Click here to load reader

Precalcaneal Congenital Fibrolipomatous Hamartoma

  • View

  • Download

Embed Size (px)

Text of Precalcaneal Congenital Fibrolipomatous Hamartoma


    Figure 2 matory der

    tant metawhere it wonly 1 cagranulocytsic Sweet some casemon, and rdifferencelymphedemlesions moteroids or pwell to oracal cortico

    The patand a numwhich posinadequatetomy and cytokines aattracting competencmalignancitrophilic d

    The diffas cellulitiwell as thogy should

    erysipeloides, and contact dermatitis. The clinical course,biopsy ndings, and additional tests will all help to establishthe denitive diagnosis.2,6

    In conclusion, we report a new case of neutrophilic der-matosis on the site of a lymphedema. This condition isa localized variant of classic Sweet syndrome with differ-ences thatalso a mildrelapses, ainammatolarge numb

    as ae paocuinan in


    n PRse coa-Ra J, Garca-Dez A. Sweets sndrome on the area of post-ectomy lymphoedema. J Eur Acad Dermatol Venereol.;20:401---5.

    T, Francs C, Marinho E, Herson S, Chosidow O.hoedema-area-restricted Sweet sndrome during G-CSFment. Lancet. 1996;347:690.itsu T, Tadaki T. Atypical neutrophilic dermatosis on ther extremity affected by postmastectomy lymphedema:rt of 2 cases. Dermatologica. 1991;183:230---3.CH, Lee HC, Lu CF, Hsiao CH, Jee SH, Tjiu JW. Neutrophilicatosis on postmastectomy lymphoedema: a localized andsevere variant of Sweet sndrome. Eur J Dermatol.;19:641---2.s A, Betlloch I. Ppulas eritematosas en el brazo de unaente mastectomizada. Actas Dermosiliogr. 2009; E, Puca RV, Brunetti G, Schwartz RA, Roucco V. Lymphede-us areas: privileged sites for tumors, infections, and immuneders. Int J Dermatol. 2007;46:662.

    irrez-Paredes, A. Gonzlez-Rodrguez,na-Gallardo, E. Jord-Cuevas

    io de Dermatologa, Hospital Clnico Universitario deia, Universidad de Valencia, Spain

    sponding author. address: ev [email protected]).



    Please Barrantes Hamartoma Dermosilioneal Congenital Fibrolipomatousma

    ma brolipomatoso precalcneoo

    cite this article as: Rubio-Flores C, Lpez-Gonzlez O, Garrido-Gutirrez C, Daz-Daz RM.

    brolipomatoso precalcneo congnito. Actasgr.2012;103:651-653.

    To the Editor:

    Precalcaneal congenital brolipomatous hamartoma (PCFH)is a rare and benign childhood skin disorder, with only a fewcases reported in the literature. It has been referred to bya variety of names, including pedal papules in the newborn,congenital piezogenic-like papules, and bilateral congenitaladipose plantar nodules.

    We present the case of a 9-month-old girl, with no rele-vant personal or family history, whose family brought her toconsultation for the presence of symmetric subcutaneousPredominantly perivascular and interstitial inam-mal neutrophilic inltrate.

    stases of the breast cancer, except in 1 caseas described in association with skin metastases2;se has been reported in a patients receivinge-colony stimulating factor.2,3 Compared to clas-syndrome, the systemic symptoms are mild (ins absent), neutrophilic leukocytosis is less com-elapses are less prevalent. The other distinctive

    is that the lesions are restricted to the site of thea. Treatment with antibiotics appears to cure the

    re rapidly than treatment with systemic corticos-otassium iodide,4 and the disorder also respondsl anti-inammatory drugs and high-potency topi-steroids.2---6

    hophysiology of this condition is not understoodber of theories have been put forward, all ofit a local disruption in cell trafcking due to

    lymphatic drainage caused by the lymphadenec-the radiation therapy. The hypothesis is thatccumulate at the site of the lymphadenectomyneutrophils to an area with reduced immuno-e and thereby favoring local development ofes, infections, and immune disorders such as neu-ermatosis.2,7

    erential diagnosis should include infections suchs, erysipelas, folliculitis, and herpes zoster, asrombophlebitis and recall phenomenon. Histol-

    rule out chronic radiodermatitis, carcinoma

    arisingin thesbeen dother commo


    1. Cohedisea

    2. GarcFragmast2006

    3. PetitLymptreat

    4. Demupperepo

    5. Lee dermless 2009

    6. Lucapaci

    7. Ruocmatodisor

    E. GutI. Moli


    CorreE-mail(E. Gu include not only the location of the lesions buter course with fewer systemic symptoms, fewernd a good response to oral antibiotics, anti-ry drugs, and topical corticosteroids. Despite theer of cases of breast cancer and of lymphedema

    complication of the different treatments usedtients, few cases of neutrophilic dermatosis havemented. This is probably due to confusion withmmatory or infectious diseases that are more

    this group of patients.2


    , Kurzrock R. Sweets sndrome revisited: a review ofncepts. Int J Dermatol. 2003;42:761---78.

    o I, Prez-Gala S, Arages M, Fernndez-Herrera J,


    nodular lesions on the plantar surface of both feet, justanterior to the heels (Fig. 1). The lesions had been presentsince birth. They had a soft consistency, were poorly cir-cumscribed, and were not adherent to the supercial ordeeper layers. The overlying skin was normal. They had adiameter of 1.5 cm at their widest point and appeared tobe asymptomatic. There were no other evident abnormali-ties. Based on these clinical features, a diagnosis of PCFHwas made. The lesions remained unchanged over more than6 months of follow-up. During this time, the patient startedto walk annot conside

    PCFH wdescribed In 1996, Lacongenitalbeen usedlated case to be rareawareness nodules.

    It is typIt has beenthan in fem

    Its paththat it mitissue1---3 asheel exhibithe fact thahistologic ehas not betradict thissuggested nective tratissue over

    PCFH tebeen reporently autosalso been schondrial i

    PCFH isric, mobilelocated onanterior tothe heel. Oon one fooskin is normbeen repordo not genan option i

    The natThe noduleand there adulthood.published little is kntaneous reassociated formation.

    HistologHistologic by collagend there was no gait disturbance. Treatment wasred necessary.

    as rst reported in 1990 by Larralde et al.,1 whothe nodules as pedal papules in the newborn.rregue et al.2 called the disorder precalcaneal

    brolipomatous hamartoma and this term has ever since. The literature contains only iso-reports and small series. Although PCFH appears, it is probably underdiagnosed because of lowamong clinicians and the benign nature of the

    ically present at birth, but it can develop later. reported to be slightly more common in malesales.3

    ogenesis is unknown. Early descriptions suggestedght be due to incomplete regression of fetal

    brolipomatous fetal tissue in the area of thets physiologic hypodermic hypertrophy. However,t fetal adipose tissue has never been detected onxamination and that a higher incidence of PCFH

    en noted in preterm infants would seem to con- theory.4,5 Other possible causes that have beeninclude a congenital alteration in the brocon-becular network of the adipose tissue3---10 and agrowth disorder.7

    nds to appear sporadically, although there havets of a familial association,3,6,7,9 with an appar-omal dominant pattern of inheritance.7,9 It hasuggested that there might be an X-linked or mito-nheritance pattern.6

    characterized by the presence of soft, symmet-, subcutaneous nodular lesions that are generally

    the mid region of the soles of both feet, just the heel, although they can also extend onton occasions, the nodules can be more prominentt than on the other.8 The color of the overlyingal. The nodules are asymptomatic and have not

    ted to cause gait disturbance. Accordingly, theyerally require treatment, but surgical excision isf they cause discomfort.ural history of PCFH is not well established.s tend to increase in size as the child grows,

    have been reports of lesions persisting well into4,5 However, because so few reports have beenon this relatively recently described disorder,own about clinical course or potential for spon-gression. There have been no reports to date ofabnormalities or potential for malignant trans-

    y is generally not necessary to diagnose PCFH.4---10

    ndings include mature adipose tissue surrounded bers of different thicknesses and normal elas-

    Figure 1 Symmetric nodules on the plantar surface of bothfeet, just anterior to the heels.

    tic bers. In addition, there may be mucin deposits at theperiphery and within the fat lobules2,4 and an increasednumber of blood vessels without associated perivascularalterations.10 The above ndings have been conrmed inultrastructural studies.3

    The differential diagnosis should include piezogenicpapules, which are generally found in adults and are causedby herniation of fat through the dermis following injury.Unlike PCHF nodules, piezogenic papules are typically mul-tiple and are often painful, particularly on walking. Othertypes of neonatal nod