Case Report
Pheochromocytoma in Pregnancy
Kacy Church and Marina Basina*
Department of Endocrinology, Stanford University School of Medicine, USA
*Corresponding author: Julia Starlinger, Department of Trauma Surgery, Medical University Vienna, General Hospital Vienna, Währinger Gürtel 18-20, 1090 Vienna, Austria, Europe
Published: 19 May, 2017
Cite this article as: Starlinger J, Ringl H, Weissinger
M, Hajdu S. Surgical Debridement
Provided for Fast Recovery in
Metaepiphyseal Haematogenous
Osteomyelitis with Adjacent
Subperiostal Abscess Formation in Two
Children. Clin Surg. 2017; 2: 1475.
Abstract
Background: Primary metaepiphyseal haematogenous osteomyelitis (HOM) with subperiostal
abscess formation at initial admission is a rare but serious condition. A considerate treatment
strategy is crucial to avoid damage to the adjacent physis.
Methods: Charts and radiographic data of two cases were reviewed and reported in a retrospective
study approved by the Institutional Review Board.
Results: Two children presented with limping, painful restriction of movement and very
mild laboratory findings. No trauma was remembered. Conventional radiography showed
no abnormalities, but subsequent MRI revealed metaepiphyseal HOM as well as an adjacent
subperiostal abscess collection.
Conclusion: Antibiotic therapy and immediate surgical debridement provided for uncomplicated
healing.
Introduction
Primary metaepiphyseal HOM (haematogenous osteomyelitis) in children is rare and is
reported to occur acute, subacute or chronic [1-5]. Causes remain unclear but a history of a fall
and/or previous illnesses (e.g. angina, tooth infection) is discussed. Diagnosis is often delayed as
symptoms are rather unspecific (e.g., fever, weakness, joint effusions, malaise, local bone pain).
Clinical investigation many reveal no findings other than local tenderness and painful limping.
Laboratory findings often tell nothing more than an elevated ESR (erythrocyte sedimentation rate),
elevated levels of CRP (C-reactive protein) or abnormalities in white blood cell count.
Conventional X-rays might be regular at the early stages of HOM. Subsequently, MRI
is necessary to or confirms HOM or rule out other serious causes of bone pain, such as Ewing
sarcoma or Langerhans cell histiocytosis. In OM MRI shows bone marrow edema in the early
stages, a fluid collection within the bones as well as increased contrast uptake at the boarders of
the inflammation in an advanced OM. In rare cases of metaepiphyseal HOM a subperiostal abscess
formation is present at initial presentation. As the infection spreads through the medullar canal the
intramedullary pressure increases. Subsequently, infection spreads through Havers and Volkmann’s
canals progressing into the subperiostal space [6]. A subperiostal abscess lies between periost and
cortex and is adjacent to an acute or subacute OM. According to the pathophysiologic concept
by Dresing et al. [6], the physis is a barrier causing the abscess to spread to the periostal space.
As the abscess formation enlarges, ruptures of this collection into the adjacent soft tissue have
been reported [7]. Furthermore, a chronic course of a subperiostal formation in childhood OM is
reported to ultimately cause pathologic fractures [8].
No consensus exists whether metaepiphyseal HOM heals with conservative treatment alone.
Antibiotic therapy alone is reported to be sufficient in the early course of HOM [9-11] Ezra et al.
[11] treated eight patients with the same underlying pathology conservatively, and they noted no
difference in duration of treatment compared to Sorensen et al. [4]. In their opinion operative treatment is reserved for cases with persistent infection. In contrast Sorensen reported on three patients with primary epiphyseal HOM
who underwent curettage besides antibiotic administration recovered
without complications. Nevertheless, in cases of progressive OM
with adjacent subperiostal abscess formation, debridement is
recommended by some authors [12]. Complications after HOM are
acute bacterial bone and joint infections harming the adjacent physis
and ultimately resulting in concomitant growth disturbance [13].
Therefore, early diagnosis is crucial especially in HOM that comes
close to the physis to prevent long-term complications.
The aim of this study is to report our therapeutic approach
emphasizing surgical repair in seldom cases of a subperiostal abscess
formation in metaepiphyseal HOM.
Case Presentation
Case 1
An eight-year-old boy presented with pain and swelling of his
right wrist. Neither the boy nor his mother remembered a previous
trauma. About two weeks ago he suffered from tonsillitis and
bronchitis and therefore received antibiotic therapy. At admission
body temperature was elevated. Laboratory tests showed a CRP-level
of 9 mg/l and ASLO (Anti-streptolysin) titre of 433 U/ml. At control
after two days CRP was 10.7 mg/l. As pain did not subside under
antiphlogistic therapy a MRI scan was performed six days after initial
presentation (Figure 1).
Our diagnosis was a metaepiphyseal OM of the distal radius
and ulna with a subperiostal abscess formation adjacent to the distal
radius.
Case 2
A nine-year-old girl presented with painful swelling and redness
of the lateral malleolus. There was no history of a previous illness or a
memorable trauma. Plain radiographs at initial presentation showed
no pathologies. Three days later the ankle joint was livid, swollen and
painful (WCC 14.300/mm3, CRP 7.0 mg/dl, ASLO titre 1612 U/ml).With elevated body temperature (37.5°C) beginning erysipelas was
suspected. She underwent MRI of her ankle joint (Figure 2).
Our diagnosis was a primary metaepiphyseal OM of the distal
fibula with an adjacent subperiostal collection.
Treatment
In an effort to limit harm to the physis as well as further joint
involvement the abscess formations were treated operatively.
Drainage and abscess lavage with normal saline solution was
performed. Further drainage of the metaepiphysis by two drill holes
was performed to release the intramedullary pressure. In case one
an antibiotic-instilling drainage was in use for five days post OP. In
case two the wound was closed with a modified Manuvac drain which
was retained for two days. Postoperatively a cast immobilization was
applied until removal of the stitches.
Cultures of the curetted material were positive for staphylococcus
aureus. Both children were given intravenous antibiotics (Augmentin
3 g per day) for eight days. Oral antibiotic therapy was continued for
further six weeks.
At follow-up both children were pain free and had no limitation
in range of motion.
Figure 1
Figure 1
Eight-year-old boy with subperiostal abscess in the distal radius.
A: Axial T1 weighted sequence showing the subperiostal abscess. The pus
collection is hypointense (asterix). Arrows mark the abscess boarders.
B: Coronal STIR sequence of the distal forearm: The subperiostal abscess
is hyperintens (bright) and marked with the asterix and located in the
subperiostal layer with a maximum diameter of 2,5 cm. Arrows point at the
abscess border. Note the marked edema in the distal radius induced by the
initial OM. Slight edema in the epiphysis of the ulna.
C: Axial STIR sequence of the distal forearm: The subperiostal abscess
is hyperintens (bright) and marked with the asterix and located in the
subperiostal layer with a maximum diameter of 2,5 cm. Arrows point at the
abscess border.
Figure 2
Figure 2
Nine-year-old girl with subperiostal abscess in the distal fibula.
A: Coronal STIR sequence (heavily T2 weighted sequence with fat
suppression): Arrows show the subperiostal fluid collection in the distal fibula
diaphysis without interruption of the periost. The Asterisk is located in the
bone marrow of the distal fibula that shows increased signal intensity as a
sign of reactive bone marrow edema.
B: Axial T1 weighted sequence with fat suppression after the injection of
gadolinium. Arrows point at the contrast enhancing abscess border.
C: Axial T2 weighted sequence. The arrows indicate the periostal layer that
is separated from the cortical bone and also represents the abscess border.
The asterisk is located within the abscess with a diameter of 12 mm.
Discussion
Metaepiphyseal HOM is a rare but typical childhood condition
after a history of bacteremia (e.g. common cold). Metaepiphyseal
HOM occurs in an acute, subacute or chronic form. Most of the
cases are classified subacute due to the subtle clinical findings and
the delayed diagnosing. Like as in case 1, antibiotic treatment during
the course of tonsillitis might have masked the early symptoms of
the osseous infection. Besides bacteremia a trauma to the physis
might be directly related to the pathogenesis. A fall or minor trauma
several days ago is not always to be remembered. These causal links
were nicely demonstrated by Morrissy et al. who established a rabbit
tibial trauma model regarding the development of OM. The model
consisted of 3 groups, the first two groups had bacteremia or trauma
alone and hardly any developed OM. But in group three, the rabbits
that had both, bacteremia and a trauma and developed significant
OM [14].
It is unclear, whether the lesions in both cases are truly
metaepiphyseal or started as metaphyseal lesions then evolving to an
epiphyseal lesion. In both of our cases, the sudden onset of symptoms
supports the hypothesis of a true primary metaepiphyseal infection.
Of note the physis reflects a barrier for fluid and pus. The periosteum
is loosely attached to the cortex and as the intramedullary pressure
increases a subperiostal collection develops [15,16].
At initial presentation conventional X-rays should be acquired
although osseous destruction is reported to be invisible until 7 to 14
days after the onset of the symptoms [6,16]. Correspondingly plain
radiographs of our patients showed no evidence of a lytic lesion or
periostal reaction. Still, Ezra et al. [11] suggest that carful assessment of
plain radiographs alone can reveal the benign features of most lesions
with a high degree of reliability thereby making additional imaging
redundant. But considerable subperiostal collections, as found in
our two patients might then be missed and treatment prolonged.
Nevertheless, MRI is reported to be an accurate tool to identify OM
[17]. Moreover, MRI shows the extent of bone involvement and is
thereby a crucial tool identify cases that profit from surgical repair
especially in patients with subperiostal abscesses and soft tissue
collections.
MRI should be obtained with a T1 and T2 weighted sequence
without fat suppression. In addition a STIR sequence as well as a
T1 sequence after the injection of gadolinium should be performed.
The T1 sequence shows good anatomic details as well as the fat
content of the bone marrow. The STIR sequence is most sensitive for
bone marrow edema and fluid collection and the T1 sequence after
injection of gadolinium with fat suppression perfectly delineates the
boarder of the inflammation.
If MRI is not immediately available ultrasound is useful to detect
a subperiostal abscess formation [15]. Ezra et al. [11] recommended
ultrasound as an available, noninvasive method. To our opinion
ultrasound provides only limited information regarding the extent
of the OM. Nevertheless, ultrasound is reported to be useful for
needle aspiration of a subperiostal abscess formation in selected cases
[15,16].
In all cases of HOM material for bacterial culture has to be
obtained by aspirate, intraoperative specimen or blood culture.
Cultures of aspirated material are reported to be negative in up to
75% [18,19]. But even in the absence of positive culture patients with
epiphyseal OM improve during the course of antibiotic treatment
[18]. Therefore, antibiotic regimen has to be started at the onset
of symptoms targeting Staphylococcus aureus, which accounts for
approximately 50%-60% of AHMO [7,18,20].
Current literature is inconclusive whether a metaepiphyseal HOM
with abscess formation heals with AB treatment alone or requires
surgical repair. In our opinion antibiotics alone may be sufficient in
metaepiphyseal HOM in the absence of a periostal collection. But a
subperiostal formation is a result of high intramedullary pressure
levels. Still, no threshold is defined at which size a surgical drainage
is indicated. Howard et al. [21] for example suggested a subperiostal
formation of 2 mm to be crucial for surgical debridement.
Nevertheless, uneventful recovery after spontaneous rupture of a
subperiostal abscess collection is reported by Mah et al. [7] Of note,
the authors at the same time consider surgery as to allow for quicker
recovery.
Besides presence of a subperiostal abscess formation, our decision
to perform surgery was based on the MRI findings regarding the
physeal affection. Two drill holes thereby released the intramedullary
pressure and limited the progression of the infection. However, no
general recommendation can be made due to the small number of
patients.
References
- Andrew TA, Porter K. Primary subacute epiphyseal osteomyelitis: a report of three cases. J Pediatr Orthop. 1985;5(2):155-57.
- Longjohn DB, Zionts LE, Stott NS. Acute hematogenous osteomyelitis of the epiphysis. Clin Orthop Relat Res. 1995;316:227-34.
- Kramer SJ, Post J, Sussman M. Acute hematogenous osteomyelitis of the epiphysis. J Pediatr Orthop. 1986;6(4):493-95.
- Sorensen TS, Hedeboe J, Christensen ER. Primary epiphyseal osteomyelitis in children. Report of three cases and review of the literature. J Bone Joint Surg Br. 1988;70(5):818-20.
- Allan C, Huntley JS. Acute distal radial epiphyseal osteomyelitis: a case report. Scott Med J. 2013; 58(2):e27-30.
- Dresing K, Sturmer KM. Diagnosis and therapy of pediatric and juvenile hematogenous osteomyelitis. Unfallchirurg. 1998;101(9):662-73.
- Mah ET, LeQuesne GW, Gent RJ, Paterson DC. Ultrasonic features of acute osteomyelitis in children. J Bone Joint Surg Br. 1994;76(6):969-74.
- Belthur MV, Birchansky SB, Verdugo AA, Mason EO Jr, Hulten KG, Kaplan SL, et al. Pathologic fractures in children with acute Staphylococcus aureus osteomyelitis. J Bone Joint Surg Am. 2012;94(1):34-42.
- Hamdy RC, Lawton L, Carey T, Wiley J, Marton D. Subacute hematogenous osteomyelitis: are biopsy and surgery always indicated? J Pediatr Orthop. 1996;16(2):220-23.
- Pääkkönen M, Kallio PE, Kallio MJ, Peltola H. Management of osteoarticular infections caused by Staphylococcus aureus is similar to that of other etiologies: analysis of 199 staphylococcal bone and joint infections. Pediatr Infect Dis J. 2012;31(5):436-38.
- Ezra E, Cohen N, Segev E, Hayek S, Lokiec F, Wientroub S, et al. Primary subacute epiphyseal osteomyelitis: role of conservative treatment. J Pediatr Orthop. 2002;22(3):333-37.
- Niethard F, Pfeil J, Bieberthaler P. Orthopädie und Unfallchirurgie.Stuttgart, Germany: Thieme; 2009;154(10):237-44.
- Paakkonen M, Peltola H. Bone and joint infections. Pediatr Clin North Am. 2013;60(2):425-36.
- Morrissy RT, Haynes DW. Acute hematogenous osteomyelitis: a model with trauma as an etiology. J Pediatr Orthop. 1989;9(4):447-56.
- Kaiser S, Rosenborg M. Early detection of subperiosteal abscesses by ultrasonography. A means for further successful treatment in pediatric osteomyelitis. Pediatr Radiol. 1994;24(5):336-39.
- Dietz HG, Bachmeyr A, Joppich I. [Osteomyelitis in children]. Orthopade. 2004;33(3):287-96.
- Totty WG. Radiographic evaluation of osteomyelitis using magnetic resonance imaging. Orthop Rev. 1989;18(5):587-92.
- Green NE, Beauchamp RD, Griffin PP. Primary subacute epiphyseal osteomyelitis. J Bone Joint Surg Am. 1981;63(1):107-14.
- Pöyhiä T, Azouz EM. MR imaging evaluation of subacute and chronic bone abscesses in children. Pediatr Radiol. 2000;30(11):763-8.
- Hempfing A, Placzek R, Göttsche T, Meiss AL. Primary subacute epiphyseal and metaepiphyseal osteomyelitis in children. Diagnosis and treatment guided by MRI. J Bone Joint Surg Br. 2003;85(4):559-64.
- Howard CB, Einhorn M, Dagan R, Nyska M. Ultrasound in diagnosis and management of acute haematogenous osteomyelitis in children. J Bone Joint Surg Br. 1993;75(1):79-82.