Ecthyma Gangrenosum in a Previously Healthy Infant · Kim KJ, Park BI, Kim YB, et al ... C-reactive...
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Vol. 39 / No. 6 / November 2012 673 Ecthyma gangrenosum (EG) is a rare but charac- teristic cutaneous and potentially fatal disorder caused by a pseudomonal infection. e characteri- stic cutaneous lesion progresses sequentially, and starting as a maculopapular eruption, advancing to a hemorrhagic bulla, and then evolving into a gangre- nous ulceration with black eschar surrounded by an erythema [1]. EG usually occurs in immunocom- promised patients, but occasionally it affects previ- ously healthy individuals. We present a case of a 10- month-old previously healthy infant with transient severe neutropenia caused by influenza B infection without bacteremia. A previously healthy 10-month-old boy was ad- mied with a 5-day history of fever, general weakness, and a red spot on the right buock. e red spot had started as a small papule, aſter a perineal diaper rash disappeared. Initially, it was thought to be part of the diaper rash, but rapidly progressed to a bulla and finally to a necrotic black eschar with surrounding erythema within only 24 hours, but without suppu- ration (Fig. 1). An initial blood test revealed leukopenia (4,000/ μL; normal range, 6,000 to 17,500/μL) with severe neutropenia (absolute neutrophil count, 320/μL; normal range, 3,150 to 6,200/μL), lymphocytosis, and monocytosis. His inflammatory markers were elevated: C-reactive protein 13.9 mg/dL (normal range, 0.08 to 1.12 mg/dL) and erythrocyte sedimen- tation rate 35 mm/hr (normal range, 0 to 9 mm/hr). All other results were in the normal range. However, a nasal smear test revealed positivity for influenza B. At the time of admission, the child was initially given intravenous third-generation cephalosporin for broad spectrum coverage, but the cutaneous lesion continued to progress. A wound culture grew Pseudo- Ecthyma Gangrenosum in a Previously Healthy Infant Su Han Koo 1 , Joon Ho Lee 1 , Heakyeong Shin 1 , Jong Im Lee 2 Departments of 1 Plastic and Reconstructive Surgery, and 2 Pathology, Dongguk University College of Medicine, Gyeongju, Korea Correspondence: Heakyeong Shin Department of Plastic and Reconstructive Surgery, Dongguk University College of Medicine, 87 Dongdae-ro, Gyeongju 780-350, Korea Tel: +82-54-770-8242, Fax: +82-54-770-8501 E-mail: [email protected] This article was presented as a poster at the Second Research and Reconstructive Forum of the Korean Society of Plastic and Reconstructive Surgeons on June 1-2, 2012 in Gwangju, Korea. No potential conflict of interest relevant to this article was reported. Received: 31 Aug 2012 • Revised: 26 Sep 2012 • Accepted: 26 Sep 2012 pISSN: 2234-6163 • eISSN: 2234-6171 http://dx.doi.org/10.5999/aps.2012.39.6.673 • Arch Plast Surg 2012;39:673-675 Copyright 2012 The Korean Society of Plastic and Reconstructive Surgeons This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Images Fig. 1. Cutaneous lesion. Typical appearance of ecthyma gangrenosum on the right buttock.
Transcript of Ecthyma Gangrenosum in a Previously Healthy Infant · Kim KJ, Park BI, Kim YB, et al ... C-reactive...
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after CO2 laser treatment with staged excisions and covered with local advancement flaps, but the patient was satisfied with the cosmetic results and there was no recurrence. Consequently, physicians should take note that insufficient treatment, including CO2 laser or any surgical excision, can cause recurrence, as in this case. We suggest that staged excision can be another treatment modality for large multiple type NLCS. References
1. Jones EW, Marks R, Pongsehirun D. Naevus superficialis lipomatosus. A clinicopathological report of twenty cases. Br J Dermatol 1975;93:121-33.
2. Hattori R, Kubo T, Yano K, et al. Nevus lipomatosus cutaneous superficialis of the clitoris. Dermatol Surg 2003;29:1071-2.
3. Kim KJ, Park BI, Kim YB, et al. Clinical experience of nevus lipomatosus cutaneous superficialis. J Korean Soc Plast reconstr Surg 1994;21:360-4.
4. Jung SJ, Kim HJ, Seo YJ, et al. A case of nevus lipomatosus cutaneous superficialis treated by CO2 laser. Korean J Dermatol 2007;45:1093-5.
5. Knuttel R, Silver EA. A cerebriform mass on the right buttock. Dermatol Surg 2003;29:780-1.
Ecthyma gangrenosum (EG) is a rare but charac- teristic cutaneous and potentially fatal disorder caused by a pseudomonal infection. The characteri- stic cutaneous lesion progresses sequentially, and starting as a maculopapular eruption, advancing to a hemorrhagic bulla, and then evolving into a gangre- nous ulceration with black eschar surrounded by an erythema [1]. EG usually occurs in immunocom- promised patients, but occasionally it affects previ- ously healthy individuals. We present a case of a 10- month-old previously healthy infant with transient severe neutropenia caused by influenza B infection without bacteremia. A previously healthy 10-month-old boy was ad- mitted with a 5-day history of fever, general weakness, and a red spot on the right buttock. The red spot had started as a small papule, after a perineal diaper rash disappeared. Initially, it was thought to be part of the diaper rash, but rapidly progressed to a bulla and finally to a necrotic black eschar with surrounding erythema within only 24 hours, but without suppu- ration (Fig. 1). An initial blood test revealed leukopenia (4,000/μL; normal range, 6,000 to 17,500/μL) with severe neutropenia (absolute neutrophil count, 320/μL; normal range, 3,150 to 6,200/μL), lymphocytosis, and monocytosis. His inflammatory markers were elevated: C-reactive protein 13.9 mg/dL (normal range, 0.08 to 1.12 mg/dL) and erythrocyte sedimen- tation rate 35 mm/hr (normal range, 0 to 9 mm/hr). All other results were in the normal range. However, a nasal smear test revealed positivity for influenza B. At the time of admission, the child was initially given intravenous third-generation cephalosporin for broad spectrum coverage, but the cutaneous lesion continued to progress. A wound culture grew Pseudo-
Ecthyma Gangrenosum in a Previously Healthy InfantSu Han Koo1, Joon Ho Lee1, Heakyeong Shin1, Jong Im Lee2
Departments of 1Plastic and Reconstructive Surgery, and 2Pathology, Dongguk University College of Medicine, Gyeongju, Korea
Correspondence: Heakyeong ShinDepartment of Plastic and Reconstructive Surgery, Dongguk University College of Medicine, 87 Dongdae-ro, Gyeongju 780-350, KoreaTel: +82-54-770-8242, Fax: +82-54-770-8501 E-mail: [email protected]
This article was presented as a poster at the Second Research and Reconstructive Forum of the Korean Society of Plastic and Reconstructive Surgeons on June 1-2, 2012 in Gwangju, Korea.
No potential conflict of interest relevant to this article was reported.
Received: 31 Aug 2012 • Revised: 26 Sep 2012 • Accepted: 26 Sep 2012pISSN: 2234-6163 • eISSN: 2234-6171http://dx.doi.org/10.5999/aps.2012.39.6.673 • Arch Plast Surg 2012;39:673-675
Copyright 2012 The Korean Society of Plastic and Reconstructive SurgeonsThis is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Images
Fig. 1. Cutaneous lesion. Typical appearance of ecthyma gangrenosum on the right buttock.
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monas aeruginosa 2 days later, but repeated blood cultures grew nothing. However, a skin biopsy showed a necrotizing small vessel with thrombus surrounded by bluish bacterial clumps (Fig. 2). In light of the wound culture result, intravenous antibiotics were changed to piperacillin plus tazobactam, and ceftazi- dime was started additively. After changing antibiotics, the buttock lesion rapidly improved. Initially the erythema around the eschar faded and the central necrosis stopped enlarging, and remained at a size of about 2.5 cm in diameter (Fig. 3). Surgical repair was planned when the lesion became stable. A local flap using an intergluteal fold line was performed for wound coverage (Fig. 4). The flap sur- vived without complication and the wound healed well. In general, EG is pathognomonic for Pseudomonas septicemia, but rarely, it occurs by direct skin inocu- lation without bacteremia [1]. In our case, a single lesion, a previous history of diaper rash and the absence of bacteremia suggest the latter etiology. Lesions progress in a characteristic sequential
manner. They start as papules and advance to hemo- rrhagic bullae, and then evolve into gangrenous ulce- rations with black eschar surrounded by an erythe- matous halo [1]. EG can be distinguished from pyo- derma gangrenosum by a greater number of suppu- rative lesions and no bacterial pathogen in cultures, and can also be distinguished from common cellulitis by the characteristic sequential pattern and the speed of disease progression, which might be a key to its early recognition and treatment. Lesions can occur anywhere, but are more common in the perineum, buttocks, axillae, and extremities [2]. The histopathologic appearance of EG is a vasculitis caused by bacterial invasion of themedia and adven- titia of the vascular wall, sparing the intima. The intra- luminal thrombus causing locoregional necrosis is caused by bacilli in the vessel walls, by circulating immune complexes, and/or by the effect of bacterial exotoxins or endotoxins [2]. This feature explains the rapid progression of the lesions to necrosis. In our case, a necrotizing small vessel with intraluminal
Fig. 3. After changing to anti-pseudomonal antibiotics. The improvement in
surrounding erythema.
Fig. 2. Histological finding. A necrotizing small vessel with thrombus
surrounded by bluish bacterial clumps (H&E, ×400).
Fig. 4. Postoperative view. (A) Immediate postoperative view, and
(B) View at 1 month postoperatively.A B
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thrombus surrounded by bluish bacterial clumps was observed (Fig. 2). Although EG occurs in 1% to 30% of cases of Pseudomonas sepsis as a first manifestation of immu- nodeficiency or in patients with chronic disease, cases have occasionally been reported in previously healthy individuals. These individuals had undetected immu- nodeficiency or were transiently immunocompromised due to drugs, infections, or some other cause. Thus, an immunological work up should be performed in suspected cases [3]. In our patient, influenza B infection caused severe neutropenia and a previous diaper rash might have aided bacterial inoculation. A subsequent immunological work up established that the infant had a normal immune system. In cases of EG, early recognition and the admi- nistration of prompt appropriate antibiotics is essen- tial due to rapid disease progression. Antibiotics including ceftazidime (alone), azlocillin, mezlocillin or piperacillin-tazobactam with aminoglycosides are known to be effective [4]. Furthermore, in several of the reported cases, after controlling the infection, wounds healed by secondary intention left notable scars. Mortality rates of from 38% to 96% have been reported in septicemic cases and of 15% in non-bacteremic cases, respectively [5]. Poor prognosis is associated with multiple lesions, delay in treatment, and neutropenia. In conclusion, this case shows that even a previ- ously healthy patient in a temporarily immunocom- promised state may be susceptible to pseudomonal infections, which cautions that the possibility of opportunistic infections warrants consideration. The characteristic clinical features of the skin lesion led to a differential diagnosis of EG. Furthermore, the rapid progression of the lesions and EG’s high mortality rate emphasize the importance of early suspicion and proper treatment even when diagnosis has not been confirmed. References
1. Boisseau AM, Sarlangue J, Perel Y, et al. Perineal ecthyma gangrenosum in infancy and early childhood: septicemic and nonsepticemic forms. J Am Acad Dermatol 1992;27:415-8.
2. Mota-Burgos A, Villa AV, Noguera-Julian A, et al. Fever and skin lesions in a healthy 6-month-old boy. Diagnosis: Ecthyma gangrenosum. Pediatr Infect Dis J 2012;31:789-94.
3. Zomorrodi A, Wald ER. Ecthyma gangrenosum:
Gynecomastia is a benign enlargement of the male breast due to the proliferation of the glandular com- ponent. It may be an incidental finding on routine
Unilateral Gynecomastia in a Tennis PlayerSang Gue Kang, Woo Jin Song, Chul Han Kim, Ju Won Kim, Min Sung TarkDepartment of Plastic and Reconstructive Surgery, Soonchunhyang University College of Medicine, Seoul, Korea
Correspondence: Sang Gue KangDepartment of Plastic and Reconstructive Surgery, Soonchunhyang University College of Medicine, 59 Daesagwan-ro, Yongsan-gu, Seoul 140-743, KoreaTel: +82-2-709-9283, Fax: +82-2-796-3543E-mail: [email protected]
This article was presented at the 68th Korean Society of Plastic and Reconstructive Surgeons on Nov 4-7, 2010 in Seoul, Korea.
No potential conflict of interest relevant to this article was reported.
Received: 31 May 2012 • Revised: 16 Jul 2012 • Accepted: 17 Jul 2012pISSN: 2234-6163 • eISSN: 2234-6171http://dx.doi.org/10.5999/aps.2012.39.6.675 • Arch Plast Surg 2012;39:675-678
Copyright 2012 The Korean Society of Plastic and Reconstructive SurgeonsThis is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Images
considerations in a previously healthy child. Pediatr Infect Dis J 2002;21:1161-4.
4. Kliegman RM, Behrman RE, Jenson HB, et al. Nelson textbook of pediatrics. 18th ed. Philadelphia: Elsevier Saunders; 2007.
5. Huminer D, Siegman-Igra Y, Morduchowicz G, et al. Ecthyma gangrenosum without bacteremia. Report of six cases and review of the literature. Arch Intern Med 1987;147:299-301.
Table 1. Endocrinologic findings
Endocrine study Patients Normal reference range
Beta-hCG (ng/mL) 0.00 <1.0 Prolactine (ng/mL) 9.87 1.8-15.9 TSH (µIU/mL) 1.92 0.25-4.0 Free T4 (ng/dL) 1.28 0.7-2.0 Testosterone (ng/dL) 624.92 241-827 FSH (mIU/mL) 4.28 1.1-13.5 LH (mIU/mL) 2.65 0.4-5.7 E2 (pg/mL) 22.85 0-44
hCG, human chorionic genodotropin; TSH, thyroid-stimulating hormone; Free, T4 free thyroxine; FSH, follicle-stimulating hormone; LH, luteinizing hormone; E2, estradiol.
