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MULTIPLE PRIMARY PRESENTATIONS OF SQUAMOUS CELL CARCINOMA ASSOCIATED WITH PALMOPLANTAR HYPERKERATOSIS :
Case Report and Review of the Literature


Prakash Patil, Shailesh Shrikhande, Praful Desai
Department of Oncosurgery, Bombay Hospital Institute of Medical Sciences, Mumbai.

Multiple primary presentation of squamous cell carcinoma is a rare clinical entity. We report a case that was treated for perianal squamous cell carcinoma 10 years ago and currently presenting with squamous cell carcinoma at two diverse sites (lung and palm). The underlying aetiology was palmoplantar hyperkeratosis.

A medline search of the literature revealed 12 cases of palmoplantar hyperkeratosis associated with bronchial carcinoma. However, this case may be the first report of a triple primary cancer in the literature.

CASE REPORT

A 54 year old family physician presented with history of cough and haemoptysis of three months duration. He was treated 10 years ago for a grade III squamous carcinoma of the perianal region by a wide excision and split skin graft. On clinical evaluation, disease was controlled locally and there was no inguinal or peripheral lymphadenopathy. The palmoplantar regions were the site of diffuse nodular thickening, unrelated to his daily work (Fig. 1). There was no history of exposure to radiation. On the left hypothenar eminence was a chronic non-healing ulcer measuring 3 x 3 cm in diameter. Rest of his physical examination was unremarkable.

Fig. 1
Fig. 1 : Diffuse modular thickening on the plantar aspects of both feet.


An X-ray chest revealed a solid peripheral mass lesion in the left lower lobe (Fig. 2). A CT scan of the chest confirmed the lesion to be extending across the oblique fissure involving a portion of the lingular lobe (Fig. 3). There were no mediastinal lymph nodes or other parenchymal lesions. A CT guided FNAC confirmed a diagnosis of squamous cell carcinoma (? second primary/? metastasis). CT scan of the abdomen did not reveal any iliac or para-aortic lymph nodes and liver was normal. Biopsy of the non-healing ulcer over the palm revealed severe dysplastic changes in the epidermis overlying the growth and therefore it was reported as a likely “second primary squamous cell carcinoma”.

The patient underwent a left lower lobectomy with wedge resection of the lingular lobe and a wide excision of the malignant ulcer over the palm with a primary split skin graft (Fig. 4). The post-operative recovery was uneventful and was discharged on the 12th post-operative day. The final histopathological diagnosis was a grade III squamous cell carcinoma of lung with disease free margins. The histopathological diagnosis of the lesion on the palm was also squamous cell carcinoma with disease free margins.

Fig. 2
Fig. 2 : X-ray chest showing a peripheral lesion in the left lower lobe.


Fig. 3
Fig. 3 : CT scan of the chest showing the lesion in the left lower lobe.

 

DISCUSSION

A diagnosis of synchronous, multicentric, squamous cell carcinoma over secondary metastasis was more plausible primarily because the disease was diagnosed 10 years after the initial surgery and also because two diverse lesions - one on the lung and one on the palm were discovered and diagnosed simultaneously.

The nodular lesions on the palm raised a suspicion of “Palmo-plantar hyperkeratosis”.

This clinical entity is a diffuse keratoderma of the palm that has been reported to be associated with internal malignancies such as oesophageal carcinomas.[1-3] The presence of palmoplantar hyperkeratosis raised the possibility of an aetiologic factor for development of squamous cell carcinoma at diverse sites.

A medline search from 1973-1999 revealed 12 cases of pulmonary carcinomas associated with palmoplantar hyperkeratosis.[1-9] However, it is possible that this may be the first reported case in literature of a triple primary squamous cell carcinoma on a background of palmoplantar hyperkeratosis. As regards treatment, we opted for resectional surgery at two different sites. This was in view of the clinical observation that tumor biology of this lesion was likely to be favourable (development of a triple primary after a gap of 10 years) with a better prognosis. However, since the prognosis of carcinoma associated with palmoplantar hyperkeratosis is poor,[1] the patient will be followed up by frequent (three monthly) review examinations.

Fig. 4
Fig. 4 : Split skin graft over the left palm after wide

Distinctive skin syndromes correlating skin manifestations and gastrointestinal malignancies have been described.[10] It should alert the physician to the possibility of an associated internal cancer.


REFERENCES
  1. Nomori H, Horio H, Iga R, Fuyuno G, Kobayashi R, Morinaga S. Squamous cell carcinoma of the lung associated with palmar hyperkeratosis. Nippon Kyobu Shikkan Gakkai Zasshi 1996; 34 (1) : 76-9.
  2. Khanna SK, Agnone FA, Leibowitz AI, Raschke RA, Trehan M. Nonfamilial diffuse palmoplantar keratoderma associated bronchial carcinoma. J Am Acad Dermatol 1993; 28 (2) : 295-7.
  3. Howel-Evans W, McConnell RB, Clarke CA, et al. Carcinoma of the esophagus with keratosis palmaris et plantaris (tylosis) : a study of two families. Quart J Med New Ser 1958; 27 (107) : 413-29.
  4. Miki Y, Kawatsu T, Matsuda K, Machino H, Kubo K. Cutaneous and pulmonary cancers associated with bowens disease. J Am Acad Dermatol 1982; 6 (1) : 26-31.
  5. Powell F, Mackey JP. Bronchial carcinoma and hyperkeratosis palmaris et plantaris. Postgrade Med J 1981; 57 (663) : 57-9.
  6. Murata Y, Kurmano K, Tani M, Saito N, Kagotani K. Acquired diffuse keratoderma of the palms and soles with bronchial carcinoma and review of the literature. Arch Dermatol 1988; 124 (4) : 497-8.
  7. Costello MJ, Gibbs RC. Palms and soles in medicine. Springfield : Thomas. 1967; 123.
  8. Kerdel FA, MacDonald DM. Palmoplantar keratoderma associated with carcinoma of bronchus. Acta Derm Venereol 1982; 62 : 178-80.
  9. Parnell DD, Johnson SAM. Tylosis palmaris et plantaris : its occurrence with internal malignancy. Arch Dermatol 1969; 100 : 7-9.
  10. Sarnitz MH. Clinical significance of skin lesions in the diagnosis of gastrointestinal malignancies. Cutis 1977; 19 (5) : 649-52, 657-60.

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