Abstract
Introduction
Management of giant hairy nevi depends on various factors including the size and anatomical area.
Case presentation
We report a case of a giant hairy nevus treated successfully with curettage at the age of 6 hrs after birth. There was partial recurrence of pigmentation and hair on long-term (10 years) follow-up.
Discussion
Although curettage is a known method of treating hairy nevi, long term results are lacking when the nevus is treated in the first few hours after birth.
Conclusion
Following curettage of hairy nevi in the first few hours after birth, recurrence of pigmentation and hair may still occur on long term follow up.
Keywords: Congenital hairy nevus, Giant hairy nevus, Curettage
Highlights
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Management of giant hairy nevi depends on various factors including the size and anatomical area.
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Managment options includes: observation, tissue expander insterion, serial excision or currettage.
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Going through such an extensive measure (curettage) have both advantages and disadvantages which makes it a dilemma, especially at an early age
1. Introduction
Management of giant hairy nevi depends on various factors including the size and anatomical area. Less extensive nevi are usually treated with tissue expansion and serial excision. More extensive nevi are usually treated by observation and regular follow-up for early detection of melanoma. Some authors consider the risk of malignancy as significant and treat the lesion with excision and artificial skin cover [1].
Another way of management of extensive hairy nevi is curettage (or dermabrasion) of the nevus in the first few weeks of life. The idea behind this management is based on the fact that there is a well-defined cleavage plane between the abnormal melanocytes and the remaining dermis which is present at birth. This plane becomes less defined within the first few weeks of life. Hence, curettage is easier and more effective in younger neonates. However, long-term results are lacking when curettage is done in the first few hours after birth [1], [2], [3].
We report a case of a giant hairy nevus treated with curettage at the age of 6 hrs after birth with long-term follow-up of 10 years. This work has been reported in line with the SCARE criteria [10].
2. Case presentation
A male infant was born with a giant hairy nevus involving an extensive area of the trunk; as well as multiple scattered areas of the remaining body (Figs. 1,2). The total body surface area involved was 45 %. No family history of the same condition, or any use of medication. After securing consent from the parents, the patient underwent curettage of the hairy nevus 6 hrs post-delivery. The raw area was covered by a synthetic collagen substitute. Post-operatively the patient was monitored in the neonatal intensive care. The estimated blood loss was 80 ml, and the patient received 50 ml of packed red blood cells. The skin substitute was removed in 5 days because of greenish discharge from the wound and dressing was continued using silver dressing. All wounds healed within 2 weeks and the immediate postoperative appearance was impressive and the parents were very satisfied.
Figs. 1, 2.
Appearance at birth.
The patient was lost for follow-up for 10 years and was called to come back for assessment. Recurrence of pigmentation and light hair were noted (Figs. 3, 4).
Figs. 3, 4.
Appearance 3 weeks after curettage.
3. Discussion
The definition of giant hairy nevi is still controversial. Some consider hairy nevi that could not be excised and the defect closed with primary closure as a giant. Others consider a diameter of 20 cm or more as a giant [3].The risk of malignancy within giant hairy nevi is also variable in the literature, although we believe it is around 1 % [3].
A review of the literature on giant hairy nevi treated by curettage [2], [4], [5], [6], [7], [8], [9] is shown in Table 1.
Table 1.
Previously reported cases of hairy nevi treated with curettage.
| Author | Year of publication | Number of the cases | Site | Size | Age at time of curettage | Follow-up time | Outcome |
|---|---|---|---|---|---|---|---|
| Johnson [4] | 1977 | 1 | Forehead & scalp | Not mentioned | 5–16 months | 17 months | Satisfactory |
| Moss [2] | 1987 | 10 | Trunk, limbs, face | 4–20 cm2 | 2 weeks-6 months | 3 months- 5 years | Acceptable on 7 patients and poor in the remaining 3 patients |
| De Mey et al. [5] | 1992 | 6 | Not mentioned | Not mentioned | Not mentioned | 3 years | Satisfactory in 5. Poor in one |
| Casanova et al. [6] | 1997 | 9 | Trunk, limbs, and face | Small (2 cm2) to large (over 20 cm2) | 1–6 weeks | 18–55 months | Fair to good |
| Kay et al. [7] | 1998 | 1 | Trunk | Not mentioned | 16 days | 4 months | Not satisfactory |
| De Raeve et al. [8] | 2002 | 16 | Trunk, limbs, and face | Not mentioned | One case was treated at I day of age. The remaining in the first 4 weeks of life | Not mentioned | Excellent in 2 patients and satisfactory in 14 patients |
| Natara et al. [9] | 2013 | 1 | Face | Not mentioned | 2 weeks | 1-year | Satisfactory |
As seen from the table, most cases had only short-term follow-ups; and none were treated in the first few hours after birth. Moss reported the first series in 1987 [2]. Curettage was done between 2 weeks and 6 months of age. The most important observation from this early series was the documentation that curettage became more difficult, and the results were worse with the delay in the time of surgical intervention. There was only one reported case in which curettage was done at the age of one day [8]. However, the long-term follow up of the patient was not specified. Our case is unique because curettage was done at 6 hrs after birth and our case also has the longest follow up in the literature. Despite this early intervention partial recurrence occurred (Figs. 5, 6).
Figs. 5, 6.
Appearance at 10 years of age.
Embryologically, normal melanocytes are neuro-ectodermal cells that migrate along the nerves to reside at the dermal-epidermal junction [8]. This migration is normally complete by birth [1]. Our case demonstrates that the abnormal melanocytes of giant hairy nevi continue to migrate after birth and hence one should expect the re-appearance of some pigmentation and hair on long-term follow-up. Despite that, the results of early curettage are still better than the appearance of untreated nevi. Another advantage of the curettage is the theoretical lower risk of malignancy since the number of abnormal melanocytes is dramatically reduced.
In giant hairy nevi with this large surface area involved, serial debridement is not an option, skin graft is a major procedure with major morbidity and unpleasing aesthetic appearance.
4. Conclusion
A 6-hour-old male infant underwent curettage of an extensive giant hairy nevus. The early postoperative result was excellent. Despite this very early intervention, partial recurrence of pigmentation and light hair were noted on long term follow up.
Performing surgery with substantial blood loss under general anesthesia, especially immediately after delivery when the possibility of as yet undiscovered congenital diseases has not been ruled out, is fraught with risks. After seeing long term result, we believe that this method of treatment should be abandoned and should not be performed because the risks vs benefits is not worth it.
Consent
Written informed consent was obtained from the three patients for publication of this case report. A copy of the written consent is available for review by Editor-In-Chief of this journal on request.
Ethical considerations
This case report conforms to the Declaration of Helsinki.
Ethical approval
Exempted from the IRB approval.
Funding source
None.
Author contribution
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1.
Felwa A. AlMarshad: Manuscript writing and editing.
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Abdullah M. AlZahrani: Manuscript writing and editing.
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3.
Nora N. AlSaud: Manuscript writing and editing.
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4.
Ali A.AlMathami: Manuscript writing and editing.
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5.
Mohammad M. Al-Qattan Patient management and manuscript editing.
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6.
Fuad K. Hashem, MD: Patient management and manuscript editing.
Guarantor
Felwa A. Almarshad.
Fuad K. Hashem.
Mohammad M. Al-Qattan.
Research registration number
This paper does not require registry. There is no additional harm to the patient nor an innovative intervention is being applied on the patient.
Declaration of competing interest
None.
References
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