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Mesenteric hernia in VLBW infants intestine, suggests that strangulation of the bowel itself is not the only cause of ischemic changes in internal hernias. Pressure of the herniated bowel loops and their mesentery compresses the vessels in the free margins of the defect and may cause ischemic changes. This pathological condition may be similar to that observed in the testicle at the time of the intestinal incarceration of inguinal hernia even when the intestine is not necrotic. The delay of diagnosis of organic intestinal obstruction leads to a fatal outcome in VLBW infants, although such infants also tend to develop functional ileus related to meconium plugs.1 We should consider the possibility of an internal hernia when VLBW infants present with acute intestinal obstruction. The usefulness of computed tomography of internal hernia has been reported,10 although whether it can be applied to VLBW infants is controversial. A suspicion of possible rare conditions causing intestinal obstruction and surgical intervention in appropriate timeframe are mandatory to rescue VLBW infants with a strangulated mesenteric hernia.

Acknowledgments The authors are grateful to all the children and parents who participated in this report. The authors also thank the medical staff working in the neonatal intensive care unit at Osaka Medical Center and Research Institute for Maternal and Child Health.

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References 1 Garza-Cox S, Keeney SE, Angel CA, Thompson LL, Swischuk LE. Meconium obstruction in the very low birth weight premature infant. Pediatrics 2004; 114: 285–90. 2 Malit M, Burjonrappa S. Congenital mesenteric defect: Description of a rare cause of distal intestinal obstruction in a neonate. Int. J. Surg. Case Rep. 2012; 3: 121–3. 3 Oretti C, Bussani R, Janes A, Demarini S. Multiple segmental absence of intestinal musculature presenting as spontaneous isolated perforation in an extremely low-birth-weight infant. J. Pediatr. Surg. 2010; 45: E25–7. 4 Shaffner Lde S, Pennell TC. Congenital internal hernia. Surg. Clin. North Am. 1971; 51: 1355–9. 5 Page MP, Ricca RL, Resnick AS, Puder M, Fishman SJ. Newborn and toddler intestinal obstruction owing to congenital mesenteric defects. J. Pediatr. Surg. 2008; 43: 755–8. 6 Tang V, Daneman A, Navarro OM, Miller SF, Gerstle JT. Internal hernias in children: Spectrum of clinical and imaging findings. Pediatr. Radiol. 2011; 41: 1559–68. 7 Ming YC, Chao HC, Luo CC. Congenital mesenteric hernia causing intestinal obstruction in children. Eur. J. Pediatr. 2007; 166: 1045–7. 8 Garignon C, Paparel P, Liloku R, Lansiaux S, Basset T. Mesenteric hernia: A rare cause of intestinal obstruction in children. J. Pediatr. Surg. 2002; 37: 1493–4. 9 Fujita A, Takaya J, Takada K et al. Transmesenteric hernia: Report of two patients with diagnostic emphasis on plain abdominal X-ray findings. Eur. J. Pediatr. 2003; 162: 147–9. 10 Blachar A, Federle MP, Dodson SF. Internal hernia: Clinical and imaging findings in 17 patients with emphasis on CT criteria. Radiology 2001; 218: 68–74.

Pott’s puffy tumor in a 12-year-old boy Gülhadiye Avcu,1 Nursen Belet,1 Senem Cengel Kurnaz,2 Arzu Karli1 and Gülnar Sensoy1 Pediatric Infectious Disease and 2Otorhinolaryngology, Ondokuz Mayis University, Samsun, Turkey

1

Abstract

Pott’s puffy tumor (PPT) is a rare complication of sinusitis characterized by subperiosteal abscess and osteomyelitis of the frontal bone. Early diagnosis and treatment is vital before it causes intracranial complications such as subdural empyema or brain abscess. Herein we describe the case of a 12-year-old patient who developed preseptal cellulitis and PPT, and was successfully treated with abscess drainage, sinus surgery and long-term antibiotic therapy.

Key words frontal osteomyelitis, Pott’s puffy tumor, sinusitis.

Pott’s puffy tumor (PPT) is defined as subperiosteal abscess accompanied by osteomyelitis after frontal sinusitis.1 PPT was first described by Sir Percivall Pott in 1775. It may develop after a complication of acute or chronic sinusitis, trauma or insect bite rarely.1 The most common symptoms are headache, periorbital edema, fever, vomiting, lethargy and swelling in the Correspondence: Gülhadiye Avcu, MD, Pediatric Infectious Disease, Ondokuz Mayıs University, Kurupelit/Samsun, Samsun, Turkey. Email: [email protected] Received 26 February 2014; revised 28 April 2014; accepted 30 April 2014. doi: 10.1111/ped.12440

frontal region.2 PPT is more common in adolescents but it can also be seen in elderly people.2 PPT is a risk factor for intracranial complications such as subdural empyema and brain abscess, and intracranial complications are more common in children than adults. Intracranial complications have been reported in 60–85% of all PPT cases.3 Early diagnosis and treatment are vital for the prevention of serious complications. In this paper, we describe the case of a 12-year-old male patient who was hospitalized with a pre-diagnosis of preseptal cellulitis, which was then changed to a diagnosis of PPT on follow up, to emphasize the importance of PPT, a rare complication of sinusitis, in differential diagnosis. © 2015 Japan Pediatric Society

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G Avcu et al.

a

Fig. 1 Swelling over the forehead.

b

Case report A 12-year-old male patient presented to hospital with the complaints of swelling, erythema and pain around his left eye. He had had watery eye (in the left eye) accompanied by fever for 3 days and had received amoxicillin clavulanic acid due to the diagnosis of upper respiratory tract infection. The patient, who experienced sudden swelling in the left eyelid, was hospitalized with a prediagnosis of preseptal cellulitis. On physical examination, bodyweight was 45 kg (25–50th percentile), height 155 cm (50th percentile), heart rate 110 beats/min, blood pressure 100/ 60 mmHg, and body temperature 37.7°C. Swelling, erythema and tenderness were present in the left periorbital region, but there was no blurring or chemosis in the eyes. Eye movement was normal, there was no proptosis or decreased visual acuity. The patient had nasal congestion. Oropharyngeal and other system examinations were normal. On laboratory evaluation, hemoglobin was 13.3 g/dL, white blood cell count 18 940/mm3, platelets 328 000/mm3, erythrocyte sedimentation rate 90 mm/h, and C-reactive protein 174 mg/L. On peripheral blood smear there was an 87% leukocyte dominance with polymorphic nuclei. Biochemistry was within normal limits. Ceftriaxone treatment was started. Non-hyperemic but fluctuating swelling (1 × 1 cm) and palpation sensitivity were observed in the left frontal region on the third day of hospitalization (Fig. 1). Cranial and orbital magnetic resonance imaging (MRI) showed mucosal thickening in the left frontal, ethmoidal and maxillary sinus with subperiosteal abscess in the left frontal region (Fig 2). Vancomycin and metronidazole were added to the ceftriaxone treatment. The patient underwent external subperiosteal abscess drainage and endo© 2015 Japan Pediatric Society

Fig. 2 (a) Cranial and (b) orbital magnetic resonance imaging showing the subperiosteal abscess collection and left frontal sinusitis.

scopic sinus surgery by the otolaryngologist on the seventh day of hospitalization. Exudate samples obtained during the procedure were not sent to culture. No production occurred in the blood culture. The patient had significant clinical improvement, and cranial MRI performed on the 18th day of hospitalization showed regression in the previously described findings. No

Pott’s puffy tumor in a 12-year-old boy serious complications occurred. After 3 weeks of i.v. antibiotics, the patient was discharged from hospital and the treatment was completed to a total of 8 weeks with amoxicillin clavulanic acid. No additional problems were observed on 2 month outpatient follow up.

Discussion Pott’s puffy tumor was first described by Sir Percivall Pott in 1775. It is usually characterized by complications of frontal sinusitis or subperiosteal abscess due to direct trauma to the frontal bone.1 It has also been reported after insect bite, albeit rarely.1 Because aeration of the frontal sinus starts at around the age of 6 and completes its development at the age of 15, adolescents are more inclined to this disease, but PPT can also be detected in younger age groups.4 Parida et al. reported that PPT (a total of five cases in 1.5 years) was observed in patients aged between 9 and 15.5 Of these cases, (except for one patient with a history of trauma) four patients had acute or chronic sinusitis in the etiology, one of whom had acute or chronic sinusitis accompanied by preseptal cellulitis. The most common symptoms are headache, swelling in the periorbital or scalp regions, fever, and purulent or non-purulent nasal discharge. Fluctuant swelling in the midline of the forehead is typical. Skin–soft-tissue infection, hematoma, benign and malignant tumors of the skin, soft tissue, bones and frontal sinus should be considered in the differential diagnosis of swelling in the forehead. In the Akiyama et al. study involving 32 adults, 55.6% of cases were diagnosed as PPT on admission whereas 44.4% were misdiagnosed with epidermoid cyst, cellulitis.6 In the present case, the patient presented to hospital with the complaints of headache, periorbital swelling and fever, and the pre-diagnosis of preseptal cellulitis was established initially. On follow up, the patient developed sensitive fluctuating swelling in the frontal region suggesting PPT, and imaging supported this diagnosis. Frontal sinus infection may lead to osteomyelitis and subperiosteal and epidural abscess. As a result of subdural dissemination, subdural collection or cerebritis may develop.2 Sinus thrombosis, subdural empyema or brain abscess may also develop as a result of hematogenous dissemination.7 In patients with suspected PPT, imaging (computed tomography or magnetic resonance imaging) should be done to determine the intracranial complications (epidural, subdural abscess, brain abscess). The complication rate has been reported to be 60%.8 The most common microorganisms are Streptococcus species such as S. milleri and S.viridans. S. pyogenes, S. pneumoniae or Staphylococcus aureus and anaerobic microorganisms such as Bacteriodes and Fusobacterium, rarely, lead to infection as well. Most of the infections are polymicrobial, and anaerobic microorganisms are dominant in intracranial complications.7 PPT caused by Haemophilus influenzae has also been reported.7 In the present case, a small amount of material was obtained via abscess drainage but the sample was not sent to culture. Broad-spectrum i.v. antibiotics, analgesics, systemic and topical decongestants are required in the management of PPT. If

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clinical improvement is not achieved, surgical intervention should be considered. The patients may not respond to medical treatment in the presence of nasal polyps or mucosal edema.9 Surgical intervention can be performed externally or endoscopically or in combination for the cleaning of the sinus from infection, subperiosteal abscess drainage or removal of the sequestrum.5 After surgery, long-term antibiotic therapy for osteomyelitis (at least 6–8 weeks) is needed. The mortality rate of frontal osteomyelitis before introduction of broad-spectrum antibiotic has been reported to be 60%.10 Parida et al. reported that all patients underwent surgical intervention, and that treatment with broad-spectrum antibiotics continued for 6–8 weeks.5 The following antibiotics were used: ceftriaxone, crystalline penicillin and metronidazole and vancomycin in one case of methicillin-resistant S. aureus, and ceftazidime in one case of Pseudomonas aeruginosa. None of the patients experienced intracranial complications. Upon detection of PPT in the present case, we revised the use of antibiotics so as to involve resistant Streptococcus, Staphylococcus species and anaerobic factors (i.v. ceftriaxone, vancomycin, metronidazole). Sinus infection was removed via surgical intervention, subperiosteal abscess was drained and thus more serious intracranial complications could be avoided. Conclusion

Given that it may lead to serious complications, the early diagnosis and treatment of PPT are emphasized, and it should be borne in mind when treating patients with a history of trauma and sinusitis who present with headache, fever and swelling in the frontal region.

References 1 Babu RP, Todor R, Kasoff SS. Pott’s puffy tumor : The forgotten entity. Case report. J. Neurosurg. 1996; 84: 110–12. 2 Karaman E, Hacizade Y, Isildak H et al. Pott’s puffy tumor. J. Craniofac. Surg. 2008; 19: 1694–7. 3 Ketenci I, Unlu Y, Tucer B et al. Pott’s puffy tumor: A dangerous sign for intracranial complications. Eur. Arch. Otorhinolaryngol. 2011; 268: 1755–63. 4 Rogo T, Schwartz RH. Pott’s puffy tumor in a 5-year-old girl with frontal sinusitis. Ear Nose Throat J. 2013; 92: 24–6. 5 Parida PK, Surianarayanan G, Ganeshan S, Saxena SK. Pott’s puffy tumor in pediatric age group: A retrospective study. Int. J. Pediatr. Otorhinolaryngol. 2012; 76: 1274–7. 6 Akiyama K, Karaki M, Mori N. Evaluation of adult Pott’s puffy tumor. Our five cases and 27 literature cases. Laryngoscope 2012; 122: 2382–8. 7 Verbon A, Husni RN, Gordon SM, Lavertu P, Keys TF. Pott’s puffy tumor due to Haemophilus influenzae: Case report and review. Infect. Dis. 1996; 23: 1305–7. 8 Holder J, Corbin D, Marquez S, Clarke H, Walcott J, Thomas R. Pott’s puffy tumor and subdural empyema following frontal sinusitis. West Indian Med. J. 1991; 40: 33–6. 9 Jung J, Lee HC, Park IH, Lee HM. Endoscopic endonasal treatment of a Pott’s puffy tumor. Clin. Exp. Otorhinolaryngol. 2012; 5: 112–15. 10 Bordley JE, Bischofberger W. Osteomyelitis of the frontal bone. Laryngoscope 1967; 77: 1234–44.

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Pott's puffy tumor in a 12-year-old boy.

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