Complete form pachydermoperiostosis in Tunisia – A case series and literature review

Alaya, Zeineb; Boussofara, L.; Bouzaouache, M.; Amri, D.; Zaghouani, Houneida; Bouajina, E.

doi:10.1016/j.ejr.2017.06.006

Cited by 4 publications

(5 citation statements)

References 26 publications

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“…Clinical history and examination along with lab investigations suggested normal thyroid function in our case. Radiographic and clinical findings in our case were similar to a case-series from Tunisia that reported marked periostosis of the long bones and pachyderma in all six cases with athralgia and clubbing in five patients 18 . In conjunction with this, our radiographical picture also showed melorheostosis of the right and left fibula.…”

Section: Discussionsupporting

confidence: 88%

Pachydermoperiostosis: a case report of initial improvement with etoricoxib

Vaidya,

Acharya,

Katila

et al. 2023

Annals of Medicine &Amp; Surgery

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Introduction and importance: Pachydermoperiostosis is a syndrome characterised by the triad of pachydermia, digital clubbing and periostosis of long bones and its scarce incidence and similarity in clinical features with acromegaly makes the diagnosis challenging. The elevated PGE2 levels have been hypothesised as one of its mechanisms and therapies have been targeted to inhibit this prostaglandin. Case presentation: A 25-year-old man with no comorbidities presented to OPD with 10-year history of bilateral pain and swelling of hands and feets associated with hyperhidrosis, grade IV clubbing and marked skin thickening on his forehead. X-ray revealed hyperostosis of metacarpals, proximal and middle phalanges and periosteal bone formation with cortical thickening of ankle joint. Tests done to rule out differentials such as thyroid acropachy, acromegaly, psoriatic arthritis were normal and clinical diagnosis of pachydermoperiostosis, a rare genetic disease characterised by pachyderma, digital clubbing, and periostosis was made. Clinical discussion: The patient was managed conservatively with Etoricoxib for six months on a follow-up basis. The symptoms were improving and a repeat X-ray showed partial improvement of soft tissue thickening and periostosis. Conclusion: Pachydermoperiostosis is a rare diagnosis with no clear consensus on management approach. Its management with selective COX-2 inhibitors such as Etoricoxib should be considered but its long term effects should be studied further.

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Section: Discussionsupporting

confidence: 88%

Pachydermoperiostosis: a case report of initial improvement with etoricoxib

Vaidya,

Acharya,

Katila

et al. 2023

Annals of Medicine &Amp; Surgery

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“…In addition to the radiographic findings seen in our case, Rastogi et al [28] also revealed spondylotic changes in spine, with loss of lumbar lordosis and new bone formation along the iliac bones. A recent case-series from Tunisia also revealed marked periostosis of the long bones in all cases [32]. In addition to knee findings seen in our case, the case reported by Guerini et al [33] showed calcification at the insertion of the quadriceps tendon at the upper edge of the left patella.…”

Section: Discussionsupporting

confidence: 82%

Pachydermoperiostosis (Touraine–Solente–Gole syndrome): a case report

et al. 2019

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Background Pachydermoperiostosis (PDP) is a rare disorder characterized by clubbing of the fingers, thickening of the skin (pachyderma), and excessive sweating (hyperhidrosis). It typically appears during childhood or adolescence, often around the time of puberty, and progresses slowly. Clinical presentations of PDP can be confused with secondary hypertrophic osteoarthropathy, psoriatic arthritis, rheumatoid arthritis, thyroid acropachy, and acromegaly. Case presentation A Mongolian male, aged 19 years, resident of a hilly district of Nepal, with history of consanguinity, presented to our outpatient department with chief complaints of pain and swelling in both hands and feet for 6 years. The pain was insidious in onset, throbbing in nature, and not relieved by over-the-counter medications. The patient also complained of profuse sweating, progressive enlargement of hands and feet, and gradual coarsening of facial features. On examination there were marked skin folds in the forehead, face, and eyelids. Clubbing and swelling of bilateral knee joints and ankle joints was also evident. He was subsequently investigated extensively for acromegaly. Insulin-like growth factor-1 level and oral glucose tolerance test were normal. Radiography of various bones showed periosteal hypertrophy with subperiosteal bone formation. Conclusions PDP should be considered as a differential diagnosis when a patient presents with hypertrophic osteoarthropathy and acromegalic features.

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“…The aesthetic surgeries for PDP that have been documented in the literature include bilateral blepharoplasties [14], tarsal wedge resections [15], frontal rhytidectomy [16,17], along with facelifts [18]. The most evident and troubling complaint of the patient is the forehead creases, which first of all manifest prematurely and leave the patient looking distressed as well as aged.…”

Section: Discussionmentioning

confidence: 99%

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2023

IJCMCR

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Pachydermoperiostosis (PDP), is an autosomal-dominant/autosomal recessive inherited disorder. PDP goes by various other names such as Rosenfeld-Kloepfer or Touraine-Solente-Gole syndrome, and primary or Idiopathic Hypertrophic osteoarthropathy) PDP is characterized by the coarsening of facial features, with furrowing of the forehead and scalp regions, new periosteal bone formation, arthralgia, and digital clubbing. The medical management is well reported in literature along with the histopathological and genetic basis of the disease. However, the approach to cosmetic deformity in the form of forehead furrows is rarely reported. A case report of a young adult male is presented here. His chief complaint at the time of presentation was the deepened furrows over his forehead and the prematurely aged look on his face. Here we describe our surgical approach and the outcome of the same.

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Complete form pachydermoperiostosis in Tunisia – A case series and literature review

Cited by 4 publications

References 26 publications

Pachydermoperiostosis: a case report of initial improvement with etoricoxib

Pachydermoperiostosis: a case report of initial improvement with etoricoxib

Pachydermoperiostosis (Touraine–Solente–Gole syndrome): a case report

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