cutaneous mucormycosis in a patient with acute lymphocytic leukemia
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unfortunately died from severe mycotic sepsis. Micro-biological cultures from bronchoalveolar lavage (BAL)performed 3 days prior to death also confirmed the presenceof the genus Mucor.Mucormycosis is an uncommon opportunistic infectioncaused by members of the order Mucorales. These organ-isms exist widely in nature and are usually saprophytic,
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Figure 1. A) Necrotic skin lesions on the mucosa of the lowerlip. B) Necrotic skin lesion on the left chest. C) Skin biopsytaken from a necrotic skin lesion, showing characteristic broad,non-septate hyphae of Mucor in the midst of neutrophilic
o hot air during sauna-bathing [3]. However, heat-strokeurn can occur even under clothing and during a heat wavenywhere in the world. In cases of hot-air sauna burn,wo patterns of skin injury, namely, mesh-like and uni-orm, and rhabdomyolysis have been reported. We observedivedoid patterns exclusively in major regions of burns,ith a uniform pattern only on the faces of our patients.habdomyolysis was minimal when estimated in terms oflevation of serum creatine kinase.uring heat stress, active vasodilator tone directed
owards cutaneous arterioles is enhanced [4]. Furthermore,iminished arteriolar flow can result from dehydration,yperviscosity and/or thrombosis. Then deoxygenation isaused by decreased arteriolar perfusion, which leads toivedoid patterns [5]. It is likely that lesions were foundnly on the upper areas of the supine bodies because ofecreased hydrostatic pressure within the cutaneous vesselsf the affected areas, due to dehydration.oth patients were old and living alone and their life styleight have contributed to heat stroke because they might
ave been unaware of the high temperatures outside. Otherisk factors can be proposed: drug intake, in particular, anti-ypertensive drugs, which decrease cardiac-pump activity,ith resultant loss of blood flow for heat exchange; clothing,
uch as dark-colored clothes that absorb incident radia-ion; parsimonious use of electricity (fan, air conditioning);requent outdoor labor; and acute increase in ambient tem-erature.t is curious that such skin injury has not been reported, tour knowledge, in the literature, but the numbers of suchases might be expected to increase in the near future, withlobal climate change [6]. �isclosure. Financial support: none. Conflict of interest:one.
1 Department of Dermatology,2 Emergency and Critical Care
enter,3 Department of Neurosurgery,aculty of Medecine,ita University, 1-1 Idaigaokaasama-machi, 879-5593 Yufu,
Yusuke NAKAMURA1
Naoko TAKEO1
Mayuko GOTO1
Yoshitaka KAI1
Yutaka HATANO1
Osamu OKAMOTO1
Sakuhei FUJIWARA1
Sanshi TANABE2
Ryuichi TAKENAKA2
Hirotaka FUDABA3
Takeshi KUBO3
Minoru FUJIKI3
. Epstein Y, Rberts WO. The pathology of heat stroke: an integra-ive view of the final common pathway. Scand J Med Sci Sports011; 21: 742-8.. Vardy DA, Khoury M, Ben-Meir P, Ben-Yakar Y, Shoenfeld Y. Fullkin thickness burns caused by contact with the pavement in a heat-troke victim. Burns 1989; 15: 115-6.. Koljonen V. Hot air sauna burns-review of their etiology and treat-ent. J Burn Care Res 2009; 30: 705-10.. Charkoudian N. Skin blood flow in adult human thermoregula-
ion: How it works, when it does not, and why. Mayo Clin Proc003; 78: 603-12.. Gibbs MB, English JC, 3rd JC, Zirwas MJ. Livedo reticularis: Anpdate. J Am Acad Dermatol 2005; 52: 1009-19.. Kalkstein LS, Greene JS. An evaluation of climate/mortality relation-
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hip in large U.S. cities and the possible impacts of a climate change.nviron Health Perspect 1997; 105: 84-93.
doi:10.1684/ejd.2014.2268
Cutaneous mucormycosis in a patient withacute lymphocytic leukemia
Mucormycosis is a rare opportunistic fungal infectioncaused by members of the order Mucorales, typicallyin severely immunocompromised hosts and causing fatalinfection [1-3]. We report a patient with acute lymphocyticleukemia who developed several cutaneous plaques withnecrosis after chemotherapy.A 70-year-old man was admitted for chemotherapy forrelapsed acute lymphocytic leukemia. On day 14 of induc-tion chemotherapy, he displayed high fever and severedyspnea. High-resolution computed tomography revealedinfiltrates in the lung. Besides antibiotic therapy, antimy-cotic therapy was started with liposomal amphotericinB (2.5 mg/kg/day, i.v.) for suspected fungal infection.This was continued over 10 days but dyspnea did notimprove very much. Liposomal amphotericin B was there-fore stopped and voriconazole (8 mg/kg/day, i.v.) wasadministered. Over the next few days, the patient developedsharply demarcated plaques with necrosis on the mucosaof the lower lip and the skin of the chest (figures 1A-B).Histopathological examination revealed broad, non-septatehyphae with right-angle branching in the midst of a neu-trophilic abscess (figure 1C). Periodic-acid-Schiff stainingalso identified the growth of fungal hyphae (figure 1D). Cul-ture of a skin sample grew a fungus, which was shown tobe of the genus Mucor. One day after diagnosis, the patient
EJD, vol. 24, n◦ 1, January-February 2014
abscess (hematoxylin and eosin, original magnification ×250).D) Periodic-acid-Schiff staining also identified growth of fun-gal hyphae (original magnification ×250).
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tolerant state [2-4]. However, the symptoms of SU arefrequently resistant to these treatments. Recently, the effec-
rowing on decaying organic materials such as vegetation,ruit, and bread [1]. In addition, they can normally be cul-ured from the stools, nasal passages and oral cavities ofealthy persons [1].utaneous mucormycoses can be broadly separated into
wo categories according to the manner in which the dis-ase manifests: primary cases, in which the skin is invadedirectly by the organism; and secondary cases, in whichhe organism is transported from a lesion in an internalrgan to a skin site by hematogenous metastasis [3]. Thelinical presentation differs between these two disease pro-esses. The primary cutaneous variant is rare and invasive,nvolving not only the cutaneous tissue but also the under-ying fat, muscle and fascial layers [5]. It typically presentss a single necrotic hemorrhagic ulcer following percu-aneous inoculation of fungal spores [6]. Growth of theungus in a pre-existing lesion produces an acute inflamma-ory response with pus, abscess formation, tissue swellingnd necrosis. The lesions may appear red and indurated butften progress to form black eschars. We believe the presentase represented secondary hematogenous spread from aulmonary site to the skin because pulmonary involvementas clinically evident before the appearance of skin lesions,
nd microbiological cultures from BAL also identified theresence of the genus Mucor.his disease often causes necrotic hemorrhagic lesionsecause Mucor species have an ability to invade the wallsf vessels and proliferate, causing thrombosis and subse-uent infarction of the involved vessel, with subsequentocal tissue destruction [1, 7, 8].iven the poor prognosis, mucormycosis usually requires
everal simultaneous modes of therapy: surgical debride-ent; antifungal therapy; and improvement of immunosup-
ressive state [1, 2]. Amphotericin B has been the first-linerug of choice for most cases of mucormycosis [1, 2, 5].nfortunately, this drug has adverse effects such as nephro-
oxicity and electrolyte disturbances. Furthermore, someatients do not respond. The liposomal form of ampho-ericin B (AmBisome®; Gilead Sciences, Foster City, CA,SA) is associated with fewer side-effects and offers dra-atically better outcomes [4].he poor prognosis in this infection is mainly due to a
ailure to achieve early diagnosis, extensive involvementnd the underlying condition [4]. However, early diagno-is of mucormycosis is difficult and many patients are onlyiagnosed post-mortem [9]. Although the standard for diag-osis of cutaneous mucormycosis is a fungal culture ofull-thickness skin tissue, culture growth of Mucorales maye time-consuming. Detection of non-septate, branchedyphae in a KOH specimen or biopsy sample stained withematoxylin and eosin should also be considered highlyignificant in reaching a diagnosis more rapidly.
ith the increasing number of diabetic patients andncreased use of corticosteroids, immunosuppressants andnti-cancer agents, the incidence of cutaneous mucormy-osis is increasing. Mucormycosis should be considered in
JD, vol. 24, n◦ 1, January-February 2014
he initial evaluation of cutaneous necrosis or ecthyma-likeesions, particularly in immunocompromised patients. �
isclosure. Financial support: none. Conflict of interest:one.
1 Department of Dermatology,Ehime University Graduate Schoolof Medicine,Toon, 791-0295 Ehime, Japan2 Department of Dermatology,Ehime Prefectural Central Hospital,Matsuyama, Ehime, Japan<[email protected]>
Ken SHIRAISHI1
Shiro SASAKI2
Yasushi SADAMOTO2
1. Wirth F, Perry R, Eskenazi A, et al. Cutaneous mucormycosis withsubsequent visceral dissemination in a child with neutropenia: a casereport and review of the pediatric literature. J Am Acad Dermatol1997; 36: 336-41.2. Fujimoto A, Nagao K, Tanaka K, et al. The first case of cuta-neous mucormycosis caused by Rhizopus azygosporus. Br J Dermatol2005; 153: 428-30.3. Kobayashi M, Hiruma M, Matsushita A, et al. Cutaneous zygomy-cosis: a case report and review of Japanese reports. Mycoses2001; 44: 311-5.4. Wali YA, Lamki ZA, Kindi HA, et al. Case report. Successful out-come of invasive nasal sinus zygomycosis in a child with relapsed acutelymphoblastic leukaemia due to liposomal amphotericin B. Mycoses2001; 44: 195-9.5. Kerr OA, Bong C, Wallis C, et al. Primary cutaneous mucormy-cosis masquerading as pyoderma gangrenosum. Br J Dermatol2004; 150: 1212-3.6. Chambers CJ, Merin MR, Fung MA, et al. Primary cutaneousmucormycosis at sites of insulin injection. J Am Acad Dermatol2011; 64: 79-81.7. Ribes JA, Vanover-Sams CL, Baker DJ. Zygomycetes in human dis-ease. Clin Microbiol Rev 2000; 13: 236-301.8. Lenane P, Keane CO, Loughlin SO. Mucor mycosis infection pre-senting as a non-healing ulcer in an immunocompromised patient. ClinExp Dermatol 2003; 28: 157-9.9. Hadaschik E, Koschny R, Willinger B, et al. Pulmonary, rhino-orbitaland cutaneous mucormycosis caused by Rhizomucor pusillus in animmunocompromised patient. Clin Exp Dermatol 2012; 37: 355-7.
doi:10.1684/ejd.2013.2240
Successful treatment with UVA rush hard-ening in a case of solar urticaria
Solar urticaria (SU) is a rare idiopathic photodermatosis.The symptoms are usually observed within ten minutesafter exposure to sunlight. The action spectra are dif-ferent among cases, ranging from ultraviolet B (UVB)to visible light [1]. SU is commonly treated with oralantihistamines, sunscreen, plasmapheresis and/or immuno-suppressants. Phototherapy with various wavelengths oflight and methods has also been performed to induce a
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tiveness of ultraviolet A (UVA) rush hardening therapy hasbeen reported [5, 6]. We herein describe a case of success-ful treatment with UVA rush hardening in a SU patient inwhom irradiation of visible light strongly contributed to thedevelopment of symptoms.