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Vol. 110. Issue 7.
Pages 619-621 (September 2019)
Vol. 110. Issue 7.
Pages 619-621 (September 2019)
Case and Research Letters
DOI: 10.1016/j.adengl.2019.07.012
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Circumscribed Plantar Hypokeratosis
Hipoqueratosis plantar circunscrita
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I. Aranguren-Lópeza,
Corresponding author
, S. Vildósola-Esturoa, I. Arias-Camisónb, A. López-Pestañaa
a Servicio de Dermatología, Hospital Universitario Donostia, San Sebastián, Guipúzcoa, España
b Servicio de Anatomía Patológica, Clínica de la Asunción, Tolosa, Guipúzcoa, España
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Table 1. Review of Cases of Circumscribed Plantar Hypokeratosis Published to Date, With Clinical Characteristics, Treatments Used, and Response to Treatment.
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To the Editor:

Circumscribed palmoplantar hypokeratosis (CPH) is a rare disease characterized by focal thinning of the stratum corneum. It manifests as a round, well-defined, depressed, erythematous lesion with a scaly border, generally located on the palms. It is generally asymptomatic and predominantly affects middle-aged or elderly women. We report 3 cases of CPH on the sole of the foot, a rare site for this disease.

Case 1. A 74-year-old diabetic man who visited our department with an asymptomatic erythematous lesion on the sole of the left foot; the lesion had well defined borders and had grown slowly over the previous 7years (Fig. 1A) . Dermatoscopy revealed a stepped scaly border, an erythematous base with punctate vessels, regular white spots occasionally surrounded by the punctate vessels, and thin white lines (Fig. 1B). Clinical and dermatoscopic data were compatible with CPH. The patient refused a biopsy and did not want to undergo treatment.

Figure 1.

Case 1. A,Clinical image. B,Dermatoscopic image showing the stepped scaly border (asterisk), regular white spots (white arrows) on an erythematous background with punctate vessels, some of which are distributed around the white spots (black square).

(0.12MB).

Case 2. An 82-year-old woman with no relevant past history visited our department with a mildly pruritic lesion on the inside surface of the right foot that had grown slowly over the previous 4years. Physical examination revealed a lesion measuring 12×10mm with well-defined borders and a depressed erythematous center. Dermatoscopy revealed similar findings to the previous case. The biopsy showed a sudden reduction in the thickness of the stratum corneum, with hypergranulosis and diffuse areas of parakeratosis with no evidence of a parakeratotic column. CPH was diagnosed and the patient was treated with betamethasone dipropionate and calcipotriol, which produced a slight improvement. Treatment was suspended after a few weeks owing to irritation. The patient refused other treatments.

Case 3 A 69-year-old woman with no relevant past history visited our department with an asymptomatic, erythematous, depressed lesion on the arch of the left foot, with scaly border, which had appeared 10years earlier (Fig. 2A) . The dermatoscopy and histology findings were similar to the previous cases, confirming the diagnosis of CPH. The patient was treated with tacrolimus ointment 0.1% for 4months with little improvement. Cryotherapy with liquid nitrogen was then used and the lesion resolved completely (Fig. 2B).

Figure 2.

Caso 3. A,Lesion before cryotherapy. B,The same lesion 6months after cryotherapy.

(0.09MB).

CPH is a benign entity of unknown etiology that was first described in 2002 by Pérez et al.1 It is thought to be due to an abnormal keratinocyte clone that does not differentiate into palmoplantar epidermis, leading to marked thinning of the stratum corneum in comparison to the surrounding normal skin.2 Most of the cases described had no past history of trauma.

CPH is a rare disease, with approximately 100 cases reported in the literature.3 More than 60% of these cases were located on the thenar prominence, on the palm or back of the hand, on the sole of the foot, and on the medial surface of the foot. These last 2 locations are rare and account for less than 15% of reported cases of CPH.3,4 A review of the literature found 16published case of CPH on the sole of the foot1,3,4 (Table 1).

Table 1.

Review of Cases of Circumscribed Plantar Hypokeratosis Published to Date, With Clinical Characteristics, Treatments Used, and Response to Treatment.

No.  Authors and Date  Age  Sex  Sole  Treatments  Clinical Course 
Pérez et al.,1 2002  68  Female  Left  Several  No change 
Rütten et al.,3 2004  64  Female  Left  None  NI 
3Berk et al.,3 200775FemaleLeft5-fluorouracil topical  No change 
Calcipotriol ointment  No change 
Clobetasol ointment  No change 
4Berk et al.,3 200749FemaleLeftFluocinolone cream  No change 
Excision  Complete resolution 
Barry et al.,3 2008  59  Male  Right  NI  NI 
6Tanioka et al.,3 200962MaleRightTopical corticosteroids  No change 
Antibiotics  No change 
Cribier et al.,3 2009  65  Male  Left  None  No change 
Kanitakis et al.,3 2011  56  Female  Right  NI  NI 
Arbesman et al.,3 2012  10  Male  Left  NI  NI 
10  Santamarina-Albertos et al.,3 2013  Male  Right  Calcipotriol topical  No change 
11  Batalla et al.,3 2013  17  Male  Right  Calcipotriol topical  Complete resolution 
12Mitkov et al.,3 201473MaleRightMometasone topical  No change 
Tacrolimus topical  No change 
Pimecrolimus topical  No change 
5-fluorouracil topical  No change 
Cryotherapy  No change 
13Pinos-León et al.,3 201455FemaleLeftTopical corticosteroids  No change 
Topical antimycotics  No change 
14  Ramos-Garibay et al.,3 2016  69  Female  Left  Calcipotriol topical  No change 
15  Ramos-Garibay et al.,3 2016  71  Female  Left  None  No change 
16  Nazzaro et al.,4 2016  80  Male  Left  NI  NI 
17  Aranguren-López et al., 2018  74  Male  Left  None  NI 
18  Aranguren-López et al., 2018  82  Female  Right  Topical betamethasone dipropionate and calcipotriol  Slight improvement 
19  Aranguren-López et al., 2018  69  Female  Left  Cryotherapy  Complete resolution 

Abbreviation: NI indicates not indicated.

The histopathology of CPH is characteristic and confirms the diagnosis.2 A sudden interruption can be observed between the normal dermis and that of the lesion, which shows marked hypokeratosis and mild hypogranulosis. No parakeratotic column can be seen at the periphery of the lesion. This differentiates the disease from porokeratosis of Mibelli, the most clinically similar entity, which must be included in the differential diagnosis.2,5

Dermatoscopy is very useful for diagnosing this dermatosis. Typical findings are peripheral stepped desquamation (corresponding to the abrupt limit between the normal and hypokeratotic epidermis), a uniform erythematous base with small punctate vessels (reflection of the congestive capillaries in the underlying dermis, which become more visible due to the epidermal thinning) and regularly distributed white spots (the pores of the acrosyringia).5 White lines on the erythematous base,5 have been recently described, which correspond to the epidermal folds. The only dermatoscopy of palmoplantar hypokeratosis on the sole of the foot published to date4 describes for the first time distribution of the punctate vessels around the acrosyringia. These recent findings were also observed in our patients.

No effective treatment for CPH exists. In some of the reported cases,6 and in one of our patients, cryotherapy achieved complete remission of the lesions. Topical calcipotriol and 5-fluorouracil have also proven effective in some cases.2

In conclusion, we report 3 cases of CPH on the sole of the foot, a rare site for this rare disease, which is probably underdiagnosed due to its asymptomatic nature. We describe the dermatoscopic characteristics of the entity, which are very useful for diagnosis, and highlight the white lines found in one of our patients, which have been reported recently. We also highlight the utility of cryotherapy, which produced complete remission of the lesion in 1 case.

Conflicts of Interest

The authors declare that they have no conflicts of interest.

References
[1]
A. Pérez, A. Rütten, R. Gold, F. Urbina, C. Misad, M.J. Izquierdo, et al.
Circumscribed palmar or plantar hypokeratosis: A distinctive epidermal malformation of the palms and sole.
J Am Acad Dermatol., 47 (2002), pp. 21-27
[2]
F. Urbina, A. Pérez, L. Requena, A. Rütten.
Hipoqueratosis circunscrita palmar o plantar. Conocimientos y controversias tras 10años de su descripción.
Actas Dermosifiliogr., 105 (2014), pp. 574-582
[3]
A. Ramos-Garibay, G. Navarrete-Franco, F. Venadero-Albarrán, A. Domínguez-Gómez, J.R. Trejo-Acuña, J.J. Salazar-DelValle, et al.
Circumscribed hypokeratosis: Report on a series of 7 Mexican cases and review of the literature.
Am J Dermatopathol., 38 (2016), pp. 399-408
[4]
G. Nazzaro, A. Ponziani, M. Brena, S. Cavicchini.
Dermoscopy confirms diagnosis of circumscribed plantar hypokeratosis.
J Am Acad Dermatol., 76 (2017), pp. S43-S45
[5]
P.T. Vilas Boas da Silva, E. Rodríguez-Lomba, J.A. Avilés-Izquierdo, C. Ciudad-Blanco, R. Suárez-Fernández.
Dermoscopic features of circumscribed palmar hypokeratosis.
JAMA Dermatol., 153 (2017), pp. 609-611
[6]
M.J. Boffa, J.S. Degaetano.
Circumscribed palmar hypokeratosis: Successful treatment with cryotherapy.
J Eur Acad Dermatol Venereol., 21 (2007), pp. 420-421

Please cite this article as: Aranguren-López I, Vildósola-Esturo S, Arias-Camisón I, López-Pestaña A. Hipoqueratosis plantar circunscrita. Actas Dermosifiliogr. 2019;110:619–621.

Copyright © 2019. Elsevier España, S.L.U. and AEDV
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