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a 56-year - old male presented with complaints of pain that had gradually been getting worse in both hip regions for the past 4 years .
he was a known case of histiocytosis , diagnosed and treated 18 years back for skin lesions .
on general physical examination , the patient had deformities of bilateral hands-"opera glass hand " or " la main en lorgnette " deformity - and feet ( fig .
1 ) . evidence of old healed skin lesions was noticed : multiple violaceous nodules and papules were seen over the back , abdominal wall , thighs and forearm ( fig .
plain radiographs of the pelvis with bilateral hips revealed gross obliteration of the joint space , osteopenia , sclerosis and lytic lucencies both on the femoral and the acetabular side on both sides : all features that suggest bilateral hip arthritis ( fig .
3 ) . a radiograph of bilateral hands revealed osteopenia , lytic defects , multiple pathological fractures and dislocations of the phalanges and arthritic changes in the wrist , carpometacarpal , metacarpophalangeal and interphalangeal joints : all features that suggest arthritis mutilans with the classical " pencil - in - a - cup " appearance ( fig .
4 ) . a lateral view of the lumbar spine showed osteopenia and incidental pathological collapse of the l1 and l2 vertebrae ( fig .
tumour work - up was negative . in view of the patient 's significant pain and functional disabilities and his demand for a pain - free , stable and mobile hip ,
cemented tha was performed ( exeter femoral stem and acetabular cup , stryker , kalamazoo , mi , usa ) ( fig .
the defect was filled using an autograft harvested from the osteotomized head and neck of the femur .
this was supplemented by a medial wall mesh ( x - change , stryker ) for containment .
after an uneventful two weeks during the postoperative period , surgeons operated on the right hip .
similar to the left hip , cemented tha using exeter femoral stem and acetabular cup , stryker , was performed .
the patient was able to sit unassisted and ambulate with a walker without any discomfort .
this disease usually presents with a myriad of cutaneous manifestations and rapidly progressing destructive arthropathy .
the cutaneous manifestations of mrh are highly heterogeneous , commonly presenting as papules and nodules , which may be erythematous , violaceous or flesh - coloured .
they can be located anywhere on the body , but , are usually seen over the dorsum of the hands and fingers , periungual regions , pinna of the ears , forearm , elbows and knees.2 ) they may present in an isolated manner or in a confluent pattern , giving a cobblestone appearance .
almost any joint may be involved in arthropathy with predilection for the peripheral joints , especially the distal interphalangeal ( dip ) joints .
radiographically , there is evidence of symmetrical well - circumscribed marginal erosions at the joint surface , relatively mild osteopenia and uncalcified soft tissue nodules .
the close radiological differential diagnoses at this stage include gout and rheumatoid arthritis . in gout , there is asymmetric joint involvement . in rheumatoid arthritis ,
dip joints are rarely involved and there is marked periarticular demineralisation . in nearly 45% of the cases , the arthropathy takes a destructive course culminating in arthritis mutilans.2 ) radiologically , the marginal erosion coalesce to involve the entire articular surface leading to loss of articular cartilage , widening of the joint space and marked resorption of sub - chondral bone leading to " pencil - in - a - cup " appearance , foreshortening of the fingers , joint subluxations and/or pathological fractures .
clinically , there might be significant shortening of the fingers leading to a paw - like hand .
as a result of the shortening of the phalanges , the excess of overlying skin and soft tissue collapses and becomes transversely folded and telescope into one another .
hence , the following etymology has developed : " la main en lorgnette of marie and lri , " " opera glass hand , " " telescoping fingers , " " concertina hand , " and " accordion hand " deformity.34 ) the differential diagnosis for arthritis mutilans include : psoriatic arthritis , rheumatoid arthritis , systemic lupus erythematosus , systemic sclerosis , juvenile idiopathic arthritis , cutaneous t cell lymphoma and idiopathic arthritis mutilans.5 ) the clinical course of mrh is classic . characteristically , after a rapid initial progression followed by waxing and waning , the marked inflammation of the joints gradually abates and generally becomes inactive . following this ,
, mrh can also be associated with fever , weight loss and weakness . cardiac , pulmonary and hepatic involvement is rare . in 25% of cases ,
mrh is associated with malignancies of the skin , breast , gastrointestinal tract , genitourinary tract , leukemias and lymphomas .
other associations of mrh include diabetes , hypothyroidism , pregnancy and rheumatoid disorders.6 ) diagnosis of mrh solely depends upon a histopathological and immunohistochemical evaluation .
histiocytic multinucleated foreign body - type giant cells with a finely granulated , ground - glass eosinophilic cytoplasm , which is periodic acid - schiff - reactive and diastase - resistant .
immunohistochemistry shows positive staining with cd4 , cd45 , cd68 , human leukocyte antigen dr ( hla - dr ) , lysozyme , and alpha-1-antitrypsin and negative staining for cd20 , s100 , and factor xiiia.7 ) the hip joints are involved in 61% of the cases.2 ) symptomatic involvement of the hip joint occurred many years after the cutaneous lesions of mrh in our patient .
variable therapeutic success has been achieved with a combination of non - steroidal anti - inflammatory agents , steroids , chemotherapeutic drugs , immuno - modulators like tumour necrosis factor - alpha inhibitors and bisphosphonates , especially alendronate during the active stage of the disease.89 ) the aftermath of mrh arthropathy lead to a significant disability in our patient due to involvement of the bilateral hands and hips .
successful tha offered significant relief from pain and disability . during arthroplasty , the points of concern were osteopenia and acetabular bone defects . to address the issue of compromised bone quality , cemented
the acetabular wall defects were reconstructed with a morselised autograft harvested from the patient 's osteotomised femoral head and neck .
additionally , we had to use a stainless steel mesh on one side for containment .
therefore , before proceeding to tha in any patient with mrh , the surgeon must be well equipped with allografts and the armamentarium required for acetabular reconstruction including mesh , ring ( muller ) and cage ( burschneider or octopus ) .
since the patient was reluctant to undergo any surgical treatment for arthritis mutilans , we did not intervene .
otherwise , arthrodesis of the small joints of the hand can be considered a valuable option.10 ) a high degree of suspicion is essential for the diagnosis of mrh , which has a myriad of clinico - radiological symptoms .
multicentric reticulohistiocytosis ( mrh ) is a rare systemic disease , which commonly manifests as muco - cutaneous papulonodules and inflammatory erosive polyarthropathy . in this research 
 , we report the clinical manifestations and management of a rare case of mrh with destructive arthropathy of bilateral hip joints and arthritis mutilans presenting with characteristic deformities . disabling hip arthropathy that occurs secondary to mrh 
 can be successfully managed with bilateral total hip arthroplasty ( tha ) . 
 osteopenia and acetabular bone defects must be anticipated during tha . 
 this case is reported due to its rare occurrence and because little literature has been published regarding tha in such patients .