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Revista chilena de pediatría

versión impresa ISSN 0370-4106

Rev. chil. pediatr. vol.91 no.3 Santiago jun. 2020

http://dx.doi.org/10.32641/rchped.v91i3.1401 

CLINICAL CASE

Painful nodules on the soles in a pediatric patient: A diagnostic challenge

Javier Sánchez-Bernal1 

Isabel Zárate-Tejero2 

Pilar Collado-Hernández2 

Mariano Ara-Martín1 

Lucía Prieto-Torres1 

1 Dermatology Department at Hospital Clínico Universitario Lozano Blesa, Zaragoza, Spain.

2 Department of pediatrics at Hospital Clínico Universitario Lozano Blesa, Zaragoza, Spain.

Abstract:

Introduction:

Idiopathic Palmoplantar Eccrine Hidradenitis (IPPH) is a rare neutrophilic derma tosis, with painful erythematous nodules of sudden onset in the plantar or palmoplantar region, in children without other underlying diseases.

Objective:

To present a case that shows the main clinical and histological characteristics of this entity.

Clinical Case:

11-year-old girl with a 48-hours history of painful erythematous-violaceous nodules on the right foot plant associated with fever of up to 38.2 °C, with no history of interest except hyperhidrosis and intense exercising on previous days. Given the clinical suspicion of IPPH, a skin biopsy was performed, which showed inflammatory neutrophil infiltration around eccrine sweat glands and neutrophilic abscesses, confirming the diagnosis. Oral NSAIDs and rest were prescribed, with resolution of the lesions in 7 days.

Conclusions:

This case demonstrates the most important aspects of this entity, in many cases underdiagnosed, since it can be confused with other pathologies that occur with painful acral nodules, but have different pathogenic and therapeutic implications. To properly identify the IPPH allows preventing an unnecessary alarm, both patients and their parents, as in dermatologists and pediatricians themselves.

Keywords: Hidradenitis; Foot Dermatoses/diagnosis; Eccrine Glands/patho logy; Child

What do we know about the subject matter of this study?

Painful plantar nodules are a condition of unknown etiology, with well-defined clinical and pathological characteristics.

What does this study contribute to what is already known?

A very representative clinical case of this condition, which is pro bably under-diagnosed, that helps the understanding of this entity, easing its management and avoiding diagnostic errors that may lead to unneeded treatments.

Introduction

Idiopathic palmoplantar eccrine hidradenitis (IPPH) is a rare neutrophilic dermatosis that was first described in 1994 by Stahr et al1 in six healthy patients, five children, and one young adult. All of them presen ted a clinical picture of painful plantar lesions and a histological study similar to neutrophilic eccrine hidra denitis (NEH) but with different characteristics. Since then, less than 50 cases have been reported, most of them in healthy patients aged between 2 and 15 years, with plantar and, sometimes, palmar involvement.

Its etiopathogenesis is still unclear. A higher in cidence has been observed in patients with history of intense physical exercise and/or use of closed shoes du ring the warm season2,3. Some theories support that, in patients with immature glands, a mechanical or ther mal trauma could produce the rupture of those glands infiltrating their content into the dermis, activating inflammatory cytokines which can attract polymor phonuclear ones2,4,5. Another hypothesis suggests that excessive sweating, either intrinsic or related to inten se physical exercise, would initiate this inflammatory cascade6,7. However, laboratory studies do not provide specific data in this regard8.

The histological findings are characteristic of the presence of intense inflammatory infiltration, predo minantly neutrophilic, around the eccrine sweat glands, both in the portion of the coil, where neutrophil absces ses can also form in the adjacent tissues and in the duct, where neutrophil accumulations can also be observed inside the duct in an inconstant but a quite specific manner8. There were no eccrine squamous syringometaplasia or typical signs of leukocytoclastic vasculitis2,8.

The objective of this report is to present a case that describes the main clinical and histological characteris tics of IPPH in order to highlight a characteristic pictu re that is probably underdiagnosed.

Clinical Case

11-year-old girl seen at the Emergency Department due to a 48-hours history of painful lesions on the right foot which prevented her from walking, with fever of up to 38.2°C and no other associated cutaneous or sys temic clinical symptoms or signs.

The presence of frequent episodes of plantar hyper-hidrosis and basketball practice in the previous days were the only data of interest referred by the patient. Physical examination showed erythematous-viola ceous nodules on the external lateral edge and forefoot area of the right foot painful on palpation (Figure 1).

Figure 1 Erythematous-violaceous nodules on the external lateral edge and forefoot area of the right foot. 

A skin biopsy was performed on one of the le sions, which showed a predominantly neutrophilic inflammatory infiltrate around the eccrine sweat glands and neutrophil abscesses near to the coil (Figure 2). With these findings, the diagnostic suspicion of IPPH was confirmed and we started treatment with oral non-steroidal anti-inflammatory drugs and rest, with gradual resolution of the lesions in 7 days, with no se quelae or recurrences after 3 years of follow-up.

Figure 2 A) HE x20: Neutrophilic inflammatory infiltrate araound the eccrine sweat glands and neu trophil abscesses near to the coil. B) HE x40: inflammatory infiltration surrounding and inside the duct of glands. 

Discusión

IPPH is a disease specific to children and young adults, clinically characterized by erythematous or erythematous-violaceous plaques and/or nodules, which are painful, of sudden onset, located in the plantar or palmoplantar regions, uni- or bilaterally2,4,8. Generally, this entity occurs in healthy patients with no history of other underlying diseases or recent me dication use8; however, it also appears with fever but without other general symptoms in some cases2,4.

When a pediatric patient presents with painful acral nodules, we must know how to recognize this picture and differentiate it from others with similar symptoms (Table 1).

Table 1 Differential diagnosis of painful acral nodules. 

The following are the main differential diagnoses to consider: traumatic plantar urticaria, characterized by recurrent outbreaks of plantar erythematous maculopapulae, pruritic or painful, which appear hours after intense physical activity and its histological stu dy shows an inflammatory infiltration in the dermis without affecting the eccrine sweat glands2,8,9; plan tar erythema nodosum of childhood, it presents very painful nodules, generally after infection or the use of medicines, which resolve more slowly, leaving post- inflammatory hyperpigmentation and in its histology we observe the characteristic septal panniculitis typical of erythema nodosum2,8,10; and the pseudomonas hot foot syndrome, which presents a clinical picture very similar to IPPH, with painful, erythematous plantar nodules of sudden appearance, but which characteris tically appears as community outbreaks in children, 6 to 48 hours after bathing in a hot or heated pool conta minated by this pathogen11.

Other entities such as erythema multiforme, perniosis, and some vasculitis and cellulitis could also be considered in the differential diagnosis2,8.

Finally, we should know how to differentiate IPPH from NEH, a polymorphous skin rash due to fever which affects the trunk, face, and limbs. This entity was initially described in patients with hemato logic malignancy receiving chemotherapy and, later, it has also been associated with other drugs, malignancies, and infections. Both present similar histolo gy, but with different characteristics such as eccrine squamous syringometaplasia in NEH, and the pre sence of neutrophil abscesses near the glandular coils in IPPH8.

Regarding the treatment, only symptomatic relief is recommended, since it is a condition that resolves spontaneously in about 3 weeks without any sequelae. It generally includes rest which may be done along with the use of non-steroidal anti-inflammatory drugs and topical corticosteroids2,6.

Conclusion

We present a case of IPPH with a typical clinical and histological picture. Also, we provide the keys to make a proper differential diagnosis with other entities with painful acral nodules, but that present a different clinical evolution and therapeutic implications, which causes unnecessary concern, both in patients and their parents, as in the doctors themselves.

Ethical Responsibilities

Human Beings and animals protection: Disclosure the authors state that the procedures were followed ac cording to the Declaration of Helsinki and the World Medical Association regarding human experimenta tion developed for the medical community.

Data confidentiality: The authors state that they have followed the protocols of their Center and Local regu lations on the publication of patient data.

Rights to privacy and informed consent: The authors have obtained the informed consent of the parents (tu tors) of the patients and/or subjects referred to in the article. This document is in the possession of the co rrespondence author.

Conflicts of Interest: Authors declare no conflict of interest regarding the present study.

Financial Disclosure: Authors state that no economic support has been asso ciated with the present study.

Referencias:

1. Stahr BJ, Cooper PH, Caputo RV. Idiopathic plantar hidradenitis: a neutrophilic eccrine hidradenitis occurring primarily in children. J Cutan Pathol. 1994;21:289-96. [ Links ]

2. López Blázquez M, Lozano Masdemont B, Mora Capín A, Gómez-Recuero Muñoz L, Campos Domínguez M. Idiopathic palmoplantar hidradenitis: is biopsy always necessary?. Acta Pediatr Esp. 2017; 75:e8-e10. [ Links ]

3. Rabinowitz LG, Cintra ML, Hood AF, Esterly NB. Recurrent palmoplantar hidradenitis in children. Arch Dermatol. 1995;131:817-20. [ Links ]

4. Housni Alaoui I, Hocar O, Akhdari N, et al. An uncommon cause of delayed walking: idiopathic palmoplantar hidradenitis. Arch Pediatr. 2015; 22:306-8. [ Links ]

5. Naimer SA, Zvulunov A, Ben-Amitai D, Landau M. Plantar hidradenitis in children induced by exposure to wet footwear. Pediatr Emerg Care. 2000;16:182-3. [ Links ]

6. Ben-Amitai D, Hodak E, Landau M, Metzker A, Feinmesser M, David M. Idiopathic palmoplantar eccrine hidradenitis in children. Eur J Pediatr. 2001;160:189-91. [ Links ]

7. Blázquez Sánchez N, Rodrigo Fernández I. Lesiones plantares recurrentes. Piel. 2007; 22:453-5. [ Links ]

8. González-Hermosa MR, González-Güemes M, González-Pérez R, Catón B, Díaz-Ramón JL, Soloeta R. Hidradenitis palmar idiopática. Actas Dermosifiliogr 2003;94:660-2. [ Links ]

9. Metzker A, Brodsky F. Traumatic plantar urticaria-an unrecognized entity?. J Am Acad Dermatol. 1988;18:144-6. [ Links ]

10. Suárez SM, Paller AS. Plantar erythema nodosum: Cases in two children. Arch Dermatol 1993;129:1064-65. [ Links ]

11. Fiorillo L, Zucker M, Sawyer D, Lin AN. The pseudomonas hot-foot syndrome. N Engl J Med. 2001;345:335-8. [ Links ]

Received: September 03, 2019; Accepted: November 19, 2019

Correspondence: Javier Sánchez Bernal. E-mail: javisanchez_5@hotmail.com.

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