ARTICLE
Auteur(s) : Luca NEGOSANTI1,
Arianna ACETI2, Tommaso BIANCHI3, Luigi
CORVAGLIA2, Francesca NEGOSANTI4, Rossella
SGARZANI1, Paolo Giovanni MORSELLI1, Riccardo
CIPRIANI1, Massimino NEGOSANTI4, Annalisa
PATRIZI4, Giacomo FALDELLA2
1Unit of Plastic Surgery, S.Orsola-Malpighi Hospital,
Via Massarenti 9, 40138 Bologna, Italy
2Unit of Neonatology and Neonatal Intensive Care,
S.Orsola-Malpighi Hospital, Via Massarenti 9, 40138 Bologna,
Italy
3Unit of Dermatology, Bellaria-Maggiore Hospital,
Bologna, Italy
4Unit of Dermatology, S.Orsola-Malpighi Hospital, Via
Massarenti 9, 40138 Bologna, Italy
accepté le 15 F�vrier 2010
Necrotizing fasciitis (NF) is rare in paediatric patients. NF is
usually monomicrobial [1]: Staphylococcus Aureus is the most common
bacterial cause, however other bacteria, especially Gram-negative,
can also be responsible [2]. Predisposing factors include minor
injuries, surgical procedures and debilitating conditions, such as
immunosuppression. NF is a life-threatening condition, with an
overall neonatal mortality of approximately 59% [3], therefore
prompt diagnosis and treatment are mandatory to avoid fatal
complications. Medical care involves the administration of
appropriate antibiotics and intravenous immunoglobulins. Surgical
debridement of the entire necrotic area is mandatory [1]. The
repair of the resultant wound is difficult in newborns: surgical
reconstruction should be avoided, in our patient the use of topical
negative pressure achieved a good result.
Topical negative pressure therapy supports wound healing by
stimulating blood flow, the formation of granulation tissue and
angiogenesis. In addition, the negative pressure within the pores
of the polyurethane foam dressing contracts the wound and draws the
wound edges closer together [4]. Topical negative pressure has
proved to be effective in children and also in newborns [2, 5]; the
indications for using it in pediatric patients have been reported
by Baharestani [11].
Materials and methods
We report the case of a term female newborn (birth weight
2943 g, Apgar score 8 at 1’, 9 at 5’) admitted to
our Neonatal Intensive Care Unit immediately after birth due to the
presence of red-violaceous nodules on her whole body and severe
anaemia (Hb 9g/dL). The histology of the cutaneous lesions
demonstrated a localization of acute myeloid leukaemia with a
monoblastic differentiation negative for nucleophosmin. The
electron microscopy reported a proliferation of immature elements
belonging to the monocytoid line. The bone marrow film histology
was suggestive of monoblastic leukaemia (severe cellular depletion,
absent megacaryopoiesis, neutrophil granulopoiesis and
erythropoiesis, presence of large elements with large round nucleus
with nucleoli and basophilic cytoplasm; myeloperoxidase was
negative).
After the histological analysis of skin lesions and bone marrow,
acute monoblastic myeloid leukaemia was diagnosed. The patient
underwent a one-week treatment with Doxorubicin, Etoposid and
Cytarabine, according to the guidelines of the Italian Association
of Haematology and Pediatric Oncology.
When the patient was 20 days old, physical examination of
the perineal area showed erythema and haemorrhagic pustules which
rapidly progressed into necrotizing fasciitis. At 23 days,
physical examination of perineal area showed a lesion of
approximately 18 cm2 involving the superficial and
deep fascial planes around labia majora, a greenish secretion, a
black eschar with an erythematous halo and rectal prolapse. The
histology of the perianal lesion reported fascial necrosis,
vascular thrombosis and myonecrosis. Swap culture revealed the
presence of Pseudomonas aeruginosa, however, blood cultures were
negative.
The infant was initially treated with systemic administration
of: broad-spectrum antibiotics, immunoglobulins, inotropic drugs
and nutritional support. Antibiotic therapy was subsequently
modified according to the result of the swab culture (Meropenem,
Vancomycin, Tobramycin). The initial treatment of the wound
consisted of surgical debridement followed by the application of
advanced dressings, such as silver polyurethane foam and hydrofiber
(figure 1).
Due to the reduced healing rate and the persistence of
Pseudomonas colonies in the wound, a topical negative pressure
device (V.A.C.®, KCI, San Antonio, TX) with V.A.C.
GranuFoam Silver® dressing was applied directly on the
wound [6]. The entire V.A.C.® dressing was changed every
48 hours. During hospitalization at the Neonatal Intensive
Care Unit, the newborn was continuously monitored: pain was
assessed and opioid analgesia was used.
The positioning of the V.A.C.® dressing on the
perineal area was challenging, the main problems were: preventing
direct suction on the anal sphinter while maintaining normal
sphinteric functions and avoiding pressure sores due to the suction
tube. A double polyurethane film covering the foam on both
sides was applied, extending from left hypocondrium to the
perineum, to allow the negative pressure to be transferred from the
suction pad to the wound. To prevent direct suction on the anal
sphinteric, a custom made polyurethane foam ring tailored from an
advanced dressing (Biatain®, Coloplast DK) was placed
between the polyurethane sheets; this was necessary to keep sub
atmospheric pressure in the wound area while maintaining normal
sphinteric functions. The suction tube on the foam was not applied
directly on the wound, in order to avoid the occurrence of pressure
sores: it was placed in the left hypocondrium (where the skin was
protected by a polyurethane layer). Figure 2 shows a template
of V.A.C. dressing multilayer positioning. Topical negative
pressure was set at – 50 mmHg according to McCord [5] for
6 days and then raised to – 75 mmHg for seven days.
The rise in negative pressure was concurrant with an increase of
the C-reactive protein (CRP) value (from 1.09 to
5.07 mg/dL), however, local signs of inflammation were
reduced.
After 13 days of negative pressure treatment, the wound was
almost healed. The V.A.C. therapy was then stopped (figure 3); at that time,
the CRP had decreased to 1.13 mg/dL. All the microbiological
cultures performed in the perineal area were negative. Definitive
closure was achieved in 5 days of application of collagen
dressing (Condress, Abiogen, Pisa). After these treatments there
were no signs of NF and the swab culture was negative for
Pseudomonas aeruginosa. When the infant was 76 days old, she
was transferred from the Neonatal Intensive Care Unit to the
Onco-haematological Unit for the leukaemia follow up. She underwent
anorectoplasty to reconstruct her anal sphincter.
Discussion
This is a rare case of the simultaneous presence of acute myeloid
leukaemia and necrotizing fasciitis due to Pseudomonas aeruginosa
in a newborn, effectively treated with topical negative pressure
therapy.
Lo described another case of a newborn affected by AML who
developed an extensive NF caused by Pseudomonas aeruginosa located
in the left cheek. The infant was treated by surgical debridement,
fasciotomy, meticulous wound care and systemic administration of
antibiotics and GM-CSF [7]. Zuloaga-Salcedo reported a case of NF
due to Enterobacter cloacae and coagulase-negative Staphylococcus
in a full-term female newborn treated with negative pressure
therapy [8]. Other authors [9, 10] reported two cases of NF due to
Pseudomonas aeruginosa in paediatric patients affected by acute
leukaemia.
Topical negative pressure has been used since 1997 in adult
wound care practice and from 2000 in paediatric patients. The
indications for negative pressure therapy in paediatric and
neonatal wounds have been discussed by Baharestani, who described
his experience with pressure settings selection, ranging from
– 50 to – 100 mmHg [11].
In our experience, the use of topical negative pressure therapy
for neonatal NF allowed us to achieve rapid wound healing after
debridement. Other advanced dressings were not useful in our
patient. Interestingly, after the increase of suctioning pressure
from – 50 to – 75 mmHg, a higher CRP value was
observed, although local signs of inflammation had reduced. This
can be explained, as previously reported [12], by the fact that
topical negative pressure determines the local release of IL-6,
IL-8 and VEGF and IL-6 induces an increase in plasma CRP
concentration [13].
In conclusion, we strongly suggest that negative pressure
therapy should be considered in the treatment of NF in newborns
when conventional wound care fails to achieve complete wound
closure.
Acknowledgements
Financial support: none. Conflict of interest: none.
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