, which are larger than the contra-lateral side. The bony structures appear normal. and the soft tissues showed changes of edema. The underlying bony structures appeared normal (Figure 1). T'he radiologic impressions were necrotizing enterocolitis and cellulitis NF right lower leg and foot). The infant's serum bilirubin was 22.8 mg% with a conjugated component of 13 mg%. His cerebrospinal fluid and blood samples grew no organisms after 48 hours. Amputation was suggested at this time, but his parents refused below-the-knee amputation. He was maintained on NEC protocol, which includes adequate hydration and the initial triple drug regimens. Four days after admission, the skin and underlying soft tissues over the distal third of the right leg and foot sloughed off with the wound oozing seropurulent fluid. The distal medial tibia was exposed to the exterior around the ankle, and an extruded piece of bone was noted about the proximal tibia (Figure 2). On culture, the wound swab grew Proteus spp, Staphylococcus aureus, and Pseudomonas spp. Repeat radiographs showed changes of whole shaft infarction of the right tibia with whole shaft involucrum formation (Figure 3). The bones of the foot appeared unaffected. Serial x-rays over a 2-month period showed persistence of the tibial changes. Because the mother refused to discuss any form of amputation, the surgeons performed only debridement and limited sequestrectomy of the tibia. The clinical condition improved remarkably after debridement of the necrotic tissue. The child had a protracted stay in the hospital and was discharged 97 days after admission with residual deformities of the distal leg and ankle, with a view to 82

Figure 2. Observe the soft tissue loss in the medial right ankle with the tibia exteriorized; note the extruded piece of bone.

subsequent plastic surgery.

DISCUSSION On presentation, the patient had signs of ascending omphalitis and early infarction of the right leg. The omphalitis no doubt arose directly from the local practice of dressing the cord stump with a herbal concoction. The concoction has possible desiccant properties as the cord stump usually drops off about 2 days later. Osteomyelitis of the right leg was a consideration. The gangrenous involvement of the subcutaneous tissues were considered unusual for the typical osteomyelitis. Sequential radiographs showed transition of bone from normal to changes of infarction without an intervening phase of lytic destruction. Without the sequential radiographs, it may be impossible radiographically to differentiate osteitis from infarction. The lower limb changes were unusual, and because JOURNAL OF THE NATIONAL MEDICAL ASSOCIATION, VOL. 84, NO. 1

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of a recent relationship between neonatal omphalitis and NF in our hospital, NF was a major consideration. Necrotizing fasciitis by definition is a severe bacterial infection of the skin, subcutaneous tissues, superficial fascia, and deep fascia with edema, infarction, necrosis, and sloughing of the affected tissue.2-4 The extremities are commonly involved.2-4 However, bone infarction is not a component of this syndrome.4 Another entity that was considered in retrospect was PF. Purpura fulminans is a rare disorder seen in children and the elderly and is characterized by bleeding into the skin with bullous eruptions in the hemorrhagic areas, infarction of the skin, and occasionally peripheral gangrene.5-6 Disseminated intravascular coagulation is a usual feature, and the lesions tend to be bilaterally symmetrical and often involve the upper or lower extremities. Frequently, there is a history of recent bacterial or viral infection. Although a disease of children, neonates are rarely involved.6-9 Patients are usually very ill and present with hyperpyrexia and tachycardia; often, they are in shock. No infective organisms are cultured from infarcted tissues as infarction is a direct result of vaso-occlusive phenomenon.10 Mortality rates range from 18% to 52%.5,6,8 Several features in our patient are not typical for PE Our patient had unilateral limb involvement, and pathogenic organisms were cultured from the wound swab. Furthermore, there was no evidence of a bleeding disorder nor was the child significantly anemic. Both conditions are almost invariably present in PE Protein C deficiency, a feature that has been reported in patients with PF,6 was not sought for as PF was a retrospective consideration. We can only speculate that the bone infarction was a result of septic emboli occluding major vascular supply to the tibia. The patient had normal hemoglobin profile on electrophoresis, and we cannot provide an explanation for the unilateral selective limb involvement although Gurses and Ozkan7 reported a case of a neonate with a focal single digit involvement. Grogan et al12 have reported osteonecrosis associated with PF following meningococcemia. Based on the clinical worry for NEC and NF and the sensitivity pattern of the cultured organisms, the hydration of the patient was very rigorous while on triple parenteral antibiotic therapy of cloxacillin, gentamicin, and metronidazole. There is no doubt that amputation would have reduced the length of hospital stay. However, the parents were emphatic on their decision against amputation. Gurses and Ozkan7 recognized and stressed JOURNAL OF THE NATIONAL MEDICAL ASSOCIATION, VOL. 84, NO. 1

Figure 3. Exam through plaster cast showing destruction of the whole tibia.

the need for psychiatric support to the family in the planning of amputation for children. The relatively benign outcome of events indicate that we were not dealing with NF, which has a near uniformly fatal outcome in our environment,1 nor were we faced with the typical PF syndrome. Acknowledgments The authors thank Ms Gloria Sturm of the Department of Radiology, University of Wisconsin Hospital and Clinics for her secretarial assistance.

Literature Cited 1. Monu JUV, Okolo AA. Neonatal necrotizing fasciitis-a complication of poor cord hygiene: report of three cases. Ann Trop Paediatr 1990;10:299-303. 2. Lally KP, Atkinson JB, Wooley, et al. Necrotizing fasciitis; a serious sequela of omphalitis in the newborn. Ann Surg. 1984;199:101-103. 3. Kosloske AM, Cushing AH, Borden TA, Woodside JR, Klein MD, Kulasinghe HP, et al. Cellulitis and necrotizing fasciitis of the abdominal wall in pediatric patients. J Pediatr

83

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Surg. 1981;16:246-251. 4. Woodside JR. Necrotizing fasciitis after neonatal circumcision. Am J Dis Child. 1980;3:301. 5. Benson PM, Lupton GP, James WD. Purpura and gangrene in a septic patient. Arch Dermatol. 1988;1 24:1851. 6. Seagle MB, Bingham HG. Purpura fulminans. Ann Plast Surg. 1988;20:576-581. 7. Gurses N, Ozkan A. Neonatal and childhood purpura fulminans: review of seven cases. Cutis. 1988;41:361-363. 8. Hjort PF, Rappaport ST, Jorgensen L. Purpura fulminans. Scand J Haematol. 1964;1:169-170. 9. Van der Horst RC. Purpura fulminans in a newborn baby.

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Arch Dis Child. 1962;37:436-441. 10. Anderson CTM, Berde CG, Sethna NF, Pribaz JJ. Meningococcal purpura fulminans: treatment of vascular insufficiency in a 2-year-old child with lumbar epidural sympathetic blockade. Anesthesiology. 1989;71:463-464. 11. Ozsoylu S, Cengiz B, Karabent A. Purpura fulminans in a case of protein C deficiency. Eur J Pediatr. 1988;147:209210. 12. Grogan DP, Love SM, Ogden JA, Miller EA, Johnson LO. Chondro-osseous growth abnormalities after meningococcemia. A clinical and histopathological study. J Bone Joint Surg. 1989;71 A:920-931.

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JOURNAL OF THE NATIONAL MEDICAL ASSOCIATION, VOL. 84, NO. 1

Neonatal bone infarction following cord sepsis: case report.

An unusual case of infarction of the tibia in a neonate who presented with sepsis following poor cord management at birth is reported. He was managed ...
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